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Unicystic Ameloblastoma: A Diagnostic Dilemma and Its Management Using Free Fibula Graft: An Unusual Case Report
Aniket Jain1, Satyajit Dandagi2, Amit Sangle3, Viquar Ahmed4, Akram Khan5
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ABSTRACT: A 20 year old patient reported with a swelling in the left posterior mandibular region since 4 months. On clinical examination, there was a hard, non-tender mass, measuring 8.5 cm by 5 cm arising from the left side of the mandible, involving the ramus,
angle and body upto the leftt lower 1st premolar tooth. Radiographic picture and fluid aspiration of the pathology with protein analysis of 4.1 gm/dl, was suggestive of a keratinizing cyst or tumor. Hemimandibulectomy was performed with safe margins and an microvascular free fibula graft was placed for mandibular reconstruction. The final diagnosis after histopathological examination was given as Unicystic
Final Year P.G Student Professor 3 Professor 4 Senior Lecturer 5 First year P.G. Student Department and Institution Dept of Oral and Maxillofacial Surgery, M A Rangoonwala College of Dental Sciences and Research Centre, Pune. Article Info: Received: April 15, 2012; Review Completed: May, 14, 2012; Accepted: June 13, 2012 Published Online: August, 2012 (www. nacd. in) © NAD, 2012 - All rights reserved Email for correspondence: email@example.com
Ameloblastoma. Facial Symmetry is well maintained with no recurrence after a systematic follow up of 18 months. Key words: Unicystic Ameloblastoma, Microvascular, Free Fibula Flap
INTRODUCTION: The most common tumour of odontogenic origin is ameloblastoma, which develops from epithelial cellular elements and dental tissues in their various phases of development. It is a slow-growing, persistent, and locally aggressive neoplasm of epithelial origin.1 Unicystic ameloblastoma is second important clinical type of ameloblastoma and accounts for 10-15% of all intraosseous ameloblastomas.2 They have been reported to occur in second and third decades of life as against its solid counterpart which occurs in fourth decade of life. Unicystic ameloblastoma most commonly occurs in posterior mandible followed by parasymphysis region, anterior maxilla and posterior maxilla.3 It is a classic example of a true neoplasm of enamel organ type tissue that lacks the potential to undergo differentiation, and hence has aptly been defined as “unicentric, nonfunctional, intermittent in growth, anatomically benign and clinically persistent” by Robinson.There are three forms of ameloblastomas, namely multicystic, peripheral, and unicystic tumors . Multicystic ameloblastoma is the most common variety and represents 86% of cases. Peripheral tumors are odontogenic
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Indian J Dent Adv 2012; 4(2) 843
chewing or articulating. lining part of the cystic cavity with or without luminal and/or mural tumor growth.3 and 1. they have been found in patients up to the age of 40. (Fig 10 and 11) Discussion: Robinson and Martinez were the first persons to describe UA in 1977. as for a solid or multicystic ameloblastoma. vigorous curettage of the bone should be avoided as it may implant foci of ameloblastoma more deeply Indian J Dent Adv 2012. al. No neck nodes were palpable. lesions occur in younger patients. It refers to those cystic lesions that show clinical.2 can be treated conservatively (careful enucleation). and protein analysis revealed 4. et. Final diagnosis of unicystic ameloblastoma was confirmed with the help of histopathologic examination of the excised specimen (Fig. suddenly developed a small swelling in lower left mandibular region which gradually increased in size to attain the present state. Facial symmetry was well maintained postoperatively. luminal ameloblastomas.2. Systemic examination was normal. tumors. 2).Unicystic Ameloblastoma Aniket Jain. Fine needle aspiration cytology of the lesion as well as incisional biopsy was performed but both were not conclusive. non-tender mass. involving the ramus. 7 and 8). with the histological characteristics of intraosseous ameloblastoma that occur solely in the soft tissues covering the tooth-bearing parts of the jaws. there was a hard. rarely.3 showing intramural growths require treated radical resection. and ameloblastomas arising in dentigerous cysts . but on histologic examination shows typical ameloblastomatous epithelium.7. Diffuse overgrowth seen in lower left posterior alveolar and vestibular region with normal overlying mucosa (Fig.13 Following enucleation. 9). difficulty in opening the mouth. but on histologic examination shows a typical ameloblastomatous epithelium lining part of the cyst cavity. whereas subgroups 1. (Fig 3 and 4) An aspirate of the fluid was obtained.2 The UAs diagnosed as subgroups 1 and 1. radiographic or gross features of jaw cyst. UA should be differentiated from odontogenic cysts and also should be recognized for the reason that the former has a higher rate of recurrence than the latter12. One year systematic follow-up did not reveal any recurrence as well as showed excellent acceptance of graft in the region mimicking a lower jaw. accounting for about 6% of ameloblastomas.10. the unusual aspect is that its not associated with an imapacted tooth. A miniplate with locking screws was used to secure the osteotomized fibula and the mandible (Fig.11 As seen in the present case.6 We present a case of a large unicystic mandibular ameloblastoma in a young male. angle and body upto the leftt lower 1st premolar tooth. Facial asymmetry due to swelling is the regular presenting feature which infrequently relates with pain. 4(2) 844 . Reconstruction was done using a microvascular free fibula graft.1 gm/dl. with or without luminal and/or mural tumor growth hence. Patient was apparently alright 4 months back. Unicystic tumors include those that have been variously referred to as mural ameloblastomas. which showed large cystic lesion in the left side of mandible extending from lower left second premolar upto the ramus involving the condyle as well. Unilocular ameloblastoma (UA) is a rare type of ameloblastoma. Radiographic examination revealed a unilocular appearance and was suggestive of a cyst or tumour. which was suggestive of a keratinizing cyst or tumor. Exposure of the lesion was done via extended risdon’s incision followed by hemimandibulectomy along with dearticuation of the TM joint on the left side (Fig.8 It is most commonly seen in individuals who are 16 to 20 years of age. Ackermann et al have provided a histological subgrouping of the Unicystic Ameloblastoma as shown in (Table 1) and a diagrammatic representation of the same shown in (Fig.(Fig 1) There was no associated pain. It refers to those cystic lesions that show clinical. 12). Occasionally. Patient was taken up for surgery under general anesthesia. 5 and 6).5 cm by 5 cm arising from the left side of the mandible. Case Report: A 20-year-old female presented to Department of Oral and Maxillofacial Surgery with the Chief complaint of painless swelling in left side of the face since 4 months which was gradually increasing in size.9 About 90% of the lesions are located in the mandible and between 50 to 80% of these cases are associated with an impacted tooth. On physical examination. Shaping of the resected fibula was done according to the preoperative template. radiographic or gross features of a mandibular cyst. measuring 8. An Orthopantomogram (OPG) was done.
et al. Chan Chiu-Po. Tsai Chi-Ying.3 have a high risk for recurrence. Wu Y.Co. Yu-Fen. Dentomaxillofac Radiol 2004. and in 1989 Hidalgo first used free fibula flap in mandibular reconstruction. Zhang F. Miller GD.122:371-374. Contemporary Oral and Maxillofacial Pathology (2nd ed) USA: Mosby 2004. Int J Oral Maxillofac Surg 2006. Jeng Seng-Feng: Segmental mandibulectomy and immediate free fibula osteoseptocutaneous flap reconstruction with endosteal implants: An ideal treatment method for mandibular ameloblastoma. Page DG. iliac crest. radius or ribs. Arimura K. 16.16 Recurrence rates are also related to the histologic subtypes of UA. Conclusion: The diagnosis of unicystic ameloblastoma was based on clinical. Reichart PA.14. Jagdeep S. 7. Shear M. Reichart PA.5% for enucleation alone. Lin Hsiu-Na. 4(1): 1116 Indian J Dent Adv 2012. Fu-Chan. scapula. requiring more aggressive surgical procedures.17(1):178-182. Reichart PA. 2. Philipsen HP. Unicystic ameloblastoma. 2:1. Shen. Philipsen HP. Unicystic ameloblastoma. Yan W. Ustad H Tanveer. Ltd 2004. Ramesh Rakesh S. 4. Head and Neck Oncology 2010. al. hence the Pathologist should examine the tissue sections carefully for better prognosis of the treatment outcome. 12.15 Recurrence is always an aspect to look after resection specially in the case of unicystic ameloblastoma. Sposto MR. Caplan J. Pizer ME. Oral Oncol 1998. Chen ZW. Li T. 77-86. 113(1):80-87. 13. Taylor GI. Odontogenic tumors and allied lesions London: Quintessence Pub. Navarro CM. Philipsen HP. Sapp JP. Co. The unicystic ameloblastoma: A clinicopathologic study of 57 cases. with those invading the fibrous wall having a rate of 35. Co. Kitano M. and 18% by marsupialization followed by enucleation. Thirteen-year follow-up of large recurrent unicystic ameloblastoma of the mandible in a 15-year-old boy. Massucato EM.7%. Lau SL. Wei. Principi SM. Quintessence Pub. Annals and Essence of Dentistry 2010. 35:681-690 17. Unicystic ameloblastoma: a review of 193 cases from the literature. Yu G: Clinicopathological features of unicystic ameloblastoma with special reference to its recurrence. Arathy Manohar. Unicystic ameloblastoma is a tumor with a strong propensity for recurrence. Subgroups 1. Ltd 2004. Normally if the tumour is small (< 5cm) the defect can be repaired with a free bone graft. Chemical cauterization with Carnoy’s solution is also advocated for subgroups 1 and 1. J Oral Pathol 1988. 9. J Craniofac Surg 2006. Manjunath Suraj. References: 1. 17: 541-546 3. Srinivasan H.18 After demonstrating that osteotomies can be performed in vascularised fibula grafts without compromising the viability of the bone segment.21-23. 15. et. Anand V. into bone. Zhonghua Kou Qiang Yi Xue Za Zhi 2002. Ham FJ. 2(4):75-77. Unicystic ameloblastoma of the mandible: A case report. Ackermann GL. 10. vascularised free fibula flap became the state of art reconstruction method after mandible ablation. Lau et al reported recurrence rates of 3.Ltd. but others only 6. Li TJ. 6. radiological and histopathologic features. Ltd 2004. Lineaweaver W: Immediate mandibular reconstruction with microsurgical fibula flap transfer following wide resection for ameloblastoma. J Oral Maxillofac Surg 2002.2. 37:210212. 16% for enucleation followed by Carnoy’s solution application. Plast Reconstr Surg 1975. The study and clinical application of the osteocutaneous flap of fibula.33:60-62. Philipsen HP. 5. Since this patient has a large bony defect. 34(5):317-325.6% for resection. In: Odontogenic tumors and allied lesions.7%.3 and 1. Co. 8. London: Quintessence Pub. 2004. Yu S. 4(2) 845 . Sugihara K: Recurrence of unicystic ameloblastoma: A case report and review of the literature. This is because the cystic wall in these cases has islands of ameloblastoma tumor cells and there may be penetration into the surrounding cancellous bone.Unicystic Ameloblastoma Aniket Jain. 14. Samman N: Recurrence related to treatment modalities of unicystic ameloblastoma: A systematic review. 11. The free vascularized bone graft: A clinical extension of microvascular techniques. Philipsen HP. 77-86. Plast Reconstr Surg 2004. However. 15 Vascularised fibula graft was first described by Taylor in 1975. 77-86. 18. Arch Pathol Lab Med 1998. 60:211-215. a free fibula osteoseptocutaneous flap was chosen to address both the bony and soft tissue defect. Microsurgery 1983. Reichart PA: Classification of odontogenic tumors and allied lesions. Reichart PA: Unicystic ameloblastoma. Odontogenic tumors and allied lesions.17 Later Chen and Yen incorporated an overlying skin paddle for composite reconstruction of the bone and soft tissue defect. Yang-Ming Chang. Gerzenshtein J. Maxillary unicystic ameloblastoma.2. Altini M. 30. Unicystic ameloblatoma of the mandible-an unusual case report and review of literature. Svirsky JA. the tumour is often larger and a large defect reconstruction is challenging and may require a microsurgical flap either from fibula. 55(5): 533-544. Chana. Odontogenic tumors and allied lesions Quintessence Pub.
Unicystic Ameloblastoma Aniket Jain.2. Figure 1: Facial Profile showing facial asymmetry on the left side Figure 5: Exposure of the lesion via extended risdon’s incision Figure 9: Histopathologic picture of the resected specimen showing typical ameloblastomatous epithelium lining part of the cystic cavity Figure 2: Diffuse overgrowth seen in lower left posterior alveolar and vestibular region Figure 6: Resected specimen after hemimandibulectomy Figure 10: 18 months post operative radiographic picture showing graft in place. 3) Intramural Table 1: Showing Akerman’s grouping of UA and their interpretation Subgroup 1 1. Intraluminal and Intramural UA Luminal and Intramural UA Indian J Dent Adv 2012. al. mimicking the lower jaw Figure 3: OPG showing extent of lesion involving Figure 7: Retrieval and shaping of fibula graft with body.2 1. et. angle and ramus upto the condyle the help of osteotomies and mini plates and screws Figure 11: 18 months post operative intraoral view showing well adaptation of graft Figure 4: Lateral view of body of mandible showing the extent of lesion Figure 8: Post operative radiograph showing free fibula Figure 12: Showing Ackerman’s grouping of UA as graft secured at the recipient site with plates and screws follows: 1) Luminal. 4(2) 846 .3 1. 2) Intraluminal.3 Interpretation Luminal UA Luminal and Intraluminal UA Luminal.
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