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Early onset or syndromic epilepsy v8.139 TMEM167A Arina Puzriakova Tag Q3_25_promote_green was removed from gene: TMEM167A.
Early onset or syndromic epilepsy v8.134 TMEM167A Achchuthan Shanmugasundram reviewed gene: TMEM167A: Rating: GREEN; Mode of pathogenicity: ; Publications: ; Phenotypes: ; Mode of inheritance:
Early onset or syndromic epilepsy v8.134 TMEM167A Arina Puzriakova Source NHS GMS was added to TMEM167A.
Source Expert Review Green was added to TMEM167A.
Rating Changed from Amber List (moderate evidence) to Green List (high evidence)
Early onset or syndromic epilepsy v8.56 TMEM167A Arina Puzriakova Classified gene: TMEM167A as Amber List (moderate evidence)
Early onset or syndromic epilepsy v8.56 TMEM167A Arina Puzriakova Added comment: Comment on list classification: There is sufficient evidence to promote this gene to Green at the next GMS panel update - 6 unrelated cases with a concordant phenotype caused by biallelic variants in this gene.
Early onset or syndromic epilepsy v8.56 TMEM167A Arina Puzriakova Gene: tmem167a has been classified as Amber List (Moderate Evidence).
Early onset or syndromic epilepsy v8.55 TMEM167A Arina Puzriakova gene: TMEM167A was added
gene: TMEM167A was added to Early onset or syndromic epilepsy. Sources: Literature
Q3_25_promote_green tags were added to gene: TMEM167A.
Mode of inheritance for gene: TMEM167A was set to BIALLELIC, autosomal or pseudoautosomal
Publications for gene: TMEM167A were set to 40924476
Phenotypes for gene: TMEM167A were set to Microcephaly, epilepsy, and diabetes syndrome, MONDO:0100328
Review for gene: TMEM167A was set to GREEN
Added comment: PMID: 40924476 (2025) - 6 individuals from 6 unrelated families with biallelic variants in the TMEM167A gene identified by WGS. Clinical features in all affected individuals include neonatal diabetes (diagnosed <6 months) and severe microcephaly. 5/6 also had epilepsy in the neonatal period. TMEM167A is highly expressed in the human pancreas and brain. Both the depletion of TMEM167A in EndoC-βH1 cells and knock-in of the p.Val59Glu patient variant in iPSC-derived β cells increased β cells sensitivity to ER stress. The p.Val59Glu variant disrupted proinsulin trafficking to the Golgi and led to dysfunction in iPSC-β cells.
Sources: Literature