Literature DB >> 1564483

Neuropsychiatric manifestations of defect in mitochondrial beta oxidation response to riboflavin.

W J Triggs1, C R Roe, W J Rhead, S K Hanson, S N Lin, L J Willmore.   

Abstract

A 29 year old woman is described with severe hyperemesis gravidarum, atypical migraine, numerous admissions to hospital for psychiatric illness, non-epileptic seizures, and valproate-induced coma. Metabolic studies and measurement of [9,10(n)-3H]palmitate oxidation by cultured fibroblasts suggested a multiple acyl-CoA dehydrogenation disorder. Treatment with riboflavin abolished headaches and abnormal behaviour and normalised the plasma free carnitine level. Subtle defects in mitochondrial beta oxidation may be a treatable cause of disordered behaviour in adults.

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Year:  1992        PMID: 1564483      PMCID: PMC1014728          DOI: 10.1136/jnnp.55.3.209

Source DB:  PubMed          Journal:  J Neurol Neurosurg Psychiatry        ISSN: 0022-3050            Impact factor:   10.154


  12 in total

1.  Application of fast atom bombardment with tandem mass spectrometry and liquid chromatography/mass spectrometry to the analysis of acylcarnitines in human urine, blood, and tissue.

Authors:  D S Millington; D L Norwood; N Kodo; C R Roe; F Inoue
Journal:  Anal Biochem       Date:  1989-08-01       Impact factor: 3.365

2.  Juvenile multiple sclerosis-like episodes associated with a defect of mitochondrial beta oxidation.

Authors:  B R Powell; N G Kennaway; W J Rhead; C J Reece; T G Burlingame; N R Buist
Journal:  Neurology       Date:  1990-03       Impact factor: 9.910

3.  Plasma carnitine and renal-carnitine clearance during pregnancy.

Authors:  G Cederblad; L Fåhraeus; K Lindgren
Journal:  Am J Clin Nutr       Date:  1986-09       Impact factor: 7.045

4.  Carnitine metabolism in the fasting rat.

Authors:  E P Brass; C L Hoppel
Journal:  J Biol Chem       Date:  1978-04-25       Impact factor: 5.157

5.  Valproate-induced coma with ketosis and carnitine insufficiency.

Authors:  W J Triggs; T P Bohan; S N Lin; L J Willmore
Journal:  Arch Neurol       Date:  1990-10

6.  Complementation analysis of fatty acid oxidation disorders.

Authors:  A Moon; W J Rhead
Journal:  J Clin Invest       Date:  1987-01       Impact factor: 14.808

7.  Riboflavin concentration in maternal and cord blood in human pregnancy.

Authors:  N W Kirshenbaum; J Dancis; M Levitz; J Lehanka; B K Young
Journal:  Am J Obstet Gynecol       Date:  1987-09       Impact factor: 8.661

8.  Multiple acyl-CoA dehydrogenase deficiency occurring in pregnancy and caused by a defect in riboflavin metabolism in the mother. Study of a kindred with seven deaths in infancy: Value of riboflavin therapy in preventing this syndrome.

Authors:  J P Harpey; C Charpentier; S I Goodman; Y Darbois; G Lefèbvre; J Sebbah
Journal:  J Pediatr       Date:  1983-09       Impact factor: 4.406

9.  Glutaric acidemia type II: heterogeneity of clinical and biochemical phenotypes.

Authors:  J P Loehr; S I Goodman; F E Frerman
Journal:  Pediatr Res       Date:  1990-03       Impact factor: 3.756

10.  Recurrent hypoglycemia associated with glutaric aciduria type II in an adult.

Authors:  G Dusheiko; M C Kew; B I Joffe; J R Lewin; S Mantagos; K Tanaka
Journal:  N Engl J Med       Date:  1979-12-27       Impact factor: 91.245

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  5 in total

Review 1.  The Role of Lipidomics in Autism Spectrum Disorder.

Authors:  Afaf El-Ansary; Salvatore Chirumbolo; Ramesa Shafi Bhat; Maryam Dadar; Eiman M Ibrahim; Geir Bjørklund
Journal:  Mol Diagn Ther       Date:  2020-02       Impact factor: 4.074

2.  Clinical and genetical heterogeneity of late-onset multiple acyl-coenzyme A dehydrogenase deficiency.

Authors:  Sarah C Grünert
Journal:  Orphanet J Rare Dis       Date:  2014-07-22       Impact factor: 4.123

3.  Mood, Personality, and Behavior Changes During Treatment with Statins: A Case Series.

Authors:  Stephanie Cham; Hayley J Koslik; Beatrice A Golomb
Journal:  Drug Saf Case Rep       Date:  2016-12

Review 4.  Riboflavin Responsive Mitochondrial Dysfunction in Neurodegenerative Diseases.

Authors:  Tamilarasan Udhayabanu; Andreea Manole; Mohan Rajeshwari; Perumal Varalakshmi; Henry Houlden; Balasubramaniem Ashokkumar
Journal:  J Clin Med       Date:  2017-05-05       Impact factor: 4.241

5.  Clinical, Biochemical, and Genetic Heterogeneity in Glutaric Aciduria Type II Patients.

Authors:  Amanat Ali; Fatmah Saeed Ali Almesmari; Nahid Al Dhahouri; Arwa Mohammad Saleh Ali; Mohammed Ahmed Ali Mohamed Ahmed Aldhanhani; Ranjit Vijayan; Amal Al Tenaiji; Aisha Al Shamsi; Jozef Hertecant; Fatma Al Jasmi
Journal:  Genes (Basel)       Date:  2021-08-27       Impact factor: 4.096

  5 in total

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