Gastrointestinal Bleeding in Hereditary Hemorrhagic Telangiectasia Accompanied with Very Severe Thrombocytopenia.

Abstract

BACKGROUND Hereditary hemorrhagic telangiectasia (HHT) is a rare autosomal-dominant disorder characterized by recurrent epistaxis and gastrointestinal bleeding (GIB). To our knowledge, the management of GIB in HHT patients accompanied with very severe thrombocytopenia has not been previously reported. CASE REPORT A 66-year-old woman with HHT who experienced recurrent epistaxis for over 5 years was admitted to the hospital due to intermittent hematemesis and melena for 2 weeks. Following admission, her platelet count dropped significantly to 1×10⁹/L. Following supportive treatments, a gastroscopy was performed when the platelet count reached 23×10⁹/L. The gastroscopy revealed over 50 angioectatic spots with active bleeding. These spots were treated by argon plasma coagulation and the bleeding was stopped. She had a favorable prognosis following discharge. CONCLUSIONS This case highlights the rare coexistence of HHT, GIB, and very severe thrombocytopenia, offering insights into proton pump inhibitor (PPI)-induced thrombocytopenia. Immediate discontinuation of the involved PPI is advised. Platelet transfusion (<20×109/L) combined with recombinant human interleukin-11 is recommended for patients at risk of GIB. If PPI therapy is necessary, it is a novel and effective strategy to switch to another PPI with a different chemical structure, accompanied by close platelet monitoring.