Thalidomide for treatment of gastrointestinal bleedings due to angiodysplasia: a case report in acquired von Willebrand syndrome and review of the literature.
BACKGROUND
Acquired von Willebrand syndrome is a rare bleeding disorder and treatment of the associated gastrointestinal (GI) bleeding due to angiodysplasia is challenging.


OBJECTIVE
The aim of this study was to present a new case on the successful use of thalidomide in a patient with acquired von Willebrand syndrome and recurrent angiodysplasia-related GI bleedings, and to conduct a literature review on the use of thalidomide in patients with GI angiodysplasia.


METHODS
A literature review was conducted, searching the electronic databases PubMed, Embase and Cochrane.


RESULTS
We present a 75-year-old woman with IgG MGUS-associated acquired von Willebrand syndrome (AVWS) who suffered from recurrent gastrointestinal (GI) bleedings and epistaxis. Treatment with immunoglobulins, desmopressin, Haemate-P and blood transfusions failed to achieve long-term haemostasis. Eventually, after these bleedings relapsed for almost a decade at 1- to 3-month intervals, thalidomide was prescribed. Since then, only one single nose bleed and one single melaena episode had occurred. She currently remains in remission of GI bleeds after 3 years and 3 months follow-up. The literature review revealed a beneficial effect of thalidomide in reducing GI bleeding due to angiodysplasia, including four case reports on inherited von Willebrand disease.


CONCLUSIONS
In conclusion, thalidomide can be a reasonable option for the treatment of recurrent GI bleeding due to angiodysplasia in AVWS and we report for the first time that refractory or relapsing GI bleeding in patients with AVWS can successfully be controlled with thalidomide.
Van Creveldkliniek, University Medical Center Utrecht, Utrecht, The Netherlands.
E T
ET
Engelen
Van Creveldkliniek, University Medical Center Utrecht, Utrecht, The Netherlands.
K P M
KP
van Galen
Van Creveldkliniek, University Medical Center Utrecht, Utrecht, The Netherlands.
R E G
RE
Schutgens
D000368:Aged; D016888:Angiodysplasia / Q000150:complications / Q000175:diagnosis*; D016208:Databases, Factual; D004844:Epistaxis / Q000209:etiology / Q000517:prevention & control; D005260:Female; D006471:Gastrointestinal Hemorrhage / Q000209:etiology* / Q000517:prevention & control; D006801:Humans; D008551:Melena / Q000209:etiology; D012008:Recurrence; D013792:Thalidomide / Q000627:therapeutic use; D014842:von Willebrand Diseases / Q000150:complications / Q000175:diagnosis*
D013792:Thalidomide
acquired von Willebrand; angiodysplasia; gastrointestinal bleed; monoclonal gammopathy of undetermined significance; thalidomide