| Literature DB >> 35859960 |
Zahra Alsahlawi1,2, Zainab Fadhul1, Ali Mahmood3, Ali Mohamed4, Mohamed Khalil3, Emtithal Aljishi1,2.
Abstract
Carnitine palmitoyltransferase II (CPT II) deficiency is a rare genetic metabolic disorder. Three forms of the disease have been described: the lethal neonatal form, the severe infantile hepatocardiomuscular form, and the myopathic form. We report a case of the infantile form of CPT II deficiency with a novel mutation. Our patient is a seven-year-old Bahraini male who was investigated by the pediatric metabolic team following the sudden death of his twin sister in infancy. A fatty acid metabolic disorder was suspected based on his echocardiogram and tandem mass spectrometry (TMS) findings. Genetic analysis was initially inconclusive. Nonetheless, he was started on a fat-free diet, L-carnitine, and medium-chain triglycerides (MCT). At nearly two years of age, the patient had a metabolic crisis precipitated by a viral illness. TMS during this time was consistent with CPT II deficiency. Sanger sequencing then identified the presence of the variant c.161T>G (p.ille54Ser) in a homozygous state, confirming the diagnosis. Although this mutation has not been reported before in previous literature concerning CPT II deficiency, it is extremely likely that this mutation is pathogenic. Although the initial work-up of the patient was inconclusive, our clinical judgment was paramount in managing the patient.Entities:
Keywords: carnitine palmitoyltransferase ii deficiency; l-carnitine; metabolic disease; novel mutation; pediatric genetic disease; preimplantation genetic diagnosis
Year: 2022 PMID: 35859960 PMCID: PMC9288837 DOI: 10.7759/cureus.26043
Source DB: PubMed Journal: Cureus ISSN: 2168-8184