Literature DB >> 21407263

Mild CFTR mutations and genetic predisposition to lactase persistence in cystic fibrosis.

Edyta Mądry1, Ewa Fidler, Agnieszka Sobczyńska-Tomaszewska, Aleksandra Lisowska, Patrycja Krzyżanowska, Andrzej Pogorzelski, Łukasz Minarowski, Beata Oralewska, Ewa Mojs, Ewa Sapiejka, Ryszard Marciniak, Dorota Sands, Anna Korzon-Burakowska, Jarosław Kwiecień, Jarosław Walkowiak.   

Abstract

Taking into account the reported incidence of hypolactasia in cystic fibrosis (CF) and the possible impact of milk products on nutritional status we aimed to assess the genetic predisposition to adult-type hypolactasia (ATH) and its incidence in CF. Single nucleotide polymorphism upstream of the lactase gene (LCT) was assessed in 289 CF patients. In subject with -13910C/C genotype (C/C) predisposing to ATH, hydrogen-methane breath test (BT) with lactose loading was conducted and clinical symptoms typical for lactose malabsorption were assessed. The percentage of CF patients with C/C was similar to that observed in healthy subjects (HS) (31.5 vs 32.5% ). Eleven out of 52 (24.5%) CF C/C patients had abnormal BT results. The recalculated frequency of lactose malabsorption was similar for the entire CF and HS populations (6.9 vs 7.2%). Similarly as in the control group, few CF patients have identified and linked to lactose consumption clinical symptoms. The frequency of LCT polymorphic variants in CF patients having and not having severe mutations of CFTR gene showed significant differences. The C allele was more frequent in homozygotes of the severe mutations than in patients carrying at least one mild/unknown mutation (P<0.0028) and in patients with at least one mild mutation (P < 0.0377). In conclusion, CF patients carrying mild CFTR mutations seem to have lower genetic predisposition to ATH. Lactose malabsorption due to ATH in CF is not more frequent than in the general population. Symptomatic assessment of lactose malabsorption in CF is not reliable.

Entities:  

Mesh:

Substances:

Year:  2011        PMID: 21407263      PMCID: PMC3137504          DOI: 10.1038/ejhg.2011.36

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   4.246


  22 in total

Review 1.  Cystic fibrosis adult care: consensus conference report.

Authors:  James R Yankaskas; Bruce C Marshall; Beth Sufian; Richard H Simon; David Rodman
Journal:  Chest       Date:  2004-01       Impact factor: 9.410

2.  Assessment of maldigestion in cystic fibrosis.

Authors:  Jaroslaw Walkowiak
Journal:  J Pediatr       Date:  2004-09       Impact factor: 4.406

3.  The cystic fibrosis heterozygote--advantage in surviving cholera?

Authors:  D M Rodman; S Zamudio
Journal:  Med Hypotheses       Date:  1991-11       Impact factor: 1.538

4.  Small intestinal brush border enzymes in cystic fibrosis.

Authors:  S Van Biervliet; E Eggermont; H Carchon; G Veereman; K Deboeck
Journal:  Acta Gastroenterol Belg       Date:  1999 Jul-Sep       Impact factor: 1.316

5.  Lactase deficiency in patients with cystic fibrosis.

Authors:  I Antonowicz; V Reddy; K T Khaw; H Shwachman
Journal:  Pediatrics       Date:  1968-09       Impact factor: 7.124

6.  Identification of a variant associated with adult-type hypolactasia.

Authors:  Nabil Sabri Enattah; Timo Sahi; Erkki Savilahti; Joseph D Terwilliger; Leena Peltonen; Irma Järvelä
Journal:  Nat Genet       Date:  2002-01-14       Impact factor: 38.330

7.  Distribution of disaccharidase activity in the small bowel of normal and lactase-deficient subjects.

Authors:  A D Newcomer; D B McGill
Journal:  Gastroenterology       Date:  1966-10       Impact factor: 22.682

8.  Adult-type hypolactasia and lactose malabsorption in Poland.

Authors:  Edyta Mądry; Aleksandra Lisowska; Jarosław Kwiecień; Ryszard Marciniak; Anna Korzon-Burakowska; Sławomira Drzymała-Czyż; Ewa Mojs; Jarosław Walkowiak
Journal:  Acta Biochim Pol       Date:  2010-12-10       Impact factor: 2.149

9.  Transcriptional regulation of the lactase-phlorizin hydrolase gene by polymorphisms associated with adult-type hypolactasia.

Authors:  M Kuokkanen; N S Enattah; A Oksanen; E Savilahti; A Orpana; I Järvelä
Journal:  Gut       Date:  2003-05       Impact factor: 23.059

10.  Lactose malabsorption and intolerance and peak bone mass.

Authors:  Michele Di Stefano; Graziamaria Veneto; Simona Malservisi; Loredana Cecchetti; Licia Minguzzi; Alessandra Strocchi; Gino Roberto Corazza
Journal:  Gastroenterology       Date:  2002-06       Impact factor: 22.682

View more
  2 in total

Review 1.  Adult lactose digestion status and effects on disease.

Authors:  Andrew Szilagyi
Journal:  Can J Gastroenterol Hepatol       Date:  2015-04

2.  Lactose malabsorption is a risk factor for decreased bone mineral density in pancreatic insufficient cystic fibrosis patients.

Authors:  Edyta Mądry; Beata Krasińska; Sławomira Drzymała-Czyż; Dorota Sands; Aleksandra Lisowska; Philip Grebowiec; Alina Minarowska; Beata Oralewska; Przemyslaw Mańkowski; Jerzy Moczko; Jarosław Walkowiak
Journal:  Eur J Hum Genet       Date:  2012-03-28       Impact factor: 4.246

  2 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.