Atypical oral features of patient with neurofibromatosis type 1: case report

Alves, Caroline S.; Santana, Lucas A. M.; Felix, Fernanda A.; Ayres, Lucas G. C.; Silva, Erika R.; Takeshita, Wilton M.

doi:10.5935/1676-2444.20210041

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CASE REPORT • J. Bras. Patol. Med. Lab. 57 • 2021 • https://doi.org/10.5935/1676-2444.20210041 copy

Atypical oral features of patient with neurofibromatosis type 1: case report

Authorship SCIMAGO INSTITUTIONS RANKINGS

ABSTRACT

The purpose of this article is to assist dental clinicians to identify neurofibromatosis type 1 and its variants. We present a rare case of a 68-year-old man affected since his childhood by the presence of generalized nodules throughout the body, causing aesthetic deformations. Interestingly, the impact in the oral cavity was not significant, presenting nodes only in the gingiva, an unusual area of manifestation. Panoramic radiograph showed a discrete enlargement of the mandibular canal. After 1 year of follow-up, the patient died due to cardiovascular complications caused by the syndrome.

Authors	Type of study	Oral clinical manifestations	Oral radiographic findings
Sarmento et al. (2017)(¹⁴14 Sarmento DJS, de Carvalho SHG, de Araújo Filho JCWP, Carvalho MV, da Silveira EJD. Florid cemento-osseous dysplasia and peripheral giant cell granuloma in a patient with neurofibromatosis 1. An Bras Dermatol. 2017; 92(2): 249–52.)	Clinical case	Giant cell lesion and florid cemento-osseous dysplasia	Not reported
Friedrich et al. (2017)(²¹21 Friedrich RE, Lehmann JM, Rother J, et al. A lateral cephalometry study of patients with neurofibromatosis type 1. J Craniomaxillofac Surg. 2017; 45(6): 809–20.)	Research	Not applicable	Asymmetry in the facial skeleton, no changes in the sella turcica
Mahmud et al. (2016)(¹⁵15 Mahmud SA, Shah N, Chattaraj M, Gayen S. Solitary encapsulated neurofibroma not associated with neurofibromatosis-1 affecting tongue in a 73-year-old female. Case Rep Dent. 2016; 2016.)	Clinical case	Neurofibroma in tongue	Not reported
Shekar et al. (2015)(⁹9 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(1): 261.)	Clinical case	Neurofibroma gingival	Not reported
Cunha et al. (2015)(¹⁸18 Cunha KS, Rozza-De-Menezes RE, Luna EB, et al. High prevalence of hyposalivation in individuals with neurofibromatosis 1: a case-control study. Orphanet J Rare Dis. 2015; 10(1).)	Research	High prevalence of hyposalivation	Not reported
Visnapuu et al. (2012)(¹⁹19 Visnapuu V, Peltonen S, Tammisalo T, Peltonen J, Happonen RP. Radiographic findings in the jaws of patients with neurofibromatosis 1. J Oral Maxillofac Surg. 2012; 70(6): 1351–7.)	Research	Not applicable	Increased diameter of the mandibular canal
Kobayashi et al. (2012)(²²22 Kobayashi R, Matsune K, Ohashi H. Fused teeth, macrodontia and increased caries are characteristic features of neurofibromatosis type 1 patients with NF1 gene microdeletion. J Pediatr Genet. 2012; 1(1): 25–31.)	Research	Not applicable	Macrodontia and excess of dental caries

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[1] CORRESPONDING AUTHOR: Lucas Alves da Mota Santana. e-mail: lucassantana.pat@gmail.com