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PMID: 17873122 Published · ppublish English Case Reports Journal Article Research Support, Non-U.S. Gov't

Antenatal mitochondrial disease caused by mitochondrial ribosomal protein (MRPS22) mutation.

Journal of medical genetics ·Vol. 44 ·No. 12 ·2007-12-00 ·Pages 784-6

Saada A, Shaag A, Arnon S, Dolfin T, Miller C, Fuchs-Telem D, Lombes A, Elpeleg O

Abstract

Three patients born to the same set of consanguineous parents presented with antenatal skin oedema, hypotonia, cardiomyopathy and tubulopathy. The enzymatic activities of multiple mitochondrial respiratory chain complexes were reduced in muscle. Marked reduction of 12s rRNA, the core of the mitochondrial small ribosomal subunit, was found in fibroblasts. Homozygosity mapping led to the identification of a mutation in the MRPS22 gene, which encodes a mitochondrial ribosomal protein. Transfection of the patient cells with wild-type MRPS22 cDNA increased the 12s rRNA content and normalised the enzymatic activities. Quantification of mitochondrial transcripts is advisable in patients with multiple defects of the mitochondrial respiratory chain.

MeSH Terms
Cardiomyopathy, Hypertrophic/congenital,genetics Cells, Cultured/metabolism Consanguinity Conserved Sequence Edema/congenital,genetics Fatal Outcome Female Fetal Diseases/diagnostic imaging,genetics Humans Infant, Newborn Kidney Diseases/congenital,genetics Mitochondria, Muscle/enzymology Mitochondrial Diseases/genetics,pathology Mitochondrial Myopathies/genetics Mitochondrial Proteins/genetics,physiology RNA, Ribosomal/metabolism Recombinant Fusion Proteins/physiology Ribosomal Proteins/genetics,physiology Transfection Ultrasonography
Chemicals
MRPS22 protein, human Mitochondrial Proteins RNA, Ribosomal RNA, ribosomal, 12S Recombinant Fusion Proteins Ribosomal Proteins
Authors & Affiliations
8 authors, click to expand affiliations / ORCID
Saada A
Metabolic Disease Unit, Hadassah - Hebrew University Medical Center, Jerusalem, 91120, Israel.
Shaag A
Arnon S
Dolfin T
Miller C
Fuchs-Telem D
Lombes A
Elpeleg O
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9 references, click to expand
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Article Info
Journal
Journal of medical genetics
Abbr.
J Med Genet
ISSN
1468-6244
Published
2007-12-00
Epub
2007-00-14
Pages
784-6
Language
English
Region
England
NLM ID
2985087R
PMCID
PMC2652816
Subset
IM
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