Solitary Angiokeratoma in the Oral Cavity: A Critical Literature Review and Report of a New Case

• Published in: Indian journal of otolaryngology, and head, and neck surgery and head, and neck surgery, 2024-08, Vol.76 (4), p.3604-3611
• Main Authors: de Lima-Souza, Reydson Alcides, Kimura, Talita de Carvalho, Scarini, João Figueira, Lavareze, Luccas, Figueiredo-Maciel, Tayná, Chone, Carlos Takahiro, Egal, Erika Said Abu, Altemani, Albina, Mariano, Fernanda Viviane
• Format: Article
• Language: English
• Subjects: Clinical Report; Head and Neck Surgery; Medicine; Medicine & Public Health; Otorhinolaryngology
• ISSN: 2231-3796; 0973-7707
• DOI: 10.1007/s12070-024-04631-w

This study aimed to perform an integrative review of solitary angiokeratomas cases in the oral cavity and to report a new case in a 39-year-old man. A modified PECOS strategy was used using PubMed, Embase, Scopus, Web of Science databases, and the reference lists of the selected articles. Case reports of oral solitary angiokeratoma published in English, Portuguese, and Spanish languages with histopathological diagnosis without the presence of systemic disorders were included. Of the 51 articles identified, 18 met the eligibility criteria. Solitary angiokeratomas have a slight male predilection, with a peak incidence in the fourth decade of life. The tongue was the most common localization (77.7%), followed by buccal mucosa (11.1%), labial mucosa (5.6%), and tonsillar pillar (5.6%). The granulomatous appearance was the most frequent clinical aspect. Surgical excision was implemented in 94.4% of the cases. The lesion presented a good prognosis, with no recurrence in 3 to 24 months. In summary, solitary angiokeratoma is a rare lesion in the oral cavity. The professional making the oral diagnosis should be familiar with the clinical manifestation of angiokeratoma and be prepared to consider it in the differential diagnosis of pigmented lesions since these lesions may be part of systemic disorders.