Life-threatening hypersplenism due to idiopathic portal hypertension in early childhood: case report and review of the literature.

Abstract:

BACKGROUND:Idiopathic portal hypertension (IPH) is a disorder of unknown etiology and is characterized clinically by portal hypertension, splenomegaly, and hypersplenism accompanied by pancytopenia. This study evaluates the pathogenic concept of the disease by a systematic review of the literature and illustrates novel pathologic and laboratory findings. CASE PRESENTATION:We report the first case of uncontrolled splenic hyperperfusion and enlargement with subsequent hypersplenism leading to life-threatening complications of IPH in infancy and emergent splenectomy. CONCLUSIONS:Our results suggest that splenic NO and VCAM-1, rather than ET-1, have a significant impact on the development of IPH, even at a very early stage of disease. The success of surgical interventions targeting the splenic hyperperfusion suggests that the primary defect in the regulation of splenic blood flow seems to be crucial for the development of IPH. Thus, beside other treatment options splenectomy needs to be considered as a prime therapeutic option for IPH.

journal_name

BMC Gastroenterol

journal_title

BMC gastroenterology

authors

Däbritz J,Worch J,Materna U,Koch B,Koehler G,Duck C,Frühwald MC,Foell D

doi

10.1186/1471-230X-10-122

subject

Has Abstract

pub_date

2010-10-20 00:00:00

pages

122

issn

1471-230X

pii

1471-230X-10-122

journal_volume

10

pub_type

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