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Syndromic sensorineural deafness due to combined oxidative phosphorylation defect

MedGen UID:
976585
Concept ID:
CN294158
Disease or Syndrome
Synonyms: Syndromic sensorineural deafness due to COXPD; Syndromic sensorineural hearing loss due to COXPD
Modes of inheritance:
Autosomal recessive inheritance
MedGen UID:
141025
Concept ID:
C0441748
Intellectual Product
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on one of the autosomes (i.e., the human chromosomes 1-22) in which a trait manifests in individuals with two pathogenic alleles, either homozygotes (two copies of the same mutant allele) or compound heterozygotes (whereby each copy of a gene has a distinct mutant allele).
 
Orphanet: ORPHA457223

Definition

A rare mitochondrial disease characterized by a variable phenotype comprising congenital sensorineural deafness, intermittent or persistent hypoglycemia, and hepatic and renal dysfunction potentially progressing to organ failure. Serum lactate levels are variably increased, deficiency of mitochondrial respiratory chain complexes I, III, and IV is observed in the liver and in fibroblasts. [from ORDO]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVSyndromic sensorineural deafness due to combined oxidative phosphorylation defect

Recent clinical studies

Diagnosis

Alsharhan H, Muraresku C, Ganetzky RD
Am J Med Genet A 2021 Aug;185(8):2519-2525. Epub 2021 May 19 doi: 10.1002/ajmg.a.62344. PMID: 34008913

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