Cyst Volume Changes Measured with a 3D Reconstruction After

Clinics and Practice 2019; volume 9:1132

Cyst volume changes measured with a 3D reconstruction after Correspondence: Antoine Nicolas Berberi, Introduction Department of Oral and Maxillofacial decompression of a mandibular Surgery, Faculty of Dental Medicine, dentigerous cyst with an Odontogenic cysts, in almost all classi- Lebanese University, P.O. Box: 5208-116 fications, are divided into two major Beirut, Lebanon. impacted third molar groups: developmental and inflammatory. E-mail: [email protected] - [email protected] Some cysts arise from the proliferation of Tel.: +961.3731173 - Fax: +961.4533060. Faouzi Riachi,1 epithelial residues left behind during odon- Key words: Dentigerous cyst; Mandibular; 1 2 togenesis; such cysts are generally Carla Maria Khairallah, Nabil Ghosn, Decompression; Three-dimension; Cone 3 Antoine Nicolas Berberi described as being developmental in Beam Computed Tomography. origin.1,2 While other types arise from 1Department of Oral Surgery, Faculty of epithelial hyperplasia of the residues due to Contributions: CMK and NG: conception and Dental Medicine, St-Joseph University; adjacent foci of inflammation as a conse- 2 design, acquisition of data, analysis and inter- Department of Oral and Maxillofacial quence of the two major dental diseases, pretation of data; FR and ANB: drafting the Radiology, Faculty of Dental Medicine, decays and periodontal disease; such cysts article, critical revision of the article; ANB: 3 St-Joseph University; Department of are generally described as being inflamma- final approval of the version to be published. Oral and Maxillofacial Surgery, Faculty tory in origin.3,4 Conflict of interest: the authors declare no of Dental Medicine, Lebanese The dentigerous cyst is the second most potential conflict of interest. University, Beirut, Lebanon common odontogenic cyst5,6 and represent 7 22.3 % of odontogenic cysts. Dentigerous Funding: none. cysts have the potential to bone resorption and can expand into the surrounding tissue Received for publication: 1 February 2019. Abstract and tooth roots causing malocclusion or Revision received: 18 February 2019. facial asymmetry8 or mandibular paresthe- Accepted for publication: 18 February 2019. The aim of this article is to describe a sia.9 The diagnosis is not based only on the only large mandibular cyst treated with decom- This work is licensed under a Creative radiographic evaluation alone, a histopatho- Commons Attribution NonCommercial 4.0 pression followed by surgical enucleation. logic exam must be conducted to confirm License (CC BY-NC 4.0). Furthermore, we described the utility of it.10 The keratocysts and unilocular cyst volume measurements by using a 3D ameloblastomas are commonly misdiag-use©Copyright F. Riachi et al., 2019 reconstruction on Cone Beam Computed nosed with dentigerous cysts because con- Licensee PAGEPress, Italy Tomography (CBCT). The dentigerous cyst ventional ameloblastoma show cystic Clinics and Practice 2019; 9:1132 is the most common cyst type of epithelial degeneration with no biologic differences. doi:10.4081/cp.2019.1132 origin, arising from remnants of odonto- The unilocular ameloblastoma represents an genic epithelium, asymptomatic and associ- ameloblastoma, which presents as a cyst ated with the crown of an unerupted or par- and has a lower recurrence rate following with a 12 months clinical and radiological tially or completely impacted tooth. conservative removal. The histopathologic follow-up. However, after a long duration and exten- detection is recognized by luminal, intralu- sion of the cyst volume it may provoke sig- minal, and mural types depending on nificant bone resorption, cortical expansion, whether only the cyst lining is affected or tooth displacement and the vitality of neigh- not.11 These cysts vary in size; if allowed to Case Report boring teeth may be affected. The regular enlarge, they may, over time, cause signifi- treatment of this lesion is enucleation and cant bony expansion and destruction of A 21-year-old male reported to the extraction of the involved tooth. large portions of the bone.12 Plus, they may department of oral surgery at the Saint Marsupialization and decompression are spread to adjacent anatomical structures Joseph University in Beirut, with the main proposed when the volume of the cyst is such as the maxillary sinus or nasal cavity complaint of swelling in his lower left jaw. well developed to release the cysticNon-commercial pres- due to the increasing intra-cystic pressure. 12 The swelling was growing for the past 3 sure and allow the bone cavity to progres- However, this cyst is asymptomatic in weeks with no history of pain, numbing or sively decrease in volume with the gradual majority9,13 and discovered on dental radi- any other complaints. Patient had no history apposition of bone. This report presents a ographs usually appearing as a well-defined of systemic or local contraindications, large dentigerous cyst related to impacted radiolucency associated with the crown of including uncontrolled diabetes, bruxism, 14 mandibular third molar of a 21-year-old an unerupted tooth. Marsupialization of smoking, or uncontrolled periodontal dis- male patient. The cyst was treated success- odontogenic cystic was described by ease. fully by decompression and later by surgical Partsch in 1892, it is a technique where a enucleation with surgical extraction of the large window is made in cystic wall and Clinical and radiological examination related molar. In conclusion, the combina- then sutured to the oral mucosa.15 tion of decompression and surgical Marsupialization has the advantage of The face was asymmetric with a small approach showed on the three-dimensional reducing the cyst volume.15 For those cases swelling on the left side of the mandible CBCT investigation a significant correla- when no eruption occurs, a period of 3 to 4 angle. The overlying skin was normal and tion between the treatment and volume months after marsupialization is suggested light swelling was palpable on the sub- reduction of the cyst. The clinical case as critical time for deciding whether to mandibular lymph nodes. The vitality test described allows us to observe bone forma- extract or conserve.15 The aim of this paper was negative for the left first and second tion after decompression and surgical enu- is to present a case report of the treatment of molars, positive on the first and second cleation was performed with less risk on a large odontogenic cyst in the retro-molar mandibular left premolars and the test of vital anatomic elements. region of the lower jaw by decompression, paresthesia was negative. Intra-orally, there

[page 12] [Clinics and Practice 2019; 9:1132] Case Report was a sensitive swelling posterior to the Treatment plan Decompression molars with a hard bony on palpation. The After the histological findings, treat- Under local anesthesia as described color of the overlying mucosa was normal ment modalities, enucleation, marsupializa- before, a triangular distal wedge incision (Figure 1A). Panoramic radiograph showed tion and decompression were discussed was performed, with the base of the triangle a radiolucent lesion, unilocular, in contact with the patient and he accepted our plan to on the distal surface of the second molar. with the impacted third molar with the do a decompression followed by enucle- Copious irrigation with physiological saline crown of the tooth and the roots of the two ation and surgical extraction of the impact- serum was realized to evacuate all the intra- other molars and the second premolar com- ed molar after the endodontic treatment of cystic fluids. The opening was adjusted to pletely involved in the lesion. Resorption the non-vital first and second left molars. suit the confectioned acrylic stent. The appear on the roots of the second molar. The The patient was informed to have a strict patient was prescribed Amoxicillin + inferior alveolar nerve is repressed to the soft food diet, to avoid any complication Clavulanic acid 1g, BID for 7 days. basal bone of the lower jaw (Figure 1B). such as mandibular fracture. Analgesic medication (400 mg ibupro- Axial and coronal cuts of the Cone Beam Computed Tomography (CBCT) showed a radiolucency, ball-shaped, measuring 30.29×43.19×37.26 mm (Figure 2A, B). The differential diagnosis was a dentigerous cyst or odontogenic keratocyst tumor or a unicystic ameloblastoma.

Biopsy The importance of the biopsy was discussed with the patient and he gave his approval. Preoperatively, the patient rinsed with 0.12% chlorhexidine gluconate oral only rinse (PerioGard; Colgate-Palmolive, Figure 1. (A) Initial photograph of the left side of the mandible showing posterior Salford, United Kingdom) 5 minutes before swelling and a normal color of the overlying mucosa; (B) Initial panoramic radiograph the surgery. Local analgesia was achieved reconstructed from the cone beam computed tomography (CBCT). by inferior alveolar nerve block 2% arti- use caine with 1:100,000 adrenaline (3M ESPE, Seefeld, Germany). Buccal mucosa was also anesthetized by infiltration close to the second molar. A sample of the intra-cystic fluid was aspired by a fine needle syringe. The aspiration revealed a clear, light-yellow fluid with shiny cholesterol crystals. Muco-periosteal flap was reflected by sulcular incision from the second premolar to the second molar with a distal buccal releasing incision, and a part of the cyst membrane was dissected and fixed in 10% formaldehyde (Merck, Darmstadt, Germany). The flap was adjusted and hermetically closed by means of single suturesNon-commercial (Vicryl® 4/0, Johnson & Johnson, medical limited, UK).

Histopathological examination The cystic border was composed of fibrous tissue, lined with a non-keratinized stratified squamous epithelium. The liquid contained a hematologic smear with polymorphous leucocytes as well as foamy or pigmented macrophages. The results came as a dentigerous cyst with no sign of malig- nity (Figure 2C, D). From a practical standpoint, dentiger- Figure 2. CBCT slices (A) axial (B) coronal to change with a new one with measures. (C, ous cysts are differentiated from keratocysts D) Histopathology slides. (C) Cystic membrane: cystic wall composed of fibrous tissue, by the absence of specific features: paraker- lined with a non-keratinized stratified squamous epithelium; (D) intra-cystic liquid: a hematologic smear with polymorphous leucocytes as well as foamy or pigmented atosis, palisaded basal layer and corrugated macrophages. surface.3

[Clinics and Practice 2019; 9:1132] [page 13] Case Report

fen; Abbott Healthcare Products Limited, bone resection was performed, on the buc- the enucleation of the remnants of the cyst Vanwall, United Kingdom) was also pre- cal and distal side of the molar with a cylin- were slowly dissected and removed. scribed, and the patient was advised to rinse dric bur. The fragmentation of the tooth was Abundant irrigation was realized to remove his mouth daily with 0.12% chlorhexidine a must, to avoid a lot of force on the lower all the debris and suturing was performed gluconate oral rinse (PerioGard; Colgate- jaw. First, the crown of the tooth was cut with a Vicryl® 4/0 (Johnson & Johnson, Palmolive, Salford, United Kingdom) dur- and removed. Second, the roots were sepa- medical limited, UK). Post-operative med- ing healing. He was instructed to leave the rated and extracted one by one with angu- ication was the same as for the decompres- stent in place and visit us twice a week to lated root elevators. After tooth removal, sion procedure (Figure 5). Four months remove the stent and disinfect it with a 0.12% chlorhexidine solution and thor- oughly clean the cavity with saline serum. Through the months, with the bone formation and mesial progression of the third molar the obturator was lifted by the pressure, so every few weeks the obturator part that goes into the cavity was shortened (Figure 3). After three months, a panoramic radiograph was taken to check the evolution of the lesion. The lesion had retracted but there was not enough bone formation to be able to remove the third molar without damage. Thus, the treatment was resumed for anoth- Figure 3. Decompression procedure: (A) acrylic drain, (B) opening of the cyst distally to er two months (Figure 4A). the second lower left molar, (C) drain stabilized inonly the mouth without any interference Another panoramic radiograph was with the occlusion. taken 5 months post-decompression. It showed mesial displacement of the third molar for about 9 mm, plus a significant use bone formation on the basal bone of the jaw which became sufficiently thick to extract the third molar and enucleate the remnants of the cyst, with minimal risk of jaw fracture (Figure 4B).

Enucleation and surgical extraction The surgical procedure was done under loco-regional analgesia as described for the decompression with the same technique and Figure 4. (A) Panoramic radiograph 3 months post-decompression; (B) Panoramic radi- product. A mucoperiosteal flap was elevat- ograph 5 months post-decompression. ed, the third molar was visible. A minimal Non-commercial

Figure 5. Enucleation surgery: (A) tooth fragmented after osteotomy, (B) cyst enucleation, (C) empty cystic cavity after tooth extraction and enucleation.

[page 14] [Clinics and Practice 2019; 9:1132] Case Report post-operatively the panoramic radiograph multi slice editing tool to remove any selec- no-apical direction). showed complete bone formation in the tion that did not correspond to the cystic The measured linear dimensions (in cystic cavity (Figure 6). cavity or to select and add a missing part of mm) were: the lesion. A 3D object was created, and the i) X: 29.12 Y: 40.58 Z: 33.23 pre-opera- Volumetric and linear measure- volume of the cyst was calculated automat- tively; 3 ments ically by the software in mm in the two ii) X: 22.12 Y: 31.51 Z: 27.32 after 4 datasets. months of decompression. The patient underwent two CBCT scans The measured volumes (Figure 7A, B) The percent reduction in maximal linear (initial and 4 months after the beginning of were: dimensions were: the treatment) using the Newtom VGI scan- i) 16,775 cc pre-operatively; i) X: 24.04% Y: 22.38% Z: 17.79%. ner with a 15×15 cm field of view and ii) 6,937 cc after 4 months of decompres- In order to assess the speed of shrinkage 0.3mm voxel size. Scan data were saved in sion. and the reduction in volume, four formulas DICOM format and imported to the 3D models of the cyst were also pre- ® Simplant Pro 15 (Materialize Dental, pared in the Blue Sky Plan® 4.2.5 (Blue Sky Leuven, Belgium) software for further Bio, LLC, Grayslake, IL, USA) software analysis. A semi-automatic segmentation and exported as stereolithography (STL) 16 technique was used to isolate the affected models. A superimposition of the two area from the bone, teeth and soft tissue. datasets was realized in the same software This was done by creating a mask in the (Figure 7C, D, E). Using the Autodesk® ® Simplant software. A mask is a selection of Meshmixer® software, the maximum linear voxels within a specified range of gray val- dimensions were measured automatically in ues. The minimum threshold was set to the the 3 planes in mm for both datasets (Figure lowest (-1024: Empty spaces) while the 7 F, G): maximum threshold was adjusted manually i) X: Coronal plane (maximal width/buc- in a way that the created mask followed the cal-lingual direction); edge of the surrounding bone structures. ii) Y: Axial plane (maximal length/antero- Figureonly 6. Panoramic radiograph 6 months The mask was edited in 3D and then manu- posterior direction); post-extraction, reconstructed from the CBCT. ally checked on every single slice using the iii) Z: Sagittal plane (maximal height/coro- use

Non-commercial

Figure 7. 3D reconstruction of the cyst area showing the inferior alveolar nerve, the second and third molars (A) pre-op, (B) 116 days per-op. Superimposition of 2 CBCTs pre-op (orange) and 116 days per-op (green): (C) top, (D) bottom and (E) left views. Maximal linear dimensions in 3D: (F) Pre-op: X: 29.12 Y: 40.58 Z: 33.23, (G) 4 months per-op: X: 22.12 Y: 31.5 Z: 27.32.

[Clinics and Practice 2019; 9:1132] [page 15] Case Report were calculated as: sion, which reduce the pressure inside the the initial size of the cystic lesion.38 i) Absolute speed of shrinkage (ml/day) = cystic cavity and permits new bone to In this report, the evolution in the bone (initial detected volume – final volume) reconstruct the bone defect. Adjacent apposition was assessed through measure- / duration of decompression = 16,775 - anatomical structures such as a tooth, the ment of bone density and the cyst volume 6,937 / 116 = 0.085 ml/day. sinus, or the inferior alveolar nerve can be after decompression. Significant increases ii) Relative speed of shrinkage (/day) = protected from damage.32 in bone apposition and a remarkable (initial detected volume – final volume) The procedure decreased the volume of decrease in the cyst cavity were found 3 ×100 / initial detected volume × dura- the cyst before enucleation, and extensive months after decompression, in agreement tion of decompression = 9.838 ×100/ surgery can be avoided, and is considered with Bodner et al.35 However, if the cyst 16,775 ×116 days = 0.51/day. the best treatment for large dentigerous volume is larger than 80 mL and the cortex iii) Reduction in volume (ml) = initial cyst.33,34 The enucleation after marsupializa- is very thin or even absent before decom- detected volume – final volume = 9,838 tion will be determined by the changes pression, secondary enucleation should be ml. within the cyst cavity. Only when sufficient delayed for another 2 to 3 months. In our iv) Relative reduction in volume (%) = bone has been created, the enucleation can case report, the total healing time was 12 reduction in volume × 100 / initial be safely achieved. However, some studies months. The patient was well motivated, detected volume = 58,64%. regarding the assessment of bone apposition compliant and had no complaint about the after marsupialization have rarely been treatment modality. reported.35 The determination of the proximity of Discussion the cyst to vital adjacent anatomic structures is extremely important in enabling the Conclusions The most common odontogenic cysts surgeon to decide between marsupializa- The decompression can be a valuable are apical and lateral cysts, followed by tion, decompression and enucleation.32 treatment for dentigerous cysts despite the dentigerous cysts and odontogenic kerato- Traditionally, 4 to 6 months, after mar- need of a second surgery and the prolonged cysts. The relative incidence of dentigerous supialization or decompression, has been treatment. From a clinical point of view, the cysts according to several studies and relat- considered as a period in which sufficient only case treated in this paper allows us to con- ed to different populations are 22.3% in bone formation to perform enucleation. 17 firm that decompression of large cyst is a France by Meninguaud et al., 33% in However, Bodner et al.35 recommended, 18 very important step before enucleation and Mexico by Mosqueda et al. and 35.5% by that cysts should be enucleated at 3 months 19 the use of 3D CBCT reconstruction was Ledesma-Montes et al., 24% in Canada by after marsupialization, based on CT scansuse 20 very useful for evaluation the decrease of Daley et al., 27% in Japan by Nakamura et with multiplanar reconstruction on 23 21 the intra-cyst volume and to observe the al., 16.6% in South Africa by Shear et patients with marsupialized. The disadvan- 22 23 bone apposition. al., 19% in Nigeria by Arotiba et al., tages of marsupialization are poor oral 24 21.3% in Germany by Kreidler et al., hygiene in the opening area, healing time 25 24.8% in Jordan by Bataineh et al. prolonged, cystic remnants left behind and Moreover, dentigerous cysts are the patient’s cooperation. The cooperation of References most common odontogenic cysts in children the patient is required for a long period of 26 and adolescents. The relative incidence of time.36,37 Pathologic epithelium left in situ 1. Browne RM. Investigative pathology of dentigerous cysts in pediatric populations is may lead to epithelial proliferation and cys- the odontogenic cysts. Boca Raton: 26 76.2% in China by Li et al., 59.7% in UK tic recurrence.37 CRC Press; 1991. 27 by Jones et al. 68.2% in Chile by One other disadvantage regarding the 2. Mehra P. Benign cysts and tumors of the 28 Ochsenius et al., 89.7% in Brazil by De marsupialization in the lower jaw, is the dif- jaw bones. In: Stucker FJ, de Souza C, 29 Souza et al. and 66% in Turkey by ficulty of cleaning and drainage, whilst in Kenyon GS, et al., eds. Rhinology and 30 Tekkesin et al. the upper jaw the drainage is gravity facial plastic surgery. Berlin, Adults have high rates of inflammatory dependent.37 Heidelberg: Springer Berlin cysts, whereas in children developmental In our case, the stent was well fit and Heidelberg; 2009, pp 395-429. 26 Non-commercial cysts are more common. The most fre- stable around the two molars, to avoid food 3. Manor E, Kachko L, Puterman MB, et quent unerupted teeth involved with impaction under it. Without being too bulky al. Cystic lesions of the jaws- a clinico- dentigerous cysts are respectively the causing patient’s discomfort and without pathological study of 322 cases and mandibular third molars, the maxillary per- interfering with the occlusion. review of the literature. Int J Med Sci manent canines, the mandibular premolars Comparing our results to a previous 2012;9:20-6. and the maxillary third molars and the first study by Song et al.,38 compared reduction 4. Koseoglu BG, Atalay B, Erdem MA. common symptom is progressive swelling, in volume in cystic diseases by 3-dimen- Odontogenic cysts: a clinical study of followed occasionally by pain if infected.31 sional CT analysis, the absolute speed of 90 cases. J Oral Sci 2004;46:253-7. The radiographic feature is an unilocu- shrinkage was 0,048 ml/day, reduction in 5.Gondim JO, Moreira Neto JJS, lar radiolucent lesion involved with the volume 0,3ml, relative speed of shrinkage Nogueira RLM, Giro EMA. crown of an impacted tooth. The lesion is 26,71/day and relative reduction in volume Conservative management of a well-defined by a radio-opaque margin. 48,5%. So, we had a relatively faster shrink- dentigerous cyst secondary to primary These cysts tend to resorb the roots of adja- age of the dentigerous cyst, even though our tooth trauma. Dental Traumatol 2008; cent teeth due to their dental follicle ori- patient was 21 years old and this same study 24:676-9. gin.31 concluded that age correlated negatively 6. Dinkar AD, Dawasaz AA, Shenoy S. Large dentigerous cysts are usually with the speed of shrinkage in dentigerous Dentigerous cyst associated with multi- treated by marsupialization or decompres- cysts and the speed of shrinkage is related to ple mesiodens: a case report. J Indian

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