Long-term treatment with thalidomide for severe recurrent hemorrhage from intestinal angiodysplasia in Glanzmann Thrombasthenia

Abstract

Gastrointestinal angiodysplasia (GIA) is the most common cause of occult gastrointestinal bleeding (GIB) requiring often hospitalization and transfusions, especially in patients with hemorrhagic disorders. Thalidomide, impairing neo-angiogenesis, has been successfully used in the management of bleeding in patients with GIA and in particular in patients with inherited bleeding disorders. Only one case of short-term treatment with thalidomide in a patient with Glanzmann thrombasthenia (GT) and recurrent GIB due to GIA has been reported so far.

We report the case of a woman with GT developing high frequency recurrent GIB due to GIA requiring repeated blood and platelet transfusions, who was treated with thalidomide obtaining a striking and stable reduction of GIB and of the requirement of platelet and blood transfusions for over 5 years. Moreover, we raise the suspicion that the association between GT and GIA may not be fortuitous.

Keywords:

• Gastrointestinal bleeding
• Glanzmann Thrombasthenia
• thalidomide

Acknowledgements

This work was supported in part by a fellowship to L.B from Fondazione Umberto Veronesi. The contribution of the following BAT-VAL study investigators is gratefully acknowledged: Ibrahim Eker (Gulhane Military Medical Faculty, Ankara, Turkey), Benilde Cosmi (S.Orsola -Malpighi University Hospital, University of Bologna, Italy), Munira Borhany and Madiha Abid (Department of Hematology, Haemostasis and Thrombosis at National Institute of Blood Disease and Bone Marrow Transplantation, Karachi, Pakistan), Paula James (Department of Medicine, Queen’s University, Kingston, Ontario, Canada), Marie Christine Alessi (Centre for CardioVascular and Nutrition research C2VN, INSERM 1263, INRA 1260, Marseille, France), Erica De Candia (Hemostasis and Thrombosis Unit, Fondazione Policlinico Agostino Gemelli IRCCS, Rome, Italy) and Cristina Santoro (Hematology, Azienda Ospedaliera Universitaria Policlinico Umberto I, Rome, Italy).

Authors’ contributions

FP, LB, TF, GZG analyzed and interpreted data; PG and FC contributed the patient for the study; FP and LB wrote the manuscript; PG and FC critically revised the manuscript.

Disclosure statement

The authors declare no competing financial interests

Additional information

Funding

This work was supported by the Fondazione Umberto Veronesi.