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Peer-reviewedCC BYClinical case reports

Hereditary Hemorrhagic Telangiectasia of the Tongue: A Rare Case Report.

  • Saede Atarbashi-Moghadam1
  • Ali Lotfi1
  • Fazele Atarbashi-Moghadam2
  • Mostafa Alam3
  • Milad Baseri3
  • Mohammadreza Kashefi Baher4
4 affiliations
  1. 1Department of Oral and Maxillofacial Pathology School of Dentistry, Shahid Beheshti University of Medical Sciences Tehran Iran.
  2. 2Department of Periodontology School of Dentistry, Shahid Beheshti University of Medical Sciences Tehran Iran.
  3. 3Department of Oral and Maxillofacial Surgery School of Dentistry, Shahid Beheshti University of Medical Sciences Tehran Iran.
  4. 4Health Research Center, Chamran Hospital Tehran Iran.
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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.

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Cite

BibTeXRIS
BibTeX
@article{atarbashimoghadam2026hereditary,
  title = {{Hereditary Hemorrhagic Telangiectasia of the Tongue: A Rare Case Report.}},
  author = {Saede Atarbashi-Moghadam and Ali Lotfi and Fazele Atarbashi-Moghadam and Mostafa Alam and Milad Baseri and Mohammadreza Kashefi Baher},
  journal = {Clinical case reports},
  year = {2026},
  month = nov,
  doi = {10.1002/ccr3.73883},
  url = {https://europepmc.org/article/MED/42997498},
  note = {CC BY},
}
RIS
TY  - JOUR
TI  - Hereditary Hemorrhagic Telangiectasia of the Tongue: A Rare Case Report.
AU  - Saede Atarbashi-Moghadam
AU  - Ali Lotfi
AU  - Fazele Atarbashi-Moghadam
AU  - Mostafa Alam
AU  - Milad Baseri
AU  - Mohammadreza Kashefi Baher
PY  - 2026
DA  - 2026/11/06
JO  - Clinical case reports
DO  - 10.1002/ccr3.73883
UR  - https://europepmc.org/article/MED/42997498
AB  - 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.
SN  - 2050-0904
ER  -