Rare multiple congenital anomalies-hypotonia-seizures syndrome type 1 (MCAHS1) - the clinical and molecular summary.

Eur J Med Genet

Poznan University of Medical Sciences, Department of Medical Genetics, Poznan, Poland; Centers for Medical Genetics GENESIS, Poznan, Poland. Electronic address:

Published: January 2023

AI Article Synopsis

  • Multiple congenital anomalies-hypotonia-seizures syndrome type 1 (MCAHS1) is a rare genetic disease that causes symptoms like intellectual disability, low muscle tone, and seizures due to defects in glycosylphosphatidylinositol (GPI) biosynthesis.
  • A male patient with these symptoms underwent whole exome sequencing, revealing two harmful genetic variants in the PIGN gene, leading to the diagnosis of MCAHS1.
  • The case also highlighted additional symptoms like temporary macrosomia and advanced bone age, suggesting MCAHS1 may share traits with disorders related to overgrowth, which could aid clinicians in diagnosing similar cases.

Article Abstract

Multiple congenital anomalies-hypotonia-seizures syndrome type 1 (MCAHS1) is a rare autosomal recessive genetic disease belonging to glycosylphosphatidylinositols biosynthesis defects (GPIBD), a group of recessive disorders characterized by intellectual disability, hypotonia, and seizures. Glycosylphosphatidylinositols (GPIs) are glycolipids that anchor and remodel cell proteins. These processes are highly conserved and fundamental in the metabolism of all eukaryotes, including humans. Here, we have reported a male patient presenting with hypotonia, intellectual disability, and epilepsy, who underwent whole exome sequencing (WES). The analysis revealed the presence of two deleterious variants in PIGN that encodes GPI ethanolamine phosphate transferase-1 - one novel (c.1247_1251delAAGTG; p.Glu416Glyfs*22), and one that has been previously reported in the medical literature (c.1434+5G>A) resulting in MCAHS1. The detailed clinical assessment followed by the medical literature review also pointed out transient macrosomia and unreported in MCAHS1 advanced bone age and postnatal tall stature. These symptoms suggest that MCAHS1 shares a phenotypic overlap with disorders associated with overgrowth. To conclude, our case report and summary of the medical literature may be helpful for clinicians and geneticists who diagnose patients presenting with hypotonia accompanied by tall stature, advanced bone age, and transient macrosomia.

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http://dx.doi.org/10.1016/j.ejmg.2022.104668DOI Listing

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