Document Detail

Beta-blocker therapy and hemophagocytic lymphohistiocytosis: a case report.
MedLine Citation:
PMID:  20634935     Owner:  NLM     Status:  In-Data-Review    
Objective. The aim of this paper is to describe a fatal case of hemophagocytic lymphohistiocytosis (HLH) in a patient with severe heart failure, who was treated with low-dose propranolol. Patient and Interventions. We report on a 7-month-old boy with Downs syndrome who was born with an unbalanced, left dominant atrioventricular septal defect and aortic coarctation. Despite coarctation repair and pulmonary artery banding he developed intractable heart failure and fever of unknown origin. Since he remained in heart failure he received a trial of low-dose propranolol to stabilize his cardiopulmonary status, which resulted in unexpected immunomodulatory effects. Measurements and Main Result. Immunoactivation was evidenced by high concentrations of procalcitonin, soluble CD 25, tumor necrosis factor alpha, and interleukin 6 and 8. Propranolol resulting in hepatic compromise as indicated by high lactate dehydrogenase and alanine aminotransferase levels. A therapeutic switch from propranolol to the beta(1)-receptor blocker metoprolol appeared to be instrumental in hemodynamic improvement and allowed discharge from hospital. However, the infant ultimately died from secondary inflammatory reactivation and intractable pulmonary obstructive disease. The autopsy results revealed HLH. Conclusion. Our case describes HLH secondary to heart failure and Downs syndrome. In this highly activated inflammatory state the beneficial hemodynamic effects of propranolol may be accompanied by immunomodulatory effects and the risk of acute liver failure. HLH occurs with a distinct pathophysiology, and specific treatment might be mandatory to increase the chance of survival.
C Müller; L B Mänhardt; C Willaschek; E M Schneider; E A Stuth; R Buchhorn
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Publication Detail:
Type:  Journal Article     Date:  2010-06-20
Journal Detail:
Title:  Cardiology research and practice     Volume:  2010     ISSN:  2090-0597     ISO Abbreviation:  Cardiol Res Pract     Publication Date:  2010  
Date Detail:
Created Date:  2010-07-16     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101516542     Medline TA:  Cardiol Res Pract     Country:  Egypt    
Other Details:
Languages:  eng     Pagination:  912757     Citation Subset:  -    
Department of Pediatrics, Caritas Krankenhaus, 97980 Bad Mergentheim, Germany.
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