Literature DB >> 4730202

Variant maple syrup urine disease in mother and daughter.

L A Zaleski, J Dancis, R P Cox, J Hutzler, W A Zaleski, A Hill.   

Abstract

Intermittent MSUD in a mother and her daughter is reported. Fibroblast cultures were studied for branched-chain keto acid decarboxylase and results show that the mother has approximately 12% while the daughter has 5% of the normal enzyme activity. Other key members in the family were also studied for enzyme activity. It appears that the child has inherited an abnormal gene from her homozygous mother and another abnormal gene from her heterozygous father.A classification based on the degree of residual enzyme activity and protein tolerance places the mother in grade III and the daughter in grade II category. Classical MSUD, where the enzyme activity is less than 2% of normal, belongs to grade I.

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Year:  1973        PMID: 4730202      PMCID: PMC1946859     

Source DB:  PubMed          Journal:  Can Med Assoc J        ISSN: 0008-4409            Impact factor:   8.262


  9 in total

1.  LATE MANIFESTING VARIANT OF BRANCHED-CHAIN KETOACIDURIA (MAPLE SYRUP URINE DISEASE).

Authors:  R KIIL; T ROKKONES
Journal:  Acta Paediatr       Date:  1964-07       Impact factor: 2.299

2.  MAPLE SYRUP URINE DISEASE. REPORT OF TWO CASES.

Authors:  D LONSDALE; R D MERCER; W R FAULKNER
Journal:  Am J Dis Child       Date:  1963-09

3.  [LEUCINOSIS: A VARIANT OF BRANCHED-CHAIN KETO-ACIDURIA].

Authors:  P DURAND; G M LAMEDICA; A M MARTINO
Journal:  Pediatrie       Date:  1965-03

4.  Clinical and biochemical observations on an apparently nonfatal variant of branched-chain ketoaciduria (maple syrup urine disease).

Authors:  M D MORRIS; B D LEWIS; P D DOOLAN; H A HARPER
Journal:  Pediatrics       Date:  1961-12       Impact factor: 7.124

5.  Thiamine-responsive maple-syrup-urine disease.

Authors:  C R Scriver; S Mackenzie; C L Clow; E Delvin
Journal:  Lancet       Date:  1971-02-13       Impact factor: 79.321

6.  A variant form of branched-chain keto aciduria.

Authors:  J L van der Horst; S K Wadman
Journal:  Acta Paediatr Scand       Date:  1971-09

7.  Biochemical studies on a variant of branched chain ketoaciduria in a 19-year-old female.

Authors:  M H Fischer; T Gerritsen
Journal:  Pediatrics       Date:  1971-11       Impact factor: 7.124

8.  A new variant of maple syrup urine disease (branched chain ketoaciduria). Clinical and biochemical evaluation.

Authors:  J D Schulman; T J Lustberg; J L Kennedy; M Museles; J E Seegmiller
Journal:  Am J Med       Date:  1970-07       Impact factor: 4.965

9.  Methylmalonic acidemia and vitamin B12 dependency.

Authors:  F Zacchello; R Tenconi
Journal:  Acta Paediatr Scand       Date:  1970-01
  9 in total
  5 in total

1.  Management of maple syrup urine disease in Canada. Committee for improvement of Hereditary Disease Management.

Authors: 
Journal:  Can Med Assoc J       Date:  1976-11-20       Impact factor: 8.262

2.  Maple syrup urine disease: branched-chain keto acid decarboxylation in fibroblasts as measured with amino acids and keto acids.

Authors:  J Dancis; J Hutzler; R P Cox
Journal:  Am J Hum Genet       Date:  1977-05       Impact factor: 11.025

3.  Successful domino liver transplantation in maple syrup urine disease using a related living donor.

Authors:  F H Feier; I K Miura; E A Fonseca; G Porta; R Pugliese; A Porta; I V D Schwartz; A V B Margutti; J S Camelo; S N Yamaguchi; A T Taveira; H Candido; M Benavides; V Danesi; T Guimaraes; M Kondo; P Chapchap; J Seda Neto
Journal:  Braz J Med Biol Res       Date:  2014-04-25       Impact factor: 2.590

4.  Maple syrup urine disease: two different forms within a single family.

Authors:  J Frézal; O Amédée-Manesme; G Mitchell; S Heuertz; F Rey; J Rey; J M Saudubray
Journal:  Hum Genet       Date:  1985       Impact factor: 4.132

5.  Inborn errors of metabolism. Vitamin-responsive genetic disease.

Authors:  S H Mudd
Journal:  J Clin Pathol Suppl (R Coll Pathol)       Date:  1974
  5 in total

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