Skip to main content

Advertisement

Log in

Demographic, Clinical and Histopathological Features of Oral Neural Neoplasms: A Retrospective Study

  • Original Paper
  • Published:
Head and Neck Pathology Aims and scope Submit manuscript

Abstract

Intraoral neural neoplasms though unusual may be clinically significant. The aim of this study was to categorize and evaluate oral neural tumors in a large oral pathology biopsy service. With IRB approval, a retrospective search of all neural neoplasms of the oral cavity in the archives of the University of Florida Oral Pathology Biopsy Service spanning from 1994 to 2015 was performed. Extraoral cases as well as cases with insufficient patient information were excluded. A total of 340 out of 164,578 submitted specimens in a 22 year period (0.2%) were included with a mean age of 43.3 years (range: 6–89), and 44% male and 56% female. The most commonly affected locations were: tongue (37.5%), palate (22%), lip (19%), and gingiva (14%). The microscopic diagnoses rendered, in descending order of frequency were: neurofibromas (NFs): 123 (36%), granular cell tumor (GCT): 108 (32%), schwannomas: 61 (17%), palisaded encapsulated neuromas: 39 (11%), benign neural lesion not otherwise specified: 8 (2%), and mucosal neuroma c/w multiple endocrine neoplasia type 2B (MEN 2B): 1 (< 0.5%). Six cases of NF reported a history of neurofibromatosis Type 1 (NF 1). Four cases showed multifocal lesions. Immunohistochemical staining was performed on equivocal cases (25% of the lesions) and all were confirmed by their S-100 positivity. Intraoral neural neoplasms, though uncommon should be in the differential diagnosis of oral soft tissue entities and specific consideration to syndromal linkage is paramount as this may impact patient management.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2
Fig. 3
Fig. 4
Fig. 5

Similar content being viewed by others

References

  1. Alotaibi O, Al Sheddi M. Neurogenic tumors and tumor-like lesions of the oral and maxillofacial region: a clinicopathological study. Saudi Dent J. 2016;28:76–9.

    Article  PubMed  PubMed Central  Google Scholar 

  2. Salla JT, Johann AC, Garcia BG, Aguiar MC, Mesquita RA. Retrospective analysis of oral peripheral nerve sheath tumors in Brazilians. Braz Oral Res. 2009;23:43–8.

    Article  PubMed  Google Scholar 

  3. Chrysomali E, Papanicolaou SI, Dekker NP, Regezi JA. Benign neural tumors of the oral cavity: a comparative immunohistochemical study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1997;84:381–90.

    Article  CAS  PubMed  Google Scholar 

  4. Accurso B, Mercado A, Allen CM. Multiple endocrine neoplasia-2B presenting with orthodontic relapse. Angle Orthod. 2010;80(3):585–90.

    Article  PubMed  Google Scholar 

  5. Katz AD, McAlpin C. Face and neck neurogenic neoplasms. Am J Surg. 1993;166:421–3.

    Article  CAS  PubMed  Google Scholar 

  6. Fletcher CDM, Bridge JA, Hogendoorn PCW, Mertens F, editors. WHO classification of tumours of soft tissue and bone: pathology and genetics of tumours of soft tissue and bone. 4th ed. Lyon: IARC Press; 2013.

    Google Scholar 

  7. Eryilmaz MA, Varsak YK, Gul Z, Etli Ö. Second primary malignant peripheral nerve sheath tumor of the paranasal sinuses after nasopharynx carcinoma. J Craniofac Surg. 2014;25:2074–6.

    Article  PubMed  Google Scholar 

  8. Depprich R, Singh DD, Reinecke P, Kübler NR, Handschel J. Solitary submucous neurofibroma of the mandible: review of the literature and report of a rare case. Head Face Med. 2009;5:24.

    Article  PubMed  PubMed Central  Google Scholar 

  9. do Nascimento GJ, de Albuquerque Pires Rocha D, Galvão HC, de Lisboa Lopes Costa A, de Souza LB. A 38-year review of oral schwannomas and neurofibromas in a Brazilian population: clinical, histopathological and immunohistochemical study. Clin Oral Investig. 2011;15:329–35.

    Article  PubMed  Google Scholar 

  10. Riccardi VM. The genetic predisposition to and histogenesis of neurofibromas and neurofibrosarcoma in neurofibromatosis type 1. Neurosurg Focus. 2007;22:E3.

    Article  PubMed  Google Scholar 

  11. Hassell DS, Bancroft LW, Kransdorf MJ, et al. Imaging appearance of diffuse neurofibroma. AJR Am J Roentgenol. 2008;190:582–8.

    Article  PubMed  Google Scholar 

  12. Shekar V, Rangdhol V, Baliah WJ, Thirunavukarasu S. An unusual oral manifestation of type 1 neurofibromatosis: a case report and review of literature. J Nat Sci Biol Med. 2015;6:261–3.

    Article  PubMed  PubMed Central  Google Scholar 

  13. Majoie CB, Hulsmans FJ, Castelijns JA, et al. Primary nerve-sheath tumours of the trigeminal nerve: clinical and MRI findings. Neuroradiology. 1999;41:100–8.

    Article  CAS  PubMed  Google Scholar 

  14. Furniss D, Swan MC, Morritt DG, et al. A 10-year review of benign and malignant peripheral nerve sheath tumors in a single center: clinical and radiographic features can help to differentiate benign from malignant lesions. Plast Reconstr Surg. 2008;121:529–33.

    Article  CAS  PubMed  Google Scholar 

  15. Kim DH, Murovic JA, Tiel RL, Moes G, Kline DG. A series of 397 peripheral nerve sheath tumors: 30-year experience at Louisiana State University Health Sciences Center. J Neurosurg. 2005;102:246–55.

    Article  PubMed  Google Scholar 

  16. Shapiro SD, Abramovitch K, Van Dis ML, et al. Neurofibromatosis: oral and radiographic manifestations. Oral Surg Oral Med Oral Pathol. 1984;58:493–8.

    Article  CAS  PubMed  Google Scholar 

  17. Jouhilahti EM, Visnapuu V, Soukka T, et al. Oral soft tissue alterations in patients with neurofibromatosis. Clin Oral Investig. 2012;16:551–8.

    Article  PubMed  Google Scholar 

  18. D’Ambrosio JA, Langlais RP, Young RS. Jaw and skull changes in neurofibromatosis. Oral Surg Oral Med Oral Pathol. 1988;66:391–6.

    Article  PubMed  Google Scholar 

  19. Sørensen SA, Mulvihill JJ, Nielsen A. Long-term follow-up of von Recklinghausen neurofibromatosis: survival and malignant neoplasms. N Engl J Med. 1986;314:1010–5.

    Article  PubMed  Google Scholar 

  20. Masocco M, Kodra Y, Vichi M, et al. Mortality associated with neurofibromatosis type 1: a study based on Italian death certificates (1995–2006). Orphanet J Rare Dis. 2011;6:11.

    Article  PubMed  PubMed Central  Google Scholar 

  21. Lin AL, Gutmann DH. Advances in the treatment of neurofibromatosis-associated tumours. Nat Rev Clin Oncol. 2013;10:616–24.

    Article  CAS  PubMed  Google Scholar 

  22. Oderich GS, Sullivan TM, Bower TC, et al. Vascular abnormalities in patients with neurofibromatosis syndrome type I: clinical spectrum, management, and results. J Vasc Surg. 2007;46:475–84.

    Article  PubMed  Google Scholar 

  23. Williams VC, Lucas J, Babcock MA, Gutmann DH, Korf B, Maria BL. Neurofibromatosis type 1 revisited. Pediatrics 2009;123:124–33.

    Article  PubMed  Google Scholar 

  24. Shilpa B. Ancient schwannoma-a rare case. Ethiop J Health Sci. 2012;22:215–8.

    CAS  PubMed  PubMed Central  Google Scholar 

  25. Kohyama S, Hara Y, Nishiura Y, Hara T, Nakagawa T, Ochiai N. A giant plexiform schwannoma of the brachial plexus: case report. J Brachial Plex Peripher Nerve Inj. 2011;6:9.

    PubMed  PubMed Central  Google Scholar 

  26. Megahed M. Plexiform schwannoma. Am J Dermatopathol. 1994;16:288–93.

    Article  CAS  PubMed  Google Scholar 

  27. Chi AC, Carey J, Muller S. Intraosseous schwannoma of the mandible: a case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2003;96:54–65.

    Article  PubMed  Google Scholar 

  28. Reed RJ, Fine RM, Meltzer HD. Palisaded, encapsulated neuromas of the skin. Arch Dermatol. 1972;106:865–70.

    Article  CAS  PubMed  Google Scholar 

  29. Koutlas IG, Scheithauer BW. Palisaded encapsulated (“solitary circumscribed”) neuroma of the oral cavity: a review of 55 cases. Head Neck Pathol. 2010;4:15–26.

    Article  PubMed  PubMed Central  Google Scholar 

  30. Fanburg-Smith JC, Meis-Kindblom JM, Fante R, Kindblom LG. Malignant granular cell tumor of soft tissue: diagnostic criteria and clinicopathologic correlation. Am J Surg Pathol. 1998;22:779–94.

    Article  CAS  PubMed  Google Scholar 

  31. Sena Costa NC, Bertini F, Carvalho YR, Almeida JD, Rodrigues Cavalcante AS. Granular cell tumor presenting as a tongue nodule: two case reports. J Med Case Rep. 2012;6:56.

    Article  PubMed  PubMed Central  Google Scholar 

  32. van de Loo S, Thunnissen E, Postmus P, van der Waal I. Granular cell tumor of the oral cavity; a case series including a case of metachronous occurrence in the tongue and the lung. Med Oral Patol Oral Cir Bucal. 2015;20:e30–33.

    Google Scholar 

  33. Rosenthal SA, Livolsi VA, Turrisi AT. Adjuvant radiotherapy for recurrent granular cell tumor. Cancer. 1990;65:897–900.

    Article  CAS  PubMed  Google Scholar 

  34. Tomson N, Abdullah A, Tan CY. Multiple granular cell tumors in a child with growth retardation. Report of a case and review of the literature. Int J Dermatol. 2006;45:1358–61.

    Article  PubMed  Google Scholar 

  35. Lee MJ, Chung KH, Park JS, Chung H, Jang HC, Kim JW. Multiple endocrine neoplasia type 2B: early diagnosis by multiple mucosal neuroma and its DNA analysis. Ann Dermatol. 2010;22:452–5.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  36. Ramalingam WV, Nair S, Mandal G. Malignant peripheral nerve sheath tumor of the oral cavity. J Oral Maxillofac Surg. 2012;70:e581–5.

    Article  CAS  PubMed  Google Scholar 

  37. Patil K, Mahima VG, Ambika L. Malignant peripheral nerve sheath tumour: an elusive diagnosis. Indian J Dent Res. 2007;18:19–22.

    Article  PubMed  Google Scholar 

Download references

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Faraj M. Alotaiby.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Alotaiby, F.M., Fitzpatrick, S., Upadhyaya, J. et al. Demographic, Clinical and Histopathological Features of Oral Neural Neoplasms: A Retrospective Study. Head and Neck Pathol 13, 208–214 (2019). https://doi.org/10.1007/s12105-018-0943-1

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12105-018-0943-1

Keywords

Navigation