A 5-month-old girl was referred to stomatology service for evaluation of a nodule on the posterior dorsum of the tongue that had been present for several weeks. One of the parents' complaints was that this lesion was affecting breastfeeding. She was not feeding properly, and her weight had stagnated while her twin brother was gaining weight. The patient's past medical history was unremarkable. His twin brother did not have the lesion. Intraoral examination revealed a well-defined nodule, with a smooth surface and soft to palpation, approximately 1.5 cm in diameter (Figure 1). Therefore, an excisional biopsy under general anesthesia was proposed, and the specimen was sent for histopathological analysis. The day after procedure she resumed breastfeeding and eating properly. Microscopic analysis revealed a well-circumscribed lesion composed of a benign and disorganized proliferation of smooth muscle bundles, consisting of spindle-shaped cells with eosinophilic cytoplasm, elongated nuclei with blunt ends (“cigar-shaped”), fine chromatin, and inconspicuous nucleoli. The muscle bundles are arranged irregularly and crisscrossing, permeating the fibrous connective tissue (Figure 2). Given this, the diagnosis was leiomyomatous hamartoma. Hamartomas in general are classified as a tissue malformation, similar to true neoplasms, but composed of mature cells and native tissues of the anatomical site where they occur, often with a predominant element, such as neurovascular hamartomas, predominantly adipose hamartomas containing smooth muscle and fat, and predominantly smooth muscle hamartomas (leiomyomatous hamartomas LH) (Phoon Nguyen et al. 2018; Sánchez-Romero et al. 2019; de Jesus et al. 2025). Oral LHs (OLH) are considered rare alterations in the oral cavity, representing < 3% of lesions affecting the tongue (Kreiger et al. 2007; Alarcón-Sánchez et al. 2024). OLHs are more frequent in the midline of the dorsum of the tongue and incisive papilla of pediatric patients. To date, 66 cases of OLH have been reported, the age of the patients ranged from 1 month to 61 years (with a mean age of 6 years); the distribution between the sexes is similar. The surgical excision is the treatment employed for OHL, and recurrence is rare during follow-up of reported cases (Kreiger et al. 2007; Gao et al. 2022; Alarcón-Sánchez et al. 2024). Following surgical excision of the lesion, one month later, she weighed the same as her brother and she has been followed up for 8 months, with no signs of recurrence. Although rare, OLH should be included in the differential diagnosis of asymptomatic nodular lesions, without a history of trauma, affecting the tongue of pediatric patients. Giuliano Saraceni Issa Cossolin: conceptualization, data curation, formal analysis, writing – original draft, supervision, investigation, writing – review and editing. Heitor Albergoni Silveira: conceptualization, investigation, writing – original draft, data curation, supervision, formal analysis, writing – review and editing. Ana Paula Abreu Vieira Alves: supervision, investigation, validation, writing – review and editing. Carla Gabriela Ermacora: investigation, validation, writing – review and editing, supervision. José Burgos Ponce: data curation, formal analysis, conceptualization, writing – review and editing, writing – original draft, supervision, validation, investigation. Camila Lopes Cardoso: conceptualization, investigation, writing – original draft, validation, writing – review and editing, formal analysis, data curation, supervision. The Article Processing Charge for the publication of this research was funded by the Coordenação de Aperfeiçoamento de Pessoal de Nível Superior - Brasil (CAPES) (ROR identifier: 00x0ma614). The parents of the patient reported in this manuscript provided written informed consent for the publication of the case details. The authors declare no conflicts of interest. Data sharing not applicable to this article as no datasets were generated or analyzed during the current study.
Cossolin et al. (Fri,) studied this question.