Literature DB >> 1256470

Sucrase-isomaltase deficiency. Absence of an inactive enzyme variant.

G M Gray, K A Conklin, R R Townley.   

Abstract

Deficiency of sucrase-isomaltase, an intestinal enzyme complex that is essential for digestion of nutritionally important carbohydrates, appears to be inherited as an autosomal recessive in 0.2 per cent of North Americans. The molecular basis of the deficiency has not been elucidated. To characterize the enzyme defect quantitatively, papain-solubilized intestinal biopsies were analyzed for total enzyme protein by radioimmunoassay and for enzyme activity by hydrolytic assay. Normal intestine had a close correlation between immunologically reactive enzyme and enzymic activity. In contrast, seven patients with sucrase-isomaltase deficiency were found to have complete absence of the enzyme protein by radioimmunoassay even though up to 10 times more intestinal protein was present than with normal tissue. This absence of an inactive enzyme variant can be explained by a major (no-sense) mutation of the structural gene or by a complete repression of the regulatory mechanism that controls structural gene function.

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Year:  1976        PMID: 1256470     DOI: 10.1056/NEJM197604012941403

Source DB:  PubMed          Journal:  N Engl J Med        ISSN: 0028-4793            Impact factor:   91.245


  10 in total

1.  Sucrase-isomaltase (palatinase) deficiency diagnosed during adulthood.

Authors:  R E Ringrose; H Preiser; J D Welsh
Journal:  Dig Dis Sci       Date:  1980-05       Impact factor: 3.199

2.  Immunochemical studies on cultured fibroblasts from patients with inherited methylmalonic acidemia.

Authors:  J F Kolhouse; C Utley; W A Fenton; L E Rosenberg
Journal:  Proc Natl Acad Sci U S A       Date:  1981-12       Impact factor: 11.205

3.  Immunochemical studies on cultured fibroblasts from patients with homocystinuria due to cystathionine beta-synthase deficiency.

Authors:  F Skovby; J Kraus; C Redlich; L E Rosenberg
Journal:  Am J Hum Genet       Date:  1982-01       Impact factor: 11.025

4.  Basic defect in the expression of adenosine deaminase in ADA-SCID disease. II. Deficiency of ADA-CRM detected in heterozygote human-Chinese hamster cell hybrids.

Authors:  E Herbschleb-Voogt; J W Scholten; P Meera Khan
Journal:  Hum Genet       Date:  1983       Impact factor: 4.132

5.  Sucrase-isomaltase deficiency in humans. Different mutations disrupt intracellular transport, processing, and function of an intestinal brush border enzyme.

Authors:  H Y Naim; J Roth; E E Sterchi; M Lentze; P Milla; J Schmitz; H P Hauri
Journal:  J Clin Invest       Date:  1988-08       Impact factor: 14.808

6.  Combined assessment of intestinal disaccharidases in congenital asucrasia by differential urinary disaccharide excretion.

Authors:  D G Maxton; S D Catt; I S Menzies
Journal:  J Clin Pathol       Date:  1990-05       Impact factor: 3.411

7.  Congenital sucrase-isomaltase deficiency presenting with failure to thrive, hypercalcemia, and nephrocalcinosis.

Authors:  John W Belmont; Barbara Reid; William Taylor; Susan S Baker; Warren H Moore; Michael C Morriss; Susan M Podrebarac; Nancy Glass; I David Schwartz
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Review 8.  Irritable bowel syndrome and diet.

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Review 9.  Heterogeneity in Metabolic Responses to Dietary Fructose.

Authors:  Ruixue Hou; Chinmayee Panda; V Saroja Voruganti
Journal:  Front Genet       Date:  2019-10-31       Impact factor: 4.599

10.  Two Novel Mutations in the SI Gene Associated With Congenital Sucrase-Isomaltase Deficiency: A Case Report in China.

Authors:  Jianli Zhou; Yuzhen Zhao; Xia Qian; Yongwei Cheng; Huabo Cai; Moxian Chen; Shaoming Zhou
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  10 in total

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