Integral Role of the Mitochondrial Ribosome in Supporting Ovarian Function: MRPS7 Variants in Syndromic Premature Ovarian Insufficiency

Genes (Basel). 2022 Nov 14;13(11):2113. doi: 10.3390/genes13112113.

Abstract

The mitochondrial ribosome is critical to mitochondrial protein synthesis. Defects in both the large and small subunits of the mitochondrial ribosome can cause human disease, including, but not limited to, cardiomyopathy, hypoglycaemia, neurological dysfunction, sensorineural hearing loss and premature ovarian insufficiency (POI). POI is a common cause of infertility, characterised by elevated follicle-stimulating hormone and amenorrhea in women under the age of 40. Here we describe a patient with POI, sensorineural hearing loss and Hashimoto's disease. The co-occurrence of POI with sensorineural hearing loss indicates Perrault syndrome. Whole exome sequencing identified two compound heterozygous variants in mitochondrial ribosomal protein 7 (MRPS7), c.373A>T/p.(Lys125*) and c.536G>A/p.(Arg179His). Both novel variants are predicted to be pathogenic via in-silico algorithms. Variants in MRPS7 have been described only once in the literature and were identified in sisters, one of whom presented with congenital sensorineural hearing loss and POI, consistent with our patient phenotype. The other affected sister had a more severe disease course and died in early adolescence due to liver and renal failure before the reproductive phenotype was known. This second independent report validates that variants in MRPS7 are a cause of syndromic POI/Perrault syndrome. We present this case and review the current evidence supporting the integral role of the mitochondrial ribosome in supporting ovarian function.

Keywords: MRPS7; Perrault syndrome; mitochondrial disease; mitochondrial ribosome; ovarian dysfunction; premature ovarian insufficiency.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Female
  • Gonadal Dysgenesis, 46,XX* / genetics
  • Gonadal Dysgenesis, 46,XX* / pathology
  • Hearing Loss, Sensorineural* / genetics
  • Hearing Loss, Sensorineural* / pathology
  • Humans
  • Mitochondrial Proteins / genetics
  • Mitochondrial Ribosomes / pathology
  • Primary Ovarian Insufficiency* / genetics
  • Ribosomal Proteins / genetics

Substances

  • Ribosomal Proteins
  • Mitochondrial Proteins

Supplementary concepts

  • Gonadal dysgenesis XX type deafness

Grants and funding

This work was supported by a CHU Rennes grant (Appel à Projets Innovations 2019 to S.J.), an Australian National Health and Medical Research Council (NHMRC) program grant (1074258 to A.H.S.), NHMRC fellowships (1054432 to E.J.T., 1062854 to A.H.S.), a Suzi Carp postdoctoral scholarship (to E.J.T.) The research conducted at the Murdoch Children’s Research Institute was supported by the Victorian government’s operational infrastructure support program. The Chair in Genomic Medicine awarded to J.C. is generously supported by The Royal Children’s Hospital Foundation.