Giant sublingual hamartoma with medial cleft tongue: a case report and literature review

Abstract

Hamartomas commonly occur in respiratory and digestive organs, such as the lungs, pancreas, and liver; they rarely occur in the oral cavity, especially in the sublingual region. This report describes a 5-month-old boy who presented with a giant sublingual hamartoma and medial cleft tongue. He underwent corrective operations at 5 months, 11 months, and 31 months of age. Histopathological analysis revealed features suggestive of hamartoma. There have been no signs of recurrence. The boy exhibited normal speech development at 3 years of age; all other oral functions were unaffected at that time. This report includes a review of relevant literature. The findings in this report and previous literature suggest that a multidisciplinary approach, carefully planned staged surgery, and rehabilitation are needed to achieve favorable outcomes in patients with hamartoma in the oral cavity.

Oral hamartoma with an advanced elevation of the tongue

“…Forty-five oral hamartomas were reported in PubMed from 1990 to 2020 and two of them were located sublingually and one of those was as large as the one described in the present patient. 2 In our H&E examination, the skeletal muscles bundles had proliferated, with only slight ossification and calcification observed. Ossification was also found in the aforementioned large hamartoma.…”

Multiple hamartomas of the oral cavity with bifid tongue and cleft palate: A unique observation

“…[5] According to the published literature, most hamartomas of the oral cavity are multifocal, relatively small, and asymptomatic, and have been frequently linked to present as a manifestation of the oro-facial-digitalsyndrome. [2] Our patient presented with feeding difficulties at birth resulting from the multifocal, relatively large lesions that obstructed the oral cavity and thus necessitated urgent surgical intervention to obviate any possible airway problems. As a thorough evaluation did not reveal any associated defects, we consider the index patient to have developed the malformation sporadically.…”

“…[1] They develop most frequently in the lung, pancreas, spleen, liver, and kidney, with only a rare occurrence in the oral cavity. [1,2] Oral hamartomas can develop as isolated lesions, or in association with other systemic syndromes. [2,3] Multiple hamartomas that develop in the same patient are usually referred to as hamartomatosis or pleiotropic hamartomas.…”

“…[1,2] Oral hamartomas can develop as isolated lesions, or in association with other systemic syndromes. [2,3] Multiple hamartomas that develop in the same patient are usually referred to as hamartomatosis or pleiotropic hamartomas. [1] Herein, we present a unique association of bifid tongue, and cleft palate with lingual and palatal hamartomas in a newborn, describing its clinical presentation and surgical management.…”

Hamartomatous lesion at the base of tongue: A rare entity

“…4 The findings in this report and previous literature suggests that hamartoma of the oral cavity requires a careful and multistep approach to achieve a favorable outcome and easy recovery of the patients. 6…”