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Subgemmal neurogenous plaque: a series of six cases and a literature overview

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Abstract

The study aims to describe six subgemmal neurogenous plaque (SNP) cases and compile the previously available data on SNP. Searches on PubMed, Web of Science, Embase, Scopus, and a two-step study selection were performed to compile data from case reports/series of SNP published in English literature. Our six patients (range 29–63 years) had SNP in the posterior lateral border of the tongue. Four of them were associated with lymphoepithelial cysts. A total of 15 studies, comprising 116 patients, were included in this overview. There was a slight predilection for female patients (56.0%) in the 5th (22.4%) and the 7th (23.9%) decades. The lateral border of the tongue (66.4%) was the most common anatomical location. The major manifestation was nodule/papule. Histologically, the presence of taste buds (n = 40/ 34.5%), superficial neural plexus (n = 32/ 27.6%), deep nerve fascicles (n = 32/ 27.6%), ganglion cells (n = 41/ 35.3%), and lymphoid tissue (n = 22/ 19.0%) was mainly reported. Surgical removal was the primary approach, with a low recurrence rate. Although largely unknown, SNP is a regular and likely frequent structure located in the tongue. The findings emphasize the importance of describing clinicopathological patterns of SNP as a differential diagnosis of neural lesions that affect the tongue.

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References

  1. McDaniel RK (1999) Subepithelial nerve plexus (with ganglion cells) associated with taste buds. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 87(5):605–609

    Article  CAS  PubMed  Google Scholar 

  2. Lopes-Santos G, Cardoso CL, Oliveira DT (2022) Subgemmal neurogenous plaque of posterolateral region in tongue: A case report and review of literature. Int J Surg Case Rep 94:107086. https://doi.org/10.1016/j.ijscr.2022.107086

  3. Fonseca FP, Latta Moreira JP, Almeida OP, Vargas PA, Mauad T (2015) Neuroepithelial structures associated with neurogenous subgemmal plaque of the tongue: an autopsy finding. Oral Surg Oral Med Oral Pathol Oral Radiol [Internet] 120(1):94–96. https://doi.org/10.1016/j.oooo.2015.02.487

    Article  PubMed  Google Scholar 

  4. Palazzolo MJ (2015) Response to “neuroepithelial structures associated with neurogenous subgemmal plaque of the tongue: an autopsy finding.” Oral Surg Oral Med Oral Pathol Oral Radiol [Internet] 120(1):96. https://doi.org/10.1016/j.oooo.2015.03.010

    Article  PubMed  Google Scholar 

  5. Gueiros LA, Leon JE, Lopes MA, de Almeida OP, Jorge J (2008) Subgemmal neurogenous plaque associated with burning tongue: report of two cases and review of the literature. Int J Oral Maxillofac Surg 37(8):773–776. https://doi.org/10.1016/j.ijom.2008.01.025

  6. Artur Ricieri Brito J, Tinôco Alvim De Souza F, César Tanos De Lacerda J, Fátima Bernardes V, Cavaliéri Gomes C, Gomez RS (2012) Asymptomatic nodule in the tongue. Oral Surg Oral Med Oral Pathol Oral Radiol [Internet]. 114(3):281–3. https://doi.org/10.1016/j.oooo.2011.11.003

  7. Val-Bernal JF, Rivadulla I, Garijo MF (2006) Lingual subgemmal neurogenous plaques with pseudoepitheliomatous hyperplasia: incidental pseudomalignant condition. Pathol Int 56(8):462–465

    Article  PubMed  Google Scholar 

  8. Pellicioli ACA, Fonseca FP, Silva RN, Gueiros LAM, de Almeida OP, Vargas PA et al (2017) Histomorphometric characterization of subgemmal neurogenous plaques. Oral Surg Oral Med Oral Pathol Oral Radiol [Internet] 123(4):477–481. https://doi.org/10.1016/j.oooo.2016.12.014

    Article  PubMed  Google Scholar 

  9. da Silva KD, Coelho LV, do Couto AM, de Aguiar MCF, Tarquínio SBC, Gomes APN et al (2020) Clinicopathological and immunohistochemical features of the oral lymphoepithelial cyst: a multicenter study. J Oral Pathol Med. 49(3):219–26

    Article  PubMed  Google Scholar 

  10. Palazzolo MJ, Fowler CB, Magliocca KR, Gnepp DR (2014) Neuroepithelial structures associated with the subepithelial nerve plexus of taste buds: a fortuitous finding resembling the juxtaoral organ of Chievitz. Oral Surg Oral Med Oral Pathol Oral Radiol [Internet] 117(4):497–501. https://doi.org/10.1016/j.oooo.2013.12.403

    Article  PubMed  Google Scholar 

  11. Soares CD, de Souza LL, Laffitte CM, Pontes HAR, de Almeida OP, Mosqueda-Taylor A et al (2023) Symptomatic subgemmal neurogenous plaque in patients with COVID-19: is there an association? J Oral Pathol Med 52(6):514–520

    Article  CAS  PubMed  Google Scholar 

  12. Alnajar H, O’Toole TR, Lin DM, Al-Khudari S, Gattuso P (2019) Subgemmal neurogenous plaque: a clinical and pathologic review with comparison to common head and neck neural tumors. Clin Pathol 12:2632010X1983018

    Article  Google Scholar 

  13. Gueiros LA, León JE, Leão JC, Lopes MA, Jorge J, de Almeida OP (2009) Subgemmal neurogenous plaque clinical and microscopic evaluation of 7 cases. Oral Surg, Oral Med Oral Pathol Oral Radiol Endodontol [Internet] 108(6):920–4. https://doi.org/10.1016/j.tripleo.2009.07.038

    Article  Google Scholar 

  14. Gonzaga AKG, Moreira DGL, Sena DAC, de Lopes MLDS, de Souza LB, Queiroz LMG (2017) Subgemmal neurogenous plaque of the tongue: a report of three cases. Oral Maxillofac Surg 21(3):351–5

    Article  PubMed  Google Scholar 

  15. Heguedusch D, Felipe L, Maciel O, Oliveira SV, Custódio M, Daumas Nunes F, et al. (2019) Clinical aspects of tongue subgemmal neurogenous plaque: surgical treatment of two symptomatic cases. J Oral Diagnosis [Internet]. 4(1):1–4. http://www.jordi.com.br/detalhe_artigo.asp?id=276. Accessed 22 Jan 2024

  16. Cunha JLS, Roza ALOC, Cruz VMS, Ribeiro JL, Cavalcante IL, Cavalcante RB et al (2022) Oral lymphoepithelial cyst: a collaborative clinicopathologic study of 132 cases from Brazil. Head Neck Pathol [Internet] 16(1):268–77. https://doi.org/10.1007/s12105-021-01352-2

    Article  PubMed  Google Scholar 

  17. Triantafyllou A, Coulter P (2004) Structural organization of subgemmal neurogenous plaques in foliate papillae of tongue. Hum Pathol 35(8):991–999

    Article  PubMed  Google Scholar 

  18. Agrawal M, Sonthalia S, Jha A, Goldust M (2018) Asymptomatic pinkish-red nodule over the posterolateral tongue. J Cutan Aesthet Surg 11(4):245–247

    Article  PubMed  PubMed Central  Google Scholar 

  19. Val-Bernal JF, Garijo MF, Fontanil N (2022) Subgemmal neurogenous plaques of the tongue: a systematic autopsy study. Rom J Morphol Embryol 63(3):545–553

    Article  PubMed  PubMed Central  Google Scholar 

  20. Watanabe IS, Guimarães JP, Boleta SA, Almeida SRY, Righeti MM, Santos TC et al (2009) Nerve endings of filliform, fungiform and vallate papillae of dorsal tongue mucosa of White-lipped peccary (Tayassu pecari): neurohistological observations. Pesqui Vet Bras 29(4):281–5

    Article  Google Scholar 

  21. Farbman AI, Hellekant G, Nelson A (1985) Structure of taste buds in foliate papillae of the rhesus monkey Macaca mulatta. Am J Anat 172(1):41–56

    Article  CAS  PubMed  Google Scholar 

  22. Baradi AF, Bourne GH (1953) Gustatory and olfactory epithelia. Int Rev Cytol 2(C):289–330

    Article  CAS  Google Scholar 

  23. Burki T, Kiho L, Scheimberg I, Phelps S, Misra D, Ward H et al (2011) Neonatal functional intestinal obstruction and the presence of severely immature ganglion cells on rectal biopsy: 6 year experience. Pediatr Surg Int 27(5):487–490

    Article  PubMed  Google Scholar 

  24. Zhang J, Jin H, Zhang W, Ding C, O’Keeffe S, Ye M et al (2019) Sour sensing from the tongue to the brain. Cell [Internet] 179(2):392-402.e15. https://doi.org/10.1016/j.cell.2019.08.031

    Article  CAS  PubMed  Google Scholar 

  25. Lee KC, Mandel L (2019) Lingual (Not Palatine) Tonsillolith: case report. J Oral Maxillofac Surg [Internet] 77(8):1650–4. https://doi.org/10.1016/j.joms.2019.03.006

    Article  PubMed  Google Scholar 

  26. Almeida LKY, Silveira HA, Ferrisse TM, Silva EV, Almeida LY, Bufalino A et al (2020) Unilateral enlargement of the lateral lingual tonsil: an immunohistochemical study and literature review. Oral Oncol 109:8–11

    Article  Google Scholar 

  27. Kennedy R (2020) Neuroepithelial structures of the oral soft tissues including the juxtaoral organ of chievitz: a literature review and audit of diagnosed cases. Head Neck Pathol [Internet] 14(4):876–83. https://doi.org/10.1007/s12105-020-01131-5

    Article  PubMed  Google Scholar 

  28. Ide F, Ito Y, Nishimura M, Kusama K, Kikuchi K (2020) So-called neuroepithelial structures in the posterolateral tongue: what can be learned from former studies? Head Neck Pathol [Internet] 14(4):1092–3. https://doi.org/10.1007/s12105-020-01180-w

    Article  PubMed  Google Scholar 

  29. Jaiswal S. Role of immunohistochemistry in the diagnosis of central nervous system tumors. Neurol India [Internet]. 2016;64(3):502. https://doi.org/10.4103/0028-3886.181547

  30. Rauso R, Colella G, Franco R, Ronchi A, Chirico F (2019) Ossified carcinoma ex pleomorphic adenoma in accessory lobe of parotid gland: complexity in clinical, imaging and histologic diagnosis and minimally invasive surgery. Oral Oncol 92:95–98

    Article  PubMed  Google Scholar 

  31. Bryan Whitaker S, Krupa JJ, Singh BB (1996) Transient lingual papillitis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 82(4):441–445

    Article  Google Scholar 

Download references

Acknowledgements

The authors thank CNPq, CAPES, FAPEMIG, and PRPq-UFMG.

Funding

The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by the Pró-Reitoria de Pesquisa da Universidade Federal de Minas Gerais (PRPq-UFMG), Coordenação de Aperfeiçoamento de Pessoal de Nível Superior (CAPES, Financial Code 001), Fundação de Amparo à Pesquisa do Estado de Minas Gerais (FAPEMIG), and Conselho Nacional de Desenvolvimento Científico e Tecnológico (CNPq).

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All authors contributed to the study’s conception and design. DPM, FAF, and MCFd A performed material preparation, data collection, and analysis. DPM and FAF wrote the first draft of the manuscript. All authors commented on previous versions of the manuscript. All authors read and approved the final manuscript.

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Correspondence to Daniela Pereira Meirelles.

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An abstract was presented at the “47º Congresso Brasileiro de Estomatologia e Patologia Oral” held in Tiradentes, Brazil, July 2022, annals to be published at TripleO.

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Meirelles, D.P., Felix, F.A., de Sousa, S.F. et al. Subgemmal neurogenous plaque: a series of six cases and a literature overview. Oral Maxillofac Surg 28, 975–983 (2024). https://doi.org/10.1007/s10006-024-01211-8

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