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Wien Klin Wochenschr. 2017 Feb;129(3-4):141-144. doi: 10.1007/s00508-016-1124-4.

Intermittent low-dose bevacizumab in hereditary hemorrhagic telangiectasia : A case report.

Author information

1
IIIrd Medical Department with Haematology, Medical Oncology, Haemostaseology, Infectious Diseases and Rheumathology, Oncologic Center, Paracelsus Medical University, Muellner Hauptstraße 48, 5020, Salzburg, Austria.
2
Salzburg Cancer Research Institute with Laboratory of Immunological and Molecular Cancer Research and Center for Clinical Cancer and Immunology Trials, Salzburg, Austria.
3
Cancer Cluster Salzburg, Salzburg, Austria.
4
Department of Otorhinolaryngology, Paracelsus Medical University, Salzburg, Austria.
5
Blood Group Serology and Transfusion Medicine, Paracelsus Medical University, Salzburg, Austria.
6
Institute of Pathology, Paracelsus Medical University, Salzburg, Austria. d.neureiter@salk.at.

Abstract

BACKGROUND:

Hereditary hemorrhagic telangiectasia is an inherited autosomal dominant disease presenting with recurrent bleeding episodes and iron deficiency anemia due to vascular malformations. Hereditary hemorrhagic telangiectasia is associated with an increased risk of stroke, gastrointestinal bleeding and pulmonary hypertension and life expectancy is significantly reduced. Excess vascular endothelial growth factor (VEGF) plays a key role in the pathophysiology of the disease.

CASE PRESENTATION:

Here we report about a male patient with hereditary hemorrhagic telangiectasia presenting with pulmonary and central nervous system involvement experiencing repetitive nosebleeds, necessitating frequent local cauterization and transfusion of more than 100 units of packed red blood cells. After initiation of temporary therapy with the anti-VEGF antibody bevacizumab at a dosage of 1 mg/kg body weight every 2 weeks, the nose bleeding episodes and the epistaxis severity score significantly decreased and long-lasting transfusion independence was achieved. Reinitiation of low-dose bevacizumab after relapse again proved effective without any documented therapy-related adverse events. In comparison to other reported anti-VEGF antibody protocols in hereditary hemorrhagic telangiectasia, our treatment approach proved to be cost-efficient.

CONCLUSION:

Intermittent low-dose therapy with bevacizumab represents an effective and cost-efficient treatment option for transfusion-dependent patients with hereditary hemorrhagic telangiectasia.

KEYWORDS:

Anemia; Bevacizumab; Nosebleeds; Telangiectasia; Transfusion; VEGF

PMID:
27878613
PMCID:
PMC5318479
DOI:
10.1007/s00508-016-1124-4
[Indexed for MEDLINE]
Free PMC Article

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