Saada A, Shaag A, Arnon S, Dolfin T, Miller C, Fuchs-Telem D, Lombes A, Elpeleg O
Metabolic Disease Unit, Hadassah - Hebrew University Medical Center, Jerusalem, 91120, Israel.
J Med Genet. 2007 Dec;44(12):784-6. doi: 10.1136/jmg.2007.053116. Epub 2007 Sep 14.
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.
三名由同一对近亲父母所生的患者出现产前皮肤水肿、肌张力减退、心肌病和肾小管病。肌肉中多种线粒体呼吸链复合物的酶活性降低。在成纤维细胞中发现线粒体小核糖体亚基的核心——12s rRNA显著减少。纯合子定位导致鉴定出MRPS22基因中的一个突变,该基因编码一种线粒体核糖体蛋白。用野生型MRPS22 cDNA转染患者细胞可增加12s rRNA含量并使酶活性正常化。对于线粒体呼吸链存在多种缺陷的患者,建议对线粒体转录本进行定量分析。