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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