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Pulmonary Lymphomatoid Granulomatosis in Griscelli Syndrome Type 2.
MedLine Citation:
PMID:  22111599     Owner:  NLM     Status:  Publisher    
Abstract/OtherAbstract:
Abstract Griscelli syndrome type 2 (GS2) is a rare autosomal-recessive disorder associated with a RAB27A gene mutation, and clinically manifesting as hypopigmentation, disseminated chronic encephalitis, and severe immunological disorders characterized by an accelerated hematological phase, also referred to as hemophagocytic syndrome (HS), or hemophagocytic lymphohistiocytosis (HLH). The authors report the diagnosis of GS2 in an 11-year-old girl with hypopigmentation, immunodeficiency, hepatosplenomegaly, severe neurological impairments, and fatal multiorgan failure. In this patient a diagnosis of pulmonary lymphomatoid granulomatosis (LG), an Epstein-Barr virus (EBV)-related lymphoproliferative disorder, was established from radiological and histological findings. Although EBV-related malignancies are common in immunocompromised patients, this is the first report of a diagnosis of pulmonary LG in a patient with GS2.
Authors:
Aleksandra Szczawinska-Poplonyk; Zdzislawa Kycler; Anna Breborowicz; Maja Klaudel-Dreszler; Malgorzata Pac; Maria Zegadlo-Mylik; Renata Langfort
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Publication Detail:
Type:  JOURNAL ARTICLE     Date:  2011-11-23
Journal Detail:
Title:  Viral immunology     Volume:  -     ISSN:  1557-8976     ISO Abbreviation:  -     Publication Date:  2011 Nov 
Date Detail:
Created Date:  2011-11-24     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  8801552     Medline TA:  Viral Immunol     Country:  -    
Other Details:
Languages:  ENG     Pagination:  -     Citation Subset:  -    
Affiliation:
1 Department of Pediatric Pneumonology, Allergology, and Clinical Immunology, 3rd Chair of Pediatrics, Poznan University of Medical Sciences , Poznan, Poland .
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