Literature DB >> 8384909

Diagnosis of fructose-1,6-bisphosphatase deficiency using leukocytes: normal leukocyte enzyme activity in three female patients.

Y S Shin1.   

Abstract

In the past 5 years we have discovered 8 boys and 3 girls who suffer from fructose-1,6-bisphosphatase deficiency. Although they all showed the typical symptoms of the deficiency such as frequent vomiting, hypoglycemia, lactic acidosis, and hepatomegaly, the age at diagnosis varied from 2 months to 4 years. All the boys revealed the deficient enzyme activity in leukocytes but none of the girls. The liver biopsy was investigated in six patients to confirm the diagnosis. These results suggest the existence of tissue-specific isoenzymes for fructose-1,6-bisphosphatase possibly with a different gene origin.

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Year:  1993        PMID: 8384909     DOI: 10.1007/bf00179991

Source DB:  PubMed          Journal:  Clin Investig        ISSN: 0941-0198


  11 in total

1.  Clinical and biochemical observations on three cases of fructose-1,6-diphosphatase deficiency.

Authors:  A B Burlina; M Poletto; Y S Shin; F Zacchello
Journal:  J Inherit Metab Dis       Date:  1990       Impact factor: 4.982

2.  Biochemical and clinical observations in four patients with fructose-1,6-diphosphatase deficiency.

Authors:  P Bührdel; H J Böhme; L Didt
Journal:  Eur J Pediatr       Date:  1990-05       Impact factor: 3.183

3.  Purification of alpha and beta subunits of phosphorylase-b-kinase in human liver and cardiac muscle by affinity chromatography and immunodetection.

Authors:  T Podskarbi; W Endres; Y S Shin
Journal:  J Inherit Metab Dis       Date:  1991       Impact factor: 4.982

4.  Diagnosis of glycogen storage disease.

Authors:  Y S Shin
Journal:  J Inherit Metab Dis       Date:  1990       Impact factor: 4.982

5.  Fructose-1,6-diphosphatase deficiency: diagnosis using leukocytes and detection of heterozygotes with radiochemical and spectrophotometric methods.

Authors:  D Alexander; M Assaf; A Khudr; I Haddad; A Barakat
Journal:  J Inherit Metab Dis       Date:  1985       Impact factor: 4.982

6.  Fasting hypoglycaemia and metabolic acidosis associated with deficiency of hepatic fructose-1,6-diphosphatase activity.

Authors:  L Baker; A I Winegrad
Journal:  Lancet       Date:  1970-07-04       Impact factor: 79.321

7.  A simple assay for amylo-1,6-glucosidase to detect heterozygotes for glycogenosis type III in erythrocytes.

Authors:  Y S Shin; R Ungar; M Rieth; W Endres
Journal:  Clin Chem       Date:  1984-10       Impact factor: 8.327

8.  Glycerol-3-phosphate excretion in fructose-1,6-diphosphatase deficiency.

Authors:  S Krywawych; G Katz; A M Lawson; S Wyatt; D P Brenton
Journal:  J Inherit Metab Dis       Date:  1986       Impact factor: 4.982

9.  A radioisotopic method for fructose-1-phosphate aldolase assay that facilitates diagnosis of hereditary fructose intolerance.

Authors:  Y S Shin; V Moro; H Doliwa; W Endres
Journal:  Clin Chem       Date:  1983-11       Impact factor: 8.327

10.  Fructose-1,6-diphosphatase deficiency: glycerol excretion during fasting test.

Authors:  P A Dremsek; M Sacher; W Stögmann; R Gitzelmann; C Bachmann
Journal:  Eur J Pediatr       Date:  1985-07       Impact factor: 3.183

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  9 in total

Review 1.  Disorders of gluconeogenesis.

Authors:  G van den Berghe
Journal:  J Inherit Metab Dis       Date:  1996       Impact factor: 4.982

2.  Fructose-1,6-bisphosphatase deficiency: severe phenotype with normal leukocyte enzyme activity.

Authors:  G T Besley; J H Walter; M A Lewis; C R Chard; G M Addison
Journal:  J Inherit Metab Dis       Date:  1994       Impact factor: 4.982

3.  Diagnosis of fructose-1,6-bisphosphatase deficiency using cultured lymphocyte fraction: a secure and noninvasive alternative to liver biopsy.

Authors:  Y Kikawa; Y S Shin; M Inuzuka; E Zammarchi; M Mayumi
Journal:  J Inherit Metab Dis       Date:  2002-02       Impact factor: 4.982

4.  Novel mutations in patients with fructose-1,6-bisphosphatase deficiency.

Authors:  B Herzog; U Wendel; A A Morris; K Eschrich
Journal:  J Inherit Metab Dis       Date:  1999-04       Impact factor: 4.982

5.  Identification of genetic mutations in Japanese patients with fructose-1,6-bisphosphatase deficiency.

Authors:  Y Kikawa; M Inuzuka; B Y Jin; S Kaji; J Koga; Y Yamamoto; K Fujisawa; I Hata; A Nakai; Y Shigematsu; H Mizunuma; A Taketo; M Mayumi; M Sudo
Journal:  Am J Hum Genet       Date:  1997-10       Impact factor: 11.025

6.  Fructose 1,6-bisphosphatase deficiency: clinical, biochemical and genetic features in French patients.

Authors:  Elise Lebigot; Anaïs Brassier; Mokhtar Zater; Dilek Imanci; François Feillet; Patrice Thérond; Pascale de Lonlay; Audrey Boutron
Journal:  J Inherit Metab Dis       Date:  2015-01-20       Impact factor: 4.982

7.  Status epilepticus due to fructose-1,6-bisphosphatase deficiency caused by FBP1 gene mutation.

Authors:  Shiyue Mei; Chao Ma; Yibing Cheng; Suyun Qian; Zhipeng Jin
Journal:  Pediatr Investig       Date:  2019-06-25

8.  Fructose-1,6-diphosphatase deficiency: a treatable neurometabolic disorder.

Authors:  Mahesh Kamate; Milind Jambagi; Prashanth Gowda; Smita Sonoli
Journal:  BMJ Case Rep       Date:  2014-09-22

9.  International practices in the dietary management of fructose 1-6 biphosphatase deficiency.

Authors:  A Pinto; M Alfadhel; R Akroyd; Y Atik Altınok; S M Bernabei; L Bernstein; G Bruni; G Caine; E Cameron; R Carruthers; B Cochrane; A Daly; F de Boer; S Delaunay; A Dianin; M Dixon; E Drogari; S Dubois; S Evans; J Gribben; G Gugelmo; C Heidenborg; I Hunjan; I L Kok; B Kumru; A Liguori; D Mayr; E Megdad; U Meyer; R B Oliveira; A Pal; A Pozzoli; R Pretese; J C Rocha; S Rosenbaum-Fabian; J Serrano-Nieto; E Sjoqvist; C Timmer; L White; T van den Hurk; M van Rijn; H Zweers; M Ziadlou; A MacDonald
Journal:  Orphanet J Rare Dis       Date:  2018-01-25       Impact factor: 4.123

  9 in total

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