MEDNIK syndrome with a frame shift causing mutation in AP1S1 gene and literature review of the clinical features.

Faruk Incecik, Atil Bisgin, Mustafa Yılmaz

Journal: Metabolic brain disease 2019;33(6):2065-2068

PMID: 30244301

Abstract

MEDNIK syndrome is an autosomal recessive rare disease as one of the most recently described copper metabolism disorder characterized by intellectual disability, ichthyosis, hearing loss, peripheral neuropathy, enteropathy and keratodermia. Here in, we reported a case presented with ichthyosis and intellectual disability with MEDNIK syndrome that confirmed by mutation analysis in a Turkish child. She was finally diagnosed with MEDNIK syndrome by clinical findings, which were confirmed by molecular genetic testing. Sequencing of AP1S1 gene showed a homozygous insertion c.364dupG (NM_001283.4), which is predicted to cause a frameshift of the reading frame (p.D122Gfs*18). To our knowledge, this is the first case of MEDNIK syndrome from Turkey. Diagnosis of MEDNIK syndrome is still challenging and we hope that this case will contribute to further understanding.

Address: Department of Pediatric Neurology, Cukurova University Faculty of Medicine, Toros mah. 78186 sok. Yeşilpark Evleri, Kat: 7, no: 13, Adana, Turkey. [email protected].; Department of Medical Genetics, Cukurova University Faculty of Medicine, Adana, Turkey.; Department of Pediatric Immunology, Cukurova University Faculty of Medicine, Adana, Turkey.

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