Atypical cellular neurothekeoma of the oral mucosa: A rare case report and literature review

Cellular neurothekeoma (CN) is a benign dermal neoplasm that most often affects the head and neck region and rarely occurs in the oral mucosa. We report a rare case of CN with atypical features on the floor of the mouth and summarize the reported cases of oral CN in English‐language literature. A 62...

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Bibliographic Details
Published in:Journal of cutaneous pathology 2024-12, Vol.51 (12), p.930-937
Main Authors: Bezerra, Hélen Kaline Farias, Nakamura, Gabriele Prospero, Mariz, Bruno Augusto Linhares Almeida, Lopes, Rodrigo Nascimento, Prado, José Divaldo, Roza, Ana Luiza Oliveira Corrêa, Vargas, Pablo Agustin
Format: Article
Language:English
Subjects:
CD34 antigen
CD63 antigen
Cell proliferation
cellular neurothekeoma
Female
floor of the mouth
Humans
Literature reviews
Middle Aged
Mouth
Mouth Mucosa - metabolism
Mouth Mucosa - pathology
Mouth Neoplasms - diagnosis
Mouth Neoplasms - metabolism
Mouth Neoplasms - pathology
Mucosa
nerve sheath myxoma
Neurothekeoma - diagnosis
Neurothekeoma - metabolism
Neurothekeoma - pathology
oral mucosa
S100 protein
Stroma
Tumor cells
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Summary:Cellular neurothekeoma (CN) is a benign dermal neoplasm that most often affects the head and neck region and rarely occurs in the oral mucosa. We report a rare case of CN with atypical features on the floor of the mouth and summarize the reported cases of oral CN in English‐language literature. A 62‐year‐old woman presented with a 6‐month history of a painless mass on the floor of the mouth. Histopathological analysis of the excised specimen revealed a proliferation of neoplastic cells with oval to spindle morphology arranged in a vaguely nested and multinodular architecture separated by scarce hyaline collagen within a predominantly myxoid‐rich stroma. The tumor cells were positive for NSE, and CD63 (NKI/C3), and negative for S100 protein, CD34, and SMA. Thus, the final diagnosis was CN. In addition, we summarized all clinicopathological data on oral CNs reported in the English‐language literature. Nineteen cases were reviewed. Among them, only one case affected the floor of the mouth of a young girl, in contrast to the present case that occurred in an elderly woman. CN is particularly rare in this location and may be a diagnostic challenge for oral pathologists due to its rarity and morphological similarity with other lesions.
ISSN:0303-6987
1600-0560
1600-0560
DOI:10.1111/cup.14693