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Hereditary Hemorrhagic Telangiectasia of the Tongue: A Rare Case Report

Year: 2026

Type: article

Abstract: Hereditary hemorrhagic telangiectasia (HHT) is a rare autosomal dominant vascular disorder that may involve the oral mucosa, providing important diagnostic clues for dental practitioners. This report describes a 54-year-old female presenting with recurrent bleeding from multiple red-purple telangiectatic papules on the dorsal surface of the tongue. Her history included recurrent epistaxis, severe iron deficiency anemia, pulmonary involvement, and a positive family history. The lesions blanched on diascopy. Symptomatic management using electrocautery excision under local anesthesia with prophylactic administration of oral amoxicillin (2 g) was performed. Histopathological examination demonstrated thin-walled vascular spaces containing erythrocytes beneath the epithelium, confirming the diagnosis of HHT. Although postoperative healing was uneventful with immediate bleeding control, recurrence occurred after 1 year, necessitating repeat surgery. This case highlights the progressive and recurrent nature of oral HHT lesions, the importance of early detection, and the need for careful dental management, including antibiotic prophylaxis before invasive procedures associated with a risk of bacteremia in patients with pulmonary involvement.

Source: Clinical Case Reports

DOI: 10.1002/ccr3.73883

Authors: Saede Atarbashi‐Moghadam, Ali Lotfi, Fazele Atarbashi‐Moghadam, Mostafa Alam, Milad Baseri, Mohammadreza Kashefi Baher

Institutions: Shahid Beheshti University, Shahid Beheshti University of Medical Sciences, Chamran Hospital

Language: English

Topic: Vascular Anomalies and Treatments

Subfield: Genetics (clinical)

Field: Medicine

Domain: Health Sciences

SDG: Good health and well-being

Open Access status: gold

Estimated APC paid: $1,820