Odontogenic Myxoma of Maxilla: Conservative Or Radical Surgery? Jalal Hamama1*, Lahcen Khalfi2, Kamal Fiqhi1 and Karim EL Khatib1

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ISSN: 2643-3907 Hamama et al. Res Rep Oral Maxillofac Surg 2018, 2:004 Volume 2 | Issue 1 Open Access Research Reports in Oral and Maxillofacial Surgery

Case Report Odontogenic Myxoma of Maxilla: Conservative or Radical Surgery? Jalal Hamama1*, Lahcen Khalfi2, Kamal Fiqhi1 and Karim EL Khatib1

1Department of Plastic and Oral Surgery, Mohammed V Military Hospital, Mohammed V University, Rabat, Morocco Check for updates 2Department of Plastic and Oral Surgery, Mohammed V Military Hospital, Sidi Mohamed Ben Abdellah University, Fes, Morocco

*Corresponding author: Jalal Hamama, Professor, Department of Plastic and Oral Surgery, Mohammed V Military Hospital, 10100 Rabat, Morocco, E-mail: [email protected]

mor, incidence rate is approximately 0.07 new cases per Abstract million people per year [7,8]. Odontogenic myxoma (OM) is a benign, locally invasive and aggressive, non-metastasizing neoplasm of the jaw bones. All reports generally agree that the tumour frequent- This article presents a rare case of odontogenic myxoma ly presents in the second or third decades of life [8-10]. occurring in the maxilla of a 42-year-old female patient with review of the literature emphasizing a huge controversy re- Most common clinical characteristics of odontogenic garding the surgical management. myxoma are swelling of the jaws with only few patients Keywords complaining of pain, paraesthesia and ulceration, 28% Odontogenic tumor, Myxoma, Maxilla, Long-term, Follow-up for pain and 56% for swelling [11]. Several studies indicating that there is a higher rate Introduction of tumour incidence in the mandible than the maxilla Odontogenic myxoma (OM) is a benign, locally inva- [12-15], and almost occurring in the premolar and mo- sive and aggressive, non-metastasizing neoplasm of the lar region of both jaws and rarely crosses the midline jaw bones. [12,16,17]. Myxomas of the head and neck can be identified in Cortical expansion and perforation are common find- two forms (I) Facial bone derived, which had been sub- ings; however, maxillary myxomas often extend into the divided in the past into true osteogenic myxoma and sinus [18]. The adequate management of Odontogenic odontogenic myxoma and (II) Soft tissue myxoma de- myxoma (OM) is a challenging problem against the high rived from perioral soft tissue, parotid gland, ear and risk of recurrence. larynx [1-3]. The aim of this paper is to report a case of odon- There has been a great deal of controversy regard- togenic myxoma that affected the left maxilla focusing ing the origin of myxomatous tumors. Virchow, in 1863, especially on surgical management. coined the term myxoma for a group of tumors that had Case Report histologic resemblance to the mucinous substance of the umbilical chord [4,5]. A 42-year-old female was referred to our department with a complaint of a growing swelling in the area of Pathogenesis is disputed. Two hypotheses have the canin and premolar molar in his left maxilla that had been suggested: Tumoral development in residual con- been noticed a few weeks before. Upon admission, she junctive tissue stem cells, or myxomatous degeneration noticed a swelling measuring about the size of a peanut of the fibrous stroma [6]. in the gingiva of upper premolar tooth. That lesion had Odontogenic myxoma of the jaws is a rare benign tu- been excised by a dentist and the histopathologist con-

Citation: Hamama J, Khalfi L, Fiqhi K, EL Khatib K (2018) Odontogenic Myxoma of Maxilla: Conserva- tive or Radical Surgery?. Res Rep Oral Maxillofac Surg 2:004. Received: December 02, 2017; Accepted: February 21, 2018; Published: February 23, 2018 Copyright: © 2018 Hamama J, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

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Figure 3: Gross examination showed grayish white, glistening smooth, gelatinous mass.

Figure 1: Intraoral image showing an overgrowth on maxillary left anterior teeth region extending from distal aspect of 23 to mesial aspect of 26.

Figure 4: Microphotograph shows mixed area of fibrous tis- Figure 2: Panoramic radiographs showing a single large sue and inconspicuous strands of odontogenic epithelium expansile radiolucent lesion without any trabeculations in in a myxoid stroma; magnification: x100. the area of bony destruction.

dunculated, bony hard in consistency, and non-pitting clude to epulis. Although, the tumour had recurred two on application of pressure with rolled out margins. months later and gradually increased to its present size. The sensory examination of the infraorbital nerve The patient’s medical history and review of systems was normal. were unremarkable. The rest of the patient’s clinical head and neck exam- On extraoral examination, a solitary swelling was vis- ination and general examination were non-contributory. ible on left side of face which was extending anteriopos- teriorly from nasio-labial fold to corner of mouth. The Panoramic radiograph revealed a single large expan- swelling was round to oval in shape and approximate- sile radiolucent lesion extending from the left first pre- ly 2 cm × 3 cm in dimension with ill-defined borders. molar to second molar and occupied the left maxillary Overlying skin appears to be normal with no secondary sinus without any trabeculations in the area of bony de- changes. On palpation, the swelling was non-tender, struction (Figure 2). firm to hard in consistency, non-compressible, non-re- Under general anesthesia the tumour was exposed ducible, and non-pulsatile, non-fluctuant and was fixed through left intraoral upper vestibular incision from 21 to underlying structure. There was no cervical mass or to 27 regions, enucleation of the tumour was performed lymphadenopathy was found. with curettage of the surrounding bone through. On intraoral examination, an overgrowth was visible Macroscopically, the surgical specimen measured on maxillary left anterior teeth region extending from dis- approximately 3 × 3.9 × 2.5 cm, on gross examination tal aspect of 23 to mesial aspect of 26 measuring approx- showed grayish white, glistening smooth, gelatinous imately 3 cm × 4 cm in diameter round to oval in shape. mass (Figure 3). With well defined borders and lobulated surface Histopathological examination of excised specimen there by obliterating the right buccal vestibule. The mu- showed tumor mass without encapsulation. It showed cosa overlying the area of the lesion was the same co- spindle and stellate shaped cells in loose, abundantly lour and texture as the surrounding mucosa (Figure 1). myxoid connective tissue stroma, closely resembling On palpation, the overgrowth was nontender, pe- the mesenchymal portion of a developing tooth.

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Some areas showed moderately dense collagen fi- bital floor and the skull base, resulting in a non-resect- bers. Few strands of inactive odontogenic epithelium able situation, especially in cases of recurrence [20]. For were seen within the connective tissue stroma. Over- the reason, wide resection with 10 to 15 mm margins is all histological appearance of the lesion revealed more generally recommended [28,29]. amount of myxoid stroma in a less fibrous and acellular Yoav LEISER took into consideration that the treat- background (Figure 4). ment protocol for OMs resembles that of ameloblas- Outcome and follow up: Healing was uneventful and toma, solid type, especially when locally aggressive follow-up after 4 months showed no recurrence of the behaviour is encountered which includes, cortical bone lesion. There have been no clinical or radiological signs perforation and major tumour expansion and infil- of recurrence over 02 years follow-up. tration in a short period of time (weeks to months). A radical primary resection of the tumour with maximal Discussion preservation of surrounding anatomic structures is nec- The treatment of odontogenic myxoma is primarily essary (healthy anatomical borders of 0.5-1 cm beyond surgical, it varies from simple enucleation and curet- the tumours radiographic borders) [30]. tage to more extensive radical surgery, regarding max- It’s recommend that further, radical surgery is war- illa it could be partial and total maxillectomies [17,19- ranted after recurrence of conservatively-treated odon- 21]. Conservative treatments have several advantages togenic myxoma, a view that believe some authors in over more radical treatments, such as segmental or the literature [8,20,31-33]. block resection, with reconstruction surgery. Conser- vative treatments are substantially less invasive, can be Reconstruction can begin immediately following the achieved by means of an intraoral surgical approach, surgical procedure or delayed until an adequate disease preserve function and aesthetics especially in the max- free period has past. In our experience, small bony de- illa because of the proximity of vital structures and the fects (smaller than 5 cm) can be reconstructed using more complex anatomy, in addition offered a shorter buccal fat pad (maxilla) [34], or using corticocancellous hospitalization time [22]. iliac crest bone graft [35]. Larger defects (more than 5 cm) usually require primary prosthetic reconstruction Several investigators have recommended that tumor (obturator) [36] followed by a final obturator [10]. Due size should determine whether a radical or more con- to the high recurrence rates of these lesions, delayed servative surgical approach should be adopted [9,20]. reconstruction is the treatment of choice, especially Boffano, et al. suggested that conservative treat- in the more locally aggressive tumours. Prosthetic re- ment by enucleation and curettage is recommended construction in maxillary lesions is highly advisable due when the diameter of an odontogenic myxoma is less to low postoperative morbidity and almost immediate than 3 cm, whereas a segmental resection with imme- phonetic and masticatory function. Furthermore pros- diate reconstruction is preferred in patients with larger thetic reconstruction by an obturator allows direct vi- tumors [23]. sualization of the primary site for recurrence detection [25]. Recently, Zanetti, et al. strongly suggested conservative treatment should involve enucleation of the lesion A follow-up period is clearly also necessary. It has with a wide curettage of normal tissue or a generous been recommended that patients should be followed amount of apparently uninvolved surrounding tissue, or closely for at least the first 2 years after surgery, which even peripheral osteotomy, as this has the advantage of represents the period during which the neoplasm is preserving vital structures and maintaining oral function most likely to recur [9,13]. [24]. Rocha, et al. suggested that 5 years of surveillance is Recurrence is considered to be directly related to the needed to confirm that the lesion has healed success- type of therapy, the rate after simple enucleation and ful excision, and periodical clinical and radiographic fol- curettage has been reported to be as high as 25% [3,19]. low-up should be maintained indefinitely [22]. Some authors report that the risk of recurrence is This review of literature shows that all datas are greater as the myxoma is not encapsulated and its myx- based on cases report, in the other hand each conser- omatous tissue infiltrates the surrounding bony tissue vative and radical surgery consist of advantages and in- without causing immediate destruction than to the cell convenients. Thus, to determine the most recommend- proliferative index which was relatively low [22,25,26]. ed surgery method is difficult. In future, it is judicious to carry out a metanalysis study which only would offer Batsakis accused incomplete removal the reason of in future a precise analysis of recurrence and allow a recurrence than the intrinsic biological behavior of the clearly guideline. tumor [27]. The aggressive nature of OM is well documented in Conclusion the literature, it can invade critical structures like the or- Management varies depending on the location and

Hamama et al. Res Rep Oral Maxillofac Surg 2018, 2:004 • Page 3 of 5 • ISSN: 2643-3907 size of the tumour, the age of the patient and individual 15. Keszler A, Dominguez FV, Giannuzio G (1995) Myxoma in experience. They range from minimally invasive excisional childhood: An analysis of 10 cases. J Oral Maxillofac Surg 53: 518-521. biopsies to an en bloc resection of a multitude of relevant structures. 16. Farman AG, Nortje CJ, Grotepass FW, Farman FJ, Van Zyl JA (1977) Myxofibroma of the jaws. Br J Oral Surg 15: 3-18. Conflicts of Interest 17. Li TJ, Sun LS, Luo HY (2006) Odontogenic myxoma: A clin- The authors declare that they have no conflicts of in- icopathologic study of 25 Cases. Arch Pathol Lab Med 130: 1799-1806. terest concerning this article. 18. Brannon RB (2004) Central odontogenic fibroma, myxoma Acknowledgements (odontogenic myxoma, fibromyxoma), and central odontogenic granular cell tumor. Oral Maxillofac Surg Clin North The authors are grateful for a cytopathologist qa- Am 16: 359-374. mouss wadie who provided a histopatholgical analysis 19. Leiser Y, Abu-el-Naaj I, Peled M (2009) Odontogenic myxo- and images. ma - A case series and review of the surgical management. References J Craniomaxillofac Surg 37: 206-209. 20. Kansy K, Juergens P, Krol Z, Paulussen M, Baumhoer D, 1. Gowda BK, Sinhasan SP, Manjula CG, George R (2011) et al. (2012) Odontogenic myxoma: Diagnostic and thera- Odontogenic myxoma of maxilla: A case report. Physicians peutic challenges in paediatric and adult patients - a case Academy 5: 29-32. series and review of the literature. J Craniomaxillofac Surg 2. Regezi JA, Kerr DA, Courtney RM (1978) Odontogenic tu- 40: 271-276. mors: Analysis of 706 cases. J Oral Surg 36: 771-778. 21. Kawase-Koga Y, Saijo H, Hoshi K, Takato T, Mori Y (2014) 3. Singaraju S, Wanjari SP, Parwani RN (2010) Odontogenic Surgical management of odontogenic myxoma: A case re- myxoma of the maxilla: A report of a rare case and review port and review of the literature. BMC Res Notes 7: 214. of the literature. J Oral Maxillofac Pathol 14: 19-23. 22. Rocha AC, Gaujac C, Ceccheti MM, Amato-Filho G, Mach- 4. Landa LE, Hedrick MH, Nepomuceno-Perez MC, Sotere- ado GG (2009) Treatment of recurrent mandibular myxoma anos GC (2002) Recurrent myxoma of the zygoma: A case by curettage and cryotherapy after thirty years. Clinics 64: report. J Oral Maxillofac Surg 60: 704-708. 149-152. 5. Jankowski M, Yeung R, Lee KM, Li TK (2002) Odontogen- 23. Boffano P, Gallesio C, Barreca A, Bianchi FA, Garzino-De- ic myxomain the Hong Kong Chinese: Clinico-radiological mo P, et al. (2011) Surgical treatment of odontogenic myx- presentation and systemic review. Dentomaxillofac Radiol oma. J Craniofac Surg 22: 982-987. 31: 71-83. 24. Zanetti LS, de Carvalho BM, Garcia IR Jr, de Barros LA, 6. Carvalho de Melo AU, de Farias Martorelli SB, Cavalcanti Dos Santos PL, et al. (2011) Conservative treatment of PH, Gueiros LA, Martorelli Fde O (2008) Maxillary odonto- odontogenic myxoma. J Craniofac Surg 22: 1939-1941. genic myxoma involving the maxillary sinus: Case report. Braz J Otorhinolaryngol 74: 472-475. 25. Martins C, Carvalho YR, do Carmo MAV (2001) Argyrophil- ic nucleolar organizer regions (AgNORs) in odontogenic 7. Manne RK, Kumar VS, Venkata Sarath P, Anumula L, myxoma (OM) and ameloblastic fibroma (AF). J Oral Pathol Mundlapudi S, et al. (2012) Odontogenic myxoma of the Med 30: 489-493. mandible. Case Rep Dent 2012: 214704. 26. Batsakis JG (1987) Myxomas of soft tissues and the facial 8. Simon EN, Merkx MA, Vuhahula E, Ngassapa D, Stoelin- skeleton. Ann Otol Rhinol Laryngol 96: 618-619. ga PJ (2004) Odontogenic myxoma: A clinicopathological study of 33 cases. Int J Oral Maxillofac Surg 33: 333-337. 27. Jihène Kourda-Boujemâa, Faten Farah-Klibi, Soumaya Rammeh, Ali Adouani, Rachida Zermani, et al. (2010) Le 9. Lo Muzio L, Nocini P, Favia G, Procaccini M, Mignogna MD myxome odontogénique: étude de quatre cas et revue de (1996) Odontogenic myxoma of the jaws: A clinical, radio- la littérature. Ann Pathol 30: 168-175. logic, immunohistochemical, and ultrastructural study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 82: 426-433. 28. R Zaininea, H Mizounib, A El Korbia, N Beltaief, S Sahtou- ta, et al. (2014) Maxillary bone myxoma. Eur Ann Otorhino- 10. Noffke CE, Raubenheimer EJ, Chabikuli NJ, Bouckaert MM laryngol Head Neck Dis 131: 257-259. (2007) Odontogenic myxoma: Review of the literature and report of 30 cases from South Africa. Oral Surg Oral Med 29. Slootweg PJ, Wittkampf AR (1986) Myxoma of the jaws: An Oral Pathol Oral Radiol Endod 104: 101-109. analysis of 15 cases. J Maxillofac Surg 14: 46-52. 11. MacDonald-Jankowski DS, Yeung R, Lee KM, Li TK (2002) 30. I Gassab, A Hamroun, K Harrathi, F Ben Mahmoud, S Odontogenic myxomas in the Hong Kong Chinese: Clini- Kadhi, et al. (2007) Myxome du maxillaire : A propos d’un co-radiological presentation and systematic review. Dento- cas. J Tun ORL 18: 44-45. maxillofac Radiol 31: 71-83. 31. Allphin AL, Manigilia AJ, Gregor RT, Sawyer R (1993) Myx- 12. Kaffe I, Naor H, Buchner A (1997) Clinical and radiological omas of the mandible and maxilla. Ear Nose Throat J 72: features of odontogenic myxoma of the jaws. Dentomaxillo- 280-284. fac Radiol 26: 299-303. 32. Schmidseder R, Groddeck A, Schunemann H (1978) Diag- 13. Barros RE, Dominguez FV, Cabrini RL (1969) Myxoma of nostic and therapeutic problems of myxomas (myxofibro- the jaws. Oral Surg Oral Med Oral Pathol 27: 225-236. mas) of the jaws. J Maxillofac Surg 6: 281-286. 14. Lu Y, Xuan M, Takata T, Wang C, He Z, et al. (1998) Odon- 33. Ogutcen-Toller M, Sener I, Kasap V, Cakir-Ozkan N (2006) togenic tumors. A demographic study of 759 cases in a Maxillary myxoma: Surgical treatment and reconstruction Chinese population. Oral Surg Oral Med Oral Pathol Oral with buccal fat pad flap: A case report. J Contemp Dent Radiol Endod 86: 707-714. Pract 7: 107-116.

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34. Demas PN, Sotereanos GC (1988) Closure of alveolar gol Head Neck Surg 117: S73-S76. clefts with corticocancellous block grafts and marrow: A ret- 36. Deron PB, Nikolovski N, den Hollander JC, Spoelstra HA, rospective study. J Oral Maxillofac Surg 46: 682-687. Knegt PP (1996) Myxoma of the maxilla: A case with ex- 35. Chiodo AA, Strumas N, Gilbert RW, Birt BD (1997) Man- tremely aggressive biologic behavior. Head Neck 18: 459- agement of odontogenic myxoma of the maxilla. Otolaryn- 464.

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