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

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Abstract

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.

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Acknowledgements

The authors are grateful to Coordenação de Aperfeiçoamento de Pessoal de Nível Superior (CAPES) - Finance Code 001 and Fundação de Amparo à Pesquisa do Estado de São Paulo (FAPESP) (19/09419-0 and 20/08431-4) for the scholarships.

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Authors and Affiliations

  1. Department of Oral Diagnosis, Piracicaba Dental School, Universidade Estadual de Campinas (UNICAMP), Piracicaba, São Paulo, Brazil

    Reydson Alcides de Lima-Souza, Talita de Carvalho Kimura, João Figueira Scarini, Luccas Lavareze & Tayná Figueiredo-Maciel

  2. Department of Pathology, School of Medical Sciences, Universidade Estadual de Campinas (UNICAMP), Rua Tessália Vieira de Camargo, 126, 13083-887, Campinas, São Paulo, Brazil

    Reydson Alcides de Lima-Souza, Talita de Carvalho Kimura, João Figueira Scarini, Luccas Lavareze, Tayná Figueiredo-Maciel, Erika Said Abu Egal, Albina Altemani & Fernanda Viviane Mariano

  3. Department of Ophthalmology and Otorhinolaryngology, Faculdade de Ciências Médica, Universidade Estadual de Campinas (UNICAMP), Campinas, São Paulo, Brazil

    Carlos Takahiro Chone

  4. Biorepository and Molecular Pathology, Huntsman Cancer Institute, University of Utah (UU), Salt Lake City, Utah, United States of America

    Erika Said Abu Egal

Authors
  1. Reydson Alcides de Lima-Souza
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  2. Talita de Carvalho Kimura
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  9. Fernanda Viviane Mariano
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Contributions

Conceptualization: Reydson Alcides de Lima Souza, Fernanda Viviane Mariano; Methodology: Reydson Alcides de Lima Souza, Talita de Carvalho Kimura, João Figueira Scarini, Luccas Lavareze; Formal analysis and investigation: Reydson Alcides de Lima Souza, Tayná Figueiredo-Maciel, Carlos Takahiro Chone, Erika Said Abu Egal; Writing - original draft preparation: Reydson Alcides de Lima Souza; Writing - review and editing: Reydson Alcides de Lima-Souza, Talita de Carvalho Kimura, João Figueira Scarini, Luccas Lavareze, Tayná Figueiredo-Maciel, Carlos Takahiro Chone, Erika Said Abu Egal, Albina Altemani, Fernanda Viviane Mariano; Supervision: Albina Altemani, Fernanda Viviane Mariano.

Corresponding author

Correspondence to Fernanda Viviane Mariano.

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Ethical Approval

The study was approved by the local Institutional Review Board (protocol number: 3.601.521) and complied with the Helsinki Declaration. The authors affirm that human research participants provided informed consent for the publication of the case.

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The authors declare no competing interests.

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This case was presented at the 44° Congresso Brasileiro de Estomatologia e Patologia Oral in July 2018 in Rio de Janeiro, Rio de Janeiro (BR).

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de Lima-Souza, R.A., Kimura, T.d., Scarini, J.F. et al. Solitary Angiokeratoma in the Oral Cavity: A Critical Literature Review and Report of a New Case. Indian J Otolaryngol Head Neck Surg (2024). https://doi.org/10.1007/s12070-024-04631-w

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