PAPILLIFEROUS KERATOAMELOBLASTOMA – A SYSTEMATIC REVIEW
Main Article Content
Keywords
Papilliferous keratoameloblastoma, Ameloblastoma, Keratoameloblastoma, Odontogenic tumour, Mandible, Histopathology.
Abstract
Background: Papilliferous keratoameloblastoma is an extremely rare keratinizing papilliferous variant within the ameloblastoma spectrum, with limited data regarding its clinicopathological profile and biological behaviour.
Objective: This systematic review aimed to evaluate the demographic, clinical, radiographic, histopathological, treatment-related, and follow-up characteristics of reported cases of papilliferous keratoameloblastoma.
Methods: A systematic literature search was performed using PubMed/MEDLINE, Scopus, Web of Science, and Google Scholar from database inception to June 2026. Human case reports and case series with histopathologically confirmed papilliferous keratoameloblastoma or definite papilliferous keratinizing ameloblastomatous features were included. Data were synthesized narratively.
Results: Eight studies reporting eight individual cases were included. Patient age ranged from 18 to 65 years, with a mean age of 43.4 years, and male predominance was observed. All cases involved the right mandible, mainly the posterior mandibular region. Clinically, most cases presented as slowly enlarging mandibular swelling. Radiographic findings were non-specific and included unilocular or multilocular radiolucent/osteolytic lesions. Histopathologically, all cases showed ameloblastomatous epithelial differentiation with papilliferous or verrucopapillary projections and keratinization. Treatment varied from excision to mandibular resection. Recurrence was reported in one case.
Conclusion: Papilliferous keratoameloblastoma is a rare, diagnostically distinct, and potentially locally aggressive variant requiring histopathological confirmation, complete surgical removal, and long-term follow-up.
References
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3. Anyanechi CE, Shetty SS. Ameloblastoma of the jaws in adult: A retrospective review of local recurrent lesions based on the resection margin in the adjacent apparent healthy tissues. Heliyon. 2023 May 13;9(5):e16243. doi: 10.1016/j.heliyon.2023.e16243. PMID: 37251853; PMCID: PMC10209404.
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9. McLean-Holden AC, Magliocca K. Ameloblastoma. PathologyOutlines.com website. https://www.pathologyoutlines.com/topic/mandiblemaxillaameloblastoma.html. Accessed June 21st, 2026.
10. Chavhan A, Pakhale A, Patil S, Hande A, Tehzeeb H, Akolkar S. Ameloblastoma With the Hybrid Desmoplastic and Plexiform Pattern: A Case Report. Cureus. 2024 Jun 4;16(6):e61686. doi: 10.7759/cureus.61686. PMID: 38975386; PMCID: PMC11226227.
11. Cadavid AMH, Teshima THN, Pinto CAL, Camillo CMC, Lourenço SV. Ameloblastoma with distinctive granular cell pattern: an 8 case study. Autops Case Rep. 2018 Oct 9;8(4):e2018052. doi: 10.4322/acr.2018.052. PMID: 30775327; PMCID: PMC6360828.
12. Sujani Madabhushi, Srighana Nadella, John Lankalis, Faizan Alawi, Nikolina Dioufa, Keratoameloblastoma: unusual keratinization in ameloblastoma – a case report and review of literature, Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology, 2026, ISSN 2212-4403, https://doi.org/10.1016/j.oooo.2026.03.012.
13. Raj V, Chandra S, Bedi RS, Dwivedi R. Keratoameloblastoma: Report of a rare variant with review of literature. Dent Res J (Isfahan). 2014 Sep;11(5):610-4. PMID: 25426155; PMCID: PMC4241617.
14. Adeyemi B, Adisa A, Fasola A, Akang E. Keratoameloblastoma of the mandible. J Oral Maxillofac Pathol. 2010 Jul;14(2):77-9. doi: 10.4103/0973-029X.72507. PMID: 21731268; PMCID: PMC3125065.
15. Kuberappa PH, Anuradha A, Kiresur MA, Bagalad BS. Papilliferous keratoameloblastoma - A rare entity: A case report with a review of literature. J Oral Maxillofac Pathol. 2020 Feb;24(Suppl 1):S2-S6. doi: 10.4103/jomfp.JOMFP_42_17. Epub 2020 Feb 28. PMID: 32189894; PMCID: PMC7069145.
16. Rathore AS, Juneja S, Khurana N, Shetty DC. Papilliferous Keratoameloblastoma: A Rare Case Report. Int J Appl Basic Med Res. 2017 Apr-Jun;7(2):139-142. doi: 10.4103/ijabmr.IJABMR_136_16. PMID: 28584748; PMCID: PMC5441264.
17. Sachdev SS, Chettiankandy TJ, Adhane YB, Sardar MA, Trimukhe A, Jain R. Papilliferous Keratoameloblastoma: Systematic review. Sultan Qaboos Univ Med J. 2023 Aug;23(3):288-296. doi: 10.18295/squmj.5.2023.021. Epub 2023 Aug 28. PMID: 37655071; PMCID: PMC10467561.
18. Netto R, Janini MER, Cortezzi W, Moura LL, de Freitas-Filho SAJ. Keratoameloblastoma of the jaws and review of international literature of 38 cases. J Clin Exp Dent. 2025 Aug 1;17(8):e1006-e1013. doi: 10.4317/jced.62973. PMID: 40950516; PMCID: PMC12424603.
19. Goh YC, Bologna-Molina R, Magliocca K, van Heerden W, Robinson L, Bilodeau EA, Hussaini HM, Soluk-Tekkesin M, Shrestha M, Vered M, Adisa AO, Li J, Gomez RS, Hunter KD, Tilakaratne WM. Papilliferous Keratoameloblastoma (PKA): Is It a Different Clinicopathological Entity or Histological Subtype of Conventional Ameloblastoma? J Oral Pathol Med. 2026 May;55(5):511-519. doi: 10.1111/jop.70115. Epub 2026 Jan 2. PMID: 41482710.
20. Bedi RS, Sah K, Singh A, Chandra S, Raj V. Keratoameloblastoma or Kerato-odontoameloblastoma: report of its soft tissue recurrence with literature review. Quant Imaging Med Surg. 2015 Dec;5(6):898-908. doi: 10.3978/j.issn.2223-4292.2015.09.01. PMID: 26807371; PMCID: PMC4700246.
21. Robinson L, Smit C, Fonseca FP, Abrahão AC, Romañach MJ, Khurram SA, Hunter KD, Speight PM, van Heerden WFP. Keratoameloblastoma: A Report of Seven New Cases and Review of Literature. Head Neck Pathol. 2022 Dec;16(4):1103-1113. doi: 10.1007/s12105-022-01470-5. Epub 2022 Jul 21. PMID: 35861917; PMCID: PMC9729669.
22. Page MJ, McKenzie JE, Bossuyt PM, Boutron I, Hoffmann TC, Mulrow CD, Shamseer L, Tetzlaff JM, Akl EA, Brennan SE, Chou R, Glanville J, Grimshaw JM, Hróbjartsson A, Lalu MM, Li T, Loder EW, Mayo-Wilson E, McDonald S, McGuinness LA, Stewart LA, Thomas J, Tricco AC, Welch VA, Whiting P, Moher D. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ. 2021 Mar 29;372:n71. doi: 10.1136/bmj.n71. PMID: 33782057; PMCID: PMC8005924.
23. Covvey JR, McClendon C, Gionfriddo MR. Back to the basics: Guidance for formulating good research questions. Res Social Adm Pharm. 2024 Jan;20(1):66-69. doi: 10.1016/j.sapharm.2023.09.009. Epub 2023 Oct 12. PMID: 37838572; PMCID: PMC11129835.
24. Norval EJ, Thompson IO, van Wyk CW. An unusual variant of keratoameloblastoma. J Oral Pathol Med. 1994 Nov;23(10):465-7. doi: 10.1111/j.1600-0714.1994.tb00446.x. PMID: 7532221.
25. Collini P, Zucchini N, Vessecchia G, Guzzo M. Papilliferous keratoameloblastoma of mandible: a papillary ameloblastic carcinoma: report of a case with a 6-year follow-up and review of the literature. Int J Surg Pathol. 2002 Apr;10(2):149-55. doi: 10.1177/106689690201000210. PMID: 12075410.
26. Mohanty N, Rastogi V, Misra SR, Mohanty S. Papilliferous keratoameloblastoma: an extremely rare case report. Case Rep Dent. 2013;2013:706128. doi: 10.1155/2013/706128. Epub 2013 Jun 3. PMID: 23862079; PMCID: PMC3686074.
27. Konda P, Bavle RM, Muniswamappa S, Makarla S, Venugopal R. Papilliferous Keratoameloblastoma of the Mandible - A Rare Case Report. J Clin Diagn Res. 2016 Aug;10(8):ZD08-11. doi: 10.7860/JCDR/2016/18862.8232. Epub 2016 Aug 1. PMID: 27656576; PMCID: PMC5028498.
28. Jacob M, Rajathi P, Mathew M, Fenn SM. Macro and microscopic study of a rare case of Papilliferous Keratoameloblastoma using a stereo zoom microscope. Indian J Pathol Microbiol. 2023 Apr-Jun;66(2):388-391. doi: 10.4103/ijpm.ijpm_714_21. PMID: 37077093.
29. Urs AB, Augustine J, Kumar P, Mohanty S, Jha S. Papilliferous keratoameloblastoma: a case report with review of the literature. Int J Oral Maxillofac Surg. 2025 Dec;54(12):1171-1176. doi: 10.1016/j.ijom.2025.07.006. Epub 2025 Aug 5. PMID: 40764150.
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Jan 09, 2017
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Dr.N.Inbanesan. (2017). PAPILLIFEROUS KERATOAMELOBLASTOMA – A SYSTEMATIC REVIEW. Journal of Population Therapeutics and Clinical Pharmacology, 24(1), 152-167. https://doi.org/10.69980/9nfxs858
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Author Biography
Dr.N.Inbanesan
Reader in Department of OMFS Karpaga vinayaga Institute of Dental sciences, Tamilnadu, India,
How to Cite
Dr.N.Inbanesan. (2017). PAPILLIFEROUS KERATOAMELOBLASTOMA – A SYSTEMATIC REVIEW. Journal of Population Therapeutics and Clinical Pharmacology, 24(1), 152-167. https://doi.org/10.69980/9nfxs858
