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Mendeliome v1.3322 CCDC188 Chirag Patel gene: CCDC188 was added
gene: CCDC188 was added to Mendeliome. Sources: Literature
Mode of inheritance for gene: CCDC188 was set to BIALLELIC, autosomal or pseudoautosomal
Publications for gene: CCDC188 were set to 41004021
Phenotypes for gene: CCDC188 were set to Male infertility due to acephalic spermatozoa, MONDO:0035153
Review for gene: CCDC188 was set to GREEN
Added comment: 2 patients from 2 unrelated families with acephalic spermatozoa syndrome. WES identified biallelic variants in CCDC188 gene (1 x homozygous, 1 x compound heterozygous). The variants (c.481C > T [p.Gln161*] and c.1022 + 1G > A [p. K325Afs*110]) were rare in gnomAD and segregated in the family with heterozygous carrier parents. Western blotting, RT-PCR, qPCR, and immunofluorescence showed depletion of CCDC188 protein (and SUN5 and PMFBP1 protein) in patient sperm. Mutations in SUN5 and PMFBP1 genes account for 75% patients with acephalic spermatozoa syndrome.
Sources: Literature
Mendeliome v1.3322 CCDC188 Chirag Patel gene: CCDC188 was added
gene: CCDC188 was added to Mendeliome. Sources: Literature
Mode of inheritance for gene: CCDC188 was set to BIALLELIC, autosomal or pseudoautosomal
Publications for gene: CCDC188 were set to 41004021
Phenotypes for gene: CCDC188 were set to Male infertility due to acephalic spermatozoa, MONDO:0035153
Review for gene: CCDC188 was set to GREEN
Added comment: 2 patients from 2 unrelated families with acephalic spermatozoa syndrome. WES identified biallelic variants in CCDC188 gene (1 x homozygous, 1 x compound heterozygous). The variants (c.481C > T [p.Gln161*] and c.1022 + 1G > A [p. K325Afs*110]) were rare in gnomAD and segregated in the family with heterozygous carrier parents. Western blotting, RT-PCR, qPCR, and immunofluorescence showed depletion of CCDC188 protein (and SUN5 and PMFBP1 protein) in patient sperm. Mutations in SUN5 and PMFBP1 genes account for 75% patients with acephalic spermatozoa syndrome.
Sources: Literature
Mendeliome v1.2883 PMFBP1 Lucy Spencer reviewed gene: PMFBP1: Rating: GREEN; Mode of pathogenicity: None; Publications: ; Phenotypes: Spermatogenic failure 31 MIM#618112; Mode of inheritance: BIALLELIC, autosomal or pseudoautosomal
Mendeliome v0.13254 PMFBP1 Zornitza Stark Marked gene: PMFBP1 as ready
Mendeliome v0.13254 PMFBP1 Zornitza Stark Gene: pmfbp1 has been classified as Green List (High Evidence).
Mendeliome v0.13254 PMFBP1 Zornitza Stark Phenotypes for gene: PMFBP1 were changed from to Male infertility with teratozoospermia due to single gene mutation, MONDO:0018394
Mendeliome v0.13253 PMFBP1 Zornitza Stark Publications for gene: PMFBP1 were set to
Mendeliome v0.13252 PMFBP1 Zornitza Stark Mode of inheritance for gene: PMFBP1 was changed from Unknown to BIALLELIC, autosomal or pseudoautosomal
Mendeliome v0.13251 PMFBP1 Zornitza Stark reviewed gene: PMFBP1: Rating: GREEN; Mode of pathogenicity: None; Publications: 33484382, 33452591, 32285443; Phenotypes: Male infertility with teratozoospermia due to single gene mutation, MONDO:0018394; Mode of inheritance: BIALLELIC, autosomal or pseudoautosomal
Mendeliome v0.0 PMFBP1 Zornitza Stark gene: PMFBP1 was added
gene: PMFBP1 was added to Mendeliome_VCGS. Sources: Expert Review Green,Victorian Clinical Genetics Services
Mode of inheritance for gene: PMFBP1 was set to Unknown