Literature DB >> 27581592

Primary Carnitine Deficiency - A Rare Treatable Cause of Cardiomyopathy and Massive Hepatomegaly.

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

Mesh:

Substances:

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


  11 in total

1.  Primary carnitine deficiency as a cause of metabolic leukoencephalopathy: Report of one case.

Authors:  Rohan R Mahale; Anish Mehta; A Timmappaya; Rangasetty Srinivasa
Journal:  Neurol India       Date:  2016 Jan-Feb       Impact factor: 2.117

2.  Carnitine levels in 26,462 individuals from the nationwide screening program for primary carnitine deficiency in the Faroe Islands.

Authors:  Jan Rasmussen; Olav W Nielsen; Nils Janzen; Morten Duno; Hannes Gislason; Lars Køber; Ulrike Steuerwald; Allan M Lund
Journal:  J Inherit Metab Dis       Date:  2013-05-08       Impact factor: 4.982

3.  Identification of mutations and evaluation of cardiomyopathy in Turkish patients with primary carnitine deficiency.

Authors:  M Kilic; R K Ozgül; T Coşkun; D Yücel; M Karaca; H S Sivri; A Tokatli; M Sahin; T Karagöz; A Dursun
Journal:  JIMD Rep       Date:  2011-09-22

4.  Genetic epidemiology of the carnitine transporter OCTN2 gene in a Japanese population and phenotypic characterization in Japanese pedigrees with primary systemic carnitine deficiency.

Authors:  A Koizumi; J Nozaki; T Ohura; T Kayo; Y Wada; J Nezu; R Ohashi; I Tamai; Y Shoji; G Takada; S Kibira; T Matsuishi; A Tsuji
Journal:  Hum Mol Genet       Date:  1999-11       Impact factor: 6.150

5.  Molecular spectrum of SLC22A5 (OCTN2) gene mutations detected in 143 subjects evaluated for systemic carnitine deficiency.

Authors:  Fang-Yuan Li; Ayman W El-Hattab; Erawati V Bawle; Richard G Boles; Eric S Schmitt; Fernando Scaglia; Lee-Jun Wong
Journal:  Hum Mutat       Date:  2010-08       Impact factor: 4.878

6.  Role of carnitine in disease.

Authors:  Judith L Flanagan; Peter A Simmons; Joseph Vehige; Mark Dp Willcox; Qian Garrett
Journal:  Nutr Metab (Lond)       Date:  2010-04-16       Impact factor: 4.169

7.  Diagnoses of newborns and mothers with carnitine uptake defects through newborn screening.

Authors:  Ni-Chung Lee; Nelson Leung-Sang Tang; Yin-Hsiu Chien; Chun-An Chen; Sho-Juan Lin; Pao-Chin Chiu; Ai-Chu Huang; Wuh-Liang Hwu
Journal:  Mol Genet Metab       Date:  2009-12-28       Impact factor: 4.797

8.  Systemic carnitine deficiency--a treatable inherited lipid-storage disease presenting as Reye's syndrome.

Authors:  P R Chapoy; C Angelini; W J Brown; J E Stiff; A L Shug; S D Cederbaum
Journal:  N Engl J Med       Date:  1980-12-11       Impact factor: 91.245

Review 9.  Primary carnitine deficiency and cardiomyopathy.

Authors:  Lijun Fu; Meirong Huang; Shubao Chen
Journal:  Korean Circ J       Date:  2013-12       Impact factor: 3.243

Review 10.  Systemic primary carnitine deficiency: an overview of clinical manifestations, diagnosis, and management.

Authors:  Pilar L Magoulas; Ayman W El-Hattab
Journal:  Orphanet J Rare Dis       Date:  2012-09-18       Impact factor: 4.123

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  2 in total

1.  Association of Abnormal Serum L-Carnitine Levels with Idiopathic Changes in Left Ventricular Geometry in Pediatric and Adolescent Patients.

Authors:  Mohsen Shahidi; Khaled Rahmani; Abdorrahim Afkhamzadeh
Journal:  Iran J Med Sci       Date:  2022-05

Review 2.  The Importance of the Fatty Acid Transporter L-Carnitine in Non-Alcoholic Fatty Liver Disease (NAFLD).

Authors:  Dragana Savic; Leanne Hodson; Stefan Neubauer; Michael Pavlides
Journal:  Nutrients       Date:  2020-07-22       Impact factor: 5.717

  2 in total

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