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Congenital Melanotic Macule of the Tongue: Report of Two Cases and Literature Review

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Abstract

Background

Congenital melanotic macule of the tongue (CMMT) has been described as a distinct entity, despite its unknown etiology. However, the diagnosis and management of affected newborns may challenge clinicians and pediatric dentists.

Methods

We document here the clinicopathological findings of two additional cases of CMMT. A literature review of CMMT reports identified across PubMed, Web of Science, Embase, and Scopus was also conducted.

Results

The patients, 2- and 4 month-old Venezuelan boys, respectively, presented at birth with a single or multiple dark-brown-pigmented macule exclusively on the dorsum of the tongue. Histopathological features revealed increased melanin pigmentation in the basal epithelial layer with overlying hyperkeratosis and pigment-laden subepithelial macrophages with normal morphological appearance. Nine studies comprising 17 cases of CMMT have been described hitherto. Most cases were from the USA and France (n = 6 each). Twelve (70.6%) patients were males, eight (50%) were white, and median age was 2.7 months. CMMT presented as brownish to black, solitary or multiple pigmentations located in the right or left region of the dorsum of the tongue, ranging in size from 3.0 to 30.0 mm.

Conclusion

An important feature for the diagnosis of CMMT is the information about the manifestation at birth and consequent proportional growth. This report intends to draw the attention of pediatricians and dentists to this apparently underdiagnosed condition for decision-making and management of affected newborns.

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Data Availability

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Acknowledgements

J.A.A.A. is the recipient of fellowship granted by Coordenação de Aperfeiçoamento de Pessoal de Nível Superior, Brazil (CAPES, finance code 001). B.A.B.A. is a research fellow of Conselho Nacional de Desenvolvimento Científico e Tecnológico (CNPq, 302627/2022-7). Mrs. E. Greene provided English editing of the manuscript.

Funding

This study was partially supported by Fundação Carlos Chagas Filho de Amparo à Pesquisa do Estado do Rio de Janeiro (FAPERJ - E-26/201.289/2022), Brazil.

Author information

Authors and Affiliations

  1. Department of Oral Surgery, Pathology and Clinical Dentistry, School of Dentistry, Universidade Federal de Minas Gerais, Belo Horizonte, Brazil

    José Alcides Almeida de Arruda

  2. Department of Oral Medicine, Dental School, Universidad Central de Venezuela, Caracas, Venezuela

    Rosanna Gómez & Mariana Villarroel-Dorrego

  3. Dermatology Programme, Biomedicine Institute, Universidad Central de Venezuela, Caracas, Venezuela

    Verónica Bracho & Ricardo Pérez-Alfonzo

  4. Department of Oral Diagnosis and Pathology, School of Dentistry, Universidade Federal do Rio de Janeiro, R. Rodolpho Paulo Rocco, n. 325, 1st floor, Rio de Janeiro, RJ, Brazil

    Israel Leal Cavalcante & Bruno Augusto Benevenuto de Andrade

  5. Department of Dentistry, Universidade de Fortaleza, Fortaleza, Brazil

    Israel Leal Cavalcante

Authors
  1. José Alcides Almeida de Arruda
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  2. Rosanna Gómez
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  3. Verónica Bracho
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  4. Israel Leal Cavalcante
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  5. Ricardo Pérez-Alfonzo
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  6. Mariana Villarroel-Dorrego
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  7. Bruno Augusto Benevenuto de Andrade
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Corresponding author

Correspondence to Bruno Augusto Benevenuto de Andrade.

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The authors have no conflicts of interest to declare.

Ethical Approval

Data were collected in accordance with guidelines from the institutional Research Ethics Board.

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No identifier information is included in the case reports, and the study meets the waiver criteria for the institutional review board of Universidad Central de Venezuela.

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Written informed consent was obtained from the patients’ parents for data collection and publication of these case reports and accompanying images.

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de Arruda, J.A.A., Gómez, R., Bracho, V. et al. Congenital Melanotic Macule of the Tongue: Report of Two Cases and Literature Review. Head and Neck Pathol 17, 581–586 (2023). https://doi.org/10.1007/s12105-023-01530-4

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  • DOI: https://doi.org/10.1007/s12105-023-01530-4

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