Document Detail


Myeloid dysplasia in familial 3-methylglutaconic aciduria.
MedLine Citation:
PMID:  16462576     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
A kindred is reported with four members affected with neurodegenerative disorder and 3-methylglutaconic aciduria. Two siblings developed thrombocytopenia heralding a myelodysplastic syndrome; in one patient it evolved into acute myeloid leukemia with monosomy 7 in the marrow. The hematologic complications have hitherto not been previously reported in other cases of 3-methylglutaconic aciduria and are thus thought to represent a new disease entity. This family adds additional evidence to the genetic heterogeneity of Mendelian disorders in which the primary mutation may have a mutator effect that could give origin to myelodysplastic syndrome and acute myeloid leukemia through acquired chromosomal changes.
Authors:
Motti Haimi; Ronit Elhasid; Ruth Gershoni-Baruch; Shai Izraeli; Ronald J A Wanders; Hanna Mandel
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Journal of pediatric hematology/oncology     Volume:  28     ISSN:  1077-4114     ISO Abbreviation:  J. Pediatr. Hematol. Oncol.     Publication Date:  2006 Feb 
Date Detail:
Created Date:  2006-02-07     Completed Date:  2006-03-31     Revised Date:  2011-10-06    
Medline Journal Info:
Nlm Unique ID:  9505928     Medline TA:  J Pediatr Hematol Oncol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  69-72     Citation Subset:  IM    
Affiliation:
Department of Pediatric Hemato-Oncology, Meyer Children's Hospital, Rambam Medical Center, Haifa, Israel.
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MeSH Terms
Descriptor/Qualifier:
Child, Preschool
Chromosomes, Human, Pair 7 / genetics
Consanguinity
Ethnic Groups / genetics
Female
Glutarates / urine*
Humans
Infant
Israel
Leukemia, Myeloid, Acute / genetics
Male
Mental Retardation / genetics
Metabolism, Inborn Errors / complications*,  urine
Monosomy
Myelodysplastic Syndromes / genetics*
Pedigree
Thrombocytopenia / genetics
Chemical
Reg. No./Substance:
0/Glutarates; 5746-90-7/3-methylglutaconic acid
Comments/Corrections
Comment In:
J Pediatr Hematol Oncol. 2006 Feb;28(2):62-3   [PMID:  16462574 ]

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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