Background: Ameloblastoma is an odontogenic tumor that originates from epithelial cells. Despite its benign classification, it exhibits local aggressiveness and the potential for unlimited growth. The 2022 World Health Organization (WHO) classification identifies five distinct subtypes of ameloblastoma, each displaying varying biological behaviors. Plexiform ameloblastoma is a subtype of conventional ameloblastoma that is characterized by a unique histopathological appearance. Unlike the typical follicular or acanthomatous patterns, plexiform ameloblastomas exhibit a network of interconnected strands or cords of odontogenic epithelium that resemble a “plexus” or network. This paper aims to present two clinical cases of conventional plexiform ameloblastoma, provide a review of the current literature, and detail the clinical, radiological, and histopathological features of this condition, as well as appropriate therapeutic approaches. Case presentation: This article presents two cases of plexiform ameloblastoma, a prevalent subtype of ameloblastoma, with one arising in the maxilla and the other in the mandible. The cases are analyzed in the context of existing literature to shed light on the unique features and clinical implications of this subtype. Conclusion: Plexiform ameloblastoma is a rare and benign odontogenic tumor that may reach gigantic proportions. Thus, ensuring an accurate diagnosis is crucial to minimizing the risk of local recurrences and obtaining an effective treatment.
Cite this paper
Oumayma, E. Y. , Malik, B. , Hicham, B. and Bouchra, T. (2025). Plexiform Ameloblastoma: Report of 2 Cases and Review of Literature. Open Access Library Journal, 12, e2857. doi: http://dx.doi.org/10.4236/oalib.1112857.
Manikkam, S., Masthan, K.M.K., Anitha, N. and Krupaa, J. (2015) Ameloblastoma. Journal of Pharmacy and Bioallied Sciences, 7, S167-S170. https://doi.org/10.4103/0975-7406.155891
Kreppel, M. and Zöller, J. (2018) Ameloblastoma—Clinical, Radiological, and Therapeutic Findings. Oral Diseases, 24, 63-66. https://doi.org/10.1111/odi.12702
Hendra, F.N., Natsir Kalla, D.S., Van Cann, E.M., de Vet, H.C.W., Helder, M.N. and Forouzanfar, T. (2019) Radical vs Conservative Treatment of Intraosseous Ameloblastoma: Systematic Review and Meta‐Analysis. Oral Diseases, 25, 1683-1696. https://doi.org/10.1111/odi.13014
Vered, M. and Wright, J.M. (2022) Update from the 5th Edition of the World Health Organization Classification of Head and Neck Tumors: Odontogenic and Maxillofacial Bone Tumours. Head and Neck Pathology, 16, 63-75. https://doi.org/10.1007/s12105-021-01404-7
Shi, H.A., Ng, C.W.B., Kwa, C.T. and Sim, Q.X.C. (2021) Ameloblastoma: A Succinct Review of the Classification, Genetic Understanding and Novel Molecular Targeted Therapies. The Surgeon, 19, 238-243. https://doi.org/10.1016/j.surge.2020.06.009
Kashyap, B., Reddy, P. and Desai, R. (2012) Plexiform Ame-loblastoma Mimicking a Periapical Lesion: A Diagnostic Dilemma. Journal of Conservative Dentistry, 15, 84-86. https://doi.org/10.4103/0972-0707.92614
Chauhan, D.S. and Guruprasad, Y. (2011) Plexiform Ameloblastoma of the Mandible. Journal of Clinical Imaging Science, 1, Article No. 61. https://doi.org/10.4103/2156-7514.91134
Castro-Silva, I.I., Israel, M.S., Lima, G.S. and de Queiroz Chaves Lourenço, S. (2011) Difficulties in the Diagnosis of Plexiform Ameloblastoma. Oral and Maxillo-facial Surgery, 16, 115-118. https://doi.org/10.1007/s10006-011-0265-x
Nwoga, M. (2022) Recurrent Tumors of Ameloblasto-ma: Clinicopathologic Features and Diagnostic Outcome. Nigerian Journal of Clinical Practice, 25, 1771-1777. https://doi.org/10.4103/njcp.njcp_82_22
Bwambale, P., Yahaya, J.J., Owor, G. and Wabinga, H. (2022) Histopathological Pat-terns and Biological Characteristics of Ameloblastoma: A Retrospective Cross-Sectional Study. Journal of Taibah University Medical Sciences, 17, 96-104. https://doi.org/10.1016/j.jtumed.2021.09.007
Sreelalita, C. and Sunil Babu, K. (2012) Plexiform Ameloblastoma. Inter-national Journal of Clinical Pediatric Dentistry, 5, 78-83. https://doi.org/10.5005/jp-journals-10005-1140
Evangelou, Z., Zarachi, A., Dumollard, J.M., Peoc’h, M., Komnos, I., Kastanioudakis, I., et al. (2020) Maxillary Ameloblastoma: A Review with Clinical, Histological and Prognostic Data of a Rare Tumor. In Vivo, 34, 2249-2258. https://doi.org/10.21873/invivo.12035
Neagu, D., Escuder-de la Torre, O., Vazquez-Mahia, I., Carral-Roura, N., Rubin-Roger, G., Penedo-Vazquez, á., et al. (2019) Surgical Manage-ment of Ameloblastoma. Review of Literature. Journal of Clinical and Experimental Dentistry, 11, 70-75. https://doi.org/10.4317/jced.55452
Maia, E.C. and Sandrini, F.A.L. (2017) Management Techniques of Ameloblastoma: A Liter-ature Review. RGO-Revista Gaúcha de Odontologia, 65, 62-69. https://doi.org/10.1590/1981-863720170001000093070
Goh, Y.C., Siriwardena, B.S.M.S. and Tilakaratne, W.M. (2021) Association of Clinicopathological Factors and Treatment Modalities in the Recurrence of Ameloblastoma: Analysis of 624 Cases. Journal of Oral Pathology & Medicine, 50, 927-936. https://doi.org/10.1111/jop.13228
Shukla, D., Bhola, N.D., Kshirsagar, K., Agrawal, P. and Wanjari, M.B. (2022) Calcium Sul-fate Dihydrate with Titanium Scaffold in Conservative Management of a Multicystic Ameloblastoma: A Case Report. Cureus, 14, e27050. https://doi.org/10.7759/cureus.27050
Laino, L., Cicciù, M., Russo, D. and Cervino, G. (2020) Surgical Strategies for Multicystic Ameloblastoma. Journal of Craniofacial Surgery, 31, e116-e119. https://doi.org/10.1097/scs.0000000000005903
Singh, M., Shah, A., Bhattacharya, A., Raman, R., Ranganatha, N. and Prakash, P. (2014) Treatment Algorithm for Ameloblastoma. Case Reports in Dentistry, 2014, Article ID: 121032. https://doi.org/10.1155/2014/121032
Peacock, Z.S. (2019) Adjunctive Strategies for Benign Maxillofacial Pathology. Oral and Maxillofacial Surgery Clinics of North America, 31, 569-578. https://doi.org/10.1016/j.coms.2019.07.002
Yoithapprabhunath, T.R., Srichinthu, K.K., Gupta, D., Singh, D., Pasupuleti, S. and Nirmal, R.M. (2022) Effectiveness of Molecular-Targeted Chemother-apy in Ameloblastomas. Indian Journal of Dental Research, 33, 323-331. https://doi.org/10.4103/ijdr.ijdr_456_22
Jhamb, T. and Kramer, J.M. (2014) Molecular Concepts in the Pathogenesis of Ameloblastoma: Implications for Therapeutics. Experimental and Mo-lecular Pathology, 97, 345-353. https://doi.org/10.1016/j.yexmp.2014.09.001
Qiao, X., Shi, J., Liu, J., Liu, J., Guo, Y. and Zhong, M. (2021) Recurrence Rates of Intraosseous Ameloblastoma Cases with Conservative or Aggressive Treatment: A Systematic Review and Meta-Analysis. Frontiers in Oncology, 11, Article ID: 647200. https://doi.org/10.3389/fonc.2021.647200
Abdel-Aziz, A. and Amin, M.M. (2012) EGFR, CD10 and Proliferation Marker Ki67 Expression in Ameloblastoma: Possible Role in Local Recurrence. Diagnostic Pathology, 7, Article No. 14. https://doi.org/10.1186/1746-1596-7-14
Yang, R., Liu, Z., Gokavarapu, S., Peng, C., Cao, W. and Ji, T. (2017) Recurrence and Cancerization of Ameloblastoma: Multivariate Analysis of 87 Recurrent Craniofacial Ameloblastoma to Assess Risk Factors Associated with Early Recurrence and Secondary Ameloblastic Carcinoma. Chinese Journal of Cancer Research, 29, 189-195. https://doi.org/10.21147/j.issn.1000-9604.2017.03.04