| Literature DB >> 27581592 |
Shivani Deswal1, Sunita Bijarnia-Mahay2, Vinamr Manocha3, Keiichi Hara4, Yosuke Shigematsu5, Renu Saxena2, Ishwar C Verma2.
Abstract
Systemic primary carnitine deficiency (CDSP) is a rare autosomal recessive disorder caused by a defect in plasma membrane uptake of carnitine due to SLC22A5 gene mutations. A nine-mo-old boy presented with hypertrophic cardiomyopathy, massive hepatomegaly and jaundice. Metabolic testing revealed very low free carnitine levels. Genetic analysis using Sanger sequencing method revealed compound heterozygous mutations in SLC22A5 gene, c. 1354 G > A (p. Glu452Lys, previously reported) and c.231_234del (novel frame-shift). Oral carnitine supplementation resulted in improved clinical outcome with ejection fraction to 75 % and normalization of liver size and enzymes after 3 mo.Entities:
Keywords: Cardiomyopathy; Carnitine; Hepatomegaly; Novel; SLC22A5
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Year: 2016 PMID: 27581592 DOI: 10.1007/s12098-016-2227-7
Source DB: PubMed Journal: Indian J Pediatr ISSN: 0019-5456 Impact factor: 1.967