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SUBPERIOSTEAL PALATAL SOFT An experimental study in growing domestic cats

SUBPERIOSTALE PALATINALE WEEFSELEXPANSIE Een experimenteel onderzoek bij groeiende katten CIP-DATA KONINKLIJKE BIBLIOTHEEK, DEN HAAG

Van Damme, Philippus Anne

Subperiosteal palatal soft tissue expansion - An experimental study in growing domestic cats / Phihppus Anne Van Damme -[SI s η ] -111 Thesis Nijmegen - With ref -With summary in Dutch ISBN: 90-9009954-9 Subject headings tissue expansion / cleft palate / experimental study / reconstructive / growth & development

С Ph A Van Damme, Nijmegen, 1996 No part of this book may be reproduced in any form, by print, photoprint or any other means without written permission from the publisher Niets uit deze uitgave mag worden verveelvoudigd en/of openbaar gemaakt door middel van druk, fotokopie, microfilm of welke andere wijze dan ook, zonder voorafgaande toestemming van de uitgever SUBPERIOSTEAL PALATAL SOFT TISSUE EXPANSION

An experimental study in growing domestic cats

Een wetenschappelijke proeve op het gebied van de Medische Wetenschappen.

PROEFSCHRIFT

ter verkrijging van de graad van doctor aan de Katholieke Universiteit Nijmegen, volgens besluit van het College van Decanen in het openbaar te verdedigen op vrijdag 6 december 1996 des namiddags om 1.30 uur precies

door

Philippus Anne van Damme geboren op 4 juni 1953 te Yerseke

1996 Druk: Drukkerij Elinkwijk b.v., Utrecht Promotores Prof.dr. H.P.M. Freihofer Prof.dr. A.M. Kuijpers-Jagtman

Copromotor Dr. J.C. Maltha

Manuscriptcommissie Prof.dr. P.H.M. Spauwen Prof.dr. P. van den Broek Prof.dr. P. Egyedi (UU) Prof.dr. P.CM. van de Kerkhof Dr. J.H.A. van Rappard

Deze studie werd verricht op de afdeling Mond- en Kaakchirurgie van het Academisch Ziekenhuis Nijmegen, St. Radboud (hoofd: Prof.dr. H.P.M. Freihofer), op het Centraal Dierenlaboratorium (hoofd: Dr. J.P. Koopman, eertijds Prof.dr. W.J.L. van der Gulden) en bij de vakgroep Orthodontie en Orale Biologie (hoofd: Prof.dr. A.M. Kuijpers-Jagtman, eertijds Prof.dr. F.P.G.M, van der Linden) van de Katholieke Universiteit Nijmegen. Dit onderzoek was onderdeel van hoofdprogramma VI "Orale Aandoeningen en Steunweefselziekten". SUBPERIOSTEAL PALATAL SOFT TISSUE EXPANSION

An experimental study in growing domestic cats

A scientific proof in the field of Medical Sciences

THESIS

submitted to fulfil the requirements of the Ph D Degree in Medical Sciences of the University of Nijmegen, according to the decision of the Board of Deans to be defended in public on Friday December 6, 1996 at 1 30 ρ m

by

Philippus Anne van Damme born June 4, 1953 in Yerseke

1996 Printed in the Netherlands by Drukkerij Ehnkwijk b ν , Utrecht Paranimfen Dr С Jansen Drs BJC Rijnders

This research-project was materially supported by

CUI, Cox Uphoff International, Carpintería, Ca , U S A , Inamed В V , Breda, The Netherlands, J Laverman Dental Laboratory В V , Ewijk, The Netherlands

The realisation and publication of this thesis was financially supported by

Byk Nederland В V (Tramal"), Zwanenburg, The Netherlands, Cavex Holland В V (CA 37 Superior Pink"), Haarlem, The Netherlands, Hoechst Pharma (Ultracain"), Amsterdam, The Netherlands, ICI Farma (Corsodyl"), Ridderkerk, The Netherlands, Inamed В V (CUIR, IntraventTni), Breda, The Netherlands, Jansen-Cilag (Durogesic"), Tilburg, The Netherlands, Johnson & Johnson Medical В V (Ethicon Division, Vicryl"), Amersfoort, The Netherlands, Knoll В V (former Boots Pharmaceuticals В V , Hilversum) (Brufen" Bruis), Amsterdam, The Netherlands, Krijnen Medical В V (WurzburgR), Beesd, The Netherlands, J Laverman Dental Laboratory В V , Ewijk, The Netherlands, Nutricia Nederland В V (Nutrison"), Zoetermeer, The Netherlands, Roche Nederland В V (former Sarva-Syntex Nederland В V , Rijswijk) (Naprosyne"), Mijdrecht, The Netherlands, J Van Straten Medische Techniek, Nieuwegein, The Netherlands

Cover CUIR custom-made tissue expanders photographed by J A Η Meeuwissen (1990) To the memory of my parents To Akke To our children Maximilian, Rebecca and Damián

Contents

Chapter 1 General introduction 13

Chapter 2 Cranio-maxillofacial tissue expansion, experimentally based or clinically empiric? A review of the literature . . 29 Published in the Journal of Cranio-Maxillofacial Surgery (1992) 20: 61-69.

Chapter 3 Two-dimensional cephalometric analysis of the effects of subperiosteal palatal soft tissue expansion in growing cats 59 Accepted for publication by the Journal of Plastic and Reconstructive Surgery, 1996.

Chapter 4 Three-dimensional morphometric analysis of the effects of subperiosteal palatal soft tissue expansion in growing cats 83 International Journal of Oral and Maxillofacial Surgery (1996). In press.

Chapter 5 Histological analysis of the effects of palatal mucoperiosteal soft tissue expansion in growing cats ... 105 Submitted to International Journal of Oral and Maxillofacial Surgery, 1996.

Chapter 6 Palatal mucoperiosteal expansion as an adjunct to palatal fistula repair. Case report and review of the literature 135 Published in Cleft Palate-Craniofacial Journal (1996) 33: 255-257.

Chapter 7 General discussion 145

Chapter 8 Summary 161

Chapter 9 Samenvatting 167

Chapter 10 Survey of recent literature 173 Acknowledgements 189

Curriculum vitae 195

Publications 197 Chapter 1

General introduction

General introduction

1.1 Prevalence of cleft lip and palate

Cleft lip and palate (CLP) comprises a complex of congenital anomalies with a prevalence in the Netherlands of approximately 175/100.000 births/year (Van den Akker et ai, 1987; NVSCA, 1996). The mean prevalence in the whole of Europe is approximately 88/100.000 births (Cornel et al., 1992). There is a variability in the prevalence throughout Europe, with higher figures in the North-West and unexplained lower figures in the South and South-East. CLP may occur unilaterally or bilaterally, and the cleft can be complete or incomplete. Isolated clefts in the palate or the lip do occur. Different combinations of clefts in lip, alveolus and palate are the most common forms (Härtel et al., 1991). Clefts may be associated with known sequences and syndromes such as Pierre Robin sequence, orofacial clefting, Van der Woude, Stickler, Shprintzen, Saethre-Chotzen, Treacher Collins, and Apert syndromes, as well as with other anomalies (Gorlin et al., 1990, Morris et al. 1993). The aetiology and pathogenesis of the disorder are multifactorial. Genetic abnormalities and/or noxious stimuli during the first weeks of gestation, when early development of the face takes place (Pfeifer, 1991), are the main causes. Disturbances in the fusion or merging of nasal and maxillary processes may give rise to various types of clefts (Härtel et al., 1991). Inadequate apoptosis or programmed cell death might be involved in the development of clefts (Vermeij-Keers et al., 1983, 1990; Mangold, 1991).

1.2 Aspects of treatment related to maxillary growth

The interdisciplinary treatment of CLP in the Netherlands is basically organized in multidisciplinary cleft palate centres, which are generally attached to university hospitals. The Nijmegen Cleft Palate Centre is one of the major centres in the Netherlands. The treatment in Nijmegen starts, if need be, with presurgical orthopaedic appliances directly after birth, according to the principles of Hotz and Gnoinski (1979), followed by closure of the cleft of the lip at the age of 3-6 months. At the age of 12-18 months,

15 Chapter 1 the soft palate cleft is closed (Kuijpers-Jagtman and Borstlap, 1992). Closure of the hard palate and alveolar cleft is delayed until the age of 8 to 12 years and is performed before eruption of the permanent canines through the alveolar (Koberg, 1973; Egyedi, 1985; Witsenburg, 1985; Borstlap et al., 1990; Witsenburg and Freihofer, 1990; Freihofer et al., 1991; Freihofer and Kuijpers-Jagtman, 1993; Freihofer et al., 1993). In other centres, however, different schedules of surgical treatment are used, based on different philosophies and strategies. Early closure of the hard palate is claimed to be beneficial for speech development, and according to several authors it does not cause serious maxillary growth retardation (Pfeifer, 1991; Correa Normando et al., 1992). On the other hand it has been reported that any kind of lip and/or palatal surgery at an early age, gives rise to retardation and impairment of growth and development of the dentomaxillary complex especially in those cases in which the bone is denuded by stripping of the mucoperiosteum and in which healing is allowed by secondary intention (Herfert, 1954; Bardach and Eisbach, 1977; Eisbach et α/. ,1978; Bardach and Mooney, 1984; Wijdeveld et al., 1988; Pfeifer, 1991; Correa Normando et ai, 1992, Noverraz et ai, 1993; Kuijpers-Jagtman, 1995; Leenstra et al., 1995b). Antero-posterior and vertical growth disturbances of the maxillo­ facial structures are less pronounced in cases where the cleft is surgically untreated. In such cases, hardly any visual impact on the profile of the non- cleft side is apparent (Bishara et al., 1976; Derijcke et ai, 1990; Mars and Houston, 1990; Correa Normando et ai, 1992). Untreated cases, however, present different degrees of problems with nursing, feeding, speech, hearing, respiration, aesthetic appearance and psycho-social development, depending on the severity of the anomaly. Iatrogenic growth disturbances of the naso- and dentomaxillary complex frequently demand further surgical treatment in the so-called tertiary phase, after completion of growth. Problems in this phase can be: residual bone defects at the site of the alveolar cleft, residual oronasal communications or fistulae, combined with absolute tissue shortage, scarred, often poorly vascularized soft tissue, retruded position of the maxilla, and transverse collapse of the maxillary segments (Freihofer, 1981; Freihofer and Brouns, 1990; Kuijpers-Jagtman and Borstlap, 1992).

16 General introduction

1.3 Surgical closure of palatal defects

1.3.1 Primary palatal closure Many techniques have been proposed for primary closure of soft-palatal defects, varying from Langenbeck's (introduced in 1861) palatoplasty to the more recent V-Y push-back methods and the Furlow double opposing Z-plasty (Furlow, 1986; Spauwen et al. 1992, Heliövaara and Ranta, 1993). Other techniques are the veloplasties according to Veau (1931), Axhausen (1952), Schweckendiek (1955), Widmaier (1959), Bardach (1967) and Kriens (1970), and their different modifications (Perko, 1979; Pfeifer, 1991). The operations may be performed as one- or two-stage procedures, meaning either combined soft- and hard-palate closure at the age of 10-20 months in one session, or closure of the soft-palate, and closure of the hard-palate in two separate sessions. In Nijmegen, primary closure of soft-palatal defects is performed at the age of 12-18 months. Closure of the defect in the hard palate and eventually in the alveolar process is carried out at the age of 8-12 years as advocated by Boyne and Sands (1972). Earlier closure, especially primary closure with bone grafting can induce serious vertical and antero-posterior growth disturbances of the nasomaxillary complex (Johanson and Ohlsson, 1961; Robinson and Wood, 1969; Koberg, 1973; Boyne and Sands, 1976; Ross, 1987).

1.3.2 Closure of remaining or recurring palatal clefts and reconstruction of the alveolar process A large number of techniques are available for the closure of the hard palate at this stage (Perko, 1979; Freihofer et al., 1993). Some ten different techniques have been developed using loco-regional flaps (Millard, 1980). Recently, the buccal fat pad (Egyedi, 1977; Voorsmit and Fennis, 1992), the tadpole flap (Watson et ai, 1988) and the conchal cartilage graft (Matsuo et ai, 1991) have been advocated as single-stage closure procedures. However, these techniques do not always yield good results. In general, the closure becomes more difficult, and the techniques less reliable, after multiple previous attempts at closure of the fistula have been performed (Freihofer et

17 Chapter 1 al., 1993). The use of distant pedicled flaps has been proposed in cases where local tissue is not available and/or vascularity seems inadequate. Examples of such flaps are the buccal musculo-mucosal flap (Nakakita et al., 1990), the temporalis muscle and myofascial flaps (Furnas, 1987; Van der Wal and Mulder, 1992; Raffaini and Costa, 1994). A lingual flap, which is usually based anteriorly may be used to fill the defect with well-vascularized tissue (Guerrerosantos and Altamirano, 1966; Pigott et al., 1984; Coghlan et ai, 1989). However, this technique has the disadvantage of being a two-stage procedure. Also, it interferes temporarily with function, particularly speech and may require intermaxillary fixation. Finally, the texture and form of many of these flaps may be unfavourable for dental rehabilitation. Free distant flaps with microvascular anastomoses, e.g. the radial forearm free fascial flap (Batchelor and Palmer, 1990), the temporo-parietal fascial flap (Raffaini and Costa, 1994; Upton et al., 1994), and other flaps have similar disadvantages and in addition, they are often hazardous, time consuming, need frequent monitoring and cause visible scars at the donor site and the site of the anastomoses. A more promising technique to meet the problems of tissue shortage in cleft surgery, might be soft tissue expansion (TE).

1.4 Tissue expansion

TE is a technique by which a gain of soft tissue is realized by the temporary implantation of a silicone balloon-like device (tissue expander) and its serial inflation with saline. This expander is inserted underneath healthy soft tissue adjacent to a defect. By gradual distension of the expander, the overlying tissues are expanded by stretching, recruitment, and/or growth of tissue (Austad et al., 1986). The gained tissue, which possesses more or less the same characteristics as the lost or absent tissues, is then available for reconstruction purposes when the expander is removed. The absence of a donor-site defect and the increased vascularity of the gained tissue may be of value in cases where scarring has compromised vascularity,

18 General introduction as in CLP (Argenta and VanderKolk, 1987) Neumann introduced TE in 1957 After having been forgotten, the technique was re introduced by Radovan in 1976, for reconstructions after breast-cancer surgery (Radovan, 1984) Since then, many new uses in plastic and reconstructive surgery have been reported TE in head and neck surgery was first time used in 1981 (Argenta et al , 1981) Applications in the oral environment for edentulous alveolar ridge augmentation purposes, were first reported in 1986 (Bonomo, 1986, Lew et al , 1986, Van Rappard, 1988, Wittkampf, 1989) Nowadays, it is a common technique frequently used by various disciplines (Van Damme et al , 1992, Anger and Gemperli, 1993) TE might be an appropriate technique for facilitating closure of palatal defects in cleft palate disorders (Van Damme, 1990) Since neither clinical nor experimental data on the subject of intra-oral TE for cleft palate closure were available in the literature at that time, a pilot-study was performed on seven domestic cats and a protocol established De Mey et al (1990) and Abramo et al (1993) have since reported on the clinical use of soft tissue expanders for closure of palatal fistulae in young cleft patients, with good results However, in these case reports, no long-term follow-up results were given Also, two case reports on the reconstruction of facial clefts with TE were recently published (Toth et al , 1990, Moore et al , 1992) An experimental animal study of Moelleken et al (1990) showed reversible defects in cranial bone after TE in miniature swine Another experimental study in miniature swine (Schmelzeisen et al , 1994) demonstrated significant growth inhibition of facial in the surroundings of a tissue expander inserted subcutaneously on the masseter muscle fascia According to these findings, it was advocated that tissue expanders be inserted only when a strict indication exists (Schmelzelsen et al , 1994) An essential improvement of the surgical therapy, especially for palatal defects already treated with unsatisfactory results, could be achieved if the use of TE techniques in the palatal region would result in genuine soft tissue growth and improved vascularity, without causing permanent growth disturbances A general point of discussion is still whether TE results in a genuine

19 Chapter I gain of tissue or in stretching and local displacement of tissue, with subsequent retraction and redisplacement after removal of the expander. This holds for any kind of TE, but it is particularly important for the intra-oral palatal environment in growing children. When it interferes with growth and development of the dento-maxillofacial complex, the hypothetical benefits of this technique are questionable. Definitive answers could not be obtained from the literature (Van Damme et al., 1992), and that experience led to the present experimental study.

1.5 Study design

The present study was designed as a prospective, longitudinal animal experiment. The prerequisites for the choice of the animal species, were based on the general principles of availability and practicality, viz. size, growth pattern, dentition, and form of the palate of the animal, allowing for extrapolation of the results of the study to the clinic of CLP surgery. Despite the fact that at the Nijmegen University and in other institutions much experience in CLP studies has been obtained in beagle dogs (Maltha, 1982; Wijdeveld et al. 1988; Bardach et al., 1994a,b; Leenstra et al., 1995a,b), they were not chosen for this study because of their non-human­ like palatal vault. Rodents were excluded because they have no deciduous or permanent dentition, as humans have. Cats were chosen for reasons of availability, practicality, and good comparability with the dentition and palatal vault with the human anatomy (Freng, 1979). The specific questions in this study are: 1. Is TE of the palatal mucoperiosteum possible? 2. Does palatal TE induce a dividend or a loan of soft tissue? 3. What is the quantity and quality of the gained tissue? 4. What are the effects on the surrounding soft tissues and bone, as well as on growth and development of the dentomaxillary structures? 5. Are these effects and the technique influenced by the presence of scars from previous palatal surgery?

20 General introduction 1.6 Literature

ABRAMO AC, VIOLA JC, ANGELO AJ (1993) Intraoperative rapid expansion in cleft palate repair Plast Reconstr Surg 91 441-445 ANGER J, GEMPERLI R (1993) Abstract Book IV International Tissue Expansion Symposium, Säo Paulo, Brazil ARGENTA LC, VANDERKOLK CA (1987) Tissue expansion in craniofacial surgery Clin Plast Surg 14 143-153 ARGENTA LC, WATANABE MJ, GRABB WC, NEWMAN MH (1981) Soft tissue expanders in head and neck surgery a new method of reconstruction Plast Surg Forum 4 244 AUSTAD ED, THOMAS SB, PASYK KA (1986) Tissue expansion dividend or loan' Plast Reconstr Surg 78 63-67 AXHAUSEN G (1952) Technik und Ergebnisse der Spaltplastiken Die Gaumenplastik München Hanser, pp 95-171 BARDACH J (1967) Roxzxczepy Wargi Gornej ι Podniebienia Warsaw, Poland Pdnstwowy Zaklad Wydawnietw Lekarskich (Cited in Bardach J, Morris HL (1991) Two-flap palatoplasty surgery and speech In Craniofacial abnormalities and clefts of the lip, alveolus and palate Interdisciplinary teamwork, principles of treatment, long term results 4th Hamburg International Symposium, Stuttgart, Germany Thieme Verlag, pp 428-430 ) BARDACH J, EISBACH KJ (1977) The influence of primary unilateral cleft lip repair on facial growth I Lip pressure Cleft Palate J 14 88-97 BARDACH J, MOONEY MP (1984) The relationship between lip pressure following lip repair and craniofacial growth an experimental study in beagles Plast ReLonstr Surg 73 544-555 BARDACH J, KELLY KM, SALYER KE (1994a) Relationship between the sequence of lip and palate repair and maxillary growth an experimental study in beagles Plast Reconstr Surg 93 269-278 BARDACH J, KELLY KM, SALYER KE (1994b) The effects of lip repair with and without soft-tissue undermining and delayed palate repair on maxillary growth an experimental study in beagles Plast Reconstr Surg 94 343-351 BATCHELOR AG, PALMER JH (1990) A novel method of closing a palatal fistula the free fascial flap Br J Plast Surg 43 359-361 BISHARA SE, KRAUSE JC, OLIN WH, WESTON D, VAN NESS J, FELLING С (1976) Facial and dental relationships of individuals with unoperated clefts of the hp and/or palate Cleft Palate J 13 238-252

21 Chapter 1

BONOMO В (1986) Subperiosteal tissue expanders tor ridge augmentation Presented at the Annual Meeting Southern Calif Soc Oral & Maxillofac Surg

BORSTLAP WA, HEIDBUCHEL KLWM? FREIHOFER HPM, KUIJPERS JAGTMAN AM (1990) Early secondary bone grafting of alveolar cleft defects J Cranio Max-Fac Surg 18 201 205 BOYNE PJ, SANDS NR (1972) Secondary bone grafting of residual alveolar and palatal clefts J Oral Surg 30 87 92 BOYNE PJ, SANDS NR (1976) Combined orthodontic-surgical management of residual palato-alveolar cleft defects Am J Orthod 70 20-37 COGHLAN K, O'REGAN B, CARTER J (1989) Tongue flap repair ol oro-nasal fistulae in cleft palate patients J Cranio Max-Fac Surg 17 255-259 CORNEL MC SPREEN JA, MEIJER I, SPAUWEN PHM, DHAR BK, TEN KATE LP (1992) Some epidemiological data on oral clefts in the northern Netherlands, 1981-1988 J Cranio-Max-Fac Surg 20 147-152 (Erratum 20 363, 1992) CORREA NORMANDO AD, DA SILVA FILHO OG, CAPELOZZA FILHO L (1992) Influence of surgery on maxillary growth in clett lip and/or palate patients J Cranio-Max-Fac Surg 20 111 118 DE MEY A, MALEVEZ C, LEJOUR M (1990) Treatment of palatal fistula by expansion Br J Plast Surg 43 362-364 DERIJCKE A, KUIJPERS-JAGTMAN AM, LEKKAS C, HARDJOWASITO W, LATIEF В (1994) Dental arch dimensions in unoperated adult cleft-palate patients An analysis of 37 cases J Craniofac Genet Dev Biol 14 69-74 EGYEDI Ρ (1977) Utilization of the buccal fat pad for closure of oro-antral and/or oro-nasal communications J Max-Fac Surg 5 241-244 EGYEDI Ρ (1985) Timing of palatal closure J Max-Fac Surg 13 177-182 EISBACH KJ, BARDACH J, KLAUSNER EC (1978) The influence of primary unilateral clett lip repair on facial growth part II Direct cephalometry ot the skull Cleft Palate J 15 109-117 FREIHOFER HPM (1981) High midface osteotomies tor the correction of secondary skeletal deformities in patients with cleft lip and palate In Long term treatment in cleft lip and palate Kehrer B, Longo ΤΗ, Graf В, Bettex M (eds) Bern Huber Verlag, pp 217 220 FREIHOFER HPM, BORSTLAP WA, KUIJPERS-JAGTMAN AM, VOORSMIT RACA, VAN DAMME PHA, HEIDBUCHEL KLWM, BORSTLAP-ENGELS VMF (1993) Timing and transplant materials for closure of alveolar cletts J Cranio-Max-Fac Surg 21 143-148

22 General introduction

FREIHOFER HPM, BROUNS JJA (1990) Midfacial movements-a reappraisal Oral Maxillofac Surg Clin North Am 2 761-773 FREIHOFER HPM, KUIJPERS-JAGTMAN AM (1993) Timing of surgical- orthodontic treatment of cleft lip and palate patients In Consensus The timing of facial osteotomies in children and adolescents - State of art in Scandinavia and The Netherlands Bjork G, Freihofer HPM, Jonsson E (eds) Karlsham, Sweden Lagerblads Trycken AB, pp 68-77 FREIHOFER HPM, VAN DAMME PHA, KUIJPERS-JAGTMAN AM (1991) Early secondary osteotomy-stabilization ot the premaxilla in bilateral clefts J Cranio-Max-Fac Surg 19 2-6 FRENG A (1979) The restorative potential of double layered mucopenosteum A study on experimental mid-palatal clefts in the cat Scand J Plast Reconstr Surg 13 313-320 FURLOW LT (1986) Cleft palate repair by double opposing Z-plasty Plast Reconstr Surg 78 724-736 FURNAS DW (1987) Temporal osteocutaneous island flaps for complete reconstruction of cleft palate defects Scand J Plast Reconstr Surg 21 119-128 GORLIN RJ, COHEN MM, LEVIN LS (1990) Syndromes of the head and neck 3rd Ed New York Oxford University Press, pp 693-783 GUERREROSANTOS J, ALTAMIRANO JT (1966) The use of lingual flaps in repair of fistulas of the hard palate Plast Reconstr Surg 38 123-128 HARTEL J, KRIENS O, KUNDT G (1991) Incidence of cleft lip, alveolus and palate forms J Cranio Max-Fac Surg 19 144-146 HELIOVAARA A, RANTA R (1993) One-stage closure oí isolated cleft palate with Veau-Wardill-Kilner V-Y push back method or the Cronin modification Int J Oral Maxillofac Surg 22 267-271 HERFERT О (1954) Experimenteller Beitrag zur Frage der Schädigung des Oberkiefer-Wachstums durch vorzeitige Gaumenspaltoperation Dtsch Zahn- Mund Kieferheilk 20 369-381 HOTZ MM, GNOINSKI WM (1979) Effects of early maxillary orthopaedics in coordination with delayed surgery for cleft lip and palate J Max-Fac Surg 7 201-210 JOHANSON B, OHLSSON A (1961) Bone grafting and dental orthopedics in primary and secondary cases of cleft lip and palate Acta Chir Scand 122 112-124 KOBERG WR (1973) Present view on bone grafting in cleft palate J Max-Fac Surg 1 185-193

23 Chapter 1

KRIENS OB (1970) Fundamental anatomic findings for an intravelar veloplasty Cleft Palate J 7 27-36 KUIJPERS-JAGTMAN AM (1995) Kieferorthopadische Aspekte von Lippen-, Kiefer- und Gaumenspalten Inform Orthod Kieferorthop 27 345-356 KUIJPERS-JAGTMAN AM, BORSTLAP WA (1992) Orthodontische en kaakchirurgische aspecten van schisis, Ned Tijdschr Tandheelkd 99 450-454 LANGENBECK В (1861) Operation der angeborenen totalen Spaltung des harten Gaumens nach einer neuen Methode Dtsch Klin 24 LEW D, CLARK R, SHAHBAZIAN Τ (1986) Use of a soft tissue expander in alveolar ridge augmentation a preliminary report J Oral Maxillofac Surg 44 516-519 LEENSTRA TS, MALTHA JC, KUIJPERS-JAGTMAN AM, SPAUWEN PHM (1995a) healing in Beagle dogs after palatal repair without denudation of bone Cleft Palate-Craniofac J, 32 363-369 LEENSTRA TS, KUIJPERS-JAGTMAN AM, MALTHA JC, FREIHOFER HPM (1995b) Palatal surgery without denudation of bone favours dentoalveolar development in dogs Int J Oral Maxillofac Surg 24 440-444 MALTHA JC (1982) The process of tooth eruption in beagle dogs PhD Thesis, University of Nijmegen, The Netherlands MANGOLD U (1991) Differentiation of fusion zones in morphogenesis of the primary palate In Craniofacial abnormalities and clefts of the lip, alveolus and palate Interdisciplinary teamwork, principles of treatment, long term results Pfeifer G (ed) 4th Hamburg International Symposium, Stuttgart, Germany Thieme Verlag, pp 142-146 MARS M, HOUSTON WJB (1990) A preliminary study of facial growth and morphology in unoperated male unilateral cleft lip and palate subjects over 13 years of age Cleft Palate J 27 7-10 MATSUO K, KIYONO M, HIROSE Τ (1991) A simple technique for closure of a palatal fistula using a conchal cartilage graft Plast Reconstr Surg 88 334-337 MILLARD Jr DR (1980) Cleft craft The evolution of its surgery III Alveolar and palatal deformities Boston Little, Brown, pp 809-840 MOELLEKEN BRW, MATHES SJ, CANN CE, SIMMONS DJ, GHAFOORI G (1990) Long-term effects of tissue expansion on cranial and skeletal bone development in neonatal miniature swine clinical findings and histomorphometric correlates Plast Reconstr Surg 86 825-834 MOORE MH, TROTT JA, DAVID DJ (1992) Soft tissue expansion in the management of the rare craniofacial clefts Br J Plast Surg 45 155-159

24 General introduction

MORRIS HL, BARDACH J, ARDINGER H, JONES D, KELLY KM, OLIN WH, WHEELER J (1993) Multidisciplinary treatment results for patients with isolated cleft palate Plast Reconstr Surg 92 842-851 NAKAKITA N, MAEDA K, ANDO S, OJIMI H, UTSUGI R (1990) Use ot a buccal musculomucosal flap to close palatal fistulae after cleft palate repair Br J Plast Surg 43 452-456 NEUMANN CG (1957) The expansion of an area of by progressive distention ot a subcutaneous balloon Plast Reconstr Surg 19 124-130 NOVERRAZ AEM, KUIJPERS-JAGTMAN AM, MARS M, VAN 'T HOF MA (1993) Timing of hard palate closure and dental arch relationships in unilateral cleft lip and palate patients A mixed - longitudinal study Cleft Palate- Cramofac J 30 391-396 NVSCA (1996) Schisis inventarisatie Nieuwe schisis baby's in Nederland NVSCA (Nederlandse Vereniging voor Schisis en Craniofaciale Afwijkingen) PERKO MA (1979) Two-stage closure of cleft palate J Max-Fac Surg 7 76-80 PFEIFER G (1991) Craniofacial abnormalities and clefts of the lip, alveolus and palate Interdisciplinary teamwork, principles of treatment, long term results 4th Hamburg International Symposium, Stuttgart, Germany Thieme Verlag PIGOTT RW, RIEGER FW, MOODIE AF (1984) Tongue flap repair of cleft palate fistulae Br J Plast Surg 37 285-293 RADOVAN С (1976) Adjacent flap development using expandable Silastic implant Presented at the annual meeting of the Am Soc Plast Reconstr Surg, Boston, Mass, USA RADOVAN С (1984) Tissue expansion in soft-tissue reconstruction Plast Reconstr Surg 74 482-490 RAFFAINI M, COSTA Ρ (1994) The temporoparietal fascial flap in reconstruction of the cranio-maxillofacial area J Cramo-Max-Fac Surg 22 261-267 ROBINSON F, WOOD В (1969) Primary bone grafting in the treatment of cleft lip and palate with special reference to alveolar collapse Br J Plast Surg 22 336-342 ROSS RB (1987) Treatment variables affecting facial growth in complete unilateral cleft hp and palate Cleft Palate J 24 5-77 SCHMELZEISEN R, SCHWIPPER V, TILKORN H, BECKER H, UBERMUTH Τ (1994) Changes in growth of the facial skeleton following implantation of tissue expanders J Cramo-Max-Fac Surg 22 (Suppl 1) 81 SCHWECKENDIEK Η (1955) Zur zweiphasigen Gaumenspaltenoperation bei primärem Velumverschluss Fortschr Kiefer Gesichtschir 1 73-76

25 Chapter 1

SPAUWEN PHM, GOORHUIS-BROUWER SM, SCHUTTE HK (1992) Cleft palate repair Furlow versus Von Langenbeck J Сгапю-Мах-Fac Surg 20 18-20 TOTH BA, GLAFKIDES MC, WANDEL A (1990) The role of tissue expansion in the treatment of atypical facial clefting Plast Reconstr Surg 86 119-122 UPTON J, FERRARO N, HEALY G, KHOURI R, MERRELL С (1994) The use of prefabricated fascial flaps for lining of the oral and nasal cavities Plast Reconstr Surg 94 573-579 VAN DAMME PHA (1990) Subperiosteal palatal tissue expansion an animal experiment in domestic cats Neth J Surg 42 164-165 VAN DAMME PHA, HEIDBUCHEL KLWM, KUIJPERS-JAGTMAN AM, MALTHA JC, FREIHOFER HPM (1992) Cranio-maxillo-facial tissue expansion, experimentally based or clinically empiric9 A review of the literature J Сгапю-Мах-Fac Surg 20 61-69 VAN DEN AKKER AMEA, HOEKSMA JB, PRAHL-ANDERSEN В (1987) Sclnsis in Nederland de vraag naar behandeling van patiënten met schisis Ned Tijdschr Tandheelkd 94 520-525 VAN DER WAL KGH, MULDER JW (1992) The temporal muscle flap for closure of large palatal defects in CLP patients Int J Oral Maxillofac Surg 21 3-5 VAN RAPPARD JHA (1988) Controlled tissue-expansion in reconstructive surgery PhD Thesis, University of Groningen, The Netherlands, pp 17-20, 60-76, 147- 156 VEAU V (1931) Division palatine, anatomie, chirurgie phonétique Pans Masson VERMEIJ-KEERS C, MAZZOLA RF, VAN DER MEULEN JC, STRICKER M (1983) Cerebro-craniofdcial and craniofacial malformations an embryological analysis Cleft Palate J 20 128-145 VERMEIJ-KEERS С (1990) Craniofacial embryology and morphogenesis normal and abnormal In Craniofacial malformations Strieker M, Van der Meulen JC, Raphael B, Mazzola R (eds) Edinburgh Churchill Livingstone, pp 27-60 VOORSMIT RACA, FENNIS JPM (1992) The buccal fat pad for closure of oro­ nasal communications in cleft patients J Сгапю-Мах-Fac Surg 20 (Suppl 1) 71 WATSON JD, REID CD, PIGOTT RW (1988) The "tadpole flap" - its role in closure of palatal fistulae Br J Plast Surg 41 485-487 WIDMAIER W (1959) Ein neues Verfahren zum Verschluss der Gaumenspalten Chirurg 30 274-278

26 General introduction

WIJDEVELD MGMM, GRUPPING EM, KUIJPERS-JAGTMAN AM, MALTHA JC (1988) Growth of the maxilla after soft tissue palatal surgery at different ages on beagle dogs A longitudinal radiographic study J Oral Maxillofac Surg 48 204-209 WITSENBURG В (1985) The reconstruction of anterior residual bone defects in patients with cleft lip, alveolus and palate J Max-Fac Surg 13 197-208 WITSENBURG B, FREIHOFER HPM (1990) Autogenous rib graft for reconstruction of alveolar bone defects in cleft patients Long term follow-up results J Cranio-Max-Fac Surg 18 55-62 WITTKAMPF ARM (1989) Short-term experience with the subperiosteal tissue expander in reconstruction of the mandibular alveolar ridge J Oral Maxillofac Surg 47 469-474

27

Chapter 2

Cranio-maxillofacial tissue expansion, experimentally based or clinically empiric? A review of the literature

Philip A. Van Damme Kiki L.W.M. Heidbüchel Anne-Marie Kuijpers-Jagtman Jaap С. Maltha Hans Peter M. Freihofer

Published in the Journal of Cranio-Maxillofacial Surgery (1992) 20: 61-69. Reprinted with permission. Parts of this chapter were presented at the 3rd International Tissue Expansion Symposium in Sapporo, Japan, October 1991 and published in Tissue Expansion Symposium, OCITES, Ohura T, Sugihara Τ (eds), Sando, Japan, Chapt. New application in aesthetic surgery (1991) 154-160.

Tissue expansion, experimentally based or clinically empiric

2.1 Summary

The literature over 10 years covering soft tissue expansion (TE) in cranio- maxillofacial surgery is reviewed. Since 1981, an evolving set of indications for the application of TE techniques have been published. However, it seems to be based much more on clinical experience (empiricism) than on the results of thorough experimental research. The direction of future research should be aimed at the effects on bone, cartilage and mucosa at the microscopic level, and at the influence on growth and development of cranio- maxillofacial structures.

31 Chapter 2

2.2 Introduction

The concept of TE in surgery, especially facial reconstructive surgery, was first introduced by Neumann (1957) It took about 25 years before a renaissance of the technique in head and neck surgery occurred (Argenta et al , 1981, 1983) Nowadays, all disciplines concerned use the technique of TE for an ever increasing variety of indications in the head, face and neck region All this kind of surgery, however, seems to have been developed, based much more on clinical experiences than on the results of experimental research Fundamental scientific questions still seem to remain unanswered (Pearl, 1984, Cohen and Dunn, 1988, Van Rappard, 1988, Schmidt et al , 1991) The ultimate general purpose of TE is to create tissue of normal texture, colour, thickness etc , with preservation of sensibility, in order to cover a certain defect without creating a donor site defect In cramo-maxillofacial surgery, the following specific reasons for TE can be distinguished firstly, to increase the amount of specific soft tissue available scalp, forehead, periorbital tissue, nose, face and neck and/or secondly, to improve the blood supply to a particular region with decreased vascularity, with the aim of improving the viability and long term stability of bone or cartilaginous grafts (Argenta and VanderKolk, 1987) The purpose of this paper is to present the state of the art in the field of TE in cramo-maxillofacial surgery The indica­ tions, results and complications of clinical applications are mentioned and related to experimental data Finally, unanswered questions are indicated and the direction of future research required is pointed out

2.3 Clinical applications

2 3 1 Scalp TE of the scalp has been performed since 1982 Numerous authors have published clinical reports on successful correction of baldness and traumatic defects (Argenta, 1984, Manders et al , 1984a, 1987a, Nordstrom, 1984, 1988a,b, Radovan, 1984, Nordstrom and Devine, 1985, Kabaker et al ,

32 Tissue expansion, experimentally based or clinically empiric

1986, 1990; Leonard and Small, 1986; Adson et al., 1987, 1988, Anderson, 1987, Fudern and Orgel, 1988; Masser, 1988; Moscona et ai, 1989; Penoff, 1990) In case reports, successful in aplasia cutis congenita and separation of craniopagus twins has been mentioned (Shively et al , 1985; Snyder, 1985, Argenta and Dingman, 1986). Even microvascular transplantation of expanded free scalp flaps between identical twins has been reported (Valaun et al , 1990). The use of TE in the treatment of post- contractures and post-burn alopecia has been presented in several case reports (Chang and Jin, 1986; Leighton et al., 1986a, Leonard and Small, 1986; Matthews and Missotten, 1986, Marks et al., 1987b; Buhrer et al , 1988; Horibe et al., 1989, Neligan and Peters, 1989, Cooper and Brown, 1990: Kasai et al., 1991) It was adjudged to be a valuable, simple, safe and reliable technique, allowing reconstruction of defects previously beyond adequate surgical repair.

2 3.2 Forehead and nose In hypertelorism, facial clefting and other congenital deformities a low hair line can be found. For expansion of the forehead and correction of the hairline, an expander was placed beneath the frontalis muscle, usually through an incision in the scalp (Argenta and VanderKolk, 1987; Hems and Godfrey, 1990). TE has also been used to create a larger forehead flap for reconstruction of the nose and to cover larger defects and the donor site defect in the meantime (Argenta and VanderKolk, 1987; Cook et al., 1987; Marchac and Pugash, 1987; Adamson, 1988; Bolton et ai, 1988; Gouet et al., 1989; Kroll, 1989; Toth et al., 1990; Cole et al., 1991). Successful reconstruction of the nose with expansion of the skin of the nose itself was reported by Hirshowitz et al. (1986), Argenta and VanderKolk (1987), Muenker (1988b), and Sasaki (1987, 1988)

2.3.3 External ear For subtotal reconstruction of the ear, after partial traumatic amputation, an area of skin was expanded to obtain cutaneous cover for a cartilaginous graft (Neumann, 1957). He described a rubber balloon which was gradually distended by insufflation with air This was the first report of successful TE.

33 Chapter 2

Ever since, several case reports have been published on auricle reconstruction with the aid of TE (O'Neal et al., 1984; Mutimer and Mulliken, 1988; Nordstrom et al., 1988c; Quaba, 1988; Hata et al., 1989; Bauer, 1990; Sasaki, 1989, 1990; Tanino and Miyasaka, 1990).

2.3.4 Eyelid and orbit The use of TE in eyelid surgery to achieve correction of cicatricial ectropion and ocular discomfort, without the need for skin grafting, was described by Victor and Hurwitz (1984), Victor et al. (1986) and Antonyshyn et al. (1988b). The possible role of TE in anophthalmic orbits has been pointed out by Lo et al. (1990) and Berry (1991).

2.3.5 Cheek and lip For central face reconstructions, cheek rotation flaps were pre-expanded (Argenta et al., 1983; Neligan and Peters, 1989; Wilmshurst and Sharpe, 1990; Iwahira and Maruyama, 1991). Reconstruction of the lip was reported by Goldstein (1984, 1990) and Hirshowitz et al. (1986). Orofacial defects as a result of rubella were reconstructed with the use of TE of the cheek by Lew et al. (1989). Intra-operative TE for rhytidectomy was proposed by Man (1989, 1990a,b) but the indications and results were questioned by several authors (Hirshowitz, 1989; Beraka, 1990; Brar, 1990; Ellenbogen and Collini, 1990; Luce, 1990; Manders et al., 1990; Olshansky, 1990; Schubert and Shons, 1990).

2.3.6 Maxilla The successful treatment of a palatal fistula by TE of the palatal mucosa in cleft palate patients was published as a case report by De Mey et al. (1990).

2.3.7 Mandible Mandibular reconstruction using TE has been reported several times (Argenta et ai, 1983; Argenta, 1984; Argenta and Dingman, 1986). It has been carried out in patients who sustained gunshot to the face and other avulsive of the mandible (Lew et ai, 1988c). TE of the gingiva was frequently used to create a subperiosteal envelope prior to augmentation of

34 Tissue expansion, experimentally based or clinically empiric the alveolar ridge with hydroxylapatite (Bonomo, 1986). Good results have been reported in preliminary reports and several case reports (Lew el al., 1986; Bock and Rose, 1987; Wittkampf, 1989, 1990; Quayle et al, 1990; Schwartz and Relie, 1990). Modifications of the technique, such as an open procedure and a successful case of reconstruction of an atrophic mandible with rib, after subperiosteal expansion, have been published (Lew et al, 1988a,b).

2.3.8 Neck In patients with burn scars of the face and neck, expansion has been used for the reconstruction of the lateral and lower third of the face (Argenta, 1984; Radovan, 1984; Thornton et al., 1987; Kessler et ai, 1989). TE was also advocated for correction of webbing of the neck (Niranjan, 1989; Miller et al., 1990; Thomson et ai, 1990) and cervical clefts (Breton and Freidel, 1989).

2.4 Clinical complications

Reported complications associated with TE were: haemorrhage, haematoma, seroma, infections, folding, deflation or leakage of the expander, exposure because of dehiscence of the incision or necrosis of the overlying tissue, resorption of the underlying bone, pain or discomfort, neurapraxia, tempora­ ry disfigurement, and psychological problems (Manders et al., 1984b; Argenta et al., 1985; Shively et ai, 1985; Snyder, 1985; Argenta and VanderKolk, 1987; Ashall and Quaba, 1987; Austad, 1987b, 1988a,b; Dickson, 1987; Gibbons, 1987; Hemmer et al, 1987; McKinney et al., 1987; Van Rappard et ai, 1987; Antonyshyn et al., 1988a,b; Donelan, 1988; Fenton, 1988; Fudern and Orgel, 1988; Neale et ai, 1988; Ehrenfeld et ai, 1989; Nordstrom, 1988c; Nordström et ai, 1988a,b; Van Rappard, 1988; Paletta et al, 1989; Ricbourg et al., 1989; Baker and Swanson, 1990a,b; Greenbaum and Greenbaum, 1990; Kawashima et al, 1990; Masser, 1990; Penoff, 1990; Cole et al, 1991). Complication rates vary with surgeon, time and topographical site. Most

35 Chapter 2 authors reported high complication rates early in their experience, with subsequent decrease (Austad, 1987b; Van Beek and Adson, 1987; Masser, 1990). The increase in use for complicated situations, on the other hand, is associated with an increased incidence of complications. Clinical complication rates vary from 0% (Argenta et al., 1983) to 69% (Antonyshyn et al., 1988a) with a mean of 25% (Zoltie et ai, 1990). The highest incidence of complications occurred in the head and neck (69%) and forehead regions (50%). Complications in the eyelid region (33%) and scalp (17%) were less common (Antonyshyn et αι., 1988a; Mayer et al., 1989). Paediatric patients had no higher complication rates with the use of scalp expansion than adults (31%) (Buhrer et al., 1988; Donelan, 1988; Nealertfl/., 1988). In 'rapid' expansion (Masson et al., 1989; Mustoe et ai, 1989) complication rates are considerably higher than in 'slow' expansion (Marks et al., 1985, 1986, 1987a; Lawrence, 1989). Sasaki (1987) judged the complication rate of intra operative sustained limited expansion (ISLE) to be negligible in the area of the head and neck. To prevent the high incidence of complications during TE related to overfilling, transcutaneous measuring of oxygen levels, or expander pressures or local perfusion was suggested in complicated situations (Hallock and Rice, 1986; Hallock, 1987b, 1988a,b; Piedla et al., 1988a,b; Gamier et al., 1989; Ricbourg et al., 1989; Canady et al., 1990; Pietilä, 1990; Esposito et ai, 1991). The injection port was reported to leak if intraluminal pressure exceeded 250 mmHg, especially if needles thicker than 23-25 gauge were used to fill the expander (Van Beek and Adson, 1988). Wound dehiscence was related to the healing time prior to expansion. Waiting 10-14 days after placement of the expander before starting expansion was reported to be beneficial (Marks et al., 1987a; Baux et al., 1988; Marks and Argenta, 1988). In some clinical cases, Austad (1987b) noticed partial necrosis of expanded head and neck flaps after expander removal and flap transposition, probably because of epinephrine in the local anaesthetic. Because of post- expansion contraction, expanded tissue flaps could decrease 56% or more

36 Tissue expansion, experimentally based or clinically empiric when raised and inset (VanderKolk et ai, 1987; Donelan, 1988). After some weeks or months, scar contracture seemed to diminish the tissue gain and more expansion than first realized was needed to achieve a sufficient amount of skin for reconstruction (Nordström et al., 1988a,с). Excessive widening of scars, particularly in the scalp, after ТЕ has been reported (Manders et ai, 1984a). However, in the scalp area, even without expansion, flap surface-areas were said to decrease to about two thirds to one half of their original magnitude in 8-12 weeks postoperatively (Nordstrom, 1984). On the other hand, long term results of scalp reconstruction after ISLE indicated not only retention of hair density in advanced flaps, but also the absence of scar widening (Muenker, 1988a; Sasaki, 1988). Some degree of late vertical tissue contracture in reconstruction of the nose was mentioned. It was suggested that this could be counteracted by means of adequate cartilage or bone support (Bolton et al., 1988; Saxby, 1990; Toth et al., 1990). Bone erosion has been reported several times (Hemmer et al., 1987; Fudern and Orgel, 1988; Paletta et ai, 1989; Penoff, 1990), however, less temporary deformity was noted in younger patients and no bone or sutural distortion could be demonstrated in infants with open sutures (Bauer and Vicari, 1988; Bauer et ai, 1990). Facial nerve neurapraxia was mentioned by Argenta et al. (1983). Although, they stated that care should be taken to avoid the facial nerve during surgical placement of the expander, they have not experienced any difficulty with underlying vasculature or nerve function during the process of expansion. According to Antonyshyn et al. (1988ab), clinical signs of nerve compression were rare.

2.5 Experimental studies

Although numerous reports can be found in the literature on TE it has to be realised that almost all known experimental human and animal studies deal with skin expansion, other than skin of the head, face and neck region. The effects of TE are distinguished for the different tissue layers and structures.

37 Chapter 2

2 5 1 Surface area increase Surface-area increase by TE is based on growth, stretching and recruitment of tissue, or a combination of these Radovan (1984) stated that overlying tissue doubled in size In human scalp expansion Nordstrom and Devine (1985) observed 39% increase in the transverse and 27% in the sagittal direction VanderKolk et al (1987) observed a 45% and 30% increase in transverse and sagittal directions respectively The area increase prior to elevation was 63%, of the postoperative area increase, approximately 30% and three months post-inset only about 23% had remained Others found an increase in area prior to elevation of the flap of only 12 5-22 5% The central portion over the expander increased 50 100%, the periphery less than 25% (Brobmann and Huber, 1985, Leighton et al , 1986b) Bartell and Mustoe (1989) showed an increase in area in response to traditional expansion of approximately 25%, McCann et al (1988) 50% and Wood and McMahon (1989) on the other hand found an increase in skin area up to 110% According to Van Rappard et al (1988b) in vivo only about 35% of the mathematically expected increase in surface area takes place A clear difference between surface area gain for different shapes of tissue expanders was observed by Brobmann and Huber (1985), Muenker (1988a) and Van Rappard et al (1988c) According to Van Rappard (1988) the expander base area chosen should be 2 5 times that of the defect to be closed However, Shively (1988), and Duits et al (1989) were of the opinion that area gain is only a function of the height of the expander and not of its base diameter When a defect was irregular, one large expander seemed to be more efficient than several smaller ones (Brobmann and Huber, 1985) A preoperative modelling method for individualized expansion and optimizing expander choice, flap size and shape has recently been advocated (Guero and Lacroix, 1991)

2 5 2 Capsule Pasyk et al (1987) and Saouma et al (1989) found a capsule surrounding silicone tissue expanders in humans, with a varying thickness from approximately 0 3-12 mm During rapid expansion a thinner capsule was produced than during 'slow' expansion (Marks et al , 1987a) In the first

38 Tissue expansion, experimentally based or clinically empiric month, the expander was enclosed by a thin capsule, composed of a layer of fibrous connective tissue with a few collagen fibres parallel to the surface of the expander (Samayoa et al., 1990). The capsule reached its maximal thickness after 2-2.5 months of expansion (Pasyk et ai, 1988). Histologically, four layers were distinguished in the capsule: inner, central, transitional and outer. The capsule was well vascularized, with a vascular outer layer and blood vessels in the transitional layer also (Cherry et al., 1983; Antonyshyn et al., 1988b). Increased vascularity was reported by Cherry et al. (1983) and Sasaki and Pang (1984). Collagen synthesis and inflammation appeared to be prominent around the expander (Leighton et ai, 1988a,b). Foreign body giant cells were occasionally found as well as calcification and bone formation (Pasyk et al., 1988; LaTrenta et al., 1988).

2.5.3 Epidermis Experimental studies revealed that the epidermis became thicker after TE (Austad et al., 1982, 1986; Pasyk et al., 1982, 1988; Austad, 1988c; VanderKolk et al., 1988), or at least not thinner (Cherry et al., 1983; Grossman et al., 1984; Brobmann and Huber, 1985). The mean thickness increased by approximately 0.03 mm (37.5% of the original thickness) (Pasyk et ai, 1988). This thickening was attributed to a 3-fold increase in epidermal mitotic activity (Austad et al., 1986). According to Pasyk et al. (1987) there was no correlation between thickness of the epidermis and time of expansion, size and volume of the inflated expander, location of insertion of the expander and age of the patient. VanderKolk et al. (1987, 1988) and Johnson et al. (1988) not only noticed thickening of the epidermis in expanded skin, but even in non-expanded, sham-operated skin. Epidermal cell mitotic increase is a well known response to any manipulation of the skin, such as shaving, 'prepping' or surgery (Francis and Marks, 1977).

2.5.4 Appendages Skin structures such as hair follicles, sweat and sebaceous glands, showed some compression, but they did not demonstrate degeneration or histological changes during TE (Cherry et ai, 1983; Austad et al., 1986; Pasyk et al.,

39 Chapter 2

1987, 1988; Van Rappard et al., 1988a). However, a redistribution of the hair follicles, increasing the distance between them by 20-25% was found (Argenta and VanderKolk, 1987).

2.5.5 Dermis Experimental studies showed a statistically significant thinning of the dermis after TE (Austad et al, 1982; Pasyk et al, 1982; Cherry et al, 1983; Brobmann and Huber, 1985; Leighton et al, 1988a). The mean thickness decreased by approximately 0.5 mm (25%) (Pasyk et al., 1988). The thinning of the dermis during expansion had no adverse effects on the blood supply (Cherry et al, 1983). Capillaries in the papillary dermis and the small vessels in the deep corium were dilated (Pasyk et al, 1987). Blood flow in the skin covering the expander however, was lower than in adjacent skin (Min et al, 1988; Timmenga et al, 1989, 1990). This is in contrast to other studies in which, in fact, even the reverse was found: dermal layers thickened markedly in expanded skin after 5 weeks of expansion (Lee et al., 1985; VanderKolk et al., 1988; Ricciardelli et al, 1989). The expanded dermis contained larger bundles of compacted collagen fibres, as well as thin collagen fibres, flattened fibroblasts, few myofibroblasts, and showed changes in collagen arrangement and distribution (Pasyk et al., 1982; Knight et al, 1990). Histological studies of elastic fibres have varied from finding no evidence of microfragmentation (Sasaki, 1987) to finding some fragmentation and disintegration of fibres (Pasyk et al, 1987). No signs of inflammatory or foreign body type responses were found (Sasaki and Pang, 1984).

2.5.6 Subcutaneous tissue The subcutaneous tissue showed a significant thinning following expansion. The mean thickness diminished approximately 1.0 mm (50%) (Pasyk et al, 1988).

2.5.7 Arteries, veins, nerves Leighton et al. (1988a,b) found a significant increase in the number of dermal blood vessels, with the greatest increase in the dermal papillae and a

40 Tissue expansion, experimentally based or clinically empiric smaller increase present in the reticular dermal layer The capillaries were not only more numerous but also dilated. Neovascularization was evidenced by Zarebski and Breton (1988). Elongation of arteries and veins was reported by Stark et al (1987), Austad and Pasyk (1987) and Hong et al. (1987). The mean elongation was about 80%, with a maximum gain of 140%, without reduction in vessel wall diameter or loss of intimai integrity. The effect of TE on peripheral nerves was studied by Milner (1989). The increase in length of the nerve was proportional to the degree of expansion All nerves showed a decrease in conduction velocity. Normal conduction velocity after TE, however, was reported by Wood and McMahon (1989) and Wood et al. (1991) Van Beek and Adson (1988) found that maximum elongation of nerve segments occurred over the centre of the expander, with no evidence of stretch or 'loan' from segments further from the expander Manders et al (1987b) reported on expansion of the human median nerve and the sciatic nerve in mongrel dogs, in which even 20 mm of length could be gained over a period of 3 months.

25 8 Fat Subcutaneous fat undergoing expansion displayed serious atrophy. The adipocytes became flattened and smaller, and they finally disappeared. Fibrous tissue, surrounding the adipocytes, was formed or even replaced the fat lobules (Pasyk et al., 1987). The adipose layer became 30-50% thinner (Leighton et al , 1988a).

2 5 9 Striated muscle The panniculus carnosus muscle (a cutaneous muscle only present in rodents) became thinner, especially during the first 2 weeks after implantation of the expander (Pasyk et al, 1982, Lee et al, 1985; Mustoe et al, 1989). Skeletal muscles under the expander also underwent some atrophy. Macroscopically, the tissue expander caused a reversible corresponding depression in the muscle (Radovan, 1984, Vistnes, 1984). Microscopically, atrophic muscle cells were surrounded by an increased number of collagen fibres or in some areas were totally replaced by fibrous tissues (Pasyk et al., 1988)

41 Chapter 2

2.5.10 Bone In a recent animal study, the effects of TE on cranial bone were evaluated and characterized by reversible defects in the underlying bone and remodelling with osteoclastic resorption and periosteal reaction with deposition of bone (Moelleken et al., 1990). Detrimental effects on palatal bone by subperiosteal TE have been mentioned in preliminary reports since 1989 (Van Damme, 1990; Van Damme et al., 1991).

2.6 Discussion

An important basic question is whether there is a 'dividend' or 'loan' (Austad et al., 1986) of tissue during expansion, since this has implications on the long term results of the reconstructions carried out with expanded tissue. When only 'loan' of tissue is achieved, the tissue is likely to contract and the reconstruction will deform. Conflicting results are found with respect to real tissue gain (Austad, 1987a; Mackay et ai, 1990). Surface-area-increase is reported over a wide range from 12,5 up to 110%, and can be derived from growth, stretching or recruitment of tissue. Even if genuine tissue growth increases the thickness of the epidermis, in our opinion, it is so little (approximately 0.03 mm) compared to the reported loss of dermis (about 0.5 mm), subcutaneous tissue (circa 1.0 mm) and fat (30-50%), that it is almost negligible. On the other hand there is a gain of tissue in the form of a fibrous capsule (about 0.3-1.2 mm). It is still questionable whether the capsule around the tissue expander has to be considered to be an advantage or a disadvantage. The reported increased vascularity in the capsule seems to be of value in cases of scarification and radiation of tissue (Saxby, 1988; Stark and Jaeger, 1989). It may also play an important role in improvement of the viability of bone or cartilaginous grafts. However, myofibroblasts located in the capsule, may be responsible for part of the contraction. Some authors (Horibe et al., 1989; Morris et ai, 1989) therefore advise capsulotomy or capsulectomy during the preparation, before insetting of the flap, to prevent or diminish the contraction, but with the risk of compromising the vascularity as well (Manders and Saggers, 1989). It is a well known fact that the body

42 Tissue expansion experimentally based or clinically empiric tries to encapsulate any 'foreign body' and to keep the 'exposure surface' as small as possible (Vistnes et al , 1978, Picha and Goldstein, 1991) TE appears to stimulate collagen formation The increase probably results from a combination of stretch and thus stress (evidenced by reorientation) along with an increase in the cellular activity resulting in an increase m extracellular components Almost all experimental studies deal with skin and subcutaneous soft tissues of animal or human origin Though only a few studies have been conducted into the effects of TE on other important tissues in cranio maxillofacial surgery e g mucosa, mucopenosteum, cartilage and bone Only Austad et al (1982) mentioned the probability that mucosa will increase in surface area when mechanically stimulated from beneath by an enlarging mass, but so far, to our knowledge, no histological data have been published on this subject

2.7 Conclusions

TE in cranio-maxillofacial surgery can be looked upon as mainly based on empiricism Many case reports of the successful clinical use of TE in cranio- maxillofacial surgery can be found but almost no long term follow-up studies have been conducted There is an ever-evolving set of indications on an empirical basis, without being based on experimental data on e g scalp- expansion, expansion of eyelids, cheeks, lips, etc Furthermore, to our knowledge, there has been only one experimental study into the effects of TE on underlying bone and none on cartilaginous structures The influence of TE on normal growth has hardly ever been studied in a prospective experimental set-up Further research into the effects of TE on mucosa, mucopenosteum, cartilage and bone has to be done, before applying the TE technique in the faces of growing individuals In our opinion experimental data have to pave the way for new clinical applications in cranio-maxillofacial surgery

Ρ S For updating of the literature see Chapter 10

43 Chapter 2

2.8 Literature

ADAMSON JE (1988) Nasal reconstruction with the expanded forehead flap Plast Reconstr Surg 81 12 20 ADSON MH, ANDERSON RD, ARGENTA LC (1987) Scalp expansion in the treatment of male pattern baldness Plast Reconstr Surg 79 906 914 ADSON MH, ANDERSON RD, ARGENTA LC (1988) Scalp expansion for male pattern baldness (Reply) Plast Reconstr Surg 81 999-1000 ANDERSON RD (1987) Expansion-assisted treatment of male pattern baldness Clin Plast Surg 14 477-490 ANTONYSHYN O, GRUSS JS, MACKINNON SE, ZUCKER R (1988a) Complications of soft tissue expansion Br J Plast Surg 41 239-250 ANTONYSHYN O, GRUSS JS, ZUCKER R, MACKINNON SE (1988b) Tissue expansion in head and neck reconstruction Plast Reconstr Surg 82 58-68 ARGENTA LC (1984) Controlled tissue expansion in reconstructive surgery Br J Plast Surg 37 520-529 ARGENTA LC, DINGMAN RO (1986) Total reconstruction of aplasia cutis congenita involving scalp skull and dura Plast Reconstr Surg 77 650-653 ARGENTA LC, MARKS MW, PASYK KA (1985) Advances in tissue expansion Clin Plast Surg 12 159-171 ARGENTA LC, VANDERKOLK CA (1987) Tissue expansion in craniofacial surgery Clin Piasi Surg 14 143-153 ARGENTA LC, WATANABE MJ, GRABB WC (1983) The use of tissue expansion in head and neck reconstruction Ann Plast Surg 11 31-37 ARGENTA LC, WATANABE MJ, GRABB WC, NEWMAN MH (1981) Soft tissue expanders in head and neck surgery a new method of reconstruction Plast Surg Forum 4 244 ASHALL G, QUABA A (1987) A hemorrhagic hazard of tissue expansion Plast Reconstr Surg 79 627-630 AUSTAD ED (1987a) The origin of expanded tissue Clin Plast Surg 14 431-433 AUSTAD ED (1987b) Complications in tissue expansion Clin Plast Surg 14 549- 550 AUSTAD ED (1988a) Evolution of the concept of tissue expansion Facial 5 277-279 AUSTAD ED (1988b) Contraindications and complications in tissue expansion Facial Plastic Surgery 5 379-382

44 Tissue expansion, experimentally based or clinically empiric

AUSTAD ED (1988c) Dermal and epidermal response to soft-tissue expansion in the pig (Discussion) Plast Reconstr Surg 81 396-397 AUSTAD ED, PASYK KA (1987) Rapid elongation of arteries and veins in rats with a tissue expander (Discussion) Plast Reconstr Surg 80 579-581 AUSTAD ED, PASYK KA, McCLATCHEY KD, CHERRY GW (1982) Histomorphologic evaluation of guinea pig skin and soft tissue after controlled tissue expansion Plast Reconstr Surg 70 704-710 AUSTAD ED, THOMAS SB, PASYK KA (1986) Tissue expansion dividend or loan? Plast Reconstr Surg 78 63-67 BAKER SR, SWANSON NA (1990a) Tissue expansion ot the head and neck Indications technique and complications Arch Otolaryngol Head Neck Surg 116 1147-1153 BAKER SR, SWANSON NA (1990b) Clinical applications of tissue expansion in head and neck surgery Laryngoscope 100 313-319 BARTELL TH, MUSTOE TA (1989) Animal models of human tissue expansion Plast Reconstr Surg 83 681-686 BAUER BS (1990) The role ot tissue expansion in reconstruction of the ear Clin Plast Surg 17 319-325 BAUER BS, VICARI FA (1988) An approach to excision of congenital giant pigmented nevi in infancy and early childhood Plast Reconstr Surg 82 1012-1021 BAUER BS, VICARI FA, RICHARD ME (1990) The role of tissue expansion in pediatric plastic surgery Clin Plast Surg 17 101-112 BAUX S, MIMOUN M, KIRSCH JM, DUBERT Τ, ZUMER L (1988) Les prothèses d'expansion cutanée et leur utilisation en dehors de la reconstruction mammaire. A propos de cinquante-quatre cas Ann Chir Plast Esthét 33 159-162 BERAKA GJ (1990) Tissue expansion for rhytidectomy (Letter) Plast Reconstr Surg 86 802 BERRY FD (1991) A modified tissue expander for socket enlargement in clinical anophthalmos Ophthal Plast Reconstr Surg 7 41-47 BOCK NJJ, ROSE R (1987) Der subperiostal Gewebeexpander als Hilfsmittel bei der Alveolarkammplastik Dtsch Ζ Mund Kiefer Gesichts Chir 11 443-447 BOLTON LL, CHANDRASEKHAR B, GOTTLIEB ME (1988) Forehead expansion and total nasal reconstruction Ann Plast Surg 21 210-216 BONOMO D (1986) Subperiosteal tissue expanders for ridge augmentation Presented at the Annual Meeting Southern Calif Soc Oral & Maxillofac Surg

45 Chapter 2

BRAR M (1990) Tissue expansion with rhytidectomy (Letter) Plast Reconstr Surg 85 996 BRETON P, FREIDEL M (1989) Bride cervical médiane congenitale Ann Chir Piasi Esthét 34 73-76 BROBMANN GF, HUBER J (1985) Effects of different-shaped tissue expanders on transluminal pressure oxygen tension histopathologic changes and skin expansion in pigs Plast Reconstr Surg 76 731-736 BUHRER DP, HUANG TT, YEE HW, BLACKWELL SJ (1988) Treatment of burn alopecia with tissue expanders in children Plast Reconstr Surg 81 512- 515 CANADY JW, SQUIER CA, KELLY KM, BARDACH J (1990) Serial measurement of blood flow in expanded tissue by laser Doppler velocimetry Otolaryngol Head Neck Surg 103 986-990 CHANG TS, JIN YT (1986) Application of tissue expansion in the treatment ot post-burn skin contractures and alopecia Eur J Plast Surg 9 7-12 CHERRY GW, AUSTAD ED, PASYK DA, McCLATCHEY KD, ROHRICH RJ (1983) Increased survival and vascularity of random-pattern skin flaps elevated in controlled expanded skin Plast Reconstr Surg 72 680-685 COHEN IK, DUNN JD (1988) Future aspects in tissue expansion Facial Plastic Surgery 5 383-384 COLE RP, GAULT DT, MAYOU BJ, DAVIS PKB (1991) Pain and forehead expansion Br J Plast Surg 44 41-43 COOK HE, LEWIS MK, STOKER NG (1987) Tissue expansion reconstruction ol soft tissue avulsions of the face report of two cases J Oral Maxillofac Surg 45 362-370 COOPER RL, BROWN D (1990) Pretansfer tissue expansion of a scalp tree flap tor burn alopecia reconstruction in a child a case report J Reconstr Microsurg 6 339-343 DE MEY A, MALEVEZ C, LEJOUR M (1990) Treatment of palatal fistula by expansion Br J Plast Surg 43 362-364 DICKSON MG (1987) Breast reconstruction by tissue expansion- a previously unreported complication (Letter) Plast Reconstr Surg 80 474-475 DONELAN Jr MB (1988) Complications of controlled tissue expansion in the pediatric burn patient (Discussion) Plast Reconstr Surg 82 846-848 DUITS EHA, MOLENAAR J, VAN RAPPARD JHA (1989) The modeling of skin expanders Plast Reconstr Surg 83 362-365

46 Tissue expansion, experimentally based or clinically empiric

EHRENFELD M, RIEDIGER D, SCHWENZER N (1989) Zur Problematik des Hautersatzes unter besonderer Berücksichtigung des Gewebeexpanders Fortschr Kiefer/GesichtsChir 34 161-164 ELLENBOGEN R, COLLINI FJ (1990) Tissue expansion for rhytidectomy (Letter) Plast Reconstr Surg 85 995 ESPOSITO G, DI CAPRIO G, ZICCARDI Ρ, SCUDERI Ν (1991) Tissue expansion in the treatment of pressure ulcers Plast Reconstr Surg 87 501-508 FENTON О (1988) Complications of soft tissue expansion (Discussion) Br J Plast Surg 41 249-250 FRANCIS AJ, MARKS R (1977) Skin stretching and epidermopoiesis Br J Exp Pathol 58 35-39 FUDEM GM, ORGEL MG (1988) Full-thickness erosion ol the skull secondary to tissue expansion for scalp reconstruction (Letter) Piasi Reconstr Surg 82 368-369 GARNIER D, COSTES Y, RICBOURG В (1989) La microvascularisation cutanee apres expansion tissulaire etude expérimentale chez le rat Ann Chir Plast Esthet 34 531-535 GIBBONS WP (1987) Caution in expanding radiated tissue (Letter) Plast Reconstr Surg 80 871 GOLDSTEIN MH (1984) A tissue expanding vermilion myocutaneous flap for lip repair Plast Reconstr Surg 73 768-770 GOLDSTEIN MH (1990) The elastic flap for lip repair Plast Reconstr Surg 85 446-452 GOUET O, BOUREAU M, PRADET G, ISELIN F (1989) Expansion du tegument facial chez l'enfant Ann Chir Plast Esthet 34 421-425 GREENBAUM SS, GREENBAUM CH (1990) Intraoperative tissue expansion using a Foley catheter following excision of a basal cell carcinoma J Dermatol Surg Oncol 16 45 48 GROSSMAN JAI, McGRAW JB, McDOUGAL HD, JACOBS J, MIEKLEY С (1984) Histopathology of human skin expansion Plast Surg Forum 7 85 GUERO SJ, LACROIX PA (1991) An experimental model of scalp expansion A method of optimizing flap size and shape Plast Reconstr Surg 87 776-779 HALLOCK GG (1987) Maximum ovennflation of tissue expanders Plast Reconstr Surg 80 567-569 HALLOCK GG (1988a) Bursting the tissue expander bubble (Letter) Plast Reconstr Surg 81 993

47 Chapter 2

HALLOCK GG (1988b) Puncture threshold prior to leakage from tissue expander reservoirs Plast Reconstr Surg 82 666-668 HALLOCK GG, RICE DC (1986) Objective monitoring for safe tissue expansion Plast Reconstr Surg 77 416-420 HATA Y, HOSOKA WA К, YANO К, MATSUKA К, ITO О (1989) Correction of congenital microtia using the tissue expander Plast Reconstr Surg 84 741-751 HEMMER KM, MARSH JL, PICKER S (1987) Calvanal erosion after scalp expansion Ann Plast Surg 19 454-459 HEMS TEJ, GODFREY A (1990) Cosmetic surgery for Cornelia de Lange syndrome Br J Plast Surg 43 489-491 HIRSHOWITZ В (1989) Stretching and tissue expansion for rhytidectomy an improved approach (Discussion) Plast Reconstr Surg 84 570-571 HIRSHOWITZ B, KAUFMAN T, ULLMAN J (1986) Reconstruction of the tip of the nose and ala by load cycling of the nasal skin and harnessing of extra skin Plast Reconstr Surg 77 316-319 HONG C, STARK GB, FUTRELL JW (1987) Elongation of axial blood vessels with a tissue expander Clin Plast Surg 14 465-467 HORIBE EK, PINTO WS, YAMAGUCHI CT (1989) The use of tissue expanders in the treatment of burn sequelae (Abstract) Plast Reconstr Surg 84 867-868 IWAHIRA Y, M ARU Y AM A Y (1991) Expanded preauricular full-thickness free skin graft Plast Reconstr Surg 87 150-152 JOHNSON PE, KERNAHAN DA, BAUER BS (1988) Dermal and epidermal response to soft-tissue expansion in the pig Plast Reconstr Surg 81 390-395 KABAKER SS, KRIDEL RWH, KRUGMAN ME, SWENSON RW (1986) Tissue expansion in the treatment of alopecia Arch Otolaryngol Head Neck Surg 112 720-725 KABAKER SS, MOSS RM, SWENSON RW (1988) Expanded pedicle scalp flaps for baldness is a delay necessary1? Facial Plast Surg 5 356-361 KASAI K, OGAWA Y, TAKEUCHI E (1991) A case of sideburn reconstruction using a temporo paneto-occipital island flap Plast Reconstr Surg 87 146-149 KAWASHIMA T, YAM ADA A, HASHIMOTO M (1990) Problems in the use of tissue expanders for scalp reconstruction (Abstract) Plast Reconstr Surg 85 834-835 KESSLER A, ORENSTEIN A, KAPLAN H, TSUR H (1989) Neck contracture as a rare complication of cervical soft tissue expansion Burns 15 392-393

48 Tissue expansion, experimentally based or clinically empiric

KNIGHT KR, McCANN JJ, VANDERKOLK CA, СОЕ SA, O'BRIEN BM (1990) The redistribution ot collagen in expanded pig skin Br J Plast Surg 43 565 570 KROLL SS (1989) Forehead Пар nasal reconstruction with tissue expansion and delayed pedicle separation Laryngoscope 99 448-452 LATRENTA GS, MCCARTHY JG, EPSTEIN M, CUTTING CB, ORENTREICH С (1988) Bone graft survival in expanded skin Plast Reconstr Surg 81 406-413 LAWRENCE WT (1989) Isolated necrosis of deep tissue as a complication of rapid tissue expansion (Letter) Plast Reconstr Surg 84 172 LEE P, SQUIER CA, BARDACH J (1985) Enhancement of tissue expansion by anticontractile agents Plast Reconstr Surg 76 604-610 LEIGHTON WD, JOHNSON ML, FRIEDLAND JA (1986a) Use of the temporary soft-tissue expander in posttraumatic alopecia Plast Reconstr Surg 77 737-743 LEIGHTON WD, RUSSELL RC, FELLER AM, ERIKSSON E, MATHUR A, ZOOK EG (1988a) Experimental pretransfer expansion of free-flap donor sites II Physiology histology and clinical correlation Plast Reconstr Surg 82 76-84 LEIGHTON WD, RUSSELL RC, MARCUS DE, ERIKSSON E, ZOOK EG (1986b) Experimental expansion of cutaneous and myocutaneous free flap donor sites anatomical physiological and histological changes Plast Surg Forum 9 262-263 LEIGHTON WD, RUSSELL RC, MARCUS DE, ERIKSSON E, SUCHY H, ZOOK EG (1988b) Experimental pretransfer expansion of free-flap donor sites I Flap viability and expansion characteristics Plast Reconstr Surg 82 69-75 LEONARD AG, SMALL JO (1986) Tissue expansion in the treatment of alopecia Br J Plast Surg 39 42-56 LEW D, AMOS EL, SHROYER JV III (1988a) The use of a subperiosteal tissue expander in rib reconstruction of an atrophic mandible J Oral Maxillotac Surg 46 229-232 LEW D, AMOS EA, UNHOLD GP (1988b) An open procedure for placement of a tissue expander over the atrophic alveolar ridge J Oral Maxillofac Surg 46 161-166 LEW D, CLARK R, SHAHBAZIAN Τ (1986) Use of a soft tissue expander in alveolar ridge augmentation a preliminary report J Oral Maxillofac Surg 44 516-519

49 Chapter 2

LEW D, SHROYER JV III, UNHOLD GP (1988c) The use ot tissue expanders in the correction of avulsive injuries of the mandible report of two cases J Oral Maxillofac Surg 46 892-899 LEW D, SHROYER JV III, UNHOLD GP, STUTES RD (1989) The use of tissue expanders in the reconstruction of orofacial defects secondary to congenital rubella case report J Oral Maxillofac Surg 47 1202-1207 LO AKM, COLCLEUGH RG, ALLEN L, VAN WYCK L, BITE U (1990) The role of tissue expanders in an anophthalmic animal model Plast Reconstr Surg 86 399-408 LUCE EA (1990) Tissue expansion for rhytidectomy (Letter) Plast Reconstr Surg 86 602-603 MACKAY DR, SAGGERS GC, KOTWAL N, MANDERS EK (1990) Stretching skin undermining is more important than intraoperative expansion Plast Reconstr Surg 86 722-730 MAN D (1989) Stretching and tissue expansion for rhytidectomy an improved approach Plast Reconstr Surg 84 561-569 MAN D (1990a) Tissue expansion tor rhytidectomy (Reply) Plast Reconstr Surg 85 828 MAN D (1990b) Tissue expansion for rhytidectomy (Reply) Plast Reconstr Surg 85 996-997 MANDERS EK, AU VK, WONG RKM (1987a) Scalp expansion for male pattern baldness Clin Plast Surg 14 469-475 MANDERS EK, GRAHAM WP, SCHENDEN MJ, DAVIS TS (1984a) Skin expansion to eliminate large scalp defects Ann Plast Surg 12 305-312 MANDERS EK, MACKAY DR, SAGGERS GC (1990) Tissue expansion for rhytidectomy (Letter) Plast Reconstr Surg 85 995-996 MANDERS EK, SAGGERS GC (1989) Effect of capsulectomy on the hemodynamics and viability of random-pattern skin flaps raised on expanded skin in the pig (Discussion) Plast Reconstr Surg 84 323-324 MANDERS EK, SAGGERS GC, DIAZ-ALONSO Ρ, FINN L, SIPIO JC, GLUMAC T, AU VK, WONG RKM, MOTTALEB M (1987b) Elongation of penferal nerve and viscera containing smooth muscle Clin Plast Surg 14 551-562 MANDERS EK, SCHENDEN MJ, FURREY JA, HETZLER PT, DAVIS TS, GRAHAM WP III (1984b) Soft-tissue expansion concepts and complications Plast Reconstr Surg 74 493-507

50 Tissue expansion experimentally based or clinically empiric

MARCHAC D, PUGASH E (1987) Expanded scalping forehead Пар tor coverage ol giant congenital nevi ot the face a report of three cases Eur J Plast Surg 10 2-7 MARKS MW, ARGENTA LC (1988) Skin expansion in reconstructive surgery Facial Plastic Surgery 5 301-311 MARKS MW, ARGENTA LC, THORNTON JW (1987a) Rapid expansion experimental and clinical experience Clin Plast Surg 14 455-463 MARKS MW, ARGENTA LC, THORNTON JW (1987b) Burn management the role of tissue expansion Clin Plast Surg 14 543-548 MARKS MW, BURNEY RE, MACKENZIE JR, KNIGHT PR (1986) Enhanced capillary blood flow in rapidly expanded random pattern flaps J Trauma 26 913-915 MARKS MW, MACKENZIE JR, BURNEY RE, KNIGHT PR, ANDERSON SH (1985) Response ol random pattern skin flaps to rapid expansion J Trauma 25 947-952 MASSER MR (1988) A twin tissue expander used in the elimination of alopecia Plast Reconstr Surg 81 444-449 MASSER MR (1990) Tissue expansion a reconstructive revolution or a cornucopia ot complications7 Br J Plast Surg 43 344-348 MASSON CL, FOYATIER JL, DESSAPT B, COMPARIN JP (1989) L'expansion tissulaire rapide Ann Chir Plast Esthet 34 521-523 MATTHEWS RN, MISSOTTEN FEM (1986) Early tissue expansion to close a traumatic defect of scalp and pericranium Br J Plast Surg 39 417-421 MAYER B, AMARANTE L, BARTH С, PITANGUY I, NASSIF Τ (1989) Gewebeexpander in der plastischen Kopf- und Haischirurgie Laryngo-Rhino- Otol 68 313 318 McCANN JJ, MITCHELL GM, O'BRIEN BMC, VANDERKOLK CA (1988) Comparative viability of expanded and unexpanded axial pattern skin flaps in pigs Br J Plast Surg 41 294-297 McKINNEY P, EDELSON R, TERRASSE A, ZUKOWSKI M (1987) Chest-wall deformity following soft-tissue expansion for breast reconstruction Plast Reconstr Surg 80 442-444 MILLER LB, KANTER M, WOLFORT F (1990) Treatment of webbed neck in Turner's syndrome with tissue expansion Ann Plast Surg 24 447-450 MILNER RH (1989) The effect of tissue expansion on peripheral nerves Br J Plast Surg 42 414-421

51 Chapter 2

MIN Ζ, SVENSSON Η, SVEDMAN Ρ (1988) On expander pressure and skin blood flow during tissue expansion in the pig Ann Plast Surg 21 134-139 MOELLEKEN BRW, MATHES SJ, CANN CE, SIMMONS DJ, GHAFOORI G (1990) Long-term effects of tissue expansion on cranial and skeletal bone development in neonatal miniature swine clinical findings and hislomorphometnc correlates Plast Reconstr Surg 86 825-834 MORRIS SF, PANG CY, MAHONEY J, LOFCHY N, KADDOURA IL, PATTERSON R, LISTA F (1989) Effect of capsulectomy on the hemodynamics and viability of random-pattern skin flaps raised on expanded skin in the pig Plast Reconstr Surg 84 314-322 MOSCONA R, ULLMANN Y, BERGMAN R, FRIEDMAN-BIRNBAUM R (1989) Postirradiation extensive cicatricial scalp alopecia treated by tissue expansion J Dermatol Surg Oncol 15 337-341 MUENKER R (1988a) Various devices available for tissue expansion and clinical experience Facial Plastic Surgery 5 291-300 MUENKER R (1988b) Nasal reconstruction with tissue expansion Facial Plastic Surgery 5 328-337 MUSTOE TA, BARTELL TH, GARNER WL (1989) Physical, biomechanical, histologic and biochemical effects of rapid versus conventional tissue expansion Plast Reconstr Surg 83 687-691 MUTIMER KL, MULLIKEN JB (1988) Correction of cryptotia using tissue expansion Plast Reconstr Surg 81 601-604 NEALE HW, HIGH RM, BILLMIRE DA, CAREY JP, SMITH D, WARDEN G (1988) Complications of controlled tissue expansion in the pediatric burn patient Plast Reconstr Surg 82 840-845 NELIGAN PC, PETERS WJ (1989) Advances in burn scar reconstruction the use of tissue expansion Ann Plast Surg 22 203-210 NEUMANN CG (1957) The expansion of an area of skin by progressive distention of a subcutaneous balloon Plast Reconstr Surg 19 124-130 NIRANJAN NS (1989) Webbing of the neck correction by tissue expansion Plast Reconstr Surg 84 985-988 NORDSTROM REA (1984) "Stretch-back" in scalp reductions for male pattern baldness Plast Reconstr Surg 73 422-426 NORDSTROM REA (1988a) Tissue expansion and flaps for surgical correction of male pattern baldness Facial Plastic Surgery 5 347-355 NORDSTROM REA (1988b) Tissue expansion and flaps for surgical correction of male pattern baldness Br J Plast Surg 41 154-159

52 Tissue expansion experimentally based or clinically empiric

NORDSTROM REA (1988c) Antibiotics in the tissue expander to decrease the rate of infection (Letter) Plast Reconstr Surg 81 137-138 NORDSTROM REA, DEVINE JW (1985) Scalp stretching with a tissue expander for closure of scalp detects Plast Reconstr Surg 75 578-581 NORDSTROM REA, PIETILA JP, RINTALA AE (1988a) Clinical experience with tissue expansion Facial Plastic Surgery 5 317-327 NORDSTROM REA, PIETILA JP, VOUTILAINEN PEJ, LILIUS GPS, VIRKKUNEN PJ, RINTALA AE (1988b) Tissue expander injection dome leakage Plast Reconstr Surg 81 26-29 NORDSTROM REA, SALO HP, RINTALA AE (1988c) Auricle reconstruction with the help of tissue expansion Facial Plastic Surgery 5 338-346 OLSHANSKY К (1990) Tissue expansion in conjunction with rhytidectomy (Letter) Plast Reconstr Surg 85 828 O'NEAL RM, ROHRICH RJ, IZENBERG PH (1984) Skin expansion as an adjunct to reconstruction of the external ear Br J Plast Surg 37 517 519 PALETTA CE, BASS J, SHEHADI SI (1989) Outer table skull erosion causing rupture of scalp expander Ann Plast Surg 23 538-542 PASYK KA, ARGENTA LC, AUSTAD ED (1987) Histopathology of human expanded tissue Clin Plast Surg 14 435-445 PASYK KA, ARGENTA LC, HASSETT С (1988) Quantitative analysis of the thickness of human skin and subcutaneous tissue following controlled expansion with a silicone implant Plast Reconstr Surg 81 516-523 PASYK KA, AUSTAD ED, McCLATCHEY KD, CHERRY GW (1982) Electron- microscopic evaluation of guinea pig skin and soft tissues 'expanded' with a self-inflating silicone implant Plast Reconstr Surg 70 37-45 PEARL RM (1984) Pathophysiology of skin flaps raised on expanded pig skin (Discussion) Plast Reconstr Surg 74 66-67 PENOFF J (1990) Skin expansion - a sword that "stretches" two ways scalp expansion and bone erosion J Craniofac Surg 1 103-105 PICHA GJ, GOLDSTEIN JA (1991) Analysis of the soft-tissue response to components used in the manufacture of breast implants rat animal model Plast Reconstr Surg 87 490-500 PIETILA JP (1990) Tissue expansion and skin circulation Simultaneous monitoring by laser Doppler flowmetry and transcutaneous oximetry Scand J Plast Reconstr Surg Hand Surg 24 135-140

53 Chapter 2

PIETILA JP, E A NORDSTROM REA, NUMMINEN MK, VIRKKUNEN PJ, VOUTILAINEN PEJ, RINTALA AE (1988a) Validity of laser Doppler flowmetry tor measuring the effect of the tissue expander pressure on skin circulation Scand J Plast Reconstr Surg 22: 135-139 PIETILA JP, NORDSTROM REA, VIRKKUNEN PJ, VOUTILAINEN PEJ, RINTALA AE (1988b) Accelerated tissue expansion with the "overfilling" technique. Plast Reconstr Surg 81 204-207 QU AB A AA (1988) Reconstruction ot a posttraumatic ear defect using tissue expansion. 30 years after Neumann. Plast Reconstr Surg 82· 521-524. QUAYLE AA, MAROUF H, HOLLAND I (1990). Alveolar ridge augmentation using a new design of inflatable tissue expander surgical technique and preliminary results Br J Oral Max Fac Surg 28: 375-382. RADOVAN С (1984). Tissue expansion in soft-tissue reconstruction. Plast Reconstr Surg 74: 482-490. RICBOURG B, HARBON S, CHARTOUNI M, MENARD PH (1989). Les prothèses d'expansion en chirurgie réparatrice et esthétique de l'extrémité céphalique. Rev Stomatol Cliir Maxillofac 90 79-83. RICCIARDELLI EJ, GOODING GS, BRIGHT DA, CUMMINGS CW (1989) Acute blood flow changes in rapidly expanded and adjacent skin Arch Otolaryngol Head Neck Surg 115: 182-186 SAMAYOA V, VALIENTE E, SERRA-RENOM JR (1990). Experimental study of the pathologic changes in the build-up of the capsule during progressive tissue expansion (Abstract). Plast Reconstr Surg 85: 662-663 SAOUMA S, BRICOUT N, SERVANT JM, BANZET Ρ (1989). Technique de l'expansion tissulaire J Chir (Pans) 126: 34-39. SASAKI GH (1987) Intraoperative sustained limited expansion (ISLE) as an immediate reconstructive technique. Clin Plast Surg 14: 563-573. SASAKI GH (1988). Intraoperative expansion as an immediate reconstructive technique. Facial Plastic Surgery 5: 362-378 SASAKI GH (1989). Correction of congenital microtia using the tissue expander (Discussion). Plast Reconstr Surg 84: 752-753 SASAKI GH (1990). Tissue expansion™ in reconstruction of acquired auricular defects Clin Plast Surg 17: 327-338 SASAKI GH, PANG CY (1984). Pathofysiology of skin flaps raised on expanded pig skin. Plast Reconstr Surg 74: 59-65. SAXBY PJ (1988) Survival of island flaps after tissue expansion: a pig model. Plast Reconstr Surg 81: 30-34.

54 Tissue expansion, experimentally based or clinically empiric

SAXBY PJ (1990) The preexpanded radial free flap (Discussion) Plast Reconstr Surg 86 302-303 SCHMIDT SC, LOGAN SE, HAYDEN JM, TAE AHN S, MUSTOE TA (1991) Continuous versus conventional tissue expansion Experimental verification of a new technique Plast Reconstr Surg 87 10-15 SCHUBERT W, SHONS AR (1990) Intraoperative tissue expansion for rhytidectomy (Letter) Plast Reconstr Surg 86 603-604 SCHWARTZ HC, RELLE RJ (1990) Extraoral placement of a subperiosteal tissue expander for reconstruction with hydroxylapatite of the severely atrophic mandibular alveolar ridge J Oral Maxillofac Surg 48 157-161 SHIVELY RE (1988) Surface-area increase in tissue expansion (Discussion) Plast Reconstr Surg 82 838-839 SHIVELY RE, BERMANT MA, BUCHOLZ RD (1985) Separation of craniopagus twins utilizing tissue expanders Plast Reconst Surg 76 765-772 SNYDER CC (1985) Separation of craniopagus twins utilizing tissue expanders (Discussion) Plast Reconstr Surg 76 773 STARK GB, HONG C, FUTRELL JW (1987) Rapid elongation of arteries and veins in rats with a tissue expander Plast Reconst Surg 80 570-578 STARK GB, JAEGER К (1989) Failed tissue expansion in a patient with osteogenesis imperfecta Ann Plast Surg 22 156-159 TANINO R, MIYASAKA M (1990) Reconstruction of microtia using tissue expander Clin Plast Surg 17 339-353 THOMSON SJ, TANNER NSB, MERCER DM (1990) Web neck deformity anatomical considerations and options in surgical management Br J Plast Surg 43 94-100 THORNTON JW, MARKS MW, IZENBERG PH, ARGENTA LC (1987) Expanded myocutaneous flaps their clinical use Clin Plast Surg 14 529-534 TIMMENGA EJF, SCHOORL R, BOS JD, KLOPPER PJ (1989) An improved model for tissue expansion and flap research in the rabbit Br J Plast Surg 42 301-305 TIMMENGA EJF, SCHOORL R, KLOPPER PJ (1990) Biomechamcal and histomorphological changes in expanded rabbit skin Br J Plast Surg 43 101-106 TOTH BA, GLAFKIDES MC, WANDEL A (1990) The role of tissue expansion in the treatment of atypical facial clefting Plast Reconstr Surg 86 119-122

55 Chapter 2

VALAURI FA, BUNCKE HJ, ALPERT BS, LINEAWEAVER WC, ARGENTA LC (1990) Microvascular transplantation of expanded free scalp flaps between identical twins Plast Reconstr Surg 85 432-436 VAN BEEK AL, ADSON ΜΗ (1987) Tissue expansion in the upper extremity Clin Plast Surg 14 535-542 VAN BEEK AL, ADSON ΜΗ (1988) Experimental pretransfer expansion of free flap donor sites I flap viability and expansion characteristics II physiology, histology, and clinical correlation (Discussion) Plast Reconstr Surg 82 85-87 VAN DAMME PHA (1990) Subperiosteal palatal tissue expansion an animal experiment in domestic cats Abstr 10th Congr Eur Ass Cranio-Max-Fac Surg, Brussels, Belgium, ρ 48 VAN DAMME PHA, FREIHOFER HPM, KUIJPERS-JAGTMAN AM, MALTHA JC, VAN 'T HOF MA (1991) Subperiosteal palatal tissue expansion an animal experiment in domestic cats Abstr 4th Eur Cranio-Fac Congr, Noordwijkerhout, The Netherlands, ρ 41 VANDERKOLK CA, McCANN JJ, KNIGHT KR, O'BRIEN BMC (1987) Some further characteristics of expanded tissue Clin Plast Surg 14 447-453 VANDERKOLK CA, McCANN JJ, MITCHELL GM, O'BRIEN BMC (1988) Changes in area and thickness ot expanded and unexpanded axial pattern skin flaps in pigs Br J Plast Surg 41 284-293 VAN RAPPARD JHA (1988) Controlled tissue-expansion in reconstructive surgery PhD Thesis, University of Groningen, The Netherlands VAN RAPPARD JHA, BORGHOUTS JMHM, VAN TWISK R, THEUVENET WJ (1987) Weefselexpansie in de reconstructieve chirurgie Ned Tijdschr Geneeskd 131 853-857 VAN RAPPARD JHA, JERUSALEM С, SONNEVELD GJ, BORGHOUTS JMHM (1988a) Histologic changes in soft tissues due to tissue expansion (in animal studies and humans) Facial Plastic Surgery 5 280-286 VAN RAPPARD JHA, MOLENAAR J, VAN DOORN К, SONNEVELD GJ, BORGHOUTS JMHM (1988b) Surface-area increase in tissue expansion Plast Reconstr Surg 82 833-837 VAN RAPPARD JHA, SONNEVELD GJ, BORGHOUTS JMHM (1988c) Geometric planning and the shape of the expander Facial Plastic Surgery 5 287-290 VICTOR WH, HURWITZ JJ (1984) Cicatricial ectropion following blepharoplasty treatment by tissue expansion Can J Ophthalmol 19 317-319

56 Tissue expansion experimentally based or clinically empiric

VICTOR WH, HURWITZ JJ, GRUSS JS (1986) The development of a new tissue expander for use in ophthalmic plastic surgery Ophthalmic Surg 17 661-665 VISTNES LM (1984) Tissue expansion in soft-tissue reconstruction (Discussion) Plast Reconstr Surg 74 491-492 VISTNES LM, KSANDER GA, KOSEK J (1978) Study of encapsulation of silicone rubber implants in animals a foreign-body reaction Plast Reconstr Surg 62 580-588 WILMSHURST AD, SHARPE DT (1990) Immediate placement of tissue expanders in the management of large excisional defects on the face Br J Plast Surg 43 150-153 WITTKAMPF ARM (1989) Short-term experience with the subperiosteal tissue expander in reconstruction of the mandibular alveolar ridge J Oral Maxillofac Surg 47 469-474 WITTKAMPF ARM (1990) Placement of tissue expanders on the mandibular ndge (Letter) J Oral Max Fac Surg 48 1352 WOOD FM, McMAHON SB (1989) The response of the peripheral nerve field to controlled soft tissue expansion Br J Plast Surg 42 682 686 WOOD RJ, ADSON MH, VAN BEEK AL, PELTIER GL, ZUBKOFF MM, BUBRICK MP (1991) Controlled expansion of peripheral nerves comparison of nerve grafting and nerve expansion/repair for canine sciatic nerve defects J Trauma 31 686-690 ZAREBSKI M, BRETON Ρ (1988) Application de l'expansion tissulaire aux lambeaux cutanés micro-anastomoses Ann Chir Plast Fsthet 33 96-99 ZOLTIE N, CHAPMAN Ρ, JOSS G (1990) Tissue expansion a unit review of non- scalp, non-breast expansion Br J Plast Surg 43 325-327

57

Chapter 3

Two-dimensional cephalometric analysis of the effects of subperiosteal palatal soft tissue expansion in growing cats

Philip A. Van Damme Jaap С. Maltha Anne Marie Kuijpers-Jagtman Hans Peter M. Freihofer Martin A. Van 't Hof

Accepted for publication by the Journal of Plastic and Reconstructive Surgery, 1996. Parts of this chapter were presented as a preliminary report at the joint meeting of the German and Dutch Associations of Oral and Maxillofacial Surgery in Cologne, Germany 1993, and published in the International Journal of Oral Maxillofacial Surgery (1994) 23: 393-394.

Cephalometric analysis

3.1 Summary

The feasibility and the possible effects of palatal soft tissue expansion (TE) in palatal repair were studied. A prospective longitudinal animal experiment was performed in 75 growing cats, assigned to 5 groups. In 31 cats, a midline defect was made and bipedicled flaps were raised at the age of 8 weeks (simulated Langenbeck operation), in order to create palatal scars. At the age of 14 weeks, custom-made tissue expanders were inserted palatally in 61 animals. TE was performed by weekly inflation in 33 cats (16 without and 17 with scars) for an eight week period. The remaining 28 cats (14 without and 14 with scars) served as sham groups. A control group was formed by 14 animals (without scars and without tissue expander). Soft tissue gain and its effects on maxillofacial growth and development were measured in the midsagittal plane on tracings from standardized lateral radiographs. The effects of the experimental interventions were evaluated for eight weeks after removal of the tissue expander. Not all the cats yielded results at all time periods. This study showed that TE of palatal mucoperiosteum is feasible. The surgically induced scars did not cause significant differences between the different groups in the midsagittal plane and the data from both expansion and sham groups could be pooled. Significant soft tissue gain was achieved by the TE technique. Iatrogenic side effects were significant antero-posterior growth retardation at the level of the bony palate, and increase in vertical growth of the anterior nasomaxillary height and the posterior skull height during active TE. After removal of the tissue expanders, some accelerated growth was found in the TE in scarred tissue group, with initial correction of the abnormal growth at the cranial base level. It is concluded that palatal TE is possible in growing cats. This technique, however, impaired maxillofacial growth and development.

61 Chapter 3

3.2 Introduction

The use of TE for facilitating the closure of palatal defects in cleft lip and palate (CLP) is new and has seldom been reported' \ The usual techniques for closure of oronasal communications in CLP are summarized by Millard4. Secondary fistulae are reported to occur as failures in 5 to 30% of cases after primary cleft repair4. Recently reported closure techniques are various loco- regional pedicled mucoperiosteal flaps, such as tadpole flaps5, buccal musculo-mucosal flaps6, buccal fat pad flaps7-8, different lingual flaps9'" and temporal muscle and/or fascial flaps1211, as well as other techniques1415. These flaps do not always yield good results. For instance, with the buccal musculo-mucosal flap, complete closure can be achieved in 35 to 70% of secondary fistulae at first attempt, depending on the size and site of the defects6. Closure generally becomes more difficult and the techniques become less reliable when multiple closure attempts have been made4 '5. There are several disadvantages to these flaps. Some of them are two- stage procedures, create a donor site defect, and use tissue of different origin, texture, and characteristics for the repair. The techniques may result in temporary functional disturbances. Lingual flaps, for example, cause temporary impairment of tongue function with speech interference, and sometimes intermaxillary fixation is needed during the healing phase. Free micro-vascularized flaps such as the radial forearm fasciocutaneous flap, are hazardous, time consuming, bulky and often cause donor site morbidity and visible scars1617. Also, each surgical intervention has a negative effect on subsequent growth and development of the dentomaxillo- facial complex18"27. A reduction in the number of palatal operations and subsequent scarring is, therefore, a desirable goal. TE has been used in cranio-maxillofacial surgery since 198128. More recently, the use of a soft tissue expander for closure of palatal fistulae has been advocated1,2. Few studies have reported on the effects of TE on growth and development in growing individuals29-30. In order to quantify the positive and/or negative effects of subperiosteal palatal TE on the antero-posterior and vertical growth of the maxilla, a prospective longitudinal animal experiment was performed and maxillary growth evaluated radiographically.

62 Cephalometric analysis

The present study evaluates the feasibility and the effects of TE on the palatal mucoperiosteum of growing cats, with and without pre-existing scars.

3.3 Materials and methods

Seventy five domestic tomcat kittens aged 8 weeks were assigned to five groups: I: tissue expansion group, TEXP (n = 16) II: sham group, SEXP (n = 14) III: tissue expansion in scar tissue group, TSCA (n = 17) IV: sham in scar tissue group, SSCA (n = 14) V: control group, CONT (n = 14). The animals were housed under normal laboratory conditions in the Central Animal Laboratory, University of Nijmegen, and received a diet consisting of granules, canned meat, and drinking water ad libitum. The cats were weighed every week and prior to any intervention. All interventions were performed under standard general anaesthesia, induced by 30 mg/kg NimatekR i.m. (Ketamine HCL 100 mg/ml, Α.U.V. Cuijk, The Netherlands) with 0.5 ml Atropine i.m. (Atropine Sulphate 0.5 mg/ml) and maintained after endotracheal intubation with a mixture of 02 (1 R 1/min), N20 (2 1/min) and 0.5-4.0% Ethrane (Enflurane, Abbott B.V., Amstelveen, The Netherlands). A simulated Langenbeck operation was performed under sterile conditions in the TSCA and SSCA groups at the age of 8 weeks. For this purpose, the oral cavity and perioral region were disinfected with BetadineR solution (Povidoniodine 10%, Dagra-Pharma B.V., Diemen, The Netherlands). Hereafter, a standardized lenticular soft tissue defect was created in the median region of the palate by excising a mucoperiosteal flap (Fig. 3-1). This flap extended from just behind the incisive papilla to the caudal margin of the hard palate. The maximum width of the flap was one third of the transverse distance between the deciduous first molars. On both sides, relaxation incisions were made adjacent to the posterior teeth. The mucoperiosteum was elevated and the soft tissue defect was closed in the

63 Chapter 3 midline, in one layer with Vicryl1* 5-0 (Ethicon, Johnson & Johnson Company, Amersfoort, The Netherlands), leaving two areas of denuded bone adjacent to the posterior teeth to heal by secondary intention. Healing was allowed for six weeks until the age of 14 weeks.

Figure 3-1: Schematic drawing of modified Langenbeck operation. Excision of lenticular part of the palatal mucoperiosteum in the midline (a), bilateral relaxation incisions. Elevation of the mucoperiosteum and closure of the midline defect (b). Three scars are created: 1 healed per primam intentionem in the midline and 2 per secundam inlentionem lateral on both sides.

At 14 weeks of age custom-made, hemispherical soft tissue expanders (CUIR, Inamed B.V., Breda, The Netherlands) were inserted in TEXP, TSCA, SEXP, and SSCA (Fig. 3-2). The tissue expanders were made of silicone, had a diameter of 12 mm at their base and a filling volume of 1 ml31,32.

64 Cephalometric analysis

!llljl!lll!!l|lli!lll!jl II ΙΙΙψΙΙΙ l!l!| I!l|ll

4 5 6 7 8 9

Figure 3-2: Lateral view of custom-made hemispheric tissue expander with self sealingfilling port. Base diameter in empty state 12.0 mm, height 6,0 mm. Filled, height 12.0 mm.

The length of the external filling tube, with an incorporated self-sealing port, was 15 mm. To introduce the expander, a standardized para-marginal palatal incision from left to right upper canine was made to create a submucoperiosteal pocket of approximately 15 χ 15 mm, by elevation of the mucoperiosteum. Both anterior palatal neurovascular bundles were coagulated, while the posterior palatal neurovascular bundles were spared. A 2 mm incision was made on the buccal side, between canine and first deciduous molar and the gingiva and mucoperiosteum were tunneled to allow through the external filling tube (Fig. 3-3). The palatal incision was closed with Vicryl" 5-0 sutures.

65 Chapter 3

Figure 3-3: Lateral view with tissue expander in situ and external filling tube tunneled between canine and first deciduous molar.

Perioperative antibiotics were administered; preoperatively 0.5 ml AlbipenR 15%, i.m. (Ampicillin-anhydrate 150 mg/ml, Gist-brocades N.V., Delft, The Netherlands), and postoperatively on day 1 and day 3, 1.0 ml AlbipenR L.A., i.m. (Ampicillin-anhydrate 100 mg/ml). The wounds were inspected daily for the first week after surgery or, when indicated, for a longer period. Healing was allowed for two weeks until the age of 16 weeks. All inserted expanders were initially filled with 0.1 ml of a radiopaque solution (AngiografinR, Meglumine-amidotrizoate 650 mg/ml; 306 mg I/ml, Schering A.G., Berlin, Germany). Hereafter, approximately 0.1 ml AngiografinR was added weekly to each tissue expander in the TEXP and TSCA groups, using a blunt 22-gauge needle, until the age of 24 weeks. In the SEXP and SSCA groups, no additional filling of the tissue expanders was performed after the initial filling. At 24 weeks the tissue expanders and the

66 Cephalometric analysis tissue surplus were surgically removed and the wound was closed with VicrylR 5-0 sutures. The animals were monitored for another eight weeks, using the same regime of recording of data. No surgery was performed in the CONT group, but the animals were monitored and assessed as the operated groups. Radiographs were taken every two weeks under standardized conditions in a specially designed cephalostat". The animals were fixed in the cephalostat with two ear rods and a pin in the mid-sagittal plane. Kodak diagnostic films Min-rR (Eastman Kodak Company, Rochester, N.Y., USA) were used. A Philips PractixR X-ray machine (Philips, The Hague, The Netherlands) was utilized and set at 20 mA, 100 kV, with an exposure time of 10 seconds. The focus-film distance was 4.5 m and the object-film distance 9 cm. Thirteen cats had to be excluded from the study due to technical failures. Sixty-two cats were followed until the age of 20 weeks and 52 cats were followed for the entire inflation period up to 24 weeks. Forty animals were studied during the eight weeks after removal of the tissue expander. All radiographs were examined independently by two investigators. The following anatomical reference points were identified and marked on tracings (Fig. 3-4): Ρ = most posterior point of the bony palate F = centre of the optic foramen 0 — most posterior point of the occiput С = intersection of the coronal suture and the external cortical bone plate N = most anterior point of the nasal bone 1 = intersection of the labial contour of the central incisor and the alveolar bone. The contour of the tissue expander was also traced. The points and structures were digitized independently by both observers, using an X-Y-digitizing tablet (Hitachi HDG-1111B, Manudax Nederland B.V., The Netherlands)34, which was connected to a computer (IBM-PC PS 2, model 70 386) with a mathematical co-processor. The projections of the points P, О, C, I on the line NF were denoted P*, О*, C*, I*.

67 Chapter 3

Figure 3-4: Schematic drawing of lateral cephalogram with points indicated. For definition of the points see text.

The following distances and angles were calculated (Fig. 3-4): PI direct distance, PI indirect distance (along the oral outline of the tissue expander), NF, I*F, P*F, 0*F, IP, PP*, CC*, 00* and angle NF/PI. The error of the cephalometric procedures was determined by measurements on repeated exposures of 15 cats of different ages. The animals were removed and then replaced in the cephalostat between the exposures. Means and standard deviations were calculated for the increments of all variables in all groups. Increments were defined as differences between two time periods. Comparison between the groups was carried out by one-way ANOVA and analysis of co-variance, and if significant differences between groups were found, Tukey's multiple comparisons test was used for evaluation of these differences. Analysis was performed for the period from 14 to 20 weeks of age for 62 cats and for the period from 14 to 24 weeks of age for 52 cats. A period

68 Cephalometru. analysis of 8 weeks after removal of the tissue expanders was also evaluated in 40 animals Intra-observer and inter-observer variability tests were performed to check the accuracy and reproducibility of the procedures

3.4 Results

3 4 1 Eiroi analysis The total error of the cephalometric procedure is composed of an error in the positioning of the animals in the cephalostat, the tracing procedure, including the definition of points and structures on the lateral cephalograms, and the measuring procedure The duplicate exposures at different ages showed a mean total error of 0 23 mm. The angle NF/PI showed a mean total error of 0 92 degrees Based on these results, the accuracy and reproducibility of the methods were considered to be acceptable

3 4 2 Effects of the interventions Mutual comparison of all groups by one-way ANOVA revealed no significant differences between comparable scarred and non-scarred groups for any of the parameters until removal of the expander Additional analysis of co- vanancc supported that scarring of the tissue itself was not decisive for the effects of TE on maxillofacial growth and development in the sagittal plane This meant that pooling of the groups TEXP and TSCA as well as of the groups SEXP and SSCA was allowed Pooling resulted in a tissue expansion group TE, consisting of 26 (20) cats and a sham group SH, consisting of 22 (18) animals (Tables 3-1, 3-2) The pooled groups TE, SH and CO (controls, η = 14) were mutually compared by one-way ANOVA, and significant differences, if any, were subsequently explored by Tukey's multiple comparisons test The differences caused by the interventions were more pronounced for the period trom 14-24 weeks of age than for the period from 14-20 weeks, indicating time-related effects

69 Chapter 3

Table 3-1: Mean increments in mm resp degrees per month Period 14-20 weeks (initial 6 weeks after insertion of TE)

Variable I and III II and IV V Pooled Intergroup TE SH CO SD difference (η = 26) (n = 22) (n = 14) ρ < 005

Pl-dir 1 79 1 93 2 32 0 49 TE < CO PI-indir 7 23 2 25 2 32 1 39 TE > SH, CO NF 3 07 3 07 2 94 0 38 I*F 2 77 3 07 3 27 0 46 TE < CO P*F 1 08 1 20 0 99 0 38 0*F 1 23 1 19 1 15 0 43 II* 1 38 1 06 1 17 0 34 TE > SH PP* 0 68 0 60 0 74 0 32 CC* 0 32 0 38 0 17 0 41 00* 1 02 0 81 071 0 69 < NF/PI 0 69 0 17 0 05 0 79 TE > CO

Table 3-2: Mean increments in tmn resp degrees per month Period 14-24 weeks (initial 10 weeks after insertion of TE)

Variable I and III II and IV V Pooled Intergroup TE SH CO SD difference (η = 20) (n = 18) (n = 14) ρ < 005

Pl-dir 1 52 1 65 1 96 0 32 TE, SH < CO Pl-indir 5 62 1 80 1 96 1 18 TE > SH, CO NF 2 53 2 64 2 57 0 32 I*F 2 30 2 71 2 85 0 31 TE < CO, SH P*F 0 86 1 07 0 92 0 26 0*F 1 06 1 12 1.26 0 30 II* 1 18 0 84 0 92 0 24 TE > SH, CO PP* 0.59 0 63 0 61 0 23 CC* 021 0 25 0 15 0 22 OO* 0 97 0 73 0 63 0 38 TE > CO < NF/PI 0 55 -0 16 -0 04 0 50 TE > SH, CO Cephalometric analysis

The changes in distance Pi-direct, which are a measure of palatal growth, indicated that TE, and to a lesser extent, surgery itself slowed down the antero-posterior growth of the maxilla (Fig. 3-5).

mm 50

Pl-direct

45

40

35

—~ group I

30 "•" group II "*" group III "•"group IV ~*~ group V 25 —ι— 14 20 24 30 36 age in weeks

Figure 3-5: Mean distance of Pl-direct in mm ± SD during the experimental period.

71 Chapter 3

The distance Pi-indirect increased, as expected, significantly faster in the TE group than in the others (Fig. 3-6).

mm 50 Pl-indirect

45

40

35

group I

30 group II group III

group IV

group V С -J I Г I I Г~ 14 20 24 30 36 age in weeks

Figure 3-6: Mean distance of Pi-indirect in mm ± SD during the experimental period.

72 Cephalometric analysis

Measurements along the 'cranial base', defined as the line NF, indicated a relative posterior change in the position of point I* due to the TE as expressed by the distance I*F The vertical dimensions in the anterior region, as represented by the distance II*, increased faster in the TE group than in the SH or the CO groups (Fig 3-7) mm 20

15

^ group I -"- group II •*" group III "•"group IV ~*~ group V 10 І4 20 24 30 36 age in weeks

Figure 3-7: Mean distance of II* in mm ± SD during the experimental period

73 Chapter 3

This also resulted in a significantly larger increase in the angle NF/PI in the TE group than in the SH and CO group after 24 weeks The distance 00*, which is an indication of the posterior skull height, appeared to increase faster in the TE group than in the control group, when the period up to 24 weeks of age was considered

Table 3-3: Mean increments in mm resp degrees per month First 4 weeks after removal of TE

Variable I II III IV V Pooled Intergroup TEXP SEXP TSCA SSCA CONT SD difference (n = 9) (n = 7) (n = 6) (n = 6) 1[n = 12) ρ < 005

PI-dir 0 89 0 83 0 79 0 71 0 97 0 28 PI-indir 0 89 0 83 0 79 0 71 0 97 0 28 NF 1 16 1 01 1 39 1 00 1 20 0 44 I*F 1 24 1 41 1 78 1 09 1 48 0 37 IV < V P*F 0 39 0 54 0 97 0 43 0 49 0 35 I < III 0*F 0 45 1 07 0 58 0 54 0 94 0 41 I < II II* 0 43 -0 00 0 42 0 17 0 39 0 49 PP* 0 20 0 14 0 50 -0 16 0 46 0 47 cc* 0 10 0 21 0 05 -0 13 -0 02 0 50 00* 0 99 0 36 0 44 0 27 0 11 0 69 < NF/PI 0 13 -0 48 -0 36 0 32 -0 37 1 15

The increments in the first four weeks after removal of the tissue expanders showed three significant differences, two of which seemed to be temporary and one (P*F) persistent (Tables 3-3, 3 4) The period from four to eight weeks after removal of the tissue expanders showed some significant differences in the increase of distances related to point F (Table 3-4) The distances anterior to point F (NF, I*F, P*F) increased significantly more in group TSCA than in the unscarred groups The distances NF and P*F in group TEXP increased significantly less than in the scarred groups TSCA and SSCA Only two parameters of the control group showed significant differences with any of the experimental data P*F increased more in the control group than in the TEXP group and I*F increased less in the control group than in group TSCA

74 Cephalometric analysis

Table 3-4: Mean increments in mm resp. degrees per month. Last 4 weeks after removal of TE.

Variable I II III IV V Pooled Intergroup TEXP SEXP TSCA SSCA CONT SD difference (n = 9) (n = 7) (n = 6) (n = 6) 1In = 12) ρ < 0.05

Pl-dir. 1.11 0.70 0.68 0.50 0.64 0.24 PI-indir. 1.11 0.70 0.68 0.50 0.64 0.24 NF 0.44 0.68 1.26 1.03 0.82 0.35 I, II < III I < IV I*F 0.79 0.65 1.48 0.96 1.02 0.30 I, II, IV, V < III P*F -0.01 0.02 0.68 0.51 0.41 0.30 I, II < III, IV I < V 0*F 0.75 0.36 1.04 0.72 0.52 0.43 II* 0.25 0.71 -0.08 0.67 0.29 0.77 PP* 0.34 0.29 0.49 0.40 0.07 0.55 CC* -0.07 0.04 0.01 -0.07 0.30 0.62 oo* 0.07 0.83 -0.17 0.58 0.83 1.04 < NF/PI -0.41 0.47 - 1.15 0.29 0.18 1.39

Table 3-5: Mean increments in mm resp. degrees per month. 8 Weeks after removal of TE.

Variable I II III IV V Pooled Intergroup TEXP SEXP TSCA SSCA CONT SD difference (n = 9) (n = 7) (n = 6) (n = 6) ([ n = 12) ρ < 0.05

Pl-dir 0.83 0.77 0.74 0.61 0.81 0.15 PI-indir. 0.83 0.77 0.74 0.61 0.81 0.15 NF 0.81 0.85 1.33 1.02 1.02 0.19 I, II, IV, V < III I*F 1.02 1.04 1.64 1.03 1.25 0.23 I, II, IV, V < III P*F 0.19 0.28 0.83 0.47 0.45 0.18 I, II, IV, V < III I < IV 0*F 0.60 0.72 0.81 0.63 0.74 0.23 II* 0.34 0.35 0.17 0.42 0.34 0.31 PP* 0.27 0.21 0.49 0.12 0.27 0.26 CC* 0.01 0.13 0.03 0.03 0.14 0.26 oo* 0.49 0.60 0.14 0.43 0.47 0.43 < NF/PI -0.14 -0.00 -0.76 0.31 -0.10 0.64

75 Chapter 3

In the eight weeks after removal of the tissue expander, the TSCA group was significantly different from the other groups for the parameters NF, I*F and P*F, with significant increases in the TSCA group (Table 3-5). Furthermore, P*F increased significantly more in the SSCA group compared to the TEXP group.

3.5 Discussion

This study has tried to evaluate the effects and further possibilities of TE of the palatal mucoperiosteum in growing cats, with and without pre-existing scars. Since extrapolation to the human clinical situation is important, an animal model was chosen with a deciduous and permanent dentition. Beagle dogs, although apparently suitable for study, were not selected for this experiment, because of their non-humanlike palatal anatomy24·31. The morphology of the palate of the domestic cat appears to be suitable for comparisons with humans2022. It is still impossible to breed non-rodent mammals with identical congenital clefts35. Creating an artificial palatal cleft by surgery has major negative effects on growth and development of the dentomaxillary complex19. This means that no reliable starting-point exists after surgery. To be able to check the validity of the TE technique in scarred tissue, as is usually present in CLP situations, a simulated Langenbeck operation36 was performed. The effects of this surgical intervention are well documented for dogs2327, and assumed to be more or less the same for cats2022. The most prominent growth disturbances due to this type of operation were found in the transverse dental arch measures, which are beyond the scope of the present study. Only minor temporary changes in the antero-posterior dimensions were found25. Also, the present study revealed no significant effect of the simulated Langenbeck operation on any of the parameters in the sagittal plane during TE. As a consequence, the Langenbeck and non- Langenbeck groups were pooled. Palatal TE may interfere with food intake, by mechanical hindrance and expansion related discomfort. This is reported to occur during and shortly

76 Cephalometric analysis after inflation3 To monitor this possible effect on nutrition, the weight gain was carefully monitored and recorded on a weekly basis No decrease or abnormal weight changes were found Complications of TE in the head and neck area are well established3 281819 Complication rates vary from 0 to 69 % depending on surgeon, time, technique and anatomical site Most authors reported high complication rates early in their experience, with a subsequent decrease In a pilot study, the various techniques were practised in order to accrue enough experience to start the experimental study Complication rates in the present study varied from 20 to 25%, depending on the group and the initial filling volume of the expander Erosions, necrosis of the soft tissues, leakage, and extrusions of the expander were the most frequent complications and were seen mainly in the scarred tissue groups In both groups with inflated tissue expanders, a considerable increase in the palatal soft tissue was found, as was expected This proves that TE of the intra oral mucopenosteum is possible, and that it yields at least a temporary tissue gain The effects of the experimental intervention became more prominent with increasing time after the intervention TE and to a lesser extent palatal surgery itself, however, caused some adverse side effects on the growth of bony structures Palatal growth was reduced in the antero-postenor direction, and the anterior naso-maxillary height was increased This is possibly due to the fact that with the increasing volume of the tissue expander, the animals were less able to close their mouths This might result in an overeruption of the maxillary incisors Furthermore, the increased intra-oral mucopenosteal tension caused by the tissue expander may cause palatal tipping of the incisors and a restraining effect on anlero posterior palatal growth The changes in the cranial base region indicated changing proportions of measuring points relative to point F, the optical foramen TE resulted in an increase in the height of the skull in the occipital region This phenomenon was quite unexpected and we can forward no explanation for it In the first weeks after removal of the tissue expanders, nearly all increments in the experimental groups returned to the normal, control values

77 Chapter 3

Later on, some significant differences between the groups developed All these differences were found in the cranial base region, and not in the parameters involved in palatal length and nasomaxillary height This means that, although the growth rates in that area soon after removal of the tissue expander returned to normal, it still is questionable whether real catch up growth takes place beyond the period of eight weeks after removal of the tissue expander These findings indicate that some of the abnormal growth in the maxillofacial complex induced by TE, can persist after tissue expander removal A limitation of this study is the fact that it provides only two- dimensional information about effects in the midsagittal plane Three- dimensional data is needed to elucidate the positive and negative effects of TE From this study it can be concluded that TE of the palatal mucopenosteum in young growing cats is possible, with and without scars of the palatal tissues During growth, however, TE interferes with the antero posterior growth of the bony structures at the palatal level In the vertical direction, the anterior facial and posterior skull height are altered by TE It also interferes with dental development as the incisors become more inferiorly and posteriorly located This may be due to interference with occlusion and traction in the soft tissues overlying the subperiosteal^ located tissue expander Some of these effects, however, seem to be temporary, since there is a relatively accelerated growth at the cranial base after removal of the tissue expander in the scarred TE group

3.6 Literature

1 DE MEY A, MALEVEZ C, LEJOUR M (1990) Treatment of palatal fistula by expansion Br J Plast Surg 43 362 364 2 ABRAMO AC, VIOLA JC, ANGELO AJ (1993) Intraoperative rapid expansion in cleft palate repair Plast Reconstr Surg 91 441-445 3 VAN DAMME PHA, FREIHOFER HPM (1996) Palatal mucopenosteal expansion as an adjunct to palatal fistula repair case report and review of the literature Cleft Palate-Craniofac J 33 255-257

78 Cephalometric analysis

4 MILLARD DR Jr (1980) Cleft craft The evolution of its surgery III Alveolar and palatal deformities Boston Little, Brown, pp 167-262, 809-865 5 WATSON JD, REID CD, PIGOTT RW (1988) The "tadpole flap" - its role in closure ot palatal fistulae Br J Plast Surg 41 485-487 6 NAKAKITA N, MAEDA K, ANDO S, OJIMI H, UTSUGI R (1990) Use of a buccal musculomucosal flap to close palatal fistulae after cleft palate repair Br J Plast Surg 43 452-456 7 EGYEDI Ρ (1977) Utilization of the buccal fat pad for closure of oro-antral and/or oro-nasal communications J Max-Fac Surg 5 241-244 8 VOORSMIT RACA, FENNIS JPM (1992) The buccal fat pad for closure of oro-nasal communications in cleft patients J Cranio-Max-Fac Surg 20 (Suppl 1) 71 9 GUERREROSANTOS J, ALTAMIRANO JT (1966) The use of lingual flaps in repair of fistulas of the hard palate Plast Reconstr Surg 38 123-128 10 PIGOTT RW, RIEGER FW, MOODIE AF (1984) Tongue flap repair of cleft palate fistulae Br J Plast Surg 37 285-293 11 COGHLAN K, O'REGAN B, CARTER J (1989) Tongue flap repair of oro­ nasal fistulae in cleft palate patients J Cranio Max-Fac Surg 17 255-259 12 VAN DER WAL KGH, MULDER JW (1992) The temporal muscle flap for closure of large palatal defects in CLP patients Int J Oral Maxillofac Surg 21 3-5 13 UPTON J, FERRARO N, HEALY G, KHOURI R, MERRELL С (1994) The use of prefabricated fascial flaps for lining of the oral and nasal cavities Plast Reconstr Surg 94 573-579 14 MATSUO K, KIYONO M, HIROSE Τ (1991) A simple technique lor closure of a palatal fistula using a conchal cartilage graft Plast Reconstr Surg 88 334- 337 15 FREIHOFER HPM, BORSTLAP WA, KUIJPERS JAGTMAN AM, VOORSMIT RACA, VAN DAMME PHA, HEIDBUCHEL KLWM, BORSTLAP-ENGELS VMF (1993) Timing and transplant materials for closure ot alveolar clefts J Cranio-Max-Fac Surg 21 143-148 16 BATCHELOR AG, PALMER JH (1990) A novel method of closing a palatal fistula the free fascial flap Br J Plast Surg 43 359-361 17 HASEGAWA K, AMAGASA T, ARAIDA T, MIYAMOTO H, MORITA К (1994) Oral and maxillofacial reconstruction using the free rectus abdominis myocutaneous flap Various modifications for reconstruction sites J Cranio- Max-Fac Surg 22 236-243

79 Chapter 3

18 BARDACH J, EISBACH KJ (1977) The influence of primary unilateral cleft lip repair on facial growth Cleft Palate J 14 88-97 19 MEIJER R, PRAHL В (1978) Influences of different surgical procedures on growth of dentomaxillary complex in dogs with artificially created cleft palate Ann Plast Surg 1 460-465 20 FRENG A (1979) The restorative potential of double layered mucopenosteum A study on experimental mid-palatal clefts in the cat Scand J Plast Reconstr Surg 13 313-320 21 FRENG A (1981) Growth of the middle face in experimental early bony fusion of the vomeropremaxillary, vomeromaxillary and mid-palatal sutural system A Roentgencephalometnc study in the domestic cat Scand J Plast Reconstr Surg 15 117-125 22 VOSS R, FRENG A (1982) Growth of the dental arches after ablation of the mid-palatal suture J Max-Fac Surg 10 259-263 23 BARDACH J, MOONEY MP (1984) The relationship between lip pressure following lip repair and craniofacial growth an experimental study in beagles Plast Reconstr Surg 73 554-555 24 WIJDEVELD MGMM, GRUPPING EM, KUIJPERS-JAGTMAN AM, MALTHA JC (1987) Mucopenosteal migration after palatal surgery in beagle dogs Int J Oral Maxillofac Surg 16 729-737 25 WIJDEVELD MGMM, GRUPPING EM, KUIJPERS-JAGTMAN AM MALTHA, JC (1988) Growth of the maxilla after soft tissue palatal surgery at dillerenl ages on beagle dogs - a longitudinal radiographic study J Oral Maxillofac Sui g 48 204-209 26 BARDACH J, KELLY KM (1990) Does interference with mucopenosteum and palatal bone affect craniofacial growth 9 An experimental study in beagles Plast Reconstr Surg 86 1093-1100 27 BARDACH J, KELLY KM, SALYER KE (1994) Relationship between the sequence of lip and palate repair and maxillary growth an experimental study in beagles Plast Reconstr Surg 93 269-278 28 VAN DAMME PHA, HEIDBUCHEL KLWM, KUIJPERS-JAGTMAN AM, MALTHA JC, FREIHOFER HPM (1992) Cramo-maxillo-facial tissue expansion, experimentally based or clinically empiric9 A review of the literature J Cranio-Max-Fac Surg 20 61-69

80 Cephalometnc analysis

29 MOELLEKEN BRW, MATHES SJ, CANN CE, SIMMONS DJ, GHAFOORI G (1990) Long-term effects of tissue expansion on cranial and skeletal bone development in neonatal miniature swine clinical findings and histomorphometnc correlates Plast Reconstr Surg 86 825-834 30 SCHMELZEISEN R, SCHWIPPER V, TILKORN H, BECKER H, UBERMUTH Τ (1994) Changes in growth of the facial skeleton following implantation of tissue expanders J Cranio-Max-Fac Surg 22 (Suppl 1) 81 31 MULLER G-H (1993) Les applications cliniques de la chimie des silicones Ann Chir Plast Esthet 38 660-666 32 LEVIER RR, HARRISON MC, COOK RR, LANE TH (1993) What is Silicone9 Plast Reconstr Surg 92 163-167 33 MALTHA JC (1982) The process of tooth eruption in beagle dogs PhD Thesis, University of Nijmegen, The Netherlands, ρ 46 34 ERIKSEN E, SOLOW В (1991) Linearity of cephalometnc digitizers Eur J Orthod 13 337-342 35 NATSUME N, MIYAJIMA K, KINOSHITA H, KAWAI Τ (1994) Incidence ot cleft lip and palate in beagles (Letter) Plast Reconstr Surg 93 439 36 LANGENBECK В (1861) Operation der angeborenen totalen Spaltung des harten Gaumens nach einer neuen Methode Dtsch Khn 24 37 COLE RP, GAULT DT, MAYOU BJ, DAVIS PKB (1991) Pain and forehead expansion Br J Plast Surg 44 41-43 38 MANDERS EK, SCHENDEN MJ, FURREY JA, HETZLER PT, DAVIS TS, GRAHAM WP III (1984) Soft-tissue expansion Concepts and complications Plast Reconstr Surg 74 493 507 39 ANTONYSHYN O, GRUSS JS, MACKINNON SE, ZUCKER R (1988) Complications of soft tissue expansion Br J Plast Surg 41 239-250

81

Chapter 4

Three-dimensional morphometric analysis of the effects of subperiosteal palatal soft tissue expansion in growing cats

Philip A. Van Damme Hans Peter M. Freihofer Jaap С. Maltha Anne Marie Kuijpers-Jagtman Martin A. Van 't Hof

International Journal of Oral and Maxillofacial Surgery (1996). In press.

Three-dimensional analysis

4.1 Summary

A prospective longitudinal study in 75 growing cats was conducted to evaluate palatal soft tissue expansion (TE) in cleft lip and palate surgery. In 31 cats, palatal scars were induced by simulated Langenbeck operation at the age of 8 weeks. At the age of 14 weeks, custom-made tissue expanders were inserted in 61 animals. TE was performed by weekly inflation in 33 cats (16 without and 17 with scars) for an eight week period. The remaining 28 cats (14 without and 14 with scars) served as sham groups. A control group was formed by 14 animals (without scars and without tissue expander). The effects of the experimental interventions were evaluated on a series of dental casts during the inflation period and until eight weeks after removal of the tissue expander. The results indicate that TE of the palatal mucoperiosteum is feasible. Till 20 weeks of age, no differences were found between both expansion and sham groups. Thereafter, significant soft tissue surface-area gain was quantified in relation to the base-surface and base-diameter of the tissue expander. Iatrogenic side effects of active TE consisted of significant transverse growth retardation in the anterior part of the bony palate and dento-alveolar structures. After removal of the tissue expanders, some accelerated growth, in the TE, scarred tissue group, was seen. It is concluded that palatal TE is possible in growing cats, with and without the presence of palatal scars, however, this technique, like other kinds of palatal surgery, induces impairment of dentomaxillary growth and development.

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4.2 Introduction

Traditional techniques for correction of palatal defects and oronasal communications in cleft hp-palate-alveolus (CLP) patients are summarized by Millard'6 Various loco-regional pedicled flaps are currently used, such as tadpole flaps43, different lingual flaps9181", buccal musculo-mucosal flaps29, buccal fat pad flaps1141, and temporal muscle and/or fascial flap171419 These flaps do not always yield good results Complete closure can be achieved in approximately 35 to 70% of cases at the first attempt, depending on the size and site of the communications29 Closure generally becomes more difficult and the techniques become less reliable, when multiple previous attempts at fistula closure have been performed There are several disadvantages to these different techniques Mostly they are two-stage procedures and create a donor site defect, and in most cases tissue of different origin, texture, and characteristics are used for the repair For example, lingual flaps may cause temporary impairment of tongue function, with speech interference, and sometimes intermaxillary fixation is needed during the healing phase Free micro vascularized flaps, such as the radial forearm fasciocutaneous flap and the rectus abdominis myocutaneous flap, are hazardous, time consuming, bulky and often cause donor site morbidity and visible scars7819 TE has been used in the cranio-maxillofacial region since 198116 The use of TE for facilitating the closure of palatal defects in CLP is new and has seldom been reported1 " 3537 Just recently, De Mey et al and Abramo et al advocated the use of a soft tissue expander for closure of palatal fistulae' " Few studies have reported on the effects of TE on growth and development in growing individuals2731 Surgical interventions on the palate have always negative effects on growth and development of the dento-maxillofacial complex161516 25 42 u 4

86 Three-dimensional analysis negative effects of subperiosteal palatal TE. Therefore a longitudinal animal experiment was performed, including a three-dimensionally morphometric soft- and hard-tissue analysis.

4.3 Material and methods

Seventy five domestic tomcat kittens were assigned to five groups; I: tissue expansion group, TEXP (n = 16) II: sham group, SEXP (n = 14) III: tissue expansion in scar tissue group, TSCA (n = 17) IV: sham in scar tissue group, SSCA (n = 14) V: control group, CONT (n = 14). The animals were housed under normal laboratory conditions in the Central Animal Laboratory, University of Nijmegen, and received a diet consisting of granules, canned meat, and drinking water ad libitum. The cats were weighed every week and prior to any intervention. All interventions were performed under standard general anaesthesia, induced by 30 mg/kg NimatekR i.m. (Ketamine HCL 100 mg/ml, Α.U.V. Cuijk, The Netherlands) with 0.5 ml Atropine i.m. (Atropine Sulphate 0.5 mg/ml) and maintained after intubation with a mixture of 02, N,0 and EthraneR (Enflurane, Abbott B.V., Amstelveen, The Netherlands). A simulated Langenbeck operation was performed under sterile conditions in the TSCA and SSCA groups at the age of 8 weeks (Langenbeck, 1861)20. For this purpose, the oral cavity and perioral region were disinfected with BetadineR solution (Povidon-iodine 10%, Dagra-Pharma B.V., Diemen, The Netherlands). A standardized lenticular soft tissue defect was created in the median region of the palate by excising a mucoperiosteal flap. This flap extended from just behind the incisive papilla to the caudal margin of the hard palate. The maximum width of the flap was one third of the transverse distance between the deciduous first molars. On both sides relaxation incisions were made adjacent to the posterior teeth. The mucoperiosteum was elevated and the soft tissue defect was closed in the midline, in one layer with VicrylR 5-0 (Ethicon, Johnson & Johnson

87 Chapter 4

Company, Amersfoort, The Netherlands), leaving two areas of denuded bone adjacent to the posterior teeth. Healing was allowed for six weeks until the age of 14 weeks. At 14 weeks of age, custom-made, hemispherical soft tissue expanders (CUIR, Inamed B.V., Breda, The Netherlands) were inserted in groups TEXP, TSCA, SEXP, and SSCA (Fig. 4-1).

СЛ

У)

χ

CD

Figure 4-1: Custom-made hemispheric tissue expander with self sealing filling port. Base diameter in empty state 12.0 mm, height 6.0 mm. Filled, height 12.0 mm.

88 Three-dimensional analysis

Figure 4-2: Schematic drawing of the position of the tissue expander

The silicone tissue expanders had a diameter of 12 mm at their base and a filling volume of 1 ml2128. They had a filling tube with an incorporated self- sealing port. To introduce the expander, a standardized para-marginal palatal incision trom left to right canine was made to create a submuco-periosteal pocket of approximately 15x15 mm, by elevation of the mucoperiosteum Both anterior palatal neurovascular bundles were coagulated, while the posterior palatal neurovascular bundles were spared. A 2 mm incision was made on the left buccal side, between canine and first deciduous molar and the gingiva and the mucoperiosteum were tunneled to allow through the external filling tube. The palatal incision was closed with VicrylR 5-0 sutures No surgery was performed in group CONT, but the animals were subjected to the same regime of recording. All inserted expanders were initially filled with 0.1 ml of a radiopacque solution (AngiografinR, Meglumine-amidotrizoate 0.650 g/ml; 306 mg I/ml, Schering A.G , Berlin, Germany). Perioperative antibiotics were administered; preoperatively 0.5 ml AlbipenR 15%, l.m. (Ampicilhn-anhydrate 150 mg/ml, Gist-brocades N.V., Delft, The Netherlands), and postoperatively on day 1 and day 3, 1.0 ml

89 Chapter 4

AlbipenR L.A., i.m. (Ampicillin-anhydrate 100 mg/ml Gist-brocades N.V., Delft, The Netherlands). The wounds were inspected daily for the first week after surgery or, when indicated, for a longer period. Healing was allowed for two weeks until the age of 16 weeks. Hereafter, approximately 0.1 ml AngiografinR was added weekly to each tissue expander in the TEXP and TSC A groups, using a blunt 22-gauge needle, until at the age of 24 weeks the maximum volume was reached (Fig. 4-3). Then the tissue expanders and the tissue surplus were surgically removed and the wound in the midline was closed with VicrylR 5-0 sutures. The animals were followed for another eight weeks, using the same regime of recording of data.

Figure 4-3: Clinical result of TE at the age of 24 weeks, after reaching the maximum volume.

Thirteen cats had to be excluded from the study due to technical failures. Sixty-two cats could be followed until the age of 20 weeks and 52 cats were followed for the entire inflation period up to 24 weeks of age. Forty animals were studied during a period of eight weeks after removal of the tissue expander.

90 Three-dimensional analysis Dental casts were made every two weeks, by means of an alginate impression (CA37R Superior Pink, Cavex Holland B.V., Haarlem, The Netherlands) in individually designed impression trays (Fig. 4-4).

Figure 4-4: Dental cast model of the same cat at the age of 24 weeks. Lateral view (a), occlusal view (b).

91 Chapter 4

The casts were analyzed and digitized using a standard Reflex Microscope (Reflex Measurement Ltd, Somerset, UK)12·32. Four observers were involved, independently identifying the following 10 defined points with a magnification of 10 times (Fig. 4-5): 1 = most posterior point of the rugae in the midline 2 and 10 = posterior intersection marginal gingiva third deciduous molar (intercuspid third premolar) on the right and left side respectively 3 and 9 = anterior intersection marginal gingiva second deciduous molar (second premolar) on the right and left side respectively 4 and 8 = posterior intersection marginal gingiva canine on the right and left side respectively 5 and 7 = posterior intersection marginal gingiva third incisor on the right and left side respectively 6 = anterior intersection interdental papilla central incisors in the midline. On the dental cast models a virtual 2.0 mm mesh grid was generated, and the Χ, Y and Z-coordinates of each nodal point were determined. In this way a 3-D field of points was created, which was limited by the 10 defined points. Also, a virtual 2-D area was denominated by these points. The following surfaces, distances and angles were calculated (Fig. 4-5): 2-D and 3-D surface areas transverse distance from the highest point of registration, 3-D minus 2-D calculation sagittal distance from the highest point of registration, 3-D minus 2-D calculation 1-6 = antero-posterior distance (APD) 2-10 = posterior transverse distance (PTD) 3-9 = middle transverse distance (MTD) 4-8 = anterior transverse distance (ATD) 2-3-4 = right lateral angle (RLA) 8-9-10 = left lateral angle (LLA). The error of the procedures was determined by repeated measurements of 304 models of different cats at different ages.

92 Three-dimensional analysis

Figure 4-5: Schematic drawing of a cast model showing the 10 defined points used for determination of the 3-D and 2-D surface areas. Deciduous dentition (a), permanent dentition (b).

4.3.1 Statistical procedures Means and standard deviations were calculated for the increments of all variables in all groups. Comparison between the groups was carried out by one-way ANOVA and analysis of co-variance, and if significant differences between groups were found, Tukey's multiple comparisons test was used for evaluation of these differences.

93 Chapter 4

Analysis was performed for the period of 14 to 20 weeks of age for 62 cats and for the period of 14 to 24 weeks of age for 52 cats In 40 cats the period of 8 weeks after removal of the tissue expanders was evaluated Intraobserver and interobserver variability tests were performed to check the accuracy and reproducibility of the procedures

4.4 Results

4 4 1 Error analysis The duplicate measurements at different ages showed a mean total error of the anterior, middle and posterior transverse distance measurements of maximally 0 2 mm The mean total error of the angle measurements was 1 6 degrees Surface area calculations showed mean total errors of 15 mm2 for 2-D surfaces, 25 mm2 for 3-D surfaces, and 17 mm2 for 3-D - 2-D differences Based on these results the accuracy and reproducibility of the methods were considered to be acceptable

442 Effects of the interventions Mutual comparison of all groups by one way ANOVA at the age of 20 weeks revealed that none of the two-dimensional parameters showed a significant difference between comparable scarred and non-scarred groups At the age of 24 weeks the anterior transverse distance (ATD) and the left lateral angle (LLA) showed significant differences between the groups (Table 4 1a) In group TSCA and SSCA, ATD was significantly smaller than in group SEXP The LLA was smaller in group CONT than in groups TEXP, TSCA and SSCA (Table 4-la) During the whole expansion period of ten weeks after insertion of the tissue expander (Table 4 lb), significant differences were observed for the increments of the variables ATD and LLA In the TSCA group there was a smaller increment of ATD than in the CONT and SEXP groups The LLA diminished significantly in the CONT and SEXP groups, whereas it increased in the TEXP group (Table 4 lb)

94 Three-dimensional analysis

Table 4-la: Values of parameters at 24 weeks in mm resp degrees

Variable 1 11 III IV V Pooled Intergroup TEXP SEXP TSCA SSCA CONT SD differences (n = 13) (n = 10) (n = 7) (n = 8) (n = 14) ρ < 005

PTD 28 90 29 59 29 00 28 40 29 06 0 90 MTD 22 00 22 74 21 82 21 94 21 78 0 73 ATD 17 87 18 70 17 07 17 44 18 07 0 85 III, IV < II RLA 176 86 175 61 176 36 177 26 175 09 2 46 LLA 177 75 174 79 176 80 177 60 173 02 2 18 V < I, III, IV

Table 4-lb: Increments o.f parame•ters from 14 to 24 weeksin mm resp degrees per month

Variable I II III IV V Pooled Intergroup TEXP SEXP TSCA SSCA CONT SD differences (n= 13) (η = 10) (η = 7) (n = 8) :(n = и) ρ < 005

PTD 1 22 1 32 1 08 1 25 1 51 0 34 MTD 1 22 1 30 1 02 1 05 1 17 0 26 ATD 0 93 1 12 0 57 0 96 1 13 0 32 III < II, V RLA 0 94 -0 63 1 07 0 45 -0 68 1 48 LLA 0 94 -1 05 0 59 -0 23 -1 11 1 28 II, V < I

Analysis of co-variance showed that scarring itself was not decisive for the three-dimensional parameters, surface area, transverse and sagittal distance. This means that pooling of the groups TEXP and TSCA as well as of the groups SEXP and SSCA was allowed, which resulted in a group TE, consisting of 29 (20) cats and a group SH, consisting of 22 (17) animals. The three groups TE, SH and CO (controls, η = 14) were mutually compared by one-way ANOVA, and significant differences, if any, were subsequently explored by means of Tukey's multiple comparisons test (Tables 4-2a, 4-2b). The effects of TE on the soft tissues were represented by the differences between three-dimensional minus two-dimensional data: surface area, transverse distance and sagittal distance.

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Table 4-2a: Differences 3-D and 2-D values at 20 weeks in mm2 resp mm

Variable I + IIIIII II + IIV V Intergroup TE SH CO differences (η = 29) (η = 22) (η = 14) ρ < 0 05

Surface 119 07+24 34 73 01+18 17 44 28 ± 6 70 CO < SH area SH, CO < ТЕ

Transverse 4 66 ± 1 60 1 68 + 1 17 1 06 + 1 90 SH, CO < ТЕ distance

Sagittal 3 80 + 0 91 1 79 ± 0 81 0 71 + 0 39 CO < SH distance SH, CO < ТЕ

Table 4-2b: Differences 3-D and 2 D values at 24 weeks in mm2 resp mm

Variable I + III II + IV V Intergroup ТЕ SH со differences (η = 20) (η = 17) (η = 14) ρ < 005

Surface 148 12 + 48 49 69 05 + 34 54 52 49 ± 17 71 SH, CO < ТЕ area

Transverse 4 83 ± 2 21 1 82 + 1 71 0 52 + 0 34 CO < SH distance SH, CO < ТЕ

Sagittal 5 14 ± 1 24 1 68 ± 0 73 1 34 + 0 59 SH, CO < ТЕ distance

At the age of 20 weeks, the pooled groups TE, SH and CO were statistically different for the 3-D - 2-D surface area, the 3-D - 2-D transverse and sagittal distances (Table 4-2a). The values were highest in the ТЕ group, and lowest in the CO group Only the transverse distance in the SH group was not significantly different from the CO group. At the age of 24 weeks, analogous findings could be observed, however, the SH and CO groups were then statistically different for the 3-D - 2-D transverse distance, and not for the 3-D - 2-D surface-area and sagittal distance (Table 4-2b)

96 Three-dimensional analysis The increments during the initial four weeks after removal of the tissue expander showed significant differences between the groups for all parameters except LLA (Table 4-3). The transverse distances in group TSC A were always increasing faster than in group TEXP. The same phenomenon was found during the last four weeks of the eight week period after removal of the tissue expander for the parameters MTD and ATD (Table 4-4). The increments during the whole period of eight weeks after removal of the tissue expander were analogous to the situation four weeks after removal of the tissue expander (Table 4-5). Group TEXP seemed to be the slowest growing group on all 3 transverse distances, and group TSC A the fastest (Table 4-5).

Table 4-3: Incrementsfirst 4 weeks after removal of TE in mm resp. degrees.

Variable I II III IV V Pooled Intergroup TEXP SEXP TSCA SSCA CONT SD differences (n = 10) (n = 7) (n = 6) (n = 6) ( η = 12) ρ < 0.05

PTD 0.48 0.56 1.04 0.82 0.56 0.32 I, V < III MTD -0.74 0.42 1.03 0.20 0.50 0.76 I < II, III, V ATD 0.55 0.45 1.74 0.98 0.46 0.46 I, II, IV, V < III RLA -5.04 -0.45 2.86 -3.40 1.38 3.13 I < II, III, V IV < III, V LLA -4.00 1.01 1.42 0.47 0.30 5.30

Soft tissue surface-area gain was defined as the difference between 3-D - 2-D surface area in the TE group minus the 3-D - 2-D surface area in the CO group. The gain could be quantified as 66% of the base-surface of the hemispherical tissue expander (r = 6, πτ2 = 113 mm2) at the age of 20 weeks, and 85% at 24 weeks. If related to the base-diameter of the tissue expander (d = 12 mm), total increments of 36% and 32%, in transverse and sagittal direction respectively, could be reached.

97 Chapter 4

Table 4-4: Increments last 4 weeks after removal of TE in mm resp degrees

Variable I II III IV V Pooled Intergroup TEXP SEXP TSCA SSCA CONT SD differences (rι = 10) (n = 6) (n = 6) (n = 6) (n = 12) ρ < 0 05

PTD 0 48 0 25 0 71 0 38 0 45 0 30 MTD 0 28 0 37 0 78 0 71 0 55 0 32 I < III ATD 0 13 -0 02 0 60 0 18 0 25 0 24 I, II, IV, V < III RLA 0 56 2 26 1 94 3 05 2 14 2 18 LLA 0 34 2 07 2 29 3 20 2 38 2 62

Table 4-5: Increments 8 weeks at'ter removal of TE in mm resi> degrees

Variable I II III IV V Pooled Intergroup TEXP SEXP TSCA SSCA CONT SD differences (n = 10) (n = 6) (n = 6) (n = 6) ( η = 12) ρ < 005

PTD 0 48 0 40 0 86 0 59 0 50 0 19 I, II, V < III MTD -0 23 0 39 0 90 0 45 0 52 0 39 I < II, III, IV, V ATD 0 34 0 22 1 16 0 58 0 36 0 25 I, II, IV, V < III RLA -2 22 0 90 2 40 -0 17 1 75 1 97 I < II, III, V LLA -1 81 1 53 1 76 1 82 0 83 2 68

Discussion

The present study was aiming at the evaluation of the possibilities and effects of TE in the palatal mucopenosteum in growing cats, with and without pre­ existing scars, using a hemispherical tissue expander with a 12 mm diameter, a 6 mm height and an external filling port Palatal TE may interfere with food intake, by mechanical hindrance and expansion related discomfort, which is reported to occur during and shortly after inflation10 To monitor this possible effect, the weight gain was carefully recorded on a weekly basis, and no detrimental effects were found Complications of TE in the head and neck area were reported firstly by Manders et al in 1984, and later on by Antonyshyn et al223 Complication rates vary from 0 to 69% depending on surgeon, time, technique and

98 I liree-dimensional analysis anatomical site Most authors reported high complication rates early in their experience, with a subsequent decrease. Complication rates in the present study varied from 20 to 25%, depending on the group and the initial filling of the expander Erosions, necrosis of the soft tissues, leakage and extrusions of the expander were the most frequent complications and were seen mainly in the scarred tissue groups To be able to check the validity of the TE technique in scarred tissue, as is usually present in CLP situations, a simulated Langenbeck operation20, was performed in order to induce palatal scars. The dentomaxillary anatomy and the effects of simulated Langenbeck operation in dogs are well documented224445, and assumed to be more or less the same for cats1516"2 The most prominent growth disturbances due to this type of surgery were found in the transverse dental arch measures. The present study revealed no significant influence of the simulated Langenbeck operation on the parameters in the transversal plane until the age of 20 weeks. As a consequence, the Langenbeck and non-Langenbeck groups were pooled for that period Initially there were only few significant differences in the transverse distances However, in time the differences became significant in the anterior palatal region, indicating a continuous influence of the combination of simulated Langenbeck operation and TE TE caused a significant increase in the palatal soft tissue surface area, as was expected It yields a temporary tissue gain of approximately 85% of the base surface of a hemispheric tissue expander after eight weeks of active expansion In the literature, tissue gain was initially related to the base dimensions of the tissue expander, and it was advised to choose the expander base area to be 1-2.5 times the defect to be closed113640 There is a wide range of area increase percentages, varying from 12 5 to 110%, based either on estimation, measuring, or on calculation Theoretically, a 100% area gain can be reached in case a hemispherical tissue expander is used (surface of a hemisphere minus surface of a circle, divided by the surface of the circle: 2πΓ2 - 7ГГ / 7гг2 — 1) Van Rappard and Shively used different mathematical formulas for the expected gain, and concluded that area gain is only a function of the height of the expander and not of its base diameter"40. In vivo Van Rappard was able to realize a surface area increase of 25%-38% of the

99 Chapter 4 mathematically expected area gain (depending on the shape and height of the tissue expander) The findings in this study for soft tissue gain in transversal and sagittal directions (36% and 32% respectively) are rather favourable and corresponding to data found by Nordstrom and Devine (1985) (39% and 27%) and VanderKolk et al (1987) (45% and 30 %)16 Palatal surgery and TE cause some adverse side effects in the bony palatal and dento alveolar structures The anterior transverse distance increment was significantly smaller in the TE in scar tissue situation than in the sham expansion and control groups, if the whole expansion period was considered However, after removal of the tissue expander significantly increased growth of almost all parameters, in the TE in scar tissue group, takes place The differences between the left and right lateral angles may be related to the fact that the filling tube was located on the left side in all cases An explanation may be found in the formation of scar tissue after the small buccal incision and tunneling of the mucopenosteum, with possible deterioration of the lateral growth at one side The first four weeks after removal of the tissue expanders, nearly all parameters in the experimental expansion without scar group were smaller than in the expansion with scar group, and some were less than the control values The effects after removal of the tissue expander were less pronounced in the second period of four weeks than in the first In general, the growth increments of the cats decreased with age However, significant differences in increments remain over the total period of eight weeks, indicating a prolonged effect It can be concluded that TE of the palatal mucopenosteum is very well possible in young cats, with and without scars of the palatal tissues The soft tissue gain as quantified by this method ot morphometric analysis, was related to the base-surface and base-diameter of the tissue expander, being 85%, 36% and 32% for surface area increase, transverse and sagittal distance increments respectively The effects on growth and development comprise mainly retardation of transverse growth of the dento-alveolar structures at the anterior palatal level, probably caused by traction of the soft tissues overlying

100 Three dimensional analysis the subperiosteal^ located tissue expander In the TE in scarred tissue group, these elfects are temporary, since there is accelerated growth in the episode after removal of the tissue expander Future histomorphological analysis will probably give more insight into tissue reactions, cellular and intercellular matrix responses to TE to explain the ccphalometncally and morphometncally found phenomena

4.6 Literature

1 ABRAMO AC, VIOLA JC, ANGELO AJ (1993) Intraoperative rapid expansion in clett palate repair Plast Reconstr Surg 91 441-445 2 ANTONYSHYN O, GRUSS JS, MACKINNON SE, ZUCKER R (1988) Complications of sott tissue expansion Br J Plast Surg 41 239-249 3 BARDACH J, EISBACH KJ (1977) The influence of primary unilateral cleft lip repaii on facial growth Cleft Palate J 14 88 97 4 BARDACH J, KELLY KM (1990) Does interference with mucopenosteum and palatal bone affect craniofacial growth } An experimental study in beagles Plast Reconstr Surg 86 1093-1100 5 BARDACH J, KELLY KM, SALYER KE (1994) Relationship between the sequence of lip and palate repair and maxillary growth an experimental study in beagles Plast Reconstr Surg 93 269-278 6 BARDACH J, MOONEY MP (1984) The relationship between lip pressuie following lip repair and craniofacial growth an experimental study in beagles Plast Reconstr Surg 73 544-555 7 BATCHELOR AG, PALMER JH (1990) A novel method of closing a palatal listula the tree fascial flap Br J Plast Surg 43 359-361 8 CHEN HC, GANOS DL, COESSENS ВС, KYUTOKU S, NOORDHOFF MS (1992) Free forearm flap closure ol difficult oronasal fistulas in cleft palate patients Plast Reconstr Surg 90 757 762 9 COGHLAN K, O'REGAN B, CARTER J (1989) Tongue flap repair of oro­ nasal tìstulae in cleft palate patients J Cranio-Max-Fac Surg 17 255-259 10 COLE RP, GAULT DT, MAYOU BJ, DAVIS PKB (1991) Pain and forehead expansion Br J Plast Surg 44 41-43 11 DE MEY A, MALEVEZ C, LEJOUR M (1990) Treatment of palatal fistula by expansion Br J Plast Surg 43 362-364

101 Chapter 4

12 DRAGE KJ, ORTH M, WINZAR CF, KILLINGBACK N (1991) Method errors recorded by inexperienced operators ot the Reflex Microscope Br J Onhod 18 309-313 13 EGYEDI Ρ (1977) Utilization ot the buccal fat pad for closure of oro antral and/or oro-nasal communications J Max-Fac Surg 5 241 244 14 FREIHOFER HPM, BORSTLAP WA, KUIJPERS-JAGTMAN AM, VOORSMIT RACA, VAN DAMME PHA, HEIDBUCHEL KLWM, BORSTLAP-ENGELS VMF (1993) Timing and transplant materials for closure ot alveolar clefts J Cranio Max-Fac Surg 21 143-148 15 FRENG A (1979) The lestorative potential ot double layered mucopenosteum A study on experimental mid-palatal clefts in the cat Scand J Plast Reconstr Surg 13 313-320 16 FRENG A (1981) Growth ol the middle face in experimental early bony fusion of the vomeropremaxilldry, vomeromaxillary and mid palatal sutural system A roentgencephalometnc study in the domestic cat Scand J Plast Reconstr Surg 15 117 125 17 FURNAS DW (1987) Temporal osteocutaneous island flaps for complete reconstruction of cleft palate defects Scand J Plast Reconstr Surg 21 119 128 18 GUERREROSANTOS J, ALTAMIRANO JT (1966) The use of lingual flaps in repair of fistulas of the hard palate Plast Reconstr Surg 38 123-128 19 HASEGAWA K, AMAGASA T, ARAIDA T, MIYAMOTO H, MORITA К (1994) Oral and maxillofacial reconstruction using the tree rectus abdominis myocutaneous flap J Cranio Max-Fac Surg 22 236-243 20 LANGENBECK В (1861) Operation des angeborenen totalen Spaltes des harten Gaumens nach einer neuen Methode Dtsch Khn 8 231 21 LEVIER RR, HARRISON MC, COOK RR, LANE TH (1993) What is Silicone9 Plast Reconstr Surg 92 163-167 22 MALTHA JC (1982) The process of tooth eruption in beagle dogs PhD Thesis, University of Nijmegen, The Netherlands, ρ 46 23 MANDERS EK, SCHENDEN MJ, FURREY JA, HETZLER PT, DAVIS TS. GRAHAM WP III (1984) Soft-tissue expansion Concepts and complications Plast Reconstr Surg 74 493-507 24 MATSUO K, KIYONO M, HIROSE Τ (1991) A simple technique for closure ot a palatal fistula using a conchal cartilage graft Plast Reconstr Surg 88 334- 337

102 Three dimensional analysis

25 MEIJER R, PRAHL В (1978) Influences of different surgical procedures on growth of dentomaxillary complex in dogs with artificially created cleft palate Ann Plast Surg 1 460-465 26 MILLARD Jr DR (1980) Cleft craft The evolution of its surgery III Alveolar and palatal deformities Boston Little, Brown & Co , pp 809-865 27 MOELLEKEN BRW, MATHES SJ, CANN CE, SIMMONS DJ, GHAFOORI G (1990) Long-term effects of tissue expansion on cranial and skeletal bone development in neonatal miniature swine clinical findings and lnstomorphometric correlates Plast Reconstr Surg 86 825-834 28 MULLER G-H (1993) Les applications cliniques de la chimie des silicones Ann Chir Plast Esthet 38 660-666 29 NAKAKITA Ν, MAEDA К, ANDO S, OJIMI H, UTSUGI R (1990) The use of a buccal musculomucosal flap to close palatal fistulae after cleft palate repair Br J Plast Surg 43 452-456 30 PIGOTT RW, RIEGER FW, MOODIE AF (1984) Tongue flap repair of cleft palate tistulae Br J Plast Surg 37 285-293 31 SCHMELZEISEN R, SCHWIPPER V, TILKORN H, BECKER H, UBERMUTH Τ (1994) Changes in growth of the facial skeleton following implantation of tissue expanders J Cramo-Max-Fac Surg 22 (Suppl 1) 81 32 SETCHELL DJ (1984) The Reflex Microscope - an assessment of the accuracy of 3-dimensional measurements using a new metrologica! instrument J Dent Res 64 493 33 SHIVELY RE (1988) Surface-area increase in tissue expansion (Discussion) Plast Reconstr Surg 82 838 839 34 UPTON J, FERRARO N, HEALY G, KHOURI R, MERRELL С (1994) The use of prefabricated fascial flaps for lining of the oral and nasal cavities Plast Reconstr Surg 94 573-579 35 VAN DAMME PHA, FREIHOFER HPM (1996) Palatal mucopenosteal expansion as an adjunct to palatal fistula repair case report and review of the literature Cleft Palate-Craniofac J 33 255-257 36 VAN DAMME PHA, HEIDBUCHEL KLWM, KUIJPERS-JAGTMAN AM, MALTHA JC, FREIHOFER HPM (1992) Cramo-maxillo-facial tissue expansion, experimentally based or clinically empiric9 A review of the literature J Cranio-Max-Fac Surg 20 61-69

103 Chapter 4

37 VAN DAMME PHA, MALTHA JC, KUIJPERS-JAGTMAN AM, FREIHOFER HPM, VAN 'T HOF MA (1995) Two-dimensional cephalometnc analysis of the effects of subperiosteal palatal soft tissue expansion in growing cats Accepted by Plast Reconstr Surg 38 VANDEPUT JJ, DROOGMANS B, TANNER JC (1995) Closure of palatal fistulas using a dermis-tat graft Plast Reconstr Surg 95 1105-1107 39 VAN DER WAL KGH, MULDER JW (1992) The temporal muscle Пар for closure of large palatal defects in CLP patients Int J Oral Maxillofac Surg 21 3 5 40 VAN RAPPARD JHA, MOLENAAR J, VAN DOORN К, SONNEVELD GJ, BORGHOUTS JMHM (1988) Surface-area increase in tissue expansion Plast Reconstr Surg 1988 82 833-837 41 VOORSMIT RACA, FENNIS JPM (1992) The buccal fat pad for closure of oio-nasal communications in cleft patients J Cramo-Max-Fac Surg 20 (Suppl 1) 71 42 VOSS R, FRENG A (1982) Growth of the dental arches after ablation of the mid palatal suture J Max-Fac Surg 10 259-263 43 WATSON JD, REID CD, PIGOTT RW (1988) The "tadpole flap" - its role in closure ot palatal fistulae Br J Plast Surg 41 485-487 44 WIJDEVELD MGMM, GRUPPING, EM, KUIJPERS-JAGTMAN AM, MALTHA JC (1987) Mucopenosteal migration after palatal surgery in beagle dogs Int J Oral Maxillofac Surg 16 729 737 45 WIJDEVELD MGMM, GRUPPING EM, KUIJPERS-JAGTMAN AM, MALTHA JC (1988) Growth of the maxilla after soft tissue palatal surgery at different ages on beagle dogs a longitudinal radiographic study J Oral Maxillofac Surg 48 204-209

104 Chapter 5

Histological analysis of the effects of palatal mucoperiosteal soft tissue expansion in growing cats

Philip A. Van Damme Jaap С. Maltha Hans Peter M. Freihofer Anne Marie Kuijpers-Jagtman

Submitted to International Journal of Oral and Maxillofacial Surgery, 1996.

Histological evaluation

5.1 Summary

The purpose of this study is to evaluate the effects of palatal mucopenosteal soft tissue expansion (TE) at the light-microscopic level Seventy-five growing tomcats were assigned to five different groups In four groups, custom-made soft tissue expanders were inserted, and in two ot these groups they were actively expanded by weekly inflation, two groups served as sham and one group as control In two groups, scarring of the palatal soft tissues was performed to mimic the human cleft palate situation Expansion of the palatal mucopenosteum was feasible, in scarred as well as non-scarred tissues, with temporary soft tissue gain There was, however, thinning of the epithelial layer, reorientation of the subepithelial collagenous fibres and development of a fibrous capsule No increased vascularity in the outer layers of the capsule was seen Contractility of the capsule was attributed to the presence of elastic fibres and myofibroblasts The bony palatal shelf was eroded by pressure induced osteoclastic activity at the base of the expander The palatine and vomeropalatine growth centres may have been compromised by the TE Scarring at the age of eight weeks did not demonstrate increased amounts of Sharpey's fibres, and no striking effects on the architecture of the soft and/or hard tissues were seen At the light-microscopic level, signs of temporary gain of soft tissues (loan) were evidenced Thinning of the original layers, with traction on the surrounding palatal and dento-alveolar structures, may be an explanation for the retardation ot growth and development during palatal mucopenosteal TE This has been demonstrated earlier both cephalometncally and morpho- metncally

107 Chapter 5

5.2 Introduction

TE of' the skin has been frequently performed since 1976 (Radovan, 1982, 1984). Histological data on skin expansion are well established (Austad et ai, 1982, 1986; Pasyk et ai, 1982, 1987, 1988; Leighton et al., 1986, 1988; Austad, 1987; VanderKolk et ai, 1988; Van Rappard et ai, 1988; Timmenga, 1992). Quantitative histological analysis after skin expansion in experimental animals and humans (Pasyk et al., 1988; Van Rappard, 1988) indicates that, in general, the epidermis thickens, because of an increase in mitotic activity, independent of site and time of expansion, and age of the patient or animal. Contradictory data have been reported regarding the dermis where thinning as well as thickening can occur (Austad et ai, 1982; Pasyk et ai, 1982, 1987, 1988; Cherry étal., 1983; Brobmann and Huber, 1985; Lee et al., 1985; Leighton et ai, 1988; VanderKolk et al., 1988; Ricciarelli et al., 1989; Van Damme et al., 1992). The subcutaneous tissues, such as fat and striated muscle, however, show a significant thinning. Many authors mention a fibrous capsule surrounding the tissue expanders, with varying thickness, and separate layers within itself, and increased vascularity (Pasyk et ai, 1987, 1988; Antonyshyn et ai, 1988; Leighton et ai, 1988; Saouma et al., 1989; Van Damme et ai, 1992). Inflammatory reactions, with occasional foreign body giant cells, calcification and bone formation, have also been reported (Leighton et al., 1988; Pasyk et ai, 1988; LaTrenta et al., 1988). The effects of TE on bone can be characterized by the presence of reversible bony defects, with osteoclastic resorption, remodelling and periosteal reaction with deposition of new bone (Moelleken et al., 1990; Van Damme et ai, 1992; Schmelzeisen et ai, 1994). The histological effects of intra-oral TE, however, are unknown, although various case reports of clinical intra-oral application of the TE tech­ niques have been published (Van Rappard, 1988; Van Damme et al., 1992; Van Damme and Freihofer, 1996). TE is used as an adjunct to augmentation of the alveolar process for prosthetic reasons (Bonomo, 1986; Lew et ai, 1986; Wittkampf, 1989), and for closure of residual oronasal fistulae in cleft palate surgery (CLP) (De Mey et ai, 1990; Abramo et al, 1993; Van Damme and Freihofer, 1996).

108 Histological evaluation

Application of inira-oral TE might be an interesting extension of the existing techniques for reconstruction of palatal defects m young cleft palate patients However, when TE interferes with growth and development in young individuals, it is not the appropriate technique Palatal surgery in animals and humans can induce major negative effects on growth and development of the dentomaxillary complex (Meijer and Prahl, 1978, Bardach and Kelly, 1990, Bardach et al , 1994, Van Damme et al , 1996a,b) Scarred tissue, as is usually present in CLP situations, was induced in the present investigation by a simulated Langenbeck operation (Langenbeck, 1861) The effects of this surgical intervention are well documented for dogs (Wijdeveld et al , 1987, 1988, 1989, 1991, In de Braekt, 1992, Leenstra et al , 1995), and are assumed to be more or less the same for cats (Freng, 1979, 1981, Voss and Freng, 1982, 1983) The most prominent growth disturbances due to this type of surgery were found in the transverse dental arch measures, while minor temporary changes were found in the antero­ posterior dimensions (Wijdeveld et al , 1988, Van Damme et al , 1996 a,b) The histomorphology of the palate of the domestic cat appears to be suitable for comparisons with humans (Freng, 1979, 1981, Voss and Freng, 1982, 1983, Provenza, 1988, Bhaskar, 1990, Burkitt et al , 1992, Ten Cate, 1994) The aim of the present histological study is to evaluate the effects of inlra-oral palatal TE on the palatal mucopenosteum and the palatal bone of growing cats, with and without pre-existing scars

5.3 Material and methods

Seventy five domestic tomcat kittens aged 8 weeks were assigned to five groups, tissue expansion group, TEXP (n = 16) sham group, SEXP (n = 14) III tissue expansion in scar tissue group, TSCA (n = 17) IV sham in scar tissue group, SSCA (n = 14) V control group, CONT (n = 14)

109 Chapter 5

The animals were housed under normal laboratory conditions in the Central Animal Laboratory, University of Nijmegen, and received a diet consisting of granules, canned meat, and drinking water ad libitum. The cats were weighed every week and prior to any intervention All interventions were performed under standard general anaesthesia, induced by 30 mg/kg NimatekR ι m. (Ketamine HCL 100 mg/ml, A U.V Cuijk, The Netherlands) with 0 5 ml Atropine ι m (Atropine Sulphate 0.5 mg/ml) and maintained after endotracheal intubation with a mixture of 02 R (1 1/min), N20 (2 1/min) and 0 5-4 0% Ethrane (Enflurane, Abbott В V , Amstelveen, The Netherlands)

Figure 5-1: Schematic drawings of simulated Langenbeck operation

A simulated Langenbeck operation was performed under sterile conditions in the TSCA and SSCA groups at the age of 8 weeks. For this purpose, the oral cavity and perioral region were disinfected with BetadineR solution (Povidon- ïodine 10%, Dagra-Pharma В V , Diemen, The Netherlands) Hereafter, a standardized lenticular soft tissue defect was created in the median region of the palate by excising a mucopenosteal flap (Fig 5-1) This flap extended from just behind the incisive papilla to the caudal margin of the hard palate.

110 Histological evaluation

The maximum width of the flap was one third of the transverse distance between the deciduous first molars. On both sides, relaxation incisions were made adjacent to the posterior teeth. The mucoperiosteum was elevated and the soft tissue defect was closed in the midline, in one layer with VicrylR 5-0 (Ethicon, Johnson & Johnson Company, Amersfoort, The Netherlands), leaving two areas of denuded bone adjacent to the posterior teeth to heal by secondary intention. Healing was allowed for six weeks until the age of 14 weeks.

ιιιψιιι ιιιψιιι ιιιψιιι ιιιψιιι ιιιψιιι ΙΙΙψΙΙι ιιιψι

4 5 6 7 8 9 Figure 5-2: Lateral view of custom-made hemispheric tissue expander with self sealing fdling port. a: Empty state, base diameter 12.0 mm, height 6,0 mm b: Filled, height 12.0 mm.

At 14 weeks of age, custom-made, hemispherical soft tissue expanders (CUIR, Inamed B.V., Breda, The Netherlands) were inserted in the TEXP, TSCA, SEXP, and SSCA groups (Fig. 5-2). The tissue expanders were made of silicone, had a diameter of 12 mm at their base, and a filling volume of

111 Chapter 5

1 ml. The length of the external filling tube, with an incorporated self-sealing port, was 15 mm. To introduce the expander, a standardized para-marginal palatal incision from left to right upper canine was made, and a submucoperiosteal pocket of approximately 15 χ 15 mm was created by elevation of the mucoperiosteum. Both anterior palatal neurovascular bundles were coagulated, while the posterior palatal neurovascular bundles were spared. A 2 mm incision was made on the buccal side, between canine and first deciduous molar and the gingiva and mucoperiosteum were tunneled to allow through the external filling tube. The palatal incision was closed with VicrylR 5-0 sutures. Perioperative antibiotics were administered; preoperatively 0.5 ml AlbipenR 15%, i.m. (Ampicillin-anhydrate 150 mg/ml, Gist-brocades N.V., Delft, The Netherlands), and postoperatively on day 1 and day 3, 1.0 ml AlbipenR L.A., i.m. (Ampicillin-anhydrate 100 mg/ml). The wounds were inspected daily for the first week after surgery or, when indicated, for a longer period. Healing was allowed for two weeks until the age of 16 weeks. All inserted expanders were initially filled with 0.1 ml of a radiopaque solution (AngiografinR, Meglumine-amidotrizoate 650 mg/ml; 306 mg I/ml, Schering A.G., Berlin, Germany). Approximately 0.1 ml AngiografinR was added weekly to each tissue expander in the TEXP and TSCA groups, using a blunt 22-gauge needle, until the age of 24 weeks (Fig. 5-3). In the SEXP and SSCA groups, no additional filling of the tissue expanders was performed after the initial filling. The tissue expanders and the tissue surplus were surgically removed at the completion of the expansion period, and the wound was closed with VicrylR 5-0 sutures. The animals were monitored for another eight weeks, using the same regime of data recording. No surgery was performed in the CONT group, but the animals were monitored and assessed as the operated groups. Thirteen cats had to be excluded from the study due to technical failures. Sixty-two cats were followed until the age of 20 weeks and 52 cats were followed for the entire inflation period up to 24 weeks. Forty animals were studied during the eight weeks after removal of the tissue expander.

112 Figure 5-3: Macroscopy, maximally filled tissue expander in situ at age 24 weeks.

The full thickness excision specimens from the midsagittal palatal region, taken in 31 animals during the simulated Langenbeck surgery at the age of 8 weeks, were available for histologic evaluation of the normal mucoperiosteum (Table 5-1). During TE and at the time of removal of the tissue expander, biopsy specimens of 35 animals were obtained. After fixation with 4% buffered formaldehyde solution, and embedding in paraffin, the biopsy specimens were sectioned sagittally and stained with Hematoxylin-Eosin (HE). For histologic evaluation, 38 animals were sacrificed according to the Central Animal Laboratory perfusion protocol. Their ages varied from 24 to 40 weeks (Table 5-1). Under general anaesthesia, the abdomen and thorax were opened and the vascular system was perfused with physiologic saline, followed by 4% neutral formaldehyde solution as a fixative. After perfusion, the maxillae were dissected and immersed in 4% formaldehyde solution for another two weeks.

113 Chapter 5

Table 5-1: Distribution of biopsies (Β) and perfused animals (Ρ).

prior to during after removal total expansion expansion expander

Group

TEXP В = 10 В = 1 Β = 11 η = 16 Ρ = 2 Ρ = 9 Ρ = 11

SEXP В = 6 Β = 6 η = 14 Ρ = 3 Ρ = 5 Ρ = 8

TSCA В = 17 В = 13 Β = 30 η = 17 Ρ = 3 Ρ = 4 Ρ = 7

SSCA В = 14 В = 6 Β = 20 η = 14 Ρ = 2 Ρ = 4 Ρ = 6

CONT η = 14 Ρ = 2 Ρ = 4 Ρ = 6

Total в = 31 Β = 35 Β = 1 Β = 67 η = 75 Ρ = 12 Ρ = 26 Ρ = 38

The maxilla-specimens were then decalcified and divided into two halfs, which were sectioned in the sagittal and transversal directions respectively. HE, tetrachrome (TC) according to Ralis and Watkins (Ralis and Watkins, 1992) and Weigert-Van Gieson (WVG) (Lillie, 1965) stainings were routinely applied. Immunohistochemical staining was performed only in expansion and control group specimens, because of the inherent costs and the limited budget. An α-smooth muscle actin antibody was used for the identification of the actin component of myofibroblasts (Skalli et ai, 1986; Desmoulière et al., 1992). Double immunofluorescence was performed using DAB (diaminobenzoe-acid), instead of FITC (fluorescein-isothiocyanate) or TRITC (tetra-rhodamine-isothiocyanate). Epithelial and connective tissue architecture was studied using routine light microscopy of the biopsy specimens. Sections of all the tissues involved, including the bone and periodontal structures, were obtained and studied.

114 Histological evaluation

5.4 Results

5 4 1 CONT group

Epithelial layer — The transversally oriented palatal rugae had the form of teeth of a saw, and pointed in posterior direction The palatal epithelium was of the stratified squamous orthokeratotic type (Fig 5-4a) On average the stratum spinosum comprised 10-15 cell layers The stratum germinativum, consisted of approximately seven layers of neatly aligned cuboidal cells In this layer melanocytes with melanin pigment were scattered in between the basal cells The epithelial ridges were separated from the lamina propria by a basement membrane and their orientation was mainly in sagittal direction The total thickness ot the epithelium varied between 250-400 μπι The gingival epithelium was of the parakeratotic type

Connective tissue layer — In HE stained sections, a three-dimensionally orientated subepithelial layer of coarse collagenous fibres (papillary layer), with a mean thickness of approximately 600 μπι, was present (Fig 5-4a) Underneath this layer a distinct less dense layer, with thinner collagenous fibres and numerous blood vessels and nerves could be noticed, the so-called reticular layer, with a mean thickness of approximately 600 μπι In WVG stained sections, elastic fibres were scarcely seen in the rete pegs and in the papillary layer (Fig 5-4b) Their number increased towards the reticular layer, and was maximal in a distinct zone at approximately 60 μπι from the palatal bone Their orientation was more or less parallel to the bony surface

Vessels and nerves — Wide open blood sinuses were noticed in the reticular layer, as well as arterioles and arteries (Fig 5-4) The blood sinuses in the palatal submucosa were less concentrated in the vicinity of the main neurovascular bundle and the lateral aspect of the palate, and they were oriented in sagittal direction The main neurovascular bundle, traversing from posterior lateral to anterior medial, was located at the lateral aspect of a distinct bony ridge The diameter of the palatal artery was approximately 500 μπι Up to four bundles of nervous tissue were identified

115 Figure 5-4: Sagittal and transverse sections of an animal from the CONTROL group at 27 weeks of age. a. Sagittal, HE staining, neg. magn. lOx. b. Transverse, WVG staining, neg. magn. 40x. c. Sagittal, TC staining, neg. magn. 40x. d. Sagittal, a-SM actin staining, neg. magn. 40x. e. Sagittal, TC staining, neg. magn. 40x. f Sagittal, a-SM actin staining, neg. magn. 40x.

116 117 Chapter 5

Οι a I periosteal laver — At the oral side of the bone a distinct separate fibrous layer was present at a distance of approximately 60 μιη from the bony surface A layer of active osteoblasts was seen in close contact to the bone Sharpey's fibres were observed along the palatal surface in the region of the above mentioned bony ridge and in the transition area of the palatal mucopenosteum to periodontal ligament (Fig 5-4a,c,d)

Bone — At the oral side, beneath the periosteum, the palatal bone was compact and of the lamellar type, without signs of resorption (Fig 5-4a,c) It was enveloping the cancellous bone in the more lateral parts and medial parts, e g the alveolar process and anteriorly near the nasal septum and vomer regions The marrow spaces were inconspicuous, with no active osteoblasts and/or osteoclasts At the nasal side, a more compact lamellar bone was observed, as a cortical plate and osteoclasts were frequently seen In some areas, no cancellous bone was present and one fused cortical bone plate was lound The sutures between the maxillary bones, the maxillary and palatine bones, and maxillary and premaxillary bones, had a normal appearance, without osteoid deposition at the edges

Periodontal ligament — The periodontal ligament was inconspicuous Signs of lateral drifting of teeth were noticed There was active resorption at the lateral aspect of the bone of the alveolar socket The periodontal ligament fibres were fading into Sharpey's fibres at the transition zone of alveolar socket and palatal shelf

Nasal periosteal layer — At the nasal side, a thin periosteum, continuous with a pseudostratified columnar ciliated epithelium with goblet cells, and glandular tissue in between, was observed (Fig 5-4a,b,c,d) Several osteoclasts were seen at the nasal side of the palatal bone WVG staining showed an abundance of elastic fibres in the cartilaginous nasal septum region

118 Histological evaluation

5 4 2 TE and SHAM groups

Epithelial layer — No visible differences between the scarred and non- scarred groups were observed during expansion at the epithelial level with any of the staimngs The palatal rugae in the TE group became more flattened with progression of the expansion The epithelium, still consisting of the stratified squamous type, with orthokeratotic cells at the surface, became thinner (Fig 5-5b) This phenomenon was more pronounced at the centre of the expander than in the periphery The stratum spinosum was more condensed, and comprised 10 cell layers on average The stratum germinativum, consisted of approximately 5 layers of aligned cuboidal cells with compressed nuclei The epithelial ridges became broader and less protrusive into the lamina propria The basement membrane had a normal appearance The mean total thickness of the epithelial layer varied from 100-300 μπι In the SHAM group no striking differences with the CONT group could be noticed After removal of the tissue expanders, normalization of the epithelial architecture and thickness became manifest within the observation period of 16 weeks after removal

Connective tissue layer — The simulated Langenbeck operation did not result in visible scars, or changes in the architecture of the connective tissue layers, in the HE-stained sections The other staimngs also failed to show connective tissue scarring In the TE group, the three dimensionally orientated papillary layer of thick collagenous fibres, showed more or less the same configuration, but was somewhat thinner, with a mean thickness of 250-400 μΐη (Fig 5-5b) The underlying reticular layer, with thinner collagenous fibres, was also thinner and the orientation of the fibres had changed into a course more parallel to the surface of the expander (Fig 5-6a) This phenomenon was best noticed using polarized light microscopy (Fig 5-6b) The mean thickness was 250-400 μίτι It was noted to be a time-dependent relationship, with progressive thinning of both distinguished layers in conjunction with time and volume of expansion

119 ^ssss

200μηη

Ψ

200 μιmη

Figure 5-5: Sagittal sections of an animal from the TE group at 24 weeks of age, with tissue expander in situ. a. Palatal side, HE staining, neg. magn. 40x. b. Oral side, HE staining, neg. magn. 40x.

120 Figure 5-6: Sagittal section of an animal from the TE group at 24 weeks of age, with tissue expander in situ. a. Capsule, HE staining, neg. magn. 40x. b. Same section, polarized light microscopy, neg. magn. 40x.

121 Chapter 5

In the SHAM group, no marked differences in the normal thickness and architecture of the original connective tissue layers were noticed. After removal of the tissue expanders, both connective tissue layers normalized with time (observed till 16 weeks after removal).

Capsule — A thick fibrous capsule developed around the expander in the TE and SHAM groups (Fig. 5-5a,b). The thickness varied between 100 and 400 μίτι. The epithelial side of the capsule was thicker than at the bony side. Increased vascularity at the periphery of the capsule was not noted. Different layers within the capsule could not be distinguished, in the HE stained sections. At the cellular level, no distinction could be made between fibroblasts and myofibroblasts. At the fibrillar level, predominantly collagenous fibres were present. Elastic fibres could be detected with WVG staining (Fig. 5-7a). They were located predominantly in the outer layers of the capsule, the transition zone, and the reticular layers. Immunohisto- chemistry on the formalin preserved sections, with antibodies against α-smooth muscle actin, showed the presence of myofibroblasts in the inner layers of the capsule, as well as the smooth muscle cells in the small arteries in the reticular layer at the outer boundary of the capsule (Fig. 5-7b). Inflammatory signs were sometimes present at the inner aspect of the capsule. Neutrophil leucocytes, macrophages, plasma cells and lymphocytes were seen mainly near the filling port entrance. In some cases, epithelialization of the inner capsular surface was seen at the filling port entrance, sometimes invading till a depth of 2500 μίτι. Foreign body giant cells were not found. New formation of bone at the tissue expander edges was frequently encountered, whereas new bone formation over the tissue expander dome was less frequent (Fig. 5-7b). After removal of the tissue expander, involution of the capsular tissue occurred. At the former expander site, inclusion of corpora aliena, such as hairs or vegetable material, was seen. Occasional nodular lymphocyte concentrations were also seen.

Vessels and nerves — With progressive expansion, the blood vessels in the reticular layer appeared more compressed (Fig. 5-5, Fig. 5-6). The normal

122 Histological evaluation capillary vascularization was less visible. The endothelium-lined blood sinuses were less open, and were more concentrated in the palatal submucosa in the region of the main neurovascular bundle. This bundle, traversing from posterior-lateral to anterior-medial, was easily recognizable and did not seem to be displaced. There were no signs of compression or alteration. The bony ridge was less pronounced than in the control group.

Oral periosteal layer — Underneath the expander, on the oral side of the palatal bone, no distinct fibrous periosteal layer could be distinguished within the outer capsular layers (Fig. 5-5a). A thin condensed layer of flattened fibroblasts, or maybe inactive osteoblasts was seen. Osteoclasts were scattered within Howship's lacunae. Sharpey's fibres were observed in the transition area between the palatal mucoperiosteum and medial periodontal ligament, and para-medially. No differences between specimen from the scarred and non-scarred groups could be detected.

Bone — The compact palatal bone was eroded at the oral side in the TE group, beneath the expander-base. Signs of osteoclastic resorption were found and osteoblastic activity was present in stress shielded areas. Howship's lacunae and multinuclear osteoclasts were seen incidentally. In some cases, the compact bone layers at the oral and nasal side were fused, forming one cortical plate. In some areas, the palatal bone was completely missing due to extensive bone resorption (Fig. 5-5a). In those cases no bony boundary was present between the oral and nasal cavities. α-Smooth muscle staining showed concentration of myofibroblasts in those areas. The cancellous bone in the more anterior, lateral and medial parts was inconspicuous. The architectural pattern of the trabeculae was not altered. The marrow spaces showed the presence of active and resting osteoblasts and in some cases osteoclasts. The sutures between the maxillary bones, the maxillary and palatine bones, and maxillary and premaxillary bones were unaltered. Formation of new bone was seen at the edges of the tissue expander, at the boundaries of the bony depression, in the form of a bony ridge. Occasionally, new bone formation was observed along the surface of the expander-dome, scattered in the inner layer of the capsule (Fig. 5-7b). In the SHAM group, similar findings were

123 Figure 5-7: Details of connective and capsular tissue of an animal from the TE group at 24 weeks of age, with tissue expander in situ. a. Elastic fibres in connective tissue, WVG staining, neg. magn. 40x. b. Myofibroblasts in capsular tissue, a-SM actin staining, neg. magn. 40x. (Note new bone formation over the tissue expander dome.) Figure 5-8: Sagittal section of an animal from the TE group after removal of the tissue expander at 37 weeks of age. a. Bony depression, TC staining, neg. magn. lOx. b. Detail of a. showing osteoid formation at peninsulas and bony islands, TC staining, neg. magn. 40x.

125 Chapter 5 noticed, in some cases even more pronounced than in the expansion groups. After removal of the tissue expander, bony ingrowth in the depressed defect developed (Fig. 5-8a). Peninsulas of lamellar bone (combined plexiform and bundle bone), surrounded by active osteoblasts, were present as centres of ossification (Fig. 5-8b) Osteoid formation could be evidenced by TC staining. Furthermore, loose connective tissue invagination in the bony defect was seen The original oral periosteal layer or capsular layer, with concentration of elastic fibres, remained as a spanning layer crossing over the defect The bony lipping extensions at the edges of the depression seemed to be rounded off by remodelling. Remodelling was evidenced by the presence of active osteoblasts and osteoclasts, and positive osteoid staining with TC.

Periodontal ligament — The periodontal ligament itself was inconspicuous. At the medial side of the alveolar socket, deposition of bone was seen, while at the lateral side signs of osteoclastic resorption were present. At the anterior and posterior sides, no changes were found.

Nasal mucoperiosteal layer — The nasal mucopenosteum, with the pseudostratified columnar ciliated epithelium with goblet cells, and glandular tissue in between, was not compromised in the presence of an intact bony boundary. During expansion, slightly depositary bone, with osteoblasts and scattered osteoclasts, was found. In the absence of a bony separation of oral and nasal cavities, loose and fibrous connective tissue as present in normal sutures, with the above-mentioned myofibroblast concentration, was noticed to connect the periosteum and capsule (Fig. 5-5a) After removal of the tissue expander, no essential changes of the nasal mucopenosteum were noticed

5.5 Discussion

This study is part of a larger series of experiments in which the effects of palatal mucoperiosteal TE in growing cats, with and without pre-existing scars, are evaluated in a two- and three-dimensional fashion. A considerable macroscopic increase of the palatal soft tissues is found

126 Histological evaluation in the groups with inflated tissue expanders. Soft tissue length increments and surface area increases were quantified by cephalometric and morphometric analyses (Van Damme et al., 1996 a,b). Alterations of bony palatal and dento-alveolar growth in sagittal, vertical and transversal directions, caused by TE, were also assessed cephalometrically and morphometrically (Van Damme et al., 1996 a,b). It was found that the scarring of the palate at the age of eight weeks did not alter palatal growth, nor the TE process significantly. The effects of scarring of the palatal soft tissues by the simulated Langenbeck operation were not appreciable at the light microscopic level. TE, and to a lesser extent palatal surgery for introduction and removal of the tissue expander, caused adverse effects on the growth of bony structures (Van Damme et ai, 1996 a,b). Palatal growth was decelerated in the antero-posterior and transversal directions, and the anterior nasomaxillary height increase accelerated during expansion. It was suggested that increased palatal mucoperiosteal tension caused by the tissue expander underneath the mucoperiosteum, may have caused the restraining effects on antero-posterior and transverse palatal growth and the palatal tipping of the teeth. At the light microscopy level, as revealed by the present study, neither signs nor symptoms of the simulated Langenbeck operation were found. There were no visible scars in the medial and lateral areas of the palate. The amount and site of Sharpey's fibres were unaltered. This is different from other studies in cats (Freng, 1979), and in beagle dogs (Wijdeveld et al., 1991). This study shows that TE of the intra-oral mucoperiosteum yields at least a temporary soft tissue gain. However, the thinning of the epithelium and connective tissues, are more suggestive of a loan, than a dividend (Austad et al., 1986). The effects of tension on the palatal mucoperiosteum exerted by the increasing tissue expander volume may be causing stretching. The flattening of the rugae and rete pegs is compatible with this. The question, whether or not recruitment and creeping substitution of soft tissues play an important role in this phenomenon, cannot be answered on the basis of this study. The formation of a fibrous capsule can be looked upon as genuine tissue gain. However, the increased vascularity as reported in the literature, was not seen in this study, and the relevance of the reported benefits of the

127 Chapter 5 presence of a capsule may be questioned (Van Damme et al , 1992) The fibrous capsule with predominantly collagenous fibres, also contained elastic fibres and myofibroblasts, which may be held responsible for potential contraction of the gained tissue, and thus for retraction of an expanded tissue flap after removal of the tissue expander The bony depression underneath the expander base showed an increase of the amount of Sharpey's fibres at its edges Osteoclastic resorption at the centre of the expander-base, leading to complete loss of cortical bone in the midline area, was seen in several cases This phenomenon may have compromised the palatine and vomeroseptal growth centres, with consequences as illustrated in Freng's studies (Freng, 1979, 1981, Voss and Freng, 1982, 1983) After removal of the tissue expanders, a bony palatal scar seems to remain Reparative connective tissue formation underneath a fibrous layer с q capsule was noticed The bony ingrowth from the bottom of the defect often showed the form of flattened mushroom formed peninsulas of lamellar bone Sixteen weeks after removal of the tissue expanders, bony healing was seldom complete Mostly, irregularities of the oral bony surface and connective tissue invaginations had remained at the former expander site Sometimes, inclusions of foreign bodies and nodular lympho-plasmocytic infiltrations were seen However, inflammatory reactions seem to be of minor importance In only a few cases, and more often in SHAM than m TE groups, neutrophil leucocytes and lymphocytes were seen in the neighbourhood of the filling port, at the inner aspect of the capsule Some of the reported side-effects seem to be temporary After removal of the tissue expander, normalization of epithelial and connective tissues takes place The capsular tissue dissolves in time Bony palatal growth normalizes or even accelerates (in the expansion in scarred tissue group), despite persistence of a bony scar

5.6 Conclusions

TE of the palatal mucoperiosteum in young growing cats, with and without scars of the palatal tissues, causes noticeable thinning of the epithelial layers, especially over the centre of the expander The architecture of the connective

128 Histological evaluation tissue layers is markedly altered Apart from thinning, reorientation of collagenous fibres is also seen A course more parallel to the surface of the expander is induced by progressive TE A fibrous capsule, with mainly collagenous fibres and myofibroblasts present around the expanders in the TE group as well as the SHAM group, seems to be caused by the presence of the expander and not by the expansion itself Inflammation within the capsule was occasionally noticed in both TE and SHAM groups in the neighbourhood of the filling port This does not seem to be important TE of the palatal mucopenosteum seems to induce a temporary gain, a loan of soft tissue, which can be used in palatal defect reconstruction There is, however, a potential risk of contraction and thus of retraction of the expanded flap Further studies are required to assess the potential résorption/contraction of this 'loaned' tissue

5.7 Literature

ABRAMO AC, VIOLA JC, ANGELO AJ (1993) Intraoperative rapid expansion in cleft palate repair Plast Reconstr Surg 91 441-445 ANTONYSHYN O, GRUSS JS, ZUCKER R, MACKINNON SE (1988) Tissue expansion in head and neck reconstruction Plast Reconstr Surg 82 58 68 AUSTAD ED (1987) The origin of expanded tissue Clin Plast Surg 14 431 433 AUSTAD ED, PASYK KA, McCLATCHEY KD, CHERRY GW (1982) Histomorphologic evaluation of guinea pig skin and soft tissue after controlled tissue expansion Plast Reconstr Surg 70 704-710 AUSTAD ED, THOMAS SB, PASYK KA (1986) Tissue expansion Dividend or loan'Plast Reconstr Surg 78 63-67 В ARD ACH J, KELLY KM (1990) Does interference with mucopenosteum and palatal bone affect craniofacial growth ? An experimental study in beagles Plast Reconstr Surg 86 1093-1100 BARDACH J, KELLY KM, SALYER KE (1994) Relationship between the sequence of lip and palate repair and maxillary growth an experimental study in beagles Plast Reconstr Surg 93 269-278 BHASKAR SN (1990) Orban's oral histology and embryology 11th ed Saint Louis Mosby, pp 203-336

129 Chapter 5

BONOMO D (1986) Subperiosteal expanders for ridge augmentation Presented at the Annual Meeting of the Southern Calif Soc Oral and Maxillofac Surg BROBMANN GF, HUBER J (1985) Effects of different-shaped tissue expanders on transluminal pressure, oxygen tension, histopathologic changes, and skin expansion in pigs Plast Reconstr Surg 76 731-736 BURKITT HG, YOUNG B, HEATH JW (1993) Wheater's functional histology A text and colour atlas Edinburgh Churchill Livingstone, ρ 236 CHERRY GW, AUSTAD ED, PASYK KA, McCLATCHEY KD, ROHRICH RJ (1983) Increased survival and vascularity of random-pattern skin flaps elevated in controlled, expanded skin Plast Reconstr Surg 72 680-685. DE MEY A, MALEVEZ C, LEJOUR M (1990) Treatment of palatal fistula by expansion Br J Plast Surg 43 362-364. DESMOULIÈRE A, RUBBIA-BRANDT L, GRAU G, GABBIANI G (1992) Heparin induces α-smooth muscle actin expression in cultured fibroblasts and granulation tissue myofibroblasts Lab Investigation 67 716-726 FREIHOFER HPM, BORSTLAP WA, KUIJPERS-JAGTMAN AM, VOORSMIT RACA, VAN DAMME PHA, HEIDBUCHEL KLWM, BORSTLAP-ENGELS VMF (1993) Timing and transplant materials for closure of alveolar clefts J Cramo-Max-Fac Surg 21 143-148 FRENG A (1979) The restorative potential of double layered mucopenosteum A study on experimental mid-palatal clefts in the cat Scand J Plast Reconstr Surg 13 313-320 FRENG A (1981). Growth of the middle face in experimental early bony fusion of the vomeropremaxillary, vomeromaxillary and mid-palatal sutural system A Roentgencephdlometnc study in the domestic cat Scand J Plast Reconstr Surg 15 117-125 IN DE BRAEKT MMH (1992) Dento-alveolar development after modified palatal surgery on dogs PhD Thesis, University of Nijmegen, The Netherlands LANGENBECK В (1861) Operation der angeborenen totalen Spaltung des harten Gaumens nach einer neuen Methode Dtsch Klin 24 LATRENTA GS, MCCARTHY JG, EPSTEIN M, CUTTING CB, ORENTREICH С (1988) Bone graft survival in expanded skin Plast Reconstr Surg 81 406-413 LEE P, SQU1ER CA, BARDACH J (1985) Enhancement of tissue expansion by anticontractile agents Plast Reconstr Surg 76 604-610

130 Histological evaluation

LEENSTRA TS, MALTHA JC, KUIJPERS-JAGTMAN AM, SPAUWEN PHM (1995) Wound healing in Beagle dogs after palatal repair without denudation of bone Cleft Palate Craniofac J 32 363-370 LEIGHTON WD, RUSSELL RC, FELLER AM, ERIKSSON E, MATHUR A, ZOOK EG (1988) Experimental pretransfer expansion of free-flap donor sites II Physiology, histology and clinical correlation Plast Reconstr Surg 82 76 84 LEIGHTON WD, RUSSELL RC, MARCUS DE, ERIKSSON E, ZOOK EG (1986) Experimental expansion of cutaneous and myocutaneous free flap donor sites, anatomical, physiological and histological changes Plast Surg Forum 9 262-263 LEW D, CLARK R, SHAHBAZIAN Τ (1986) The use of a soft tissue expander in alveolai ridge augmentation a preliminary report J Oral Maxillofac Surg 44 516-519 LILLIE RD (1965) Histopathologic techmc and practical histochemistry 3rd ed New York McGraw-Hill, ρ 539 MEIJER R, PRAHL В (1978) Influences of different surgical procedures on growth of dentomaxillary complex in dogs with artificially created cleft palate Ann Plast Surg 1 460-465 MILLARD Jr DR (1980) Cleft craft The evolution of its surgery III Alveolar and palatal deformities Boston Little, Brown, pp 809 865 MOELLEKEN BRW, MATHES SJ, CANN CE, SIMMONS DJ, GHAFOORI G (1990) Long term effects of tissue expansion on cranial and skeletal bone development in neonatal miniature swine clinical findings and histomorphometnc correlates Plast Reconstr Surg 86 825 834 PASYK KA, ARGENTA LC, AUSTAD ED (1987) Histopathology of human expanded tissue Clin Plast Surg 14 435-445 PASYK KA, ARGENTA LC, HASSETT С (1988) Quantitative analysis of the thickness ol human skin and subcutaneous tissue following controlled expansion with a silicone implant Plast Reconstr Surg 81 516-523 PASYK KA, AUSTAD ED, McCLATCHEY KD, CHERRY GW (1982) Electron microscopic evaluation of guinea pig skin and soft tissues 'expanded' with a sell-inflating silicone implant Plast Reconstr Surg 70 37-45 PROVENZA DV (1988) Fundamentals of oral histology and embryology 2nd ed Philadelphia Lea & Febiger, pp 20-84, 238-253 RADOVAN С (1982) Breast reconstruction after using the temporary expander Plast Reconstr Surg 69 195-206

131 Chapter 5

RADOVAN С (1984) Tissue expansion in soft tissue reconstruction Plast Reconstr Surg 74 482-490 RALIS ZA. WATKINS G (1992) Modified tetrachrome method for osteoid and defectively mineralized bone in paraffin sections Biotechn Histochem 67 339-345 RICCIARDELLI EJ, GODING GS, BRIGHT DA, CUMMINGS CW (1989) Acute blood flow changes in rapidly expanded and adjacent skin Arch Otolaryngol Head Neck Surg 115 182-186 SAOUMA S, BRICOUT M, SERVANT JM, BANZET Ρ (1989) Technique de l'expansion tissulaire J Chir (Pans) 126 34-39 SCHMELZEISEN R, SCHWIPPER V, TILKORN H, BECKER H, UBERMUTH Τ (1994) Changes in growth of the facial skeleton following implantation of tissue expanders J Cranio-Max-Fac Surg 22 (Suppl 1) 81 SKALLI O, ROPRAZ P, TRZECIAK A, BENZONANA G, GILLESSEN D, GABBIANI G (1986) A monoclonal antibody against α-smooth muscle actin a new probe for smooth muscle differentiation J Cell Biol 103 2787-2796 TEN CATE AR (1994) Oral histology, development, structure and function 4rd ed Saint Louis Mosby, 423-424 TIMMENGA EJF (1992) Skin expansion, an experimental study in the rabbit PhD Thesis, University of Amsterdam, The Netherlands VAN DAMME PHA (1994) Soft-tissue expansion in cleft palate surgery Plast Reconstr Surg 93 1307 VAN DAMME PHA, FREIHOFER HPM (1996) Palatal mucopenosteal expansion as an adjunct to palatal repair Case report and review of the literature Cleft Palate-Craniofac J 33 255-257 VAN DAMME PHA, FREIHOFER HPM, MALTHA JC, KUIJPERS-JAGTMAN AM, VAN 'T HOF MA (1996b) Three-dimensional morphometnc analysis of the effects of subperiosteal palatal soft tissue expansion in growing cats Int J Oral Maxillofac Surg In press VAN DAMME PHA, FREIHOFER HPM, VAN Τ HOF MA, KUIJPERS- JAGTMAN AM, MALTHA JC, SPIJKERS JMC (1994) Radiologic analysis of the effects of subperiosteal palatal soft-tissue expansion in growing cats Int J Oral Maxillofac Surg 23 393-394 VAN DAMME PHA, HEIDBUCHEL KLWM, KUIJPERS-JAGTMAN AM, MALTHA JC, FREIHOFER HPM (1992) Cranio-maxillo-facial tissue expansion, experimentally based or clinically empiric9 A review of the literature J Cranio-Max-Fac Surg 20 61-69

132 Histological evaluation

VAN DAMME PHA, MALTHA JC, KUIJPERS-JAGTMAN AM, FREIHOFER HPM, VAN 'T HOF MA (1996a). Two-dimensional cephalometric analysis of the effects of subperiosteal palatal soft tissue expansion in growing cats. Plast Reconstr Surg. In press. VANDERKOLK CA, McCANN JJ, MITCHELL GM, O'BRIEN BM (1988). Changes in area and thickness ol expanded and unexpanded axial pattern skin tlaps in pigs. Br J Plast Surg 41: 284-293. VAN RAPPARD JHA, JERUSALEM C, SONNEVELD GJ, BORGHOUTS JMHM (1988). Histologic changes in solt tissues due to tissue expansion (in animal studies and in humans). Facial Plast Surg 5: 280-286. VAN RAPPARD JHA (1988) Controlled tissue-expansion in reconstructive surgery, PhD Thesis, University of Groningen, The Netherlands. VOSS R, FRENO A (1982) Growth of the dental arches after ablation of the mid- palatal suture. A study in the domestic cat J Max-Fac Surg 10: 259-263. VOSS R, FRENG A (1983). Concomitant transverse growth of the maxillary base and dental arch in experimental submucous mid-palatal clefts. A biometrical study in the domestic cat J Max-Fac Surg 11: 257-262. WIJDEVELD MGMM, GRUPPING EM, KUIJPERS-JAGTMAN AM, MALTHA JC (1987). Mucopenosteal migration after palatal surgery in beagle dogs. A longitudinal radiographic study. Int J Oral Maxillotac Surg 16: 729-737. WIJDEVELD MGMM, GRUPPING EM, KUIJPERS-JAGTMAN AM, MALTHA JC (1988) Growth of the maxilla after soft tissue palatal surgery at different ages on beagle dogs - a longitudinal radiographic study J Oral Maxillofac Surg 48: 204-209. WIJDEVELD MGMM, GRUPPING EM, KUIJPERS-JAGTMAN AM, MALTHA JC (1989). Maxillary arch dimensions after palatal surgery at different ages on beagle dogs. J Dent Res 68: 1105-1109. WIJDEVELD MGMM, MALTHA JC, GRUPPING EM, DE JONGE J, KUIJPERS- JAGTMAN AM (1991). A histological study of tissue response to simulated palatal surgery at different ages on beagle dogs Arch Oral Biol 36: 837-843. WITTKAMPF ARM (1989) Short-term experience with the subperiosteal tissue expander in reconstruction of the mandibular alveolar ridge. J Oral Maxillofac Surg 47. 469-474.

133

Chapter 6

Palatal mucoperiosteal expansion as an adjunct to palatal fístula repair Case report and review of the literature

Philip A. Van Damme Hans Peter M. Freihofer

Published in Cleft Palate-Craniofacial Journal (1996) 33: 255-257.

Adjunct to fistula repair

6.1 Summary

Case report of conventional palatal soft tissue expansion (TE) in an attempt at cleft palate fistula closure, with a standard tissue expander This technique may be an alternative to a tongue flap to promote closure of persistent oronasal fistula

137 Chapter 6

6.2 Introduction

Intraoral TE for augmentation ot the alveolar process in preprosthetic surgery has been established (Lew et al , 1986) Palatal mucoperiosteal expansion with non-custom-made tissue expanders, however, has never been previously reported Indeed, conventional or rapid palatal TE was recently judged to be impossible (Abramo et al , 1993, Van Damme, 1994) In clinical reports, the use of Foley catheters and custom-made tissue expanders has been reported (De Mey et al , 1990, Abramo et al , 1993) In an animal experiment, custom-made tissue expanders were successfully utilized for subperiosteal palatal TE (Van Damme et al , 1991, 1993) The purpose of this report is to suggest that palatal TE with standard expanders can be helpful, although not necessarily completely successful, in closing residual palatal defects of cleft palate patients under unfavourable circumstances

6.3 Case report

A 37-year-old male patient was referred to the Nijmegen Centre for cleft lip and palate management He presented with an operated right-sided cleft lip and palate (UCLP), with a persistent oronasal fistula He had had eight previous operations at other centres including, at the age of 24 years, closure of the palatal oronasal communication with a costal bone graft Because of sequestration of part of the graft, a fistula had developed Initially, there were no functional problems However, in time, the patient complained of liquid leakage into the nose, and requested closure of the fistula A late secondary closure technique was indicated with nasal and oral mucopenosteal-layer closure and interposition of an autogenous chinbone graft for reconstruction of the alveolar process (Freihofer et al , 1993) The operation and immediate postoperative period were uneventful However, in spite of adjustment of a protection device, palatal dehiscence with exposure of the transplant occurred, which finally led to necrosis of the graft and a residual palatal defect (Fig 6-1) Since the patient had a fixed dental bridge, a bucco-

138 Adjunct to fistula repair

Figure 6-1: Palatal oronasal fistula (arrow) in 37-year-old male patient with right side UCLP after multiple operations before TE. vestibular-flap technique to close the defect was considered undesirable. The feasibility for conventional palatal TE with standard tissue expanders was studied on a cast model (Fig. 6-2). A spherical Intravent tissue expander (CUI-IOS-01, CUI Corporation, Carpintería CA), with an expansion volume of 1 mL and a diameter of 12 mm, with a remote filling tube was chosen. This was inserted after tunneling the palatal mucoperiosteum on the left side via a small incision in the front of the vestibulum and another near the second molar. Initially, it was filled with 0.4 mL physiologic salt solution. Healing was undisturbed. Two weeks postoperatively, serial inflation was started twice a week until, after eight sessions a volume of 1.8 mL was reached (Fig. 6-3). The tissue expander was removed along the lateral incision-line (Fig. 6-4), and a unilateral Veau soft tissue flap was created and fixed in place with VicrylR 4-0 sutures after excision of the wound edges in the acceptor area. A gauze pack, impregnated

139 Chapter 6

Figure 6-2: Study of feasibility of various standard tissue expanders.

Figure 6-3: Palatal situation after eight inflations to a volume of 1.8 ce, just before removal of the expander. Notice the remote filling tube at the buccal aspect of the molar teeth (arrow).

140 Adjunct to fistula repair

Figure 6-4: Removal of the inflated tissue expander via a lateral paramarginal incision and preparing of the unilateral Veau flap. IUI

Figure 6-5: Palatal situation six days after expander removal and rotation of the unilateral Veau flap, just after withdrawal of the protective covering.

141 Chapter 6 with iodoform/Vaseline was used as a protective covering. Six days later, it was removed and the flap was vital, with no sign of dehiscence (Fig. 6-5). Unfortunately, a small oronasal fistula developed 12 days after removal of the expander along the acceptor margin of the canine tooth. The patient noticed fluid in the nose after mouth rinsing, but he declined further surgical intervention. His general dental practitioner is making a palatal sealing plate/frame to correct this problem.

6.4 Discussion

Several techniques have been used to close residual palatal oronasal fistula (Millard, 1980; Voorsmit and Fennis, 1992). The initial success rate varies from 36% to 69% and diminishes with the second, third or further attempts. Whenever a two-stage procedure is chosen (e.g., a tongue-flap, Guerrcrosantos and Altamirano, 1966), conventional palatal TE should be considered as a realistic alternative. However, this is a multistage procedure with serial inflations and two operations, with insertion and removal of the tissue expander and with mobilization and fixation of the flap. This is compensated by the fact that tongue movements are unrestricted, and taste and sensation of the tongue are not compromised. In this case, the surgery and inflations were well tolerated, with little patient discomfort. There was also no pain during inflation or thereafter, and no mechanical hindrance to eating, despite the increasing volume of the expander. The overlying soft tissues became shiny and appeared to become thinner. After removal of the tissue expander, a fibrous capsule was seen. This seemed to restrict the mobility and pliability of the expanded tissue. Clinically, the vascularity of the expanded tissue was not compromised, in contrast to the recipient palatal site near the right canine tooth. The recurrent fistula developed in the area of the recipient wound edge, apparently, not to be considered so much a failure of the TE procedure, but rather caused mainly by the poor vascular condition of the recipient area. A more extended resection of the wound edges of the mucosa surrounding the defect might

142 Adjunct to fistula repair have avoided this complication The patient's smoking (15 cigarettes per day), however, and the scarring from previous surgery may have reduced the chances of success In conclusion, conventional palatal mucopenosteal TE with standard expanders is a realistic option as an adjunct to recurrent cleft palate oronasal fistula closure However, it does not guarantee independence of the quality and vascularity of the adjacent tissue at the recipient site

6.5 Literature

ABRAMO AC, VIOLA JC, ANGELO AJ (1993) Intraoperative rapid expansion in clett palate repair Plast Reconstr Surg 91 441-445 DE MEY A MALEVEZ C, LEJOUR M (1990) Treatment of palatal fistula by expansion Br J Plast Surg 43 362-364 FREIHOFER HPM, BORSTLAP WA, KUIJPERS-JAGTMAN AM, VOORSMIT RACA, VAN DAMME PHA, HEIDBUCHEL KLWM, BORSTLAP-ENGELS VMF (1993) Timing and transplant materials for closure of alveolar clefts J Cranio Max-Fac Surg 21 143-148 GUERREROSANTOS J, ALTAMIRANO JT (1966) The use of lingual Haps in repair of fistulas of the hard palate Plast Reconstr Surg 38 123-128 LEW D, CLARK R, SHAHBAZIAN Τ (1986) Use of a soft tissue expander in alveolar ridge augmentation a preliminary report J Oral Maxillofac Surg 44 516 519 MILLARD Jr DR (1980) Cleft craft The evolution of its surgery III Alveolar and palatal deformities Boston Little, Brown & Co, pp 809 865 VAN DAMME PHA (1994) Soft tissue expansion in cleft palate repair Plast Reconstr Surg 93 1307-1308 VAN DAMME PHA (1991) Soft tissue expansion in cleft palate surgery an animal study III International Tissue Expansion Symposium, Sapporo, Japan VAN DAMME PHA (1993) Palatal expansion experimental and clinical IV International Tissue Expansion Symposium, Säo Paulo, Brazil VOORSMIT RACA, FENNIS JPM (1992) The buccal fat pad for closure of oro­ nasal communications in clett patients J Cranio Max Fax Surg 20 (suppl 1) 71

143

Chapter 7

General discussion

General discussion

7.1 Feasibility of palatal mucoperiostcal tissue expansion

The present study shows that subperiosteal palatal soft tissue expansion (ТЕ) in growing cats is technically feasible Several conditions, however, have to be fulfilled Different types of tissue expanders were used in a pilot study in order to select the most appropriate model for the young growing cat It appeared that the diameter could not exceed 12 mm, and that the size in the empty situation should be as small as technically possible Commercially available tissue expanders did not fulfil these requirements Custom made expanders were therefore designed and developed in close cooperation with CUI, Carpintería, USA Initially, hemispheric tissue expanders with an incorporated self-sealing filling port were tried However, complications such as erosions and necrosis of the overlying mucopenosteum and extrusion of the tissue expanders were seen too frequently, probably due to shape-rigidity, excess height and consequent wrinkling of the tissue expander envelope To overcome these problems, hemispheric tissue expanders with a remote self sealing filling tube were selected for the present study Critical conditions for success are the site of the incisions for insertion ot the tissue expander, the time span ot healing of the incisions before inflation of the tissue expander, and the (initial) filling volume of the expander The incisions in the pilot study were initially made paramarginally at the lateral side of the palate, in the region of the future lateral expansion limit This led to dehiscence and premature ending of the expansion due to extrusion of the expander An incision in the anterior region, more distant from the future limits of expansion, and a small lateral incision for passing through the remote filling tube, proved to be more favourable and was used in the present study Healing should be allowed for at least a two-week period, prior to starting the serial inflations The initial filling volume should be individually adjusted to the vascularity status of the overlying soft tissues The expander should be filled until paleness of the mucopenosteum appears and then some filling fluid should be removed until the paleness again disappears The same is also true for the serial fillings Protection of the inserted tissue expander and the remote filling tube is

147 Chapter 7 advisable In order to prevent the animals from chewing on the tube, the tubes were fixed to the mucosa with a VicrylK (5-0) suture There were few dietary restrictions No fresh fish, with fish bones, or other sharp and/or hard substances should be incorporated in the diet This should apply not only to animals, but also humans Weight loss and difficulties with eating, because of the ever increasing volume of the tissue expander and the consequently decreasing intra oral volume, were anticipated but not encountered Mouth closure, nose breathing and social behaviour of the animals were unaltered There were no signs of pain or discomfort in the animals A major draw-back of this study in growing non-rodent animals is introduced by the shedding of teeth coinciding with the timing of removal of the tissue expander The initial morphometnc measurement points before and during expansion were related to the deciduous dentition, whereas after removal of the tissue expanders, different reference points related to the permanent dentition were used Since the permanent canines and molars erupt at the palatal side of their predecessors, the palatal surface area was decreasing, instead of increasing by the TE This is why two separate time intervals were observed, the first during TE in the deciduous dentition period, and the second after removal of the tissue expander and with a permanent dentition The in-between period could not be reliably evaluated

7.2 Dividend or loan?

Soft tissue gain which can be used to close certain palatal defects without creation of a donor site defect was the intention of this experiment Cephalometnc analysis on lateral headplates revealed significantly more soft tissue length increase in the expansion groups than in the sham and control groups For example, a 40% mean increase in the length of the soft tissue was seen in the expansion groups, which is about 25 to 30% more than in the sham and control groups respectively (Van Damme et al 1994, 1996a) TE was apparent not only orally but also nasally, at the palatal base of

148 General discussion the expander, the so-called 'downward expansion' (Van Rappard, 1988) A depressed erosion of the bony palate underneath the centre of the base of the expander was noticed clinically during removal of the tissue expander This phenomenon is often referred to as a 'bathtub depression' (Moelleken et al , 1990) This means that the positive effects of expansion on soft tissue gain are accompanied by negative effects exerted in the opposite direction on the palatal bone Three dimensional analysis revealed statistically significant higher surface area increments in the TE groups than in the control and sham groups (Van Damme et al , 1996b) The figures were related to the base surface area value of the empty expanders (r = 6, 7гг=113 04 mm2) The proportional gain in the experimental groups was 85% after 8 weeks ot active expansion The transverse gain was 36%, while the sagittal gain was 32% The sagittal gain figures in the 3-D morphometnc analysis correspond reasonably well with the figures from the 2 D cephalometnc analysis (25 30%) The dilemma of whether a dividend or loan of tissue after ТЕ is established, was first stated by Austad et al , in 1986 Many authors have discussed this question (Austad, 1987, Van Rappard, 1988, Van Damme et al , 1992) TE has been compared to pregnancy, morbid obesity and subcutaneous benign tumour growth (Cullen and Powell, 1989) The skin surplus that appears e g with pregnancy, fades away with time after delivery in young women In elderly people, the surplus will partly remain, depending on the (lack of) elasticity of the skin and subcutaneous tissue Tissue gain can be caused either by new-forming, recruitment, creeping substitution and/or stretching of tissue, or a combination of these phenomena Proliferation of cells (hyperplasia), or larger cells (hypertrophia), would be indications of a real dividend Creep or creeping substitution may also be considered as a dividend Recruitment of cells from the direct surroundings of the tissue expander and the expanded tissue, is a disputable form of gain Stretching of tissues is most probably a loan, since soft tissue is likely to contract during elevation ol the expanded flap and during the healing phase afterwards with traction on the wound edges and sutures A reconstruction with this stretched tissue is likely to deform, because of traction and retraction At the light microscopy level, thinning of the epithelial layer right above the expander

149 Chapter 7 dome is seen (Van Damme et al.. 1996c) There is a flattening and rounding off of the originally saw-teeth like rugae. The connective tissue layer which is normally present, is also thinner. However, a newly formed extra layer around the tissue expander, the capsule, was present. This can be looked upon as tissue gain, but it is still debatable whether or not this capsular layer is beneficial. To keep the surface exposed to a foreign body as small as possible, seems to be the rule of the animal and human biology (Vistnes et al., 1978) The capsule is composed of fibroblasts and bundles of collagenous fibres, but it also contains elastic fibres and myofibroblasts (Cherry et al., 1983, Antonyshyn et ai, 1988, Pasyk et ai, 1988, Van Damme el al , 1992, 1996c) These latter components may be responsible for the major part of the contraction of the tissue after removal of the tissue expander Unlike Cherry et al (1983), no increase in the vascularity of the outer border of the capsule was seen in this study

7.3 Quality and quantity of tissue gain

The quality of the 'gained' soft tissues was evaluated clinically during the TE process and during removal of the tissue expanders The median most expanded mucopenosteum was excised as an ovaloid and assessed as a biopsy The excised biopsies together with specimens from sacrificing several animals, were studied al the light microscopy level Clinically, the palatal tissues overlying the active expanders appeared progressively glistening, thinner and stretched. The rugae were slightly broader and flattened. The sagittal distance between the transverse rugae increased with the increasing tissue expander volume. The tissue between the rugae was thinner, the tissue expander was sometimes almost shining through. At removal of the tissue expander, an encapsulated space was encountered. A distinct capsular tissue layer was apparent. The expanded tissue at the lateral sides of the excision was pliable during transposition and the vascularity was uncompromised. The quality of the soft tissues was sufficient to withstand the tensile forces of the sutures used to approximate the wound edges. With time, there were no more dehiscences in the expansion groups with or without pre-existing scars, than

150 General discussion in the sham groups It is debatable whether or not the quality of the expanded soft tissues can be appreciated histologically The aspect of the individual cells in the distinguished layers seems to be unaltered They are at most somewhat compressed, depending on the location in relation to the tissue expander dome The architecture of the soft tissues overlying the tissue expander was changed The epithelial ridges were flattened, and protruded less into the papillary connective tissue layer The epithelium appeared to be stressed and stretched, and was markedly thinner The three-dimensional criss-cross aspect of the collagenous fibres in the papillary layer seemed to be altered A course more parallel to the surface of the tissue expander dome is noticed The same phenomenon was seen in the reticular layer, and may be explained by tensile forces causing stress and strain The formation of a separate encapsulating fibrous connective tissue layer, is seen around any foreign body that is incorporated in humans and experimental animals (Vistnes et al , 1978) The fact that there were no striking differences between the capsules formed in the expansion and sham groups, suggests that the TE process itself is not decisive in the development and thickness of the capsule The orientation of the fibres in the capsule, however, does seem to depend on the same forces of stress and stretch as mentioned above The quantity of the 'gained' soft tissue was studied in the two- dimensional cephalometnc analysis and in the three-dimensional morphometnc analysis of the cast models The difference between the soft tissue length increment measured along the oral surface and the increment in the growing bony palatal shelf length was initially expressed as a percentage (Van Damme et al , 1994), and amounted to approximately 30% This may be taken as the quantity of soft tissue length gain evoked by the TE However, the increase in the palatal bone length in the expansion groups is significantly smaller than in the control group (15%) When the palatal bone length increase in the expansion groups is compared to the increases in the control and sham groups, differences of 25 to 30%, are found These figures seem to be more similar to measurements of the soft tissue length gain found in the cephalometnc

151 Chapter 7 analysis Criticism to this concept is based on the fact that the gain is based on site specific anatomical points When the distance between these points is smaller, a larger gain percentage would have been scored Perhaps it would have been better to relate the soft tissue length gain to the base diameter or the height of the expander (Shively, 1986, 1988, Patel, 1986, Van Rappard et al , 1988, Duits et al , 1989) Related to a base diameter of 12 mm a percentage of 63% could have been noted In the three-dimensional analysis, the palatal soft tissue surface area increase was related to the base surface oí the empty expander After eight weeks of expansion, a soft tissue area increase of 85% of the base surface was registered Subdivided into sagittal length increase and transverse width increase, percentages of 32 and 36% respectively, were found These figures are similar to the two-dimensionally found measurements, and the literature data (Nordstrom and Devine, 1985, VanderKolk et al , 1987) For example, in human (scalp) expansion, increase percentages in transverse and sagittal direction of 39-45% and 29-30% respectively, were found (Nordstrom and Devine, 1985, VanderKolk et al , 1987) Area increase is reported to vary from 12 5 to 110% (Brobmann and Huber, 1985, McCann et al, 1988, Bartell and Mustoe, 1989, Wood and McMahon, 1989)

7.4 Side effects of soft tissue expansion

Alterations of growth and development of the palatal and dento-alveolar structures, induced by palatal mucopenosteal TE were significant in the sagittal, vertical, and transversal directions In the cephalometric analysis of the effects of palatal TE, significant antero-postenor growth retardation at the level of the bony palate was found during active expansion The incisor point seemed to be less protruded in the anterior direction, when compared to the stable optical foramen In the vertical direction, however, the incisor point showed a more inferior position, indicating an accelerated anterior nasomaxillary height increase These phenomena may be attributed to the increasing tensile forces exerted on the palatal mucopenosteum by the underlying tissue expander volume, causing overeruption and palatal tipping

152 General discussion ot the incisors The increasing volume of the expander may have caused clinically unnoticed mouth closure and occlusion disturbances resulting in the overeiuption of teeth It remains uncertain to what extent the palatal marginal incision from cuspid to cuspid for the introduction of the tissue expander plays a role In the three-dimensional study, no striking sagittal (antero-postenor) differences between the expansion and control groups were noted On the other hand, in the transversal direction, significant anterior palatal width restriction, with unilateral dento-alveolar arch deformation was found to occur during active expansion These phenomena were best explained by tensile forces of the palatal mucopenosteum induced by the increasing expander volume, the presence of the simulated Langenbeck operation scars, and the effect of the unilateral filling port tunnel Histological analysis showed epithelial thinning and reorientation of fibres in the connective tissue layers, indicating the forementioned tensile forces in the palatal mucopenosteum The number of Sharpey's fibres in the palatal dento-alveolar transition area also increased, indicative of transverse tensile forces with possible restriction of transverse growth and development Underneath the expander base, a marked bony depression was observed, sometimes resulting in a local interruption of the bony boundary between the nasal and oral cavities This may have caused negative effects on the vomero- palatine and palatal growth centres Earlier studies have demonstrated the detrimental effects of midpalatal suture alterations on palatal growth and development in cats (Freng, 1979, 1981, Voss and Freng, 1982, 1983) After removal of the tissue expanders, especially in the scarred TE group, acceleration of transverse palatal growth and longitudinal cranial base growth was observed Explanations for these phenomena remain unclear and cannot be forwarded on the basis of the 'during expansion' data and histological data Before and during active TE, no significant effects of the simulated Langenbeck operation at the age of eight weeks, were seen Histologically, no distinction between scarred and non-scarred animals could be made

153 Chapter 7

7.5 Influence of scars

Scars in the palatal soft tissues were induced to mimic the clinical human situation of cleft lip and palate patients (CLP). In CLP, the soft palate is usually closed at the age of 12 to 18 months by means of an operation according to Langenbeck (1861) or Furlow (1986). The hard palate cleft is often closed at the age of 5 years, to improve speech at an early age. The scars that go with these operations may influence later surgery for closure of residual oronasal fistulae and alveolar clefts. It was hypothesized that it might also influence palatal TE capability. Scarring of the palatal soft tissues at the age of 8 weeks, had no significant effects on palatal and dento-alveolar growth, in both the sagittal and transversal directions till the age of 20 weeks. This was in contrast to earlier studies performed on beagles (Wijdeveld et al., 1987, 1988). Palatal and dento-alveolar growth disturbances in mainly the transversal direction, were reported after simulated Langenbeck operation (Wijdeveld et al., 1989, 1991). The present study failed to show objective histologic, morphometric or cephalometric effects of scarring of the palatal tissues. However, the complication rate was highest in the sham scar group. Most erosions, leakages, extrusions and signs of infection around the tissue expanders, were seen in this group. A final explanation cannot be forwarded on the basis of this study. It may be suggested that the dead space around the non-expanded tissue expanders may lead to invasion of micro-organisms within the capsule causing irritation of the scarred tissue. Perhaps the cats used their claws to scratch the irritation.

7.6 Application in clinical CLP setting

Whether or not palatal mucoperiosteal TE may be considered as a realistic adjunct to palatal fistula closure in young growing cleft palate (CLP) patients, is discussed. Extrapolation of the findings of the present studies to the human clinical situation is tempting, but highly speculative. The main theme of this

154 General discussion discussion is how to interpret the results of the present study in relation to CLP-palients in general and young growing individuals in particular Is TE indeed an appropriate technique to be used in primary closure of a palatal cleft (or defect)9 Based on the experimental findings, palatal mucopenosteal TE is feasible and an expected gain of up to 85% of the expander base surface may be anticipated Soft tissue gain in both transverse and sagittal directions will be approximately 30% This might be beneficial and allow easier closure of the cleft or covering a defect, with less traction on the wound edges and less denudation of the palatal bone Since there is no donor site defect there is no denuded palatal bone The Bardach beagle studies have demonstrated the detrimental effects on transverse palatal growth of denudation of the bone (Bardach and Kelly, 1990, Bardach et al , 1994) Mucopenosteal elevation caused (almost) no or at least considerably less growth disturbances than denudation Palatal mucopenosteal TE may be compared with long-standing (or chronic) elevation of the mucopenosteum However, a significant antero­ posterior and transverse palatal growth disturbance is also to be expected This growth disturbance may be temporary, and compensation by catch-up growth may take place in the long run The present study has shown accelerated growth in the eight-week period after removal of the tissue expander, especially in the scarred tissue group It is debatable whether TE induced growth disturbances are less pronounced compared to the restrictions caused by conventional cleft/defect closure procedures Practical objections must also be elucidated and incorporated in the decision making TE is a multistage procedure with two separate operations and multiple out-patient clinic filling sessions in between This is time consuming tor both patients and On the other hand, if this investment pays off in time by avoiding tertiary surgical interventions such as Le Fort I (II and III) advancement osteotomies, it will be worthwhile It is still impossible to induce identical clefts in non rodent animals by means of gene manipulation or medication (Natsume et al , 1994) Surgical induction of a palatal cleft does not compare to naturally developing clefts and causes significant bias of the scientific cleft closure experiments (Meijer and Prahl, 1978) Prospective human cleft palate treatment/growth alteration

155 Chapter 7 experiments take many years and have intrinsic ethical implications The use of palatal mucopenosteal TE techniques in adults with persistent oronasal fistulae, as an adjunct to standard cleft palate surgery, overcomes part of this problem With the patient's informed consent of the techniques and status of the TE technique, one patient was considered for TE (Van Damme and Freihofer, 1996) This patient was scheduled for closure of the persistent fistula by means of a lingual flap (2-stage procedure) It proved that human palatal mucopenosteal TE is also feasible, but that the quality and vascularity of the surrounding tissues remain as important as ever In conclusion it may be stated that TE of palatal tissues in growing patients has not yet sufficiently proven to be non detrimental for dento- maxillofacial growth and development However, in adult non-growing patients with persistent oronasal fistulae it may be a valuable adjunct to standard closure techniques, and a realistic alternative for a pedicled tongue flap

7.7 Literature

ANTONYSHYN O, GRUSS JS, ZUCKER R, MACKINNON SE (1988) Tissue expansion in head and neck reconstruction Plast Reconstr Surg 82 58 68 AUSTAD ED, THOMAS SB, PASYK KA (1986) Tissue expansion dividend or loan > Plast Reconstr Surg 78 63-67 AUSTAD ED (1987) The origin of expanded tissue Clin Plast Surg 14 431-433 BARDACH J, KELLY KM (1990) Does interference with mucopenosteum and palatal bone affect craniofacial growth 9 An experimental study in Beagles Plast Reconstr Surg 86 1093-1100 BARDACH J, KELLY KM, SALYER KE (1994) Relationship between the sequence of lip and palate repair and maxillary growth an experimental study in Beagles Plast Reconstr Surg 93 269 278 BARTELL TH, MUSTOE TA (1989) Animal models of human tissue expansion Plast Reconstr Surg 83 681 686 BROBMANN GF, HUBER J (1985) Effects of different shaped tissue expanders on transluminal pressure, oxygen tension, histopathologic changes, and skin expansion in pigs Plast Reconstr Surg 76 731-736

156 General discussion

CHERRY GW, AUSTAD ED, PASYK KA, McCLATCHEY KD, ROHRICH RJ (1983) Increased survival and vascularity of random-pattern skin Haps elevated in controlled, expanded skin Plast Reconstr Surg 72 680-685 CULLEN KW, POWELL В (1989) Tissue expanders in surgery Br J Clin Pract 43 75-77 DUITS EHA, MOLENAAR J, VAN RAPPARD JHA (1989) The modeling of skin expanders Plast Reconstr Surg 83 362 365 FRENG A (1979) The restorative potential of double layered mucopenosteum A study on experimental mid-palatal clefts in the cat Scand J Plast Reconstr Surg 13 313-320 FRENG A (1981) Growth of the middle face in experimental early bony fusion of the vomeropremaxillary, vomeromaxillary and mid palatal sutural system A Roentgencephalometnc study in the domestic cat Scand J Plast Reconstr Surg 15 117-125 FURLOW LT (1986) Cleft palate repair by double opposing Z-plasty Plast Reconstr Surg 78 724-735 LANGENBECK В (1861) Operation der angeborenen totalen Spaltung des harten Gaumens nach einer neuen Methode Dtsch Klin 24 McCANN JJ, MITCHELL GM, O'BRIEN BM, VANDERKOLK CA (1988) Comparative viability of expanded and unexpanded axial pattern skin flaps in pigs Br J Plast Surg 41 294-297 MEIJER R, PRAHL В (1978) Influences of different surgical procedures on growth of dentomaxillary complex in dogs with artificially created cleft palate Ann Plast Surg 1 460-465 MOELLEKEN BRW, MATHES SJ, CANN CE, SIMMONS DJ, GHAFOORI G (1990) Long-term effects of tissue expansion on cranial and skeletal bone development in neonatal miniature swine clinical findings and histomorphometnc correlates Plast Reconstr Surg 86 825-834 NATSUME N, MIYAJIMA K, KINOSHITA H, KAWAI I (1994) Incidence of cleft lip and palate in Beagles (Letter) Plast Reconstr Surg 93 439 NORDSTROM REA, DEVINE JW (1985) Scalp stretching with a tissue expander for closure of scalp defects Plast Reconstr Surg 75 578 581 PASYK KA, ARGENTA LC, HASSETT С (1988) Quantitative analysis of the thickness of human skin and subcutaneous tissue following controlled expansion with a silicone implant Plast Reconstr Surg 81 516-523 PATEL P-K (1986) Estimating the tissue-expander volume a poor man's recipe (Letter) Plast Reconstr Surg 78 426-427

157 Chapter 7

SHIVELY RE (1986) Skin-expander volume estimator Plast Reconstr Surg 77 482 483 SHIVELY RE (1988) Surface-area increase in tissue expansion (Discussion) Plast Reconstr Surg 82 838-839 VAN DAMME PHA (1994) Soft tissue expansion in cleft palate surgery Plast Reconstr Surg 93 1307 VAN DAMME PHA, FREIHOFER HPM (1996) Palatal mucopenosteal expansion as an adjunct to palatal fistula repair case report and review of the literature Cleft Palate-Craniolac J 33 255 257 VAN DAMME PHA, FREIHOFER HPM, MALTHA JC, KUIJPERS-JAGTMAN AM VAN 'T HOF MA (1996b) Three-dimensional morphometnc analysis ot the effects ot subperiosteal palatal soft tissue expansion in growing cats Int J Oral Maxillofac Surg In press VAN DAMME PHA, FREIHOFER HPM, VAN'T HOF MA, KUIJPERS JAGTMAN AM, MALTHA JC, SPIJKERS JMC (1994) Radiologic analysis ol the effects ot subperiosteal palatal soft-tissue expansion in growing cats Int J Oral Maxillofac Surg 23 393 394 VAN DAMME PHA, HEIDBUCHEL KLWM, KUIJPERS-JAGTMAN AM, MALTHA JC, FREIHOFER HPM (1992) Cranio-maxillo-facial tissue expansion, experimentally based or clinically empiric9 A review ot the literature J Cranio-Max-Fac Surg 20 61-69 VAN DAMME PHA, MALTHA JC, FREIHOFER HPM, KUIJPERS-JAGTMAN AM (1996c) Histological analysis ot the effects of palatal mucopenosteal soft tissue expansion in growing cats Int J Oral Maxillofac Surg Submitted VAN DAMME PHA, MALTHA JC, KUIJPERS-JAGTMAN AM, FREIHOFER HPM, VAN'T HOF MA (1996a) Two-dimensional cephalometnc analysis of the effects ot subperiosteal palatal soft tissue expansion in growing cats Plast Reconstr Surg In press VANDERKOLK CA, McCANN JJ, KNIGHT KR, O'BRIEN BM (1987) Some further characteristics of expanded tissue Clin Plast Surg 14 447-453 VAN RAPPARD JHA (1988) Controlled tissue-expansion in reconstructive surgery, PhD Thesis, University of Groningen, The Netherlands VAN RAPPARD JHA, MOLENAAR J, VAN DOORN К, SONNEVELD GJ, BORGHOUTS JMHM (1988) Surface-area increase in tissue expansion Plast Reconstr Surg 82 833-837

158 General discussion

VISTNES LM, KSANDER GA, KOSEK J (1978). Study of encapsulation of silicone rubber implants in animals A foreign-body reaction. Plast Reconstr Surg 62 580-588 VOSS R, FRENG A (1982). Growth of the dental arches after ablation of the mid- palatal suture. A study in the domestic cat. J Max-Fac Surg 10: 259-263. VOSS R, FRENG A (1983). Concomitant transverse growth of the maxillary base and dental arch in experimental submucous mid-palatal clefts. A biometrical study in the domestic cat. J Max-Fac Surg 11 257-262 WIJDEVELD MGMM, GRUPPING EM, KUIJPERS-JAGTMAN AM, MALTHA JC (1987). Mucopenosteal migration alter palatal surgery in beagle dogs. A longitudinal radiographic study Int J Oral Maxillofac Surg 16. 729-737 WIJDEVELD MGMM, GRUPPING EM, KUIJPERS-JAGTMAN AM, MALTHA JC (1988) Growth of the maxilla after soft tissue palatal surgery at different ages on beagle dogs - a longitudinal radiographic study J Oral Maxillofac Surg 48: 204-209. WIJDEVELD MGMM, GRUPPING EM, KUIJPERS-JAGTMAN AM, MALTHA JC (1989). Maxillary arch dimensions after palatal surgery at different ages on beagle dogs. J Dent Res 68 1105-1109. WIJDEVELD MGMM, MALTHA JC, GRUPPING EM, DE JONGE J, KUIJPERS- JAGTMAN AM (1991) A histological study of tissue response to simulated palatal surgery at different ages on beagle dogs. Arch Oral Biol 36: 837-843. WOOD FM, McMAHON SB (1989). The response of the peripheral nerve field to controlled soft tissue expansion. Br J Plast Surg 42: 682-686

159

Chapter 8

Summary

Summary

In this thesis an animal study is described on the feasibility and the effects of palatal mucopenosteal sott tissue expansion (TE) in growing cats This application and its results are related to clinical human cleft palate repair The main idea was to develop a technique to close palatal defects and oronasal communications in an easier or better fashion than the well established ones, by gaining tissue from the direct surroundings, without creation of a donor site defect, and with less negative effects on growth and development of the dento-maxillofacial complex The effects ot TE in scarred and non scarred tissues were compared Chapter 1 is an introduction which reviews the problems ot cleft lip, alveolus and palate deformity The cleft related oronasal communications are to be closed to improve nutrition, respiration, speech and hearing, and dental arch development The necessary operations are phased in time and modified several times, but nevertheless, they are still known to induce retardation and restriction of dento-maxillofacial growth Denudation of palatal bone with healing by secondary intention and the inherent scarring, are detrimental in this respect TE was thought to be a potential improvement of the present cleft closure techniques Because there were hardly any scientific data available to support this assumption, it was deemed necessary to carry out a study in growing animals The most important questions to be answered are 'what is the effect of palatal submucopenosteal TE on scarred and non-scarred soft tissues ('is there a real gain of tissue9')9' and 'what is the effect on the surrounding hard tissues ('is there marked retardation in their growth and development9')9' Chapter 2 presents a review of the literature (from 1981 to 1992) dealing with cranio maxillofacial TE TE in skin has been extensively investigated and is an established technique The application of (sub)mucosal and (sub)mucopenosteal TE appears to be mainly based on empirical data and lacks support of experimental studies The deficiencies in scientific knowledge and points of attention for future research are indicated A survey of more recent literature (1991 to 1996) is provided in Chapter 10 Chapter 3 deals with the cephalometnc analysis of the effects of palatal submucopenosteal TE in scarred and non scarred tissues Seventy-five

163 Chapter 8 growing domestic torn cats, assigned to five groups, were used Tracings of series standardized lateral cephalograms were digitized The soft tissue gain and the effect on the adjacent and distant bony structures were quantified for the period of active expansion and the period after removal of the tissue expander and part of the gained tissue A statistically significant soft tissue gain can be achieved Palatal sagittal growth was significantly retarded and anterior nasomaxillary height increase was accelerated during expansion There were no significant differences between scarred and non scarred tissues After removal of the tissue expander initial correction of abnormal growth was noticed, in particular in the scarred tissue group In Chapter 4, the results of the three-dimensional morphometric analysis of the effects of palatal submucopenosteal TE in scarred and non-scarred tissues are described Series of dental casts of 75 young growing domestic torn cats, assigned to five groups, were studied and digitized, using a Reflex Microscope The soft tissue gain and the effects on the surrounding bony and dental structures were quantified After eight weeks of active expansion a soft tissue area gain of 85% of the base surface of the expander could be realized The soft tissue gain in sagittal direction was 32%, whereas the soft tissue increase in transversal direction was 36% of the base diameter During expansion significant deceleration of (anterior) transverse and sagittal growth was evident There were no significant differences between scarred and non- scarred tissues After removal of the tissue expander acceleration of growth was noticed, in particular in the scarred tissue group In Chapter 5, the results of the histological analysis are described From the 75 young growing cats used in this study, 67 biopsy specimens were taken and 38 cats were sacrificed for more extensive histological evaluation Mucopenosteal expansion led to thinning of the epithelium and the original connective tissues layers overlying the expander A fibrous capsule developed Its benefits can be questioned as elastic fibres and contractile myofibroblasts were present and no increase in vascularity could be established A depression of the palatal bone directly under the expander base, caused by osteoclastic bone resorption, was another debatable finding This seems to be the main explanation for the deceleration of palatal growth during active expansion All these phenomena seem to be reversible and they

164 Summary normalize in time after removal of the tissue expander Chapter 6 describes a case report of clinical application of the palatal mucopenosteal expansion technique as an adjunct to cleft palate repair in a 37 years old patient Conventional TE with a standard tissue expander was performed and proved to be a feasible and realistic technique However, the final outcome seemed to be dependent on the quality and the vascularity of the ad|acent tissues The technique could be an alternative to a lingual flap in the closure of persistent oronasal fistulae in adult patients In Chapter 7 the data from the previous chapters are mutually compared and related to each other A critical evaluation of their value for application in the clinical setting of cleft palate repair in the growing individual is presented It is concluded that palatal mucopenosteal TE is a feasible technique in growing domestic cats and adults However, good tissue quality and vascularity seem to be prerequisites for a successful use In the animal study hemispherical expanders with a diameter of 12 mm were used After eight weeks of active expansion a mean soft tissue area gain of 85% of the expander base surface was reached The gain in the sagittal direction was 32%, whereas in the transversal direction 36% increase was noted At histological level, thinning of the epithelium and the original connective tissue layers was evidenced The orientation of the collagenous fibres in the reticular layer had changed to a direction more parallel to the surface of the expander Also, a fibrous capsule had developed, however without increased vascularity of the capsular tissue A statistically significant retardation of both sagittal and transverse growth of the bony palate was observed during expansion On the other hand, the vertical growth of the anterior nasomaxillary height was accelerated These phenomena disappeared and normalized in the time span after removal of the tissue expander and part of the gained tissue In the previously scarred tissue animals acceleration of sagittal and transverse growth was recorded Whether this accelerated growth has to be interpreted as 'catch-up' growth, which would lead to the normal size and measures of unoperated controls, is open to question

165 Chapter 8

Further research is indicated to decide whether or not extrapolation of these animal data to the clinical setting of cleft palate repair in growing individuals is allowed. In adult patients this TE technique can be used as an alternative to a pediclcd tongue flap.

166 Chapter 9

Samenvatting

Samenvatting

In dit proefschrift wordt een dierexperimenteel onderzoek beschreven met betrekking tot de toepassingsmogelijkheden en de effecten van weefselexpansie van het palatinale mucoperiosteum in groeiende katten. Er wordt een verband gelegd met de klinische behandeling van cheilognatho- palatoschisis bij mensen. De achtergrondgedachte hierbij was het ontwikkelen van een techniek om palatumdefecten en oronasale communicaties gemakkelijker en/of beter te kunnen sluiten, met weefsel gewonnen uit de directe omgeving zonder een donorgebied defect te creëren en met minder negatieve uitwerking op de groei en ontwikkeling van het dento-maxillofaciale complex dan de klassieke methoden. De effecten van weefselexpansie in wel en niet verlittekend weefsel werden met elkaar vergeleken. In Hoofdstuk 1 worden de globale achtergronden van cheilognatho- palatoschisis en de chirurgische behandeling daarvan beschreven. De oronasale communicaties worden gesloten teneinde de voeding, de ademhaling, de spraak en het gehoor, en de ontwikkeling van de tandboog te verbeteren. De noodzakelijke chirurgische ingrepen worden in de tijd gefaseerd uitgevoerd en zijn diverse malen gemodificeerd, maar ondanks dat veroorzaken zij nog altijd vertraging en vermindering van de dentomaxillaire groei. Dit wordt waarschijnlijk veroorzaakt doordat steeds ontblote botgebieden ontstaan die vervolgens door secundaire heling genezen. De daarbij behorende vorming van littekenweefsel zou een negatief effect op de groei en ontwikkeling hebben. Weefselexpansie zou kunnen leiden tot een mogelijke verbetering van de bekende sluitingstechnieken. Aangezien er echter nauwelijks wetenschappelijke gegevens zijn die deze veronderstelling onderbouwen is besloten een dierexperimenteel onderzoek hiernaar uit te voeren. De belangrijkste vraagstellingen zijn: "wat is het effect van weefselexpansie van palatinaal mucoperiosteum dat al dan niet verlittekend is ("is er echte weefselwinst?")?" en "wat is het effect op de omgevende harde weefsels ("is er een vertraging in hun groei en ontwikkeling?")?". Hoofdstuk 2 geeft een overzicht van de literatuur (van 1981 tot 1992) over cranio-maxillofaciale weefselexpansie. Weefselexpansie van de huid is uitgebreid onderzocht en is een geaccepteerde techniek. Het gebruik van (sub)mucosale of (sub)mucoperiostale weefselexpansie blijkt voornamelijk

169 Chapter 9 gebaseerd op gegevens die proefondervindelijk zijn verkregen en mist een wetenschappelijke onderbouwing door experimenteel onderzoek Er wordt aandacht besteed aan de leemte in kennis en aan aandachtspunten voor toekomstig experimenteel wetenschappelijk onderzoek Een overzicht van meer recente literatuur (van 1991 tot 1996) is weergegeven in Hoofdstuk 10 Hoofdstuk 3 beschrijft de cefalometnsche analyse van de effecten van palatinale submucopenostale weefselexpansie in al of niet verlittekend weefsel Er werden 75 groeiende katten gebruikt die verdeeld waren over vijf groepen Metingen werden verricht aan tracings van series gestandaardiseerde midsagittale schedelrontgenopnamen De weefselwinst in de weke delen en de effecten op de afmetingen van de benige structuren werden gekwantificeerd voor de periode van actieve weefselexpansie en de periode na verwijdering van de weefselexpander εη een deel van het gewonnen weefsel Een significante weefselwinst in de weke delen lijkt te kunnen worden gerealiseerd De palatinale lengtegroei blijkt door de weefselexpansie te worden geremd en de voorste gelaatshoogte neemt meer toe door de weefselexpansie Er bleken geen significante verschillen te bestaan tussen de groepen met en zonder littekens Na het verwijderen van de weefselexpander lijkt correctieve groei op te treden, vooral in de littekengroep In Hoofdstuk 4 worden de resultaten van de driedimensionale morfo metri sehe analyse van de effecten van palatinale mucopenostale weefselexpansie beschreven Series gebitsmodellen van 75 groeiende katten die waren verdeeld over vijf groepen zijn met behulp van een reflex microscoop gedigitaliseerd De weefselwinst van de weke delen en de effecten op omgevende benige en dentale structuren werden gekwantificeerd Na acht weken actieve weefselexpansie werd een winst bereikt van 85% van het basisoppervlak van de gebruikte weefselexpander De winst in sagittale richting was 32% en in transversale richting 36% Tijdens expansie was er een vertraging van (antérieure) transversale en sagittale groei Er werden geen significante verschillen tussen de litteken- en niet-httekengroepen geconstateerd Na verwijdering van de weefselexpander was er sprake van versnelling van groei, vooral in de littekengroep In Hoofdstuk 5 worden de resultaten van een histologische analyse beschreven Van de 75 groeiende katten die in deze studie werden gebruikt

170 Samenvatting werden 67 biopsieen genomen en bestudeerd 38 katten werden opgeofferd voor een uitgebreide histologische evaluatie Weefselexpansie van het palatinale mucopenosteum leidde tot het dunner worden van het epitheel en het oorspronkelijke bindweefsel boven de weefselexpander Er ontstond een fibreus kapsel Het nut hiervan kan in twijfel worden getrokken aangezien er elastische vezels en myofibroblasten in werden aangetroffen en er geen toename in de vasculansatie kon worden waargenomen Er ontstond een depressie in het palatinale bot direct onder de basis van de weefselexpander, veroorzaakt door osteoclastische botresorptie Dit lijkt de voornaamste verklaring voor de groeivertraging tijdens actieve weefselexpansie Deze verschijnselen lijken alle reversibel en normaliseren na verwijdering van de weefselexpander Hoofdstuk 6 beschrijft de klinische toepassing van de palatinale mucopenostale weefselexpansietechniek bij een poging tot het sluiten van een persisterende oronasale communicatie in een 37-jange patient Hierbij werd gebruik gemaakt van een standaard weefselexpander en conventionele expansie Technisch bleek deze ingreep mogelijk, maar het uiteindelijke resultaat is waarschijnlijk afhankelijk van de kwaliteit van het omringende weefsel en de vaatvoorziening Deze techniek zou een alternatief kunnen zijn voor een gesteelde tonglap bij het sluiten van persisterende oronasale communicaties in volwassen patiënten In Hoofdstuk 7 worden de gegevens uit de vorige hoofdstukken onderling vergeleken en geïntegreerd, waarna een kritische beschouwing van de waarde ervan voor toepassing in de klinische setting van schisischirurgie bij groeiende individuen wordt gegeven Dit leidt tot de conclusie dat palatinale mucopenostale weefselexpansie een technisch goed toepasbare methode is in groeiende katten en volwassen patiënten Een voorwaarde voor een succesvolle toepassing lijkt wel te zijn dat het omringende weefsel van een goede kwaliteit is en dat er een goede vaatvoorziening aanwezig is In het dierexperiment werd gebruik gemaakt van halve-bolvormige weefselexpanders met een diameter van 12 mm Na acht weken actieve weefselexpansie was de winst in het oppervlak van de weke delen gemiddeld ongeveer 85% van het basisoppervlak van de weefselexpander De

171 Chapter 9 weefselwinst in de lengterichting was 32% en in breedterichting 36%. Op histologisch niveau werd geconstateerd dat het epitheel en het bindweefsel boven de weefselexpander dunner werden tijdens de weefselexpansie. De oriëntatie van de collagene vezels in het bindweefsel was gewijzigd in een verloop meer parallel aan het oppervlak van de weefselexpander. Tevens ontstond een fibreus kapsel, zonder duidelijke toename van de vascularisatie van het weefsel. Zowel de lengte- als breedte-groei van het benige palatum bleek tijdens expansie significant vertraagd. De groei van de voorste gelaatshoogte was daarentegen versneld. Deze verschijnselen verdwenen en normaliseerden in de loop van de tijd na verwijdering van de weefselexpander en een deel van het gewonnen weefsel. Vooral in de situaties waarin littekenweefsel aanwezig was trad een versnelling van de lengte- en breedtegroei op in die periode. Of deze groeiversnellling kan worden beschouwd als "catch-up growth", waardoor uiteindelijk weer normale afmetingen bereikt kunnen worden, wordt uit dit onderzoek niet duidelijk. Nader onderzoek zal moeten uitwijzen of extrapolatie van de dierexperimentele gegevens naar de klinische situatie bij schisischirurgie in groeiende individuen verantwoord is. Bij volwassen patiënten kan de techniek worden aangewend als alternatief voor een gesteelde tonglap.

172 Chapter 10

Survey of recent literature

Recent literature

Because Chapter 2, reviewing the literature on cranio-maxillofacial soft tissue expansion (TE) (Van Damme et al , 1992), was prepared before 1992, it seems relevant to supplement the data with recent literature on experimental and cranio-maxillofacial applications of TE. As is rather common with 'new techniques', they are continously introduced (Neumann, 1957; Radovan, 1976), then become popular and wide-spread (Radovan, 1982, 1984; Van Rappard, 1988; Van Damme et al., 1992), and finally are assigned the place they deserve and receive less and less attention. This leads to fewer publications on their use and importance. TE has now attained an important place in cranio-maxillofacial reconstructive surgery, since the first experiments about 15 years ago (Argenta et al, 1981; Kawashima et al., 1994, Mclvor et al., 1994, Nordstrom, 1996). Expanded tissue used in reconstruction is usually excellently matched for colour, thickness, elasticity and other physical properties This is particularly important in reconstructions in the visible parts of the head, face and neck region Various options using either conventional, slow, rapid or intraoperative expansion are available (Wee et al., 1992; Schneider et al , 1993). Combination of conventional and intraoperative expansion is judged to be favourable regarding pain and placement time (Iwahira and Maruyama. 1993a) Combinations of different surgical reconstructive modalities with TE techniques have been advocated by Antonyshyn et al (1993). Clinical studies and case reports were addressing to new indications, new applications, modifications of techniques, and long-term evaluation of results. Burn sequelae are most frequently addressed to as an indication for application of TE techniques (Robson et ai, 1992; Spence, 1992, Zellweger and Kunzi, 1991; Datubo-Brown et al , 1994; Foyatier et ai, 1993, 1995, Echinard and Dantzer, 1995, Rose, 1995), also in children (Bauer et al., 1993, Neale et al., 1993; Hudson and Grobbelaar, 1995). The use of previously expanded full-thickness skin grafts represents a good modified solution in many situations and is deemed to be an excellent reconstructive option in children. Alopecia has become an established indication for scalp expansion in children and adults (Wang et al., 1991; Van der Veen and Pulles, 1992; Van

175 Chapter 10

Damme, 1992, Konior, 1993, Trott, 1993, Maladry et al , 1994) Often reported applications in cranio-maxillofacial surgery are forehead and nasal reconstructions (Galli et al, 1991, Webster and Marshall, 1991, Iwahira and Maruyama, 1993b, Echinard and Dantzer, 1995, Edgerton, 1995, Khoun et al , 1995, Romo et al , 1995) Expanded adjacent forehead tissue and distant tissue flaps, as island or free flaps, are frequently used (Denny, 1992) There is an increasing number of publications in dermatologie surgical oncology and otolaryngology journals on TE before, during and after removal of facial skin cancer and congenital lesions (Baker and Swanson, 1990, 1994, Johnson et al , 1990, Ehlert and Thomas, 1991, Romo and Goldberg, 1992, Auletta et al , 1993, Carruthers, 1993, Frodel and Whitaker, 1993, Greenbaum, 1993, Vergnes et al , 1993, Mclvor et al , 1994) In particular rapid intraoperative TE has gained popularity among these disciplines This technique aims at the achievement of gradual skin stretching and relatively easy closure of skin defects, which could not be closed with undermining alone However, in stretching skin, undermining seems to be more important than intraoperative expansion, and spreading of the scar afterwards was noted to be correlated to the degree to which the skin was expanded (MacKay et al , 1990) Cheek reconstruction is reported to be difficult, particularly if expanded cervical tissue is used and advanced beyond the mandibular border (Manders, 1992, Neale et al , 1993) Gravity and scarring are thought to cause ectropion of the eyelid and distortion of the lips Alternative methods using expanded prefabricated musculocutaneous free flaps were reported by Mayou et al (1992), and Igawa et al (1995) An experimental study in rats showed that pre-expanded free fat grafts survived better than non-expanded fat grafts (Von Heimburg et al , 1994) Prefabricated pedicle flaps and grafts in minipigs, however, were depending on the panniculus carnosus muscle, and clinical applications are difficult to foresee because of limitations of this animal model (Shehadi et al , 1994) The presence of the panniculus carnosus muscle in rabbits and minipigs. can bias the extrapolation of data to humans (Caffee, 1992, Timmenga, 1992, Shehadi et al , 1994) Facilitation of face lifting by stretching and TE, which was earlier

176 Reeene literature considered to be controversial, was still published (Man, 1992; Nicoletis and Gholam, 1992). The effects on craniofacial growth of orbital expansion of anophfhalmic sockets have been studied in patients by Tucker et al. (1995). Experimental growth studies dealt with orbital expansion in growing cats after orbital evisceration (Buchman et al., 1994). It was concluded that severe asymmetry and constriction in the orbital and midfacial regions was ameliorated by graded TE. It directed craniofacial bone growth, resulting in normal bony histology and improved facial form. The effects on craniofacial growth, caused by expansion of the maxillary sinus in rabbits, were studied by Persing et al. (1994). It was found that insertion and inflation of the tissue expander resulted in a significant increase in posterior facial height, followed by a significant increase in the length of the anterior cranial base. Experimental facial nerve expansion was performed in a porcine model by Malis el al. (1995). No clinical and histological signs of neural degeneration were seen. Endo and Nakayama (1993) published a histologic examination of elongated peripheral nerves in rats. They achieved an average increase in length of 88%. The nerves showed separated but intact axons, and some loss of myelin. Van der Wey et al. (1993, 1994, 1995, 1996), perfor­ med laser Doppler flowmetry controlled nerve expansion and found this control measure essential for the prevention of nerve damage (Van der Wey, 1995). This method of objective monitoring has been advocated more generally by Hal lock and Rice (1993). Subperiosteal TE in alveolar ridge augmentation was reviewed by Quayle and McCord (1992), and studied experimentally in dogs by Tominaga et al. (1993, 1994). It was found that the mandibular periosteum was repla­ ced by fibrous connective tissue during expansion. The capsule formation after subperiosteal expansion was much faster than that after subcutaneous expansion. A larger increase in vascularity was noted in the subperiosteal expansion group and the subperiosteal control group than in the subcutaneous expansion group. A thick fibrous capsule, with minimal inflammatory response, was observed one week after full inflation. Leaving the fully infla­ ted subperiosteal expanders in place for more than one month induced

177 Chapter 10 resorption ot the underlying bone The firm fibrous capsule formed by TE prevented the migration of implanted hydroxyapatite particles When the newly formed capsule overlying the bone was removed before implantation of hydroxyapatite, bony union of the particles located along the surface of the mandible was observed after 1 or 2 months In these specimens, however, also marked bone resorption of the mandibular cortex, was noted, which was not the case if the capsule was left in place No evidence of bone or osteoid formation on the expanded periosteal side was noted in either group These results indicate that the subperiosteal expanded bed is a good recipient site for onlayed implants A discussion on this matter was given by Lew (1994), and osseointegration was reviewed by Wilkes (1994) Lew and Fuseler (1993) investigated the mitotic activity of fibroblasts, and the development of extracellular matrix in pen-expander capsules in rats They observed an increased vascularity, an increase in fibroblastic proliferation and collagen production The role of contractile myofibroblasts in the capsules around tissue expanders and implants has been evaluated by Coleman et al (1993) Capsules in rats, pigs and humans showed a characteristic layered structure with myofibroblasts being the predominant cell type They framed the hypothesis that capsular contracture is analogous to wound contraction However, according to their opinion, removal of the capsule will not have any effect on future stretch back or scarring In contrast to this Azzolmi et al (1992) believe that the fibrous capsule around the expander is to be completely removed to obtain the best results in head and neck reconstructions They claim that this can be achieved without vascular risk using a special technique A completely different approach to the capsule was proposed by Cariou et al (1992) and Heymans et al (1993) They raised the pen implant capsule either as an island flap or a free flap in rats for reconstruction purposes The thickness of the capsule is related to time of expansion, and volume and surface of the expander (Bern et al , 1992) Infections around the expanders were noted to have an effect on the texture of the capsule (Coleman et al , 1993) Bacteria and fungi can be found both inside and outside the expander (Liang et al , 1993, Coady et al , 1995) To maximize the gain from tissue expanders suggestions on how to cut

178 Recent literature the flap after expansion were given by Zide and Karp (1992) and discussed by Manders (1992). The latter is concerned about the creation of more scar. Another possibility to gain more tissue is overinflation of the tissue expander (Hallock, 1995). Increased turnover of keratocytes in the basal cell layer of the epidermis was shown during expansion in humans by Olenius et al. (1993), suggesting an increase in epidermal thickness. However, skin thickness appeared to decrease during expansion, which seemed to be permanent at least until six months after the second operation (Olenius et al., 1994). Long-term histopathologic evaluation of human expanded skin by Matturri et al. (1992), revealed no striking differences with earlier reports. Twelve months after expansion, the histomorphology of the expanded skin appeared to be normal. An interesting note on the physical behaviour of the gained tissue following standard TE was delivered by Brody (1993). He is of the opinion that it should not be characterized as creep, but as 'stepped stress relaxation'. Reconstruction of rare facial clefts with the aid of TE was reported by Butow and De Witt (1990), Moore et al. (1992), Thorne (1993), and Ozgur et al. (1994). There are no data on the effects on bone growth in these cases. Clinical application in cleft palate repair was reported by Abramo et al. in 1993. Intraoperative expansion was judged to increase the surface area by distension of the palate and recruitment of the surrounding palatal tissue in young children. Comments and experimental work on this subject have been published (Van Damme, 1994, 1996; Van Damme et ai, 1994; Van Damme and Freihofer, 1996). In conclusion, there have been few experimental studies in the field of cranio-maxillofacial surgery addressing the effects of TE on bone, cartilage and mucosa at a microscopic level, or the influence on growth and development of cranio-maxillofacial structures. The present study aimed to evaluate palatal mucoperiosteal TE, its benefits and its side effects on growth and development of the dento-maxillofacial complex. So far, to the best of our knowledge, no analogous study has been performed or published.

179 Chapter IO

Literature

ABRAMO AC, VIOLA JC, ANGELO AJ (1993) Intraoperative rapid expansion in cleft palate repair Plast Recons.tr Surg 91 441-445 ANTONYSHYN OM, PALETZ JL, WILSON KL (1993) Reconstruction of composite facial defects the combined application of multiple reconstructive modalities Can J Surg 36 441-452 AULETTA MJ, MATARASSO SL, GLOGAU RG, TROMOVITCH TA (1993) Comparison of skin hooks and Foley catheters for immediate tissue expansion J Dermatol Surg Oncol 19 1084-1088 AZZOLINI A, RIBERTI C, CAVALCA D (1992) Skin expansion in head and neck reconstructive suigery Plast Reconstr Surg 90 799-807 BAKER SR, SWANSON NA (1990) Rapid intraoperative tissue expansion in reconstruction of the head and neck Arch Otolaryngol Head Neck Surg 116 1431-1434 BAKER SR, SWANSON NA (1994) Reconstruction of midfacial defects following surgical management of skin cancer the role of tissue expansion J Dermatol Surg Oncol 20 133 140 BAUER BS, VICARI FA, RICHARD ME, SCHWED R (1993) Expanded full-thickness skin grafts in children case selection, planning, and management Plast Reconstr Surg 92 59-69 BERN S, BURD A, MAY JW (1992) The biophysical and histologic properties of capsules formed by smooth and textured silicone implants Plast Reconstr Surg 89 1037 1042 BRODY GS (1993) Biological creep (Letter) Plast Reconstr Surg 92 1202-1203 BUCHMAN SR, BARTLETT SP, WORNOM IL 3RD, WHITAKER LA (1994) The role of pressure on regulation of craniofacial bone growth J Cramotac Surg 5 2-10 BUTOW KW, DE WITT TW (1990) Bilateral oblique facial cleft tissue expansion with primary reconstruction J Dent Assoc S Afr 45 507-511 CAFFEE HH (1992) The biophysical and histologic properties of capsules formed by smooth and textured silicone implants (Discussion) Plast Reconstr Surg 89 1043-1044 CARIOU JL, HILLIGOT P, ARROUVEL C, BANZET Ρ (1992) Neo-lambeau de membrane pen-prothétique a pédicule axial Etude expérimentale de l'influence du temps et du volume expanse Ann Chir Plast Esthet 37 139-144

180 Recent literature

CARRUTHERS A (1993) Tissue expansion and Mohs micrographie surgery J Dermatol Surg Oncol 19 1106-1109 COLEMAN DJ, SHARPE DT, NAYLOR IL, CHANDER CL, CROSS SE (1993) The role of contractile fibroblasts in the capsules around tissue expanders and implants Br J Plast Surg 46 547-556 COADY MS, GAYLOR J, KNIGHT SL (1995) Fungal growth within a silicone tissue expander case report Br J Plast Surg 48 428-430 DATUBO-BROWN DD, KHALID KN, LEVICK PL (1994) Tissue-expanded visor (lap in burn surgery Ann Plast Surg 32 205-208 DENNY AD (1992) Expanded midline forehead flap for coverage of nonnasal facial defects Ann Plast Surg 29 576-578 ECHINARD C, DANTZER E (1995) La reconstruction du nez dans les brûlures pan-taciales profondes Ann Chir Plast Esthét. 40 238-250 EDGERTON MT (1995) Facial reconstruction with prefabricated induced expanded (PIE) supraclavicular skin flaps (Discussion) Plast Reconstr Surg 95 1016-1017 EHLERT TK, THOMAS JR (1991) Rapid intraoperative tissue expansion for closure of facial defects Arch Otolaryngol Head Neck Surg 117 1043-1049 ENDO Τ, ΝΑΚΑ Y AMA Y (1993) Histologic examination of peripheral nerves elongated by tissue expanders Br J Plast Surg 46 421-425 FOYATIER JL, COMPARIN JP, LATARJET J, DELAY E, SPITALIER P, MASSON CL (1993) Forum l'expansion tissulaire Réparation des séquelles de brûlure de la face par expansion cutanée cervicale Ann Chir Plast Esthét 38 27-33 FOYATIER JL, GOUNOT N, COMPARIN JP, DELAY E, MASSON CL, LATARJET J (1995) Les greffes de peau totale préalablement expansée principes techniques Indications dans la reparation des séquelles de brûlures a propos de 22 cas Ann Chir Plast Esthét 40 279-285 FRODEL Jr JL, WHITAKER DC (1993) Primary reconstruction of congenital facial lesion defects with tissue expansion J Dermatol Surg Oncol 19 1110-1116 GALLI A, BERRINO P, SANTI Ρ (1991) A systematic aesthetic approach to primary closure of the donor site following transposition of vertical forehead flaps Aesthetic Plast Surg 15 245-250 GREENBAUM SS (1993) Intraoperative tissue expansion with the Foley catheter J Dermatol Surg Oncol 19 1079-1083

181 Chapter 10

HALLOCK GG (1995) Safety ot clinical ovennflation ol tissue expanders Plast Reconstr Surg 96 153-157 HALLOCK GG, RICE DC (1993) Increased sensitivity in objective monitoring of tissue expansion Plast Reconstr Surg 91 217-222 HEYMANS M, LENGELE B, LAHLALI N, VANWIJCK С (1993) A pen-implant capsule flap Br J Plast Surg 46 456-459 HUDSON DA, GROBBELAAR АО (1995) The use of tissue expansion in children with burns of the head and neck Burns 21 209-211 IGAWA HH, MINAKAWA HM, SUGIHARA T, HOMMA К (1995) Cheek reconstruction with an expanded prefabricated musculocutaneous free flap case report Br J Plast Surg 48 569-571 IWAHIRA Y, MARUYAMA Y (1993a) Combined tissue expansion clinical attempt to decrease pain and shorten placement time. Plast Reconstr Surg 91 408-415 IWAHIRA Y, MARUYAMA Y (1993b) Expanded unilateral forehead flap (sail flap) for coverage of opposite forehead defect Plast Reconstr Surg 92 1052-1056 JOHNSON TM, BROWN MD, SULLIVAN MJ, SWANSON NA (1990) Immediate intraoperative tissue expansion J Am Acad Dermatol 22 283-287 KAWASHIMA T, YAMADA A, UEDA K, ASATO H, HARII К (1994) Tissue expansion in (acial reconstruction Plast Reconstr Surg 94 944-950 KHOURI RK, OZBEK MR, HRUZA GJ, YOUNG VL (1995) Facial reconstruction with prefabricated induced expanded (PIE) supraclavicular skin flaps Plast Reconstr Surg 95 1007-1015, Discussion 1016-1017 KONIOR RJ (1993) Advances in surgical hair restoration. Facial Plast Surg 9 37-48 LEW D (1994) Subperiosteal tissue expansion for mandibular augmentation with hydroxylapatite particles an experimental study (Discussion) J Oral Maxillofac Surg 52 950-951 LEW D, FUSELER JW (1993) The effect ot pulsed expansion of subfascially placed expanders on the extent and duration of mitosis in the capsule and rat integument J Oral Maxillofac Surg 51 154-158 LIANG MD, NARAYANAN K, RAVILOCHAN K, ROCHE К (1993) The permeability of tissue expanders to bacteria an experimental study Plast Reconstr Surg 92 1294-1297

182 Recent literature

MACKAY DR, SAGGERS GC, KOTWAL Ν, MANDERS ΕΚ (1990) Stretching skin undermining is more important than intraoperative expansion Plast Reconstr Surg 86 722-730 MALADRY D, BRABANT B, BERARD V, DUPUIS Ρ, MITZ V (1994) Secondary expansion of a totally replanted scalp for aesthetic adjustment Plast Reconstr Surg 94 1052-1054 MALIS DJ, MACMILLAN JG, KELLEY JL, McGATH JH (1995) Tissue expansion of the facial nerve in an animal model Ear Nose Throat J 74 261-270 MAN D (1992) Stretching and tissue expansion tor face lift Facial Plast Surg 8 52-58 MANDERS EK (1992) Maximizing gain from rectangular tissue expanders (Discussion) Plast Reconstr Surg 90 505-506 MATTURRI L, AZZOLINI A, RIBERTI C, LAVEZZI AM, CAVALCA D, VERCESI F, AZZOLINI С (1992) Long-term histopathologic evaluation of human expanded skin Plast Reconstr Surg 90 636-642 MAYOU BJ, GAULT DT, CROCK JG (1992) Tissue expanded tree flaps Br J Plast Reconstr Surg 45 413 417 McIVOR NP, FONG MW, BERGER KJ, FREEMAN JL (1994) Use of tissue expansion in head and neck reconstruction J Otolaryngol 23 46-49 MOORE MH, TROTT JA, DAVID DJ (1992) Soft tissue expansion in the management of the rare craniofacial clefts Br J Plast Surg 45 155-159 NEALE HW, KURTZMAN LC, GOH KB, BILLMIRE DA, YAKUBOFF KP, WARDEN G (1993) Tissue expanders in the lower face and anterior neck in pediatric burn patients limitations and pitfalls Plast Reconstr Surg 91 624 631 NEUMANN CG (1957) The expansion of an area of skin by progressive distention of a subcutaneous balloon Plast Reconstr Surg 19 124-130 NICOLETIS C, GHOLAM D (1992) L'etage frontal dans le lifting cervico-facial technique du "cimier de casque" associée à une expansion rapide Ann Chir Plast Esthet 37 27-34 NORDSTROM REA (1996) Tissue expansion Boston Butterworth-Heinemann OLENIUS M, DALSGAARD CJ, WICKMAN M (1993) Mitotic activity in expanded human skin Plast Reconstr Surg 91 213-216 OLENIUS M, WICKMAN M, MALM M, JURELL G (1994) Skin thickness in expanded human breast skin Plast Reconstr Surg 93 1428-1432

183 Chapter 10

OZGUR FF, KOCABALKAN O, GURSU KG (1994) Tissue expansion in median facial cleft reconstruction a case report Int J Oral Maxillofac Surg 23 137-139 PERSING JA, GAMPPER TJ, MORGAN E, WOLCOTT Ρ (1994) Experimental expansion of the maxillary sinus J Craniofac Surg 5 1115 QUAYLE AA, McCORD JF (1992) Current status of tissue expanders in alveolar ridge augmentation a review Implant-Dent 1 177-181 RADOVAN С (1976) Adjacent flap development using expandable Silastic implant Presented at the annual meeting of the Am Soc Plast Reconstr Surg, Boston, Mass, USA RADOVAN С (1982) Breast reconstruction after mastectomy using the temporary expander Plast Reconstr Surg 69 195 206 RADOVAN С (1984) Tissue expansion in sott-tissue reconstruction Plast Reconstr Surg 74 482-490 ROBSON MC, BARNETT RA, LEITCH IO, HAYWARD PG (1992) Prevention and treatment of postburn scars and contracture World J Surg 16 87-96 ROMO Τ 3RD, GOLDBERG J (1992) Versatile use of skin expanders in facial plastic surgery Arch Otolaryngol Head Neck Surg 118 333-337 ROMO Τ 3RD, JABLONSKI RD, SHAPIRO AL, McCORMICK SA (1995) Long term nasal mucosal tissue expansion use in repair of large nasoseptal peiforations Arch Otolaryngol Head Neck Surg 121 327-331 ROSE EH (1995) Aesthetic restoration ot the severely disfigured face in burn victims a comprehensive strategy Plast Reconstr Surg 96 1573-1585, Discussion 1586-1587 SCHNEIDER MS, WYATT DB, KONVOLINKA CW, HASSANEIN KM, HIEBERT JM (1993) Comparison of rapid versus slow tissue expansion on skin flap viability Plast Reconstr Surg 92 1126-1132 SHEHADI SI, DONOVAN P, STEIGMAN CK, VOGLER CA, VOGLER GA (1994) Implantation and expansion of split-thickness skin grafts a new source of prefabricated pedicle flaps and grafts Plast Reconstr Surg 93 1449-1458 SPENCE RJ (1992) Experience with novel uses of tissue expanders in burn reconstruction of the tace and neck Ann Plast Surg 28 453 464 THORNE CH (1993) Craniofacial clefts Clin Plast Surg 20 803-814 TIMMENGA EJF (1992) Skin expansion an experimental study in the rabbit PhD Thesis, University of Amsterdam, The Netherlands TOMINAGA K, MATSUO T, KUGA Y, MIZUNO A (1993) An animal model lor subperiosteal tissue expansion J Oral Maxillofac Surg 51 1244-1249

184 Recent literature

TOMINAGA K, MATSUO T, KUGA Y, MIZUNO A (1994) Subperiosteal tissue expansion tor mandibular augmentation with hydroxylapatite particles an experimental study J Oral Maxillofac Surg 52 945-950, Discussion 950-951 TROTT JA (1993) A preliminary report on a strategy for treatment of male pattern baldness bilateral vertical flaps plus tissue expansion Br J Plast Surg 46 611-615 TUCKER SM, SAPP N, COLLIN R (1995) Orbital expansion of the congenitally anophthalmic socket Br J Ophthalmol 79 667-671 VAN DAMME PHA (1992) De chirurgische behandeling van litteken-alopecia op de behaarde hoofdhuid door weefselexpansie (Letter) Ned Tijdschr Gcneeskd 136 1034-1035 VAN DAMME PHA (1994) Soft-tissue expansion in cleft palate surgery (Letter) Plast Reconstr Surg 93 1307-1308 VAN DAMME PHA (1996) Long-term nasal mucosal tissue expansion (Letter) Arch Otolaryngol Head Neck Surg in press VAN DAMME PHA, FREIHOFER HPM (1996) Palatal mucopenosteal expansion as an adjunct to palatal fistula repair case report and review of the literature Cleft Palate-Craniotac J 33 255-257 VAN DAMME PHA, FREIHOFER HPM, VAN 'T HOF MA, KUIJPERS-JAGTMAN AM, MALTHA JC, SPIJKERS JM (1994) Radiologic analysis of the effects of subperiosteal palatal soft-tissue expansion in growing cats Int J Oral Maxillofac Surg 23 393-394 VAN DAMME PHA, HEIDBUCHEL KLWM, KUIJPERS JAGTMAN AM, MALTHA JC, FREIHOFER HPM (1992) Cranio-maxillo-tacial tissue expansion, experimentally based or clinically empiric7 A review ot the literature J Cramo-Max-Fac-Surg 20 61-69 VAN DER VEEN R, PULLES HJW (1992) De chirurgische behandeling van litteken-alopecia op de behaarde hoofdhuid door weerselexpansie Ned Tijdschr Geneeskd 136 627-631 VAN DER WEY LP (1995) Peripheral nerve elongation by laser Doppler flowmetry controlled expansion an experimental study in rabbits PhD Thesis, University of Nijmegen, The Netherlands VAN DER WEY LP, POLDER TW, HOOGBERGEN MM, SPAUWEN PHM (1993) A model lor monitoring nerve blood flow during expansion by laser Doppler flowmetry in the rabbit J Neurol Sc 117 79-82

185 Chapter IO

VAN DER WEY LP, POLDER TW, MERKS MHJH, STEGEMAN DF, VINGERHOETS DHM, GABREELS FESTEN AAWM, SPAUWEN PHM, GABREELS FJM (1994) Peripheral nerve elongation by laser Doppler flowmetry controlled expansion functional and neurophysiological aspects J Neurol Sc 124 149-155 VAN DER WEY LP, GABREELS-FESTEN AAWM, MERKS MHJH, POLDER TW, STEGEMAN DF, SPAUWEN PHM, GABREELS FJM (1995) Peripheral nerve elongation by laser Doppler flowmetry controlled expansion morphological aspects Acta Neuropathol 89 166 171 VAN DER WEY LP, POLDER TW, STEGEMAN DF, GABREELS-FESTEN AAWM, SPAUWEN PHM, GABREELS FJM (1996) Peripheral nerve elongation by laser Doppler flowmetry-monitored expansion an experimental basis for future application in the management of peripheral nerve defects Plast Reconstr Surg 97 568 576 VAN RAPPARD JHA (1988) Controlled tissue expansion in reconstructive surgery PhD Thesis, University of Groningen, The Netherlands VERGNES P, TAIEB A, MALEVILLE J, LARREGUE M, BONDONNY JM (1993) Repeated skin expansion for excision of congenital giant nevi in infancy and childhood Plast Reconstr Surg 91 450-455 VON HEIMBURG D, LEMPERLE G, DIPE B, KRUGER S (1994) Free transplantation ot fat autografts expanded by tissue expanders in rats Br J Plast Reconstr Surg 47 470 476 WANG SC, CHEN FL, LI JY (1991) Soft tissue expansion in the treatment of scar alopecia in preschool children Chin Med J Engl 104 164-167 WEBSTER HR, MARSHALL DR (1991) Nasal augmentation in chondrodysplasia punctata using tissue expansion Br J Plast Surg 44 384-385 WEE SS, LOGAN SE, MUSTOE TA (1992) Continuous versus intraoperative expansion in the pig model Plast Reconstr Surg 90 808-814 WILKES GH (1994) Osseointegration and the plastic surgeon a time lor reflection (Editorial) Plast Reconstr Surg 93 582-584 ZELLWEGER G, KUNZI W (1991) Tissue expanders in reconstruction of burn sequelae Ann Plast Surg 26 380-388 ZIDE BM, KARP NS (1992) Maximizing gain from rectangular tissue expanders Plast Reconstr Surg 90 500-504, Discussion 505-506

186

Acknowledgements

This study would have been impossible without the help and support of various institutions, companies, and especially numerous interested and cooperative individuals Unfortunately it is impossible to thank them all personally, by mentioning their name and their special task or part in this study In general I would like to express my sincere gratitude to all of those who participated in any way in this Ph D -project I feel honoured to mention the never-ending efforts, love and care, of my parents Anna Nijboer and Johannes Cornells Van Damme, who both passed away during the period of preparing this thesis, in 1990 and 1993 respectively I thank them wholeheartedly for everything they did. Prof Dr Η Ρ M Freihofer Dear Hans-Peter, thank you for allowing me into your training programme from 1984 till 1988, and sharing your surgical skills and knowledge with me, until now You did teach me in the way you were taught by Obwegeser and Tessier, in cranio-maxillofacial surgery, with great interest in cleft lip and palate surgery, periorbital surgery, trauma and posttraumatic reconstructive surgery Actually you invented this 'train', tissue expansion in cleft palate surgery in 1987, and you kept 'conducting' it ever since Fortunately, I know, you like to go by 'train', and I sincerely hope you liked this 'trip' Dr J С Maltha Dear Jaap, without your knowledge, experience, inventiveness and real help, there would have been no animal experiment and no thesis at all Prof Dr A M Kuijpers-Jagtman Dear Anne Marie, your dedication to the patients with cleft lip and palate disorder and your genuine interest and experience in research, was very stimulating You both put the 'train' on the 'rails' and kept it 'rolling' Thank you both very much Dr M A van't Hof Dear Martin, your critical view and mathematical wisdom are unique to me Thank you for the 'powerful' statistical analysis and grouping of all data Dr J H A ridder van Rappard Dear Julien, thank you very much for introducing me to the fascinating world of tissue expansion The 'circus' of international tissue expansion symposia in Japan and Brazil, I will never forget Your enthusiasm and passion, your pushing and our discussions, motivated me to complete this book

189 Chapter 10

Mr. S.J.A.M. Nottet. Dear Servaas, we will always remember the difficulties in digitizing the outline of a tissue expander, and the morphometric analysis of dental casts with a reflex microscope. Nevertheless, we had a fruitful cooperation and I thank you for all the work you did. Mr. Th.H.M. Arts, Mr. P.H.G. Philipsen, Mr. A. Peters, Mr. A.J. Peters. Dear Theo, Fred, Albert and Ton, thank you very much for your invaluable assistance during the animal experiment. In the beginning you were all 'afraid' of cats, eventually you started liking them and at the end of the experiment you really missed them. You and your department are to be complimented for your genuine animal care. Drs. K.L.W.M. Heidbüchel, Drs. J.M.C. Spijkers, Drs. G.F. Robinson, Drs. E.F.E. Westerling. Dear Kiki, Judith, George, and Eefje, thank you for your active participation in literature searches, tracings, digitizing and histological analysis. Ms. M.P.A.C. Helmich, Mr. R.E.M. van Rheden. Dear Pia and René, thank you for all the histological labour, the embedding, sectioning and staining of the material for histological analysis. Ms. A.M.H. Cornelissen. Dear Anne thank you for the myofibroblast staining. Mr. J.A.D. Laverman. Dear Jan, I thank you and your co-workers for preparing the 900 dental casts. It has been a tremendous amount of work, within your already busy schedule. Dr. P.CM. de Wilde. Dear Peter, thank you for the histological analysis of the clinical case of tissue expansion. Mr. R. Draijer, Mr. H.G. van Eist, Mr. R. van der Vaart (Inamed В.V., Breda). Dear Rob, Harry and Rob, I would like to thank you personally for your moral and material support during this project. Mr. L.J.H. Hofman. Dear Louis, thank you for your repeatedly performed literature surveys and your advice for building-up a personal 'eference-retrieval system. Your co-workers Drs. Roos M.J. Schattenberg and Hans Koning are also thanked. Mr. J.A.C, van de Noort. Dear Hans, thank you for helping me with the retrieval of the original articles. Prof. Dr. H. Boersma, is thanked for his advices in cephalometric analysis.

190 Acknowledgements

Prof. Dr. W.J.L. van der Gulden, is acknowledged for his part in the choice of the cat as experimental animal. Dr. A. De Mey, Drs. T.F.J.M.C. Specken, are thanked for their willingness to share their knowledge on the clinical application of palatal tissue expansion in cleft palate cases. Drs. A.R.M. Wittkampf. Dear Albert, thank you for sharing your experiences and showing me your results of tissue expansion for alveolar ridge augmentation. Mr. J.A.H. Meeuwissen, Mr. H.A. ten Dam, Ms A. Naus. Dear Jan, Henny, and Annie, thank you for the photographs and slides of the animal and clinical cases. Ms M.F.A. Reeb, Mr. CA. de Bruin, Mr. F.J.W. Schraven, Mr. H. van Wayenburg. Dear Mary, Cor, Frans, Hans, thank you for all the slides and photographs, that I needed for my presentations, publications and this book. My collegues Drs. I. Bruaset, Dr. R.A.C.A. Voorsmit, Drs. W.A. Borstlap, Drs. J.M. Kwakman and Drs. M.A.W. Merkx. Dear Ingolv, Ralph, Wilfred, Joke, and Thijs, thank you for your patience and your willingness to take over part of my tasks and duties, and for allowing me time to complete this project. The residents, assistants, nursing and administrative personnel, and secretaries at the Department of Oral and Maxillofacial Surgery, University Hospital Nijmegen, St. Radboud, I thank you all for your compassion and I apologize for sometimes being agitated and hasty, during this project. Ms. P. van Veelen, Ms. M.A. van Gastel-Goossens, Ms. J. Baas. Dear Petra, Ria and Jeannette, thank you for your secretarial support and computer (WP) assistance during the preparation of the manuscripts and the tables. Dr. E.N. Robertson, Dr. A.E. Loewenthal, Dr. A. Whitford. Dear Eric, Anton and Anne, thank you for our corrections of the English text, and the linguistic and editorial advice. Ms. J.M.J. Verhoeven. Dear Jacqueline, thank you for the final editing of the whole text and all the figures and tables of this Ph.D.-thesis. Together with Jaap С. Maltha you gave form to this book. Drs. B.J.C. Rijnders, Dr. С. Jansen. Dear Bernard and Kees, thank you

191 Acknowledgements for your friendship and moral support before, during and hopefully after the defence of this thesis. My family, Dearest Akke, Maxim, Rebec and Dam, thank you for your confidence and patience. I owe you a lot of time, attention and love.

192

Curriculum vitae

The author Phihppus Anne Van Damme was born on June 4, 1953, in Yerseke, The Netherlands. He went to high-school in Goes, Lyceum for Zeeland, from 1965 to 1971. He studied dentistry at the University of Utrecht, from 1971, till completion with the Dental Degree, in 1977. During this period he was assistant/instructor in Histology, Preventive Orthodontics, and Anatomy He fulfilled his military duty in the Royal Dutch Navy, as lieutenant- dentist, from 1977 to 1978 He started medical school at the University of Utrecht in 1977, and completed his Medical Degree, in 1983. Throughout this period, he practised general dentistry Fiom 1983 to 1984, he was resident General Surgery and House Officer in the Joannes de Deo Foundation Hospital, in Utrecht (M.M. Plomp-Van Harmelcn, MD., J M. Wakelkamp, M.D., and the late В J.R Reijmers, MD) From 1984 until 1988, he was resident in training at the Department of Oral and Maxillofacial Surgery, University Hospital Nijmegen St. Radboud (Head Prof. H.P.M Freihofer, M.D., D M.D., Ph.D.). In 1988, he qualified and registered as an Oral and Maxillofacial Surgeon Since then he has been a Consultant/University Lecturer at the Department of Oral and Maxillofacial Surgery, University Hospital Nijmegen. Since 1991, he has been a member of the International Scientific Committee of the International Tissue Expansion Symposia in Japan (1991) and Brazil (1993) Fields of special interest are tissue expansion, cleft palate surgery, traumatology and posttraumatic periorbital reconstructive surgery, orthognathic surgery, craniomandibular disorders and facial pain, oncology and pathology of the mucous membranes. Various papers on these items have been presented and published

195

Publications in relation to soft tissue expansion and/or cleft palate surgery

VAN DAMME PHA, HEIDBUCHEL KLWM (1989) Quantitative analysis of the thickness of human skin and subcutaneous tissue following controlled expansion with a silicone implant (Letter) Plast Reconstr Surg 4 705 VAN DAMME PHA (1990) Subperiosteal palatal tissue expansion an animal experiment in domestic cats Abstr 2nd Symp IAMFST, Luxemburg VAN DAMME PHA (1990) Subperiosteal palatal tissue expansion an animal experiment in domestic cats Poster 10th Congr EACMFS, Brussels VAN DAMME PHA (1990) Subperiosteal palatal tissue expansion an animal experiment in domestic cats Abstr 10th Congr EACMFS, Brussels, ρ 48 FREIHOFER HPM, VAN DAMME PHA, KUIJPERS JAGTMAN AM (1990) The rationale for early osteotomy and stabilization of the premaxilla in CLP Abstr 10th Congr EACMFS, Brussels, ρ 277 VAN DAMME PHA (1990) Subperiosteal palatal tissue expansion an animal experiment in domestic cats Abstr 3e Symp Exp Onderz Heelk Spec (SEOHS), Leiden, ρ 5 VAN DAMME PHA (1990) Subperiosteal palatal tissue expansion an animal experiment in domestic cats Neth J Surg 42 164-165 VAN DAMME PHA (1990) Subperiostal palatinale weefselexpansie een dierexperiment bij katten Abstr 34e Najaarsverg Ned Ver Mondz & Kaak chir , Ameland, ρ 63 VAN DAMME PHA, FREIHOFER HPM, KUIJPERS-JAGTMAN AM, MALTHA JC, VAN 'T HOF MA (1991) Subperiosteal palatal tissue expansion an animal experiment in domestic cats Abstr 4th Eur Cranio-facial Congr, Noordwijkerhout, Abstr 41, ρ 75 VAN DAMME PHA, FREIHOFER HPM, KUIJPERS-JAGTMAN AM, MALTHA JC, VAN Τ HOF MA (1991) Soft-tissue expansion in cleft palate surgery an animal study Abstr 3rd Int Tissue Expansion Symposium, Sapporo, Japan, Abstr 14, ρ 26 VAN DAMME PHA, FREIHOFER HPM, KUIJPERS-JAGTMAN AM, MALTHA JC, VAN 'T HOF MA (1991) Soft-tissue expansion in cleft palate surgery an animal study In Tissue Expansion Symposium, OCITES Ohura T, Sugihara Τ (eds), Sando, Japan, Chapt Head and Neck, pp 46 48

197 Publications

VAN DAMME PHA, HEIDBUCHEL KLWM, KUIJPERS-JAGTMAN AM, MALTHA JC, FREIHOFER HPM (1991) Tissue expansion in aesthetic cranio maxillo-facial surgery Experimental rationalism or clinical empiricism' In Tissue Expansion Symposium, OCITES Ohura T, Sugihara Τ (eds), Sando, Japan, Chapt New application in aesthetic surgery, pp 154-160 FREIHOFER HPM, VAN DAMME PHA, KUIJPERS-JAGTMAN AM (1991) Early secondary osteotomy stabilization or the premaxilla in bilateral clefts J Cranio-Max-Fac Surg 19 2-7 FREIHOFER HPM, BORSTLAP WA, KUIJPERS-JAGTMAN AM, VOORSMIT RACA, VAN DAMME PHA, BORSTLAP-ENGELS VMF, HEIDBUCHEL KLWM (1992) Bone grafting a cleft When'' What'' With'' J Cranio-Max-Fac Surg 20 (Suppl 1) 68 VAN DAMME PHA, HEIDBUCHEL KLWM, KUIJPERS JAGTMAN AM, MALTHA JC, FREIHOFER HPM (1992) Cranio-maxillo-fdual tissue expansion, experimentally based or clinically empiric9 A review of the literature J Cranio-Max-Fac Surg 20 61-69 VAN DAMME PHA (1992) De chirurgische behandeling van litteken-alopecia op de behaarde hoofdhuid door weefselexpansie (Letter) Ned Tijdschr Geneeskd 136 1034-1035 VAN DAMME PHA, FREIHOFER HPM, VAN Τ HOF MA, KUIJPERS- JAGTMAN AM, MALTHA JC, SPIJKERS JMC (1992) Radiologische analyse van de effecten van subperiostale palatinale tissue expansion bij katten 36e Najaarsverg Ned Ver Mondz & Kaakchir , Deventer, Abstr 30, ρ 57 VAN DAMME PHA, FREIHOFER HPM, VAN 'T HOF MA, KUIJPERS- JAGTMAN AM, MALTHA JC, SPIJKERS JMC (1992) Radiologische analyse van de effecten van subperiostal palatinale tissue expansion bij katten Abstr Ned Ver Schisis Cranio-Fac Afwijkingen Leeuwarden FREIHOFER HPM, BORSTLAP WA, KUIJPERS-JAGTMAN AM, VOORSMIT RACA, VAN DAMME PHA, HEIDBUCHEL KLWM, BORSTLAP-ENGELS VMF (1993) Timing and transplant materials for closure of alveolar clefts a clinical comparison of 296 cases J Cranio-Max-Fac Surg 21 143-148 FREIHOFER HPM, VAN DAMME PHA, KUIJPERS-JAGTMAN AM, HEIDBUCHEL KLWM (1993) The osteotomy-stabilization of the premaxilla 7th Int Congr Cleft Palate Rel Craniotac Anom Broadbeach, Australia Abstr 320, ρ 185

198 Publications

WALJI S, VAN DAMME PHA (1993). Aseptische necrose na Le Fort I segmentale osteotomie. Abstr 37e Najaarsverg. Ned. Ver. Mondz. & Kaakchir. Groningen, p. 46. VAN DAMME PHA (1994). Soft-tissue expansion in cleft palate surgery. (Letter) Plast Reconstr Surg 93' 1307. VAN DAMME PHA, FREIHOFER HPM, VAN 'T HOF AM, KUIJPERS- JAGTMAN AM, MALTHA JC, SPIJKERS JMC (1994) Radiological analysis of the etfects of subperiosteal palatal soft-tissue expansion in growing cats Int J Oral Maxillofac Surg 21. 393-394. VAN DAMME PHA, FREIHOFER HPM (1996) Palatal mucopenosteal expansion as an adjunct to palatal fistula repair: case report and review of the literature. Cleft Palate-Cramofac J 33 255-257. VAN DAMME PHA, MALTHA JC, FREIHOFER HPM, KUIJPERS-JAGTMAN AM, VAN 'T HOF MA (1996) Three-dimensional morphomeiric analysis ot the effects of subperiosteal palatal soft tissue expansion in growing cats. Int J Oial Maxillofac Surg. In press. VAN DAMME PHA (1996) Long-term nasal mucosal tissue expansion use (Letter). Arch Otolaryngol Head Neck Surg In press VAN DAMME PHA, MALTHA JC, KUIJPERS-JAGTMAN AM, FREIHOFER HPM, VAN 'T HOF MA (1996). Two-dimensional cephalometnc analysis of the elfects of subperiosteal palatal soft tissue expansion in growing cats. Plast Reconstr Surg. In press. VAN DAMME PHA, FREIHOFER HPM, MALTHA JC, KUIJPERS-JAGTMAN AM, VAN 'T HOF MA (1996). Experimental palatal mucoperiosteal tissue expansion. Abstr. E A.C M.F.S. Zurich. J Cranio Max-Fac Surg 24 (Suppl 1): 119-120. VAN DAMME PHA, FREIHOFER HPM, MALTHA JC, KUIJPERS-JAGTMAN AM (1996). Histological analysis ot the effects of palatal mucoperiosteal soft tissue expansion in growing cats. Int J Oral Maxillofac Surg. Submitted.

199

STELLINGEN behorend bij het academisch proefschrift

SUBPERIOSTEAL PALATAL SOFT TISSUE EXPANSION An experimental study in growing domestic cats

Nijmegen, 6 december 1996

Philip A. Van Damme 1 Fistulas in the hard palate are much more difficult to close than they seem (P Randall in R M Goldwyn's The unfavorable result in plastic surgery D R Millard Jr , Cleft Craft The evolution of its surgery III Alveolar and palatal deformities, 1980, ρ 815)

2 As anywhere else in the body, if local tissue is not available, distant tissue must be brought in to fill the defect (D R Millard Jr , Cleft Craft The evolution of its surgery HI Alveolar and palatal deformities, 1980, ρ 833)

3 Subperiostal palatinale weefselexpansie is in het dierexperiment weldegelijk mogelijk (Dit proefschrift)

4 Nu het mogelijk blijkt, met behulp van weefselexpansie, de hoeveelheid en de beschikbaarheid van loco-regionaal weefsel voor defect-sluitings technieken te vergroten, is stelling 2 niet primair (Dn proefschrift)

5 De ontwikkeling en klinische toepassing van tissue expansion in de cranio- maxillofaciale chirurgie heeft een voornamelijk empirische basis (Dit proefschrift)

6 Dé oorzaak voor de maxillaire groeivertraging en uiteindelijke groeiachterstand bij de geopereerde schisis patiënten, blijft vooralsnog, ondanks uitgebreid dier­ experimenteel onderzoek, een controversieel item (Dit proefschrift)

7 Subperiostal palatinale weefsel expansie interfereert met zowel voor- achterwaartse, verticale als zijdelingse groei van het naso- en dento-maxillaire complex (Dit proefschrift)

8 De verdediging van de op vermoedelijke winst berekende weefselexpansie op St Nicolaas (6 december) is bij uitstek speculatief

9 De niet, met-compleet of pervers doorgebroken, niet-functionele en/of caneuze verstandskiezen dienen voor het stadium van volledige afvorming van de wortel te worden verwijderd

10 Indien het spreekwoordelijk Zeeuwse "Luctor et Emergo" van toepassing is op verstandskiezen dient de indicatie tot verwijdering daarvan niet "zuinig" gesteld te worden 11. "Promoveren in Porsche" is wezenlijk iets anders dan het schrijven van een proefschrift over het produkt 911 van Dr Ing h с Ferdinand Porsche

12 De "gnathiater" is die specialist, waaraan dringend behoefte is binnen het multidisciplinaire Cranio-Mandibulaire-Dysfunctie team

13 Als je toch dood gaat, is de manier waarop, eigenlijk belangrijker dan het, tevoren bekende, feit op zich, ook voor de nabestaanden

14 Het is later dan je denkt (J С Van Damme, 1925-1993)

15. De aandacht voor de correspondentie naar aanleiding van "ZHZMU"- verwijsstroken en de zorg waarmee de verwijzing en de verwezen patient omkleed is, zijn navenant

16 In de reconstructieve mond-, kaak- en aangezichtschirurgie is "micropolitiek" gewichtiger dan microchirurgie

17 "Mondheelkunde (en chirurgische prothetiek)" is een gedateerde, obsolete benaming voor de actuele mond-, kaak- en aangezichtschirurgie

18 Omdat de grootmoeder van Roodkapje ongeschonden uit de wolf tevoorschijn kwam, valt aan te nemen dat hij tenminste een door "de wolf" aangetast gebit had

19 Het besturen van een Porsche vergt zelfdiscipline, nauwgezetheid en durf Deze automobielen zouden derhalve van overheidswege aan (medisch) specialisten beschikbaar gesteld moeten worden om de genoemde kwaliteiten te verwerven en te behouden

20 "Dubbel gekwalificeerd en gepromoveerd" het verwijderen van een verstandskies blijft vervelend

21. Denkt aleer gij doende zijt en doende denkt dan nog (Guido Gezelle, 1830-1899), in combinatie met de lijfspreuk van Bernhard R К (Von) Langenbeck (1810-1887) "Nunquam retrorsum" ofwel "niemals zurückgewandt", vormt een goede uit­ gangspositie voor onderzoek en (schisis) chirurgie

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