Literature DB >> 646432

Homocystinuria in New South Wales.

B Wilcken, G Turner.   

Abstract

Homocystinuria was studied in 27 patients from 15 families in New South Wales. All 2 had biochemical findings consistent with cystathionine synthetase deficiency. One patient was ascertained by newborn screening, but the remaining index cases were detected because of symptoms: poor eyesight 6, mental retardation 3, thromboses 2, skeletal abnormalities 2, and urinary infection1. 9 patients, one-third of all cases, were mildly affected: either they had no features of the disease, or these did not occur until the late teens. Pyridoxine responsiveness was found in 8 sibships, and clinically there were two distinct kinds of response. For patients born in the decade 1960-69 the ascertainment rate for the total population was 1:58 000. The true incidence must be much higher. Our series indicates that homocystinuria occurs more frequently than has heretofore been thought, and that mild cases are common. It is likely that cases are often missed in current newborn screening programmes.

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Year:  1978        PMID: 646432      PMCID: PMC1545146          DOI: 10.1136/adc.53.3.242

Source DB:  PubMed          Journal:  Arch Dis Child        ISSN: 0003-9888            Impact factor:   3.791


  8 in total

1.  Metabolic abnormalities detected in a survey of mentally backward individuals in Northern Ireland.

Authors:  N A CARSON; D W NEILL
Journal:  Arch Dis Child       Date:  1962-10       Impact factor: 3.791

2.  HOMOCYSTINURIA: AN ENZYMATIC DEFECT.

Authors:  S H MUDD; J D FINKELSTEIN; F IRREVERRE; L LASTER
Journal:  Science       Date:  1964-03-27       Impact factor: 47.728

3.  Frequency of inborn errors of metabolism, especially PKU, in some representative newborn screening centers around the world: a collaborative study.

Authors: 
Journal:  Humangenetik       Date:  1975-12-23

4.  Homocystinuria. Reduced folate levels during pyridoxine treatment.

Authors:  B Wilcken; B Turner
Journal:  Arch Dis Child       Date:  1973-01       Impact factor: 3.791

5.  Combined vitamin responsiveness in homocystinuria.

Authors:  G Morrow; L A Barness
Journal:  J Pediatr       Date:  1972-11       Impact factor: 4.406

6.  Detection of abnormal sulphur-containing amino acid excretion in a mass urine-screening programme.

Authors:  B Wilcken; B Turner; D A Brown
Journal:  Med J Aust       Date:  1972-06-03       Impact factor: 7.738

7.  Studies of the mechanism of pyridoxine-responsive homocystinuria.

Authors:  M R Seashore; J L Durant; L E Rosenberg
Journal:  Pediatr Res       Date:  1972-03       Impact factor: 3.756

8.  HOMOCYSTINURIA DUE TO CYSTATHIONINE SYNTHETASE DEFICIENCY: THE MODE OF INHERITANCE.

Authors:  J D FINKELSTEIN; S H MUDD; F IRREVERRE; L LASTER
Journal:  Science       Date:  1964-11-06       Impact factor: 47.728

  8 in total
  9 in total

1.  Glutathionuria: gamma-glutamyl transpeptidase deficiency.

Authors:  E C Wright; J Stern; R Ersser; A D Patrick
Journal:  J Inherit Metab Dis       Date:  1980       Impact factor: 4.982

2.  Homocystinuria (cystathionine synthase deficiency). Results of treatment in late-diagnosed patients.

Authors:  H Gröbe
Journal:  Eur J Pediatr       Date:  1980-12       Impact factor: 3.183

3.  Urinary homocystine levels in a newborn infant with cystathionine synthase deficiency.

Authors:  T Watanabe; Y Kuroda; E Naito; M Ito; E Takeda; K Toshima; M Miyao; T Tomita; S Furukawa
Journal:  Eur J Pediatr       Date:  1987-07       Impact factor: 3.183

4.  The natural history of homocystinuria due to cystathionine beta-synthase deficiency.

Authors:  S H Mudd; F Skovby; H L Levy; K D Pettigrew; B Wilcken; R E Pyeritz; G Andria; G H Boers; I L Bromberg; R Cerone
Journal:  Am J Hum Genet       Date:  1985-01       Impact factor: 11.025

5.  CT of cerebral venous sinus thrombosis in a child with homocystinuria.

Authors:  F J Schwab; R G Peyster; C B Brill
Journal:  Pediatr Radiol       Date:  1987

6.  Ocular complications in homocystinuria--early and late treated.

Authors:  J P Burke; M O'Keefe; R Bowell; E R Naughten
Journal:  Br J Ophthalmol       Date:  1989-06       Impact factor: 4.638

7.  Changing incidence of neonatal hypermethioninaemia: implications for the detection of homocystinuria.

Authors:  P D Whiteman; B E Clayton; R S Ersser; P Lilly; J W Seakins
Journal:  Arch Dis Child       Date:  1979-08       Impact factor: 3.791

Review 8.  Guidelines for the diagnosis and management of cystathionine beta-synthase deficiency.

Authors:  Andrew A M Morris; Viktor Kožich; Saikat Santra; Generoso Andria; Tawfeg I M Ben-Omran; Anupam B Chakrapani; Ellen Crushell; Mick J Henderson; Michel Hochuli; Martina Huemer; Miriam C H Janssen; Francois Maillot; Philip D Mayne; Jenny McNulty; Tara M Morrison; Helene Ogier; Siobhan O'Sullivan; Markéta Pavlíková; Isabel Tavares de Almeida; Allyson Terry; Sufin Yap; Henk J Blom; Kimberly A Chapman
Journal:  J Inherit Metab Dis       Date:  2016-10-24       Impact factor: 4.982

9.  Classical homocystinuria: A common inborn error of metabolism? An epidemiological study based on genetic databases.

Authors:  Giovana R Weber Hoss; Fernanda Sperb-Ludwig; Ida V D Schwartz; Henk J Blom
Journal:  Mol Genet Genomic Med       Date:  2020-03-30       Impact factor: 2.183

  9 in total

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