Tacrolimus in Gastrointestinal Bleeding in a Young Boy With Hereditary Hemorrhagic Telangiectasia

JPGN Reports · 5 authors, 3 centres

AI SUMMARY

FIDELITY 100%

POPULATIONA pediatric patient with hereditary hemorrhagic telangiectasia (HHT) type 1 and gastrointestinal bleeding.

INTERVENTIONTacrolimus therapy with target trough levels of 2–4 µg/L.

COMPARISONClinical course was observed before, during, and after discontinuation and restart of tacrolimus.

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AGENT 01 · STRUCTURED SUMMARY

TRACE TO SOURCE

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TL;DR

This is a case report of a young boy with HHT type 1 and gastrointestinal bleeding that could not be managed by endoscopy. Treatment with tacrolimus led to a remarkable decrease in blood transfusion needs and an improvement in his quality of life.

Full summary

758 CHARS

This case report describes an 8-month-old boy diagnosed with HHT type 1 who presented with melena and iron deficiency anemia. Tacrolimus was initiated at age 2.5 years. During 14 months of tacrolimus therapy, gastrointestinal bleeding decreased in frequency, and the patient required no blood transfusions (though 3 iron infusions were needed). His quality of life improved significantly. When tacrolimus was stopped for 2 months due to parotitis, bleeding increased, requiring 3 blood transfusions and 1 iron transfusion, and his symptoms worsened. After restarting tacrolimus, his condition improved again. The mean interval between blood transfusions was 65 days on tacrolimus versus 34.5 days off tacrolimus. Adverse effects included mild ear infections.

PICO

PPOPULATION

A pediatric patient with hereditary hemorrhagic telangiectasia (HHT) type 1 and gastrointestinal bleeding.

IINTERVENTION

Tacrolimus therapy with target trough levels of 2–4 µg/L.

OOUTCOME

Frequency of gastrointestinal bleeding, need for blood and iron transfusions, and quality of life (abdominal pain, fatigue, behavioral problems).

STUDY TYPE

case_report

SPECIALTY

pediatrics

SUMMARISED BY

AI pipeline

FIDELITY CHECK

100% · A

ORIGINAL PAPER

PMC10191567

Tacrolimus in Gastrointestinal Bleeding in a Young Boy With Hereditary Hemorrhagic Telangiectasia

JPGN Reports

READ THE FULL PAPER

ABSTRACT

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.

CITATION EXPORT

Jessica M. Pruijsen, Steven Kroon, Johannes J. Mager et al.. Tacrolimus in Gastrointestinal Bleeding in a Young Boy With Hereditary Hemorrhagic Telangiectasia. JPGN Reports. (2021). https://doi.org/10.1097/PG9.0000000000000133

AUTHORS

5 · 3 CENTRES

Jessica M. Pruijsen, Steven Kroon, Johannes J. Mager, Laura B. Bungener, Hubert P. J. van der Doef

From the *Pediatric Gastroenterology and Hepatology, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands
†Department of Pulmonology, St. Antonius Hospital Nieuwegein, The Netherlands
‡Department of Laboratory Medicine, Transplantation Immunology, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands.