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Tacrolimus in Gastrointestinal Bleeding in a Young Boy With Hereditary Hemorrhagic Telangiectasia
Jessica M Pruijsen1, Steven Kroon2, Johannes J Mager2
1From the Pediatric Gastroenterology and Hepatology, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands.
Hereditary hemorrhagic telangiectasia (HHT) is an autosomal dominant disease in which gastrointestinal bleeding is a rare presenting symptom in children. Gastrointestinal bleeding in children is treated locally by endoscopy. When a focus of bleeding cannot be reached by endoscopy, management of these patients can be challenging. Previous reports showed a favorable outcome of treatment with tacrolimus in an adult HHT patient with liver vascular malformations and epistaxis and in a HHT patient with pulmonary hypertension. We report the first pediatric HHT patient who benefited from tacrolimus treatment. Our case demonstrated a remarkable decline in blood transfusions and better quality of life during the period of tacrolimus treatment.
Hereditary hemorrhagic telangiectasia (HHT) is an autosomal dominant disease in which gastrointestinal bleeding is a rare presenting symptom in children. Gastrointestinal bleeding in children is treated locally by endoscopy. When a focus of bleeding cannot be reached by endoscopy, management of these patients can be challenging. Previous reports showed a favorable outcome of treatment with tacrolimus in an adult HHT patient with liver vascular malformations and epistaxis and in a HHT patient with pulmonary hypertension. We report the first pediatric HHT patient who benefited from tacrolimus treatment. Our case demonstrated a remarkable decline in blood transfusions and better quality of life during the period of tacrolimus treatment.
