Literature DB >> 27067077

Missed Newborn Screening Case of Carnitine Palmitoyltransferase-II Deficiency.

Andrew C Edmondson1,2, Jennifer Salant2, Lynne A Ierardi-Curto1,2, Can Ficicioglu3,4.   

Abstract

Carnitine palmitoyltransferase-II (CPT-II) deficiency can be detected through newborn screening with tandem mass spectrometry. We report a 4-year-old patient with rhabdomyolysis due to CPT-II deficiency, which was initially missed by newborn screening. The patient presented with a 2-day history of fevers, upper respiratory infection, diffuse myalgia, and tea-colored urine. Her medical history was notable for frequent diffuse myalgia when ill. She was demonstrated to have homozygous mutation c.338C>T, p. S113L in CPT2, which is typically found in the adult-onset, myopathic form of the disease. An unknown number of CPT-II deficient patients with normal newborn screening have not yet presented to medical care with the adult-onset, myopathic form of disease. We conclude that (1) not all cases of CPT-II deficiency are currently detected through newborn screening, even when blood is appropriately collected on day 2 of life and (2) CPT-II deficiency should be kept on the differential for patients presenting with rhabdomyolysis, even if the newborn screening results were normal.

Entities:  

Year:  2016        PMID: 27067077      PMCID: PMC5413452          DOI: 10.1007/8904_2016_528

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  18 in total

1.  Recurrent rhabdomyolysis and acute respiratory failure due to carnitine palmityltransferase deficiency.

Authors:  K H Smolle; P Kaufmann; R Gasser
Journal:  Intensive Care Med       Date:  2001-07       Impact factor: 17.440

2.  A near-miss: very long chain acyl-CoA dehydrogenase deficiency with normal primary markers in the initial well-timed newborn screening specimen.

Authors:  Inderneel Sahai; Joyce C Bailey; Roger B Eaton; Thomas Zytkovicz; David J Harris
Journal:  J Pediatr       Date:  2010-11-12       Impact factor: 4.406

3.  Pitfalls of neonatal screening for very-long-chain acyl-CoA dehydrogenase deficiency using tandem mass spectrometry.

Authors:  Ina Schymik; Michaela Liebig; Martina Mueller; Udo Wendel; Ertan Mayatepek; Arnold W Strauss; Ronald J A Wanders; Ute Spiekerkoetter
Journal:  J Pediatr       Date:  2006-07       Impact factor: 4.406

Review 4.  Long-term major clinical outcomes in patients with long chain fatty acid oxidation disorders before and after transition to triheptanoin treatment--A retrospective chart review.

Authors:  Jerry Vockley; Deborah Marsden; Elizabeth McCracken; Stephanie DeWard; Amanda Barone; Kristen Hsu; Emil Kakkis
Journal:  Mol Genet Metab       Date:  2015-06-18       Impact factor: 4.797

Review 5.  A diagnostic approach to recurrent myalgia and rhabdomyolysis in children.

Authors:  Eunice K Chan; Andrew J Kornberg; Monique M Ryan
Journal:  Arch Dis Child       Date:  2015-01-29       Impact factor: 3.791

Review 6.  Rhabdomyolysis.

Authors:  John M Sauret; George Marinides; Gordon K Wang
Journal:  Am Fam Physician       Date:  2002-03-01       Impact factor: 3.292

7.  Carnitine-palmitoyltransferase 2 deficiency: novel mutations and relevance of newborn screening.

Authors:  Sabine Illsinger; Thomas Lücke; Michael Peter; Jos P N Ruiter; Ronald J A Wanders; Marcus Deschauer; Ingrid Handig; Wim Wuyts; Anibh M Das
Journal:  Am J Med Genet A       Date:  2008-11-15       Impact factor: 2.802

Review 8.  Carnitine palmitoyltransferases 1 and 2: biochemical, molecular and medical aspects.

Authors:  Jean-Paul Bonnefont; Fatima Djouadi; Carina Prip-Buus; Stephanie Gobin; Arnold Munnich; Jean Bastin
Journal:  Mol Aspects Med       Date:  2004 Oct-Dec

9.  A retrospective ESI-MS/MS analysis of newborn blood spots from 18 symptomatic patients with organic acid and fatty acid oxidation disorders diagnosed either in infancy or in childhood.

Authors:  H Kobayashi; Y Hasegawa; M Endo; J Purevsuren; S Yamaguchi
Journal:  J Inherit Metab Dis       Date:  2007-07-20       Impact factor: 4.982

10.  Postanalytical tools improve performance of newborn screening by tandem mass spectrometry.

Authors:  Patricia L Hall; Gregg Marquardt; David M S McHugh; Robert J Currier; Hao Tang; Stephanie D Stoway; Piero Rinaldo
Journal:  Genet Med       Date:  2014-05-29       Impact factor: 8.822

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  5 in total

Review 1.  Carnitine palmitoyltransferase II deficiency with a focus on newborn screening.

Authors:  Go Tajima; Keiichi Hara; Miori Yuasa
Journal:  J Hum Genet       Date:  2018-12-04       Impact factor: 3.172

Review 2.  Laboratory diagnostic approaches in metabolic disorders.

Authors:  Ruben Bonilla Guerrero; Denise Salazar; Pranoot Tanpaiboon
Journal:  Ann Transl Med       Date:  2018-12

3.  Performance of Expanded Newborn Screening in Norway Supported by Post-Analytical Bioinformatics Tools and Rapid Second-Tier DNA Analyses.

Authors:  Trine Tangeraas; Ingjerd Sæves; Claus Klingenberg; Jens Jørgensen; Erle Kristensen; Gunnþórunn Gunnarsdottir; Eirik Vangsøy Hansen; Janne Strand; Emma Lundman; Sacha Ferdinandusse; Cathrin Lytomt Salvador; Berit Woldseth; Yngve T Bliksrud; Carlos Sagredo; Øyvind E Olsen; Mona C Berge; Anette Kjoshagen Trømborg; Anders Ziegler; Jin Hui Zhang; Linda Karlsen Sørgjerd; Mari Ytre-Arne; Silje Hogner; Siv M Løvoll; Mette R Kløvstad Olavsen; Dionne Navarrete; Hege J Gaup; Rina Lilje; Rolf H Zetterström; Asbjørg Stray-Pedersen; Terje Rootwelt; Piero Rinaldo; Alexander D Rowe; Rolf D Pettersen
Journal:  Int J Neonatal Screen       Date:  2020-06-27

4.  Utility of Genetic Testing for Confirmation of Abnormal Newborn Screening in Disorders of Long-Chain Fatty Acids: A Missed Case of Carnitine Palmitoyltransferase 1A (CPT1A) Deficiency.

Authors:  Leah Dowsett; Lauren Lulis; Can Ficicioglu; Sanmati Cuddapah
Journal:  Int J Neonatal Screen       Date:  2017-04-28

5.  Newborn Screening for Mitochondrial Carnitine-Acylcarnitine Cycle Disorders in Zhejiang Province, China.

Authors:  Duo Zhou; Yi Cheng; Xiaoshan Yin; Haixia Miao; Zhenzhen Hu; Jianbin Yang; Yu Zhang; Benqing Wu; Xinwen Huang
Journal:  Front Genet       Date:  2022-03-14       Impact factor: 4.599

  5 in total

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