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Familial prevalence of coeliac disease: a twenty-year follow-up study
Linköping University, Department of Clinical and Experimental Medicine, Pediatrics. Linköping University, Faculty of Health Sciences.
Linköping University, Department of Clinical and Experimental Medicine, Pediatrics. Linköping University, Faculty of Health Sciences.
Linköping University, Department of Molecular and Clinical Medicine, Pediatrics. Linköping University, Faculty of Health Sciences.
Linköping University, Department of Clinical and Experimental Medicine, Pediatrics. Linköping University, Faculty of Health Sciences.
2003 (English)In: Scandinavian Journal of Gastroenterology, ISSN 0036-5521, E-ISSN 1502-7708, Vol. 38, no 1, 61-65 p.Article in journal (Refereed) Published
Abstract [en]

BACKGROUND:

The genetic predisposition of coeliac disease (CD) is well known. Previous studies of first-degree relatives of coeliac patients have shown that as many as 10% have the disease. In 1981, we published a study in which all first-degree relatives of 32 index patients with CD were investigated by small-bowel biopsy. We found 2 relatives (2%) with CD. The present study is a re-investigation of all first-degree relatives of the same index patients performed 20-25 years after the first study to reveal any new cases of CD in this high-risk population.

METHODS:

All 120 first-degree relatives were screened for CD by means of serological markers of CD. The relatives with positive markers were submitted to small-bowel biopsy.

RESULTS:

Eight new cases of CD were found among the relatives. Two had been investigated by small-bowel biopsy 20 years previously, when they had only minor mucosal changes not classified as CD. The other six new cases of CD were found among offspring of the index patients and were born after completion of the previous study. Thus no new case of CD was found among those relatives who had a completely normal small-bowel biopsy 20-25 years previously.

CONCLUSIONS:

The high prevalence of CD among first-degree relatives of coeliac patients (8.3% in this study) supports the need to screen for CD in this high-risk population. Even relatives with only mild enteropathy should be followed carefully, since some may subsequently develop CD.

Place, publisher, year, edition, pages
2003. Vol. 38, no 1, 61-65 p.
Keyword [en]
Coeliac disease, Family study, Serological markers
National Category
Medical and Health Sciences
Identifiers
URN: urn:nbn:se:liu:diva-46758DOI: 10.1080/00365520310000456OAI: oai:DiVA.org:liu-46758DiVA: diva2:267654
Available from: 2009-10-11 Created: 2009-10-11 Last updated: 2017-12-13Bibliographically approved
In thesis
1. Childhood coeliac disease: clinical aspects of heredity, diagnosis and dietary therapy
Open this publication in new window or tab >>Childhood coeliac disease: clinical aspects of heredity, diagnosis and dietary therapy
2003 (English)Doctoral thesis, comprehensive summary (Other academic)
Abstract [en]

Childhood coeliac disease (CD) is defined as a lifelong disorder, in which the small bowel mucosa is abnormal as a result of exposure to gluten in the diet. The mucosal damage improves on treatment with a gluten-free diet, but recurs within two years of reintroduction of gluten. If there is no mucosal relapse within two years of gluten challenge the condition is called transient gluten intolerance. We describe a case of CD, in whom small bowel biopsy performed yearly demonstrated that mucosal relapse took 14 years of gluten challenge. This is an exceptionally long time questioning the existence of the condition transient gluten intolerance.

Small bowel biopsy is the golden standard in diagnosing CD but an uncomfortable investigation in a child. An effective sedation of the child is necessary. In a randomised study of intranasal versus intravenous midazolam as sedation of children undergoing small bowel biopsy we showed that both administration routes are effective and safe. Intranasal midazolam produced nasal discomfort. Thus, the intravenous route of administration is to be preferred.

In a previous study at our clinic 20-25 years ago all first-degree relatives (n=100) of 32 index patients with CD were investigated with small bowel biopsy. Two cases of CD were found. In the present reinvestigation of the relatives (n=120) using serological screening, 8 new cases of CD were found. The overall prevalence of CD in the first-degree relatives is 8.3%. Six of the new cases were children of the index patients. The remaining 2 newly diagnosed cases had had some enteropathy, not classified as CD, in the previous study. This is the first study of first-degree relatives of coeliacs followed for as long as 20-25 years. The study shows that there is a strong genetic component in the aetiopathogenesis of CD.

The compliance to gluten-free diet, among 29 adult coeliacs, who had been diagnosed in childhood (one group before and one group after 4 years of age) was assessed by dietmy questionnaire and serological markers. The compliance was 80% in the patients diagnosed before 4 years and 36% in those diagnosed later in childhood. This significant difference indicates that a diagnosis in early childhood makes it easier for a coeliac patient to keep to a strict gluten-free diet. This is a clinically important observation.

In a double-blind randomised multi-centre study of oats in the gluten-free diet during a study period of one year, children with CD were carefully monitored using small bowel biopsy and serological markers. The results indicate that oats are tolerated by coeliac children. This is important, since additional oats improve the palatability and fibre content of the fairly low-fibre standard glutenfree diet. This makes it easier for a patient with CD to adhere to the gluten-free diet, which is very important in order to minimise the risk for potential complications.

Place, publisher, year, edition, pages
Linköping: Linköpings universitet, 2003. 77 p.
Series
Linköping University Medical Dissertations, ISSN 0345-0082 ; 787
National Category
Medical and Health Sciences
Identifiers
urn:nbn:se:liu:diva-26656 (URN)11221 (Local ID)91-7373-546-9 (ISBN)11221 (Archive number)11221 (OAI)
Public defence
2003-05-08, Sal K1, Kåkenhus, Bredgatan 33, Norrköping, 09:00 (Swedish)
Opponent
Available from: 2009-10-08 Created: 2009-10-08 Last updated: 2012-10-09Bibliographically approved

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Högberg, LottaFälth-Magnusson, KarinGrodzinsky, EwaStenhammar, Lars

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