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Living donor liver transplantation for Dorfman-Chanarin syndrome with 1 year follow-up: case report.

Abstract：

:A 27-year-old Japanese man underwent liver transplantation because of uncompensated cirrhosis due to Dorfman-Chanarin syndrome (DCS). At birth, the patient displayed ichthyosis and liver dysfunction. Moreover, mental retardation appeared and intracytoplasmic vacuoles were observed within peripheral blood neutrophils. A fatty liver was also noticed, leading to the diagnosis of DCS. When he was referred to our hospital, his American Society of Anesthesiologists score was 3. The findings of computed tomography showed liver atrophy, splenomegaly, and ascites. The Child-Pugh score was B, and the Model for End-stage Liver Disease score was 14. The pathophysiology was DCS with uncompensated liver cirrhosis. Therefore, living donor liver transplantation (LDLT) was performed from the patient's brother. The histological appearance of the resected liver revealed macrovesicular steatosis in most hepatocytes with excess fibrous tissue in the portal areas. These findings were compatible with nonalcoholic steatohepatitis. Although the patient's mental retardation and characteristic appearance have not improved, good liver function has been maintained since LDLT. An outpatient protocol liver biopsy performed at 12 months after LDLT did not show recurrence of macrovesicular steatosis.

journal_name

Transplant Proc

journal_title

Transplantation proceedings

authors

Takeda K,Tanaka K,Kumamoto T,Morioka D,Endo I,Togo S,Shimada H

doi

10.1016/j.transproceed.2010.07.105

subject

Has Abstract

pub_date

2010-11-01 00:00:00

pages

3858-61

issue

9

eissn

0041-1345

issn

1873-2623

pii

S0041-1345(10)01300-X

journal_volume

42

pub_type

杂志文章

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