Literature DB >> 2434882

Dihydrobiopterin synthesis defect: an adult with diurnal fluctuation of symptoms.

K Tanaka, M Yoneda, T Nakajima, T Miyatake, M Owada.   

Abstract

A deficiency of dihydrobiopterin synthesis was found in a 27-year-old man with mild mental retardation, rigid spasticity, hyperreflexia, dystonia, myoclonus, and delay in the initiation of action, since age 10. Symptoms improved after sleep. Urine contained large amounts of neopterin and a trace of biopterin. Dihydropteridine reductase activity in red blood cells was normal. CSF levels of HVA and 5-HIAA were low. Tetrahydrobiopterin administration lowered serum phenylalanine and improved the symptoms.

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Year:  1987        PMID: 2434882     DOI: 10.1212/wnl.37.3.519

Source DB:  PubMed          Journal:  Neurology        ISSN: 0028-3878            Impact factor:   9.910


  6 in total

Review 1.  Neurochemistry and defects of biogenic amine neurotransmitter metabolism.

Authors:  K Hyland
Journal:  J Inherit Metab Dis       Date:  1999-06       Impact factor: 4.982

2.  Successful treatment of childhood onset symptomatic dystonia with levodopa.

Authors:  N A Fletcher; P D Thompson; J W Scadding; C D Marsden
Journal:  J Neurol Neurosurg Psychiatry       Date:  1993-08       Impact factor: 10.154

3.  Tyrosine hydroxylase and levodopa responsive dystonia.

Authors:  N A Fletcher; I J Holt; A E Harding; T G Nygaard; J Mallet; C D Marsden
Journal:  J Neurol Neurosurg Psychiatry       Date:  1989-01       Impact factor: 10.154

Review 4.  Hereditary spastic paraparesis in adults associated with inborn errors of metabolism: a diagnostic approach.

Authors:  F Sedel; B Fontaine; J M Saudubray; O Lyon-Caen
Journal:  J Inherit Metab Dis       Date:  2007-10-22       Impact factor: 4.982

5.  Lessons from a remarkable family with dopa-responsive dystonia.

Authors:  G Harwood; R Hierons; N A Fletcher; C D Marsden
Journal:  J Neurol Neurosurg Psychiatry       Date:  1994-04       Impact factor: 10.154

Review 6.  Movement disorders and inborn errors of metabolism in adults: a diagnostic approach.

Authors:  F Sedel; J-M Saudubray; E Roze; Y Agid; M Vidailhet
Journal:  J Inherit Metab Dis       Date:  2008-05-30       Impact factor: 4.750

  6 in total

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