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Childhood coeliac disease: clinical aspects of heredity, diagnosis and dietary therapy
Linköping University, Department of Molecular and Clinical Medicine, Pediatrics. Linköping University, Faculty of Health Sciences.
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: urn:nbn:se:liu:diva-26656Local ID: 11221ISBN: 91-7373-546-9 (print)OAI: oai:DiVA.org:liu-26656DiVA: diva2:247205
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
List of papers
1. Very late mucosal relapse in a girl with coeliac disease
Open this publication in new window or tab >>Very late mucosal relapse in a girl with coeliac disease
1993 (English)In: Acta Paediatrica, ISSN 0803-5253, E-ISSN 1651-2227, Vol. 82, no 10, 887-889 p.Article in journal (Refereed) Published
Abstract [en]

Small bowel biopsy in a 4–year-old girl with symptoms suggestive of coeliac disease revealed subtotal villous atrophy. The mucosa healed on a gluten-free diet. From the age of 7 years, the girl was challenged with gluten. Annual biopsies showed normal or nearly normal mucosa specimens. At 21 years of age, after 14 years of gluten challenge, a mucosal relapse was found and a gluten-free diet was reinstituted. A biopsy one year later showed a normal mucosa. From this case report it is apparent that a patient with a past history of subtotal villous atrophy, which after a preceding period of gluten-free diet does not recur within two years of gluten challenge, must be followed carefully, so as not to miss a late relapse.

National Category
Medical and Health Sciences
Identifiers
urn:nbn:se:liu:diva-84482 (URN)10.1111/j.1651-2227.1993.tb17636.x (DOI)
Available from: 2012-10-09 Created: 2012-10-09 Last updated: 2017-12-07Bibliographically approved
2. Anti-endomysium and anti-gliadin antibodies as serological markers for a very late mucosal relapse in a coeliac girl
Open this publication in new window or tab >>Anti-endomysium and anti-gliadin antibodies as serological markers for a very late mucosal relapse in a coeliac girl
1997 (English)In: Acta Paediatrica, ISSN 0803-5253, E-ISSN 1651-2227, Vol. 86, no 3, 335-336 p.Article in journal, Letter (Refereed) Published
Abstract [en]

No abstract is available for this article.

National Category
Medical and Health Sciences
Identifiers
urn:nbn:se:liu:diva-84483 (URN)10.1111/j.1651-2227.1997.tb08905.x (DOI)
Available from: 2012-10-09 Created: 2012-10-09 Last updated: 2017-12-07Bibliographically approved
3. Intranasal versus intravenous administration of midazolam to children undergoing small bowel biopsy
Open this publication in new window or tab >>Intranasal versus intravenous administration of midazolam to children undergoing small bowel biopsy
1995 (English)In: Acta Paediatrica, ISSN 0803-5253, E-ISSN 1651-2227, Vol. 84, no 12, 1429-1431 p.Article in journal (Refereed) Published
Abstract [en]

Sixty-three children under the age of 9 years were randomized to receive intravenous (group A, n= 33) or intranasal (group B, n= 30) midazolam as sedation for small bowel biopsy. Mean doses of midazolam given to produce adequate sedation were 0.31 mg (kg body weight)−1 in group A and 0.34 mg (kg body weight)−1 in group B (NS). Four children in group A and 10 children in group B required additional doses to maintain adequate sedation throughout the biopsy procedure (p <0.05). There was no significant difference between the groups regarding the median procedure time (7 min in group A, 8.5 min in group B) or median fluoroscopy time (5 s in group A, 4 s in group B). All children in group B showed signs of discomfort from the nose when given midazolam intranasally. In conclusion, this study indicates that intravenous administration of midazolam is preferable to the intranasal route.

National Category
Medical and Health Sciences
Identifiers
urn:nbn:se:liu:diva-84484 (URN)10.1111/j.1651-2227.1995.tb13582.x (DOI)
Available from: 2012-10-09 Created: 2012-10-09 Last updated: 2017-12-07Bibliographically approved
4. Familial prevalence of coeliac disease: a twenty-year follow-up study
Open this publication in new window or tab >>Familial prevalence of coeliac disease: a twenty-year follow-up study
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.

Keyword
Coeliac disease, Family study, Serological markers
National Category
Medical and Health Sciences
Identifiers
urn:nbn:se:liu:diva-46758 (URN)10.1080/00365520310000456 (DOI)
Available from: 2009-10-11 Created: 2009-10-11 Last updated: 2017-12-13Bibliographically approved
5. Better dietary compliance in patients with coeliac disease diagnosed in early childhood
Open this publication in new window or tab >>Better dietary compliance in patients with coeliac disease diagnosed in early childhood
2003 (English)In: Scandinavian Journal of Gastroenterology, ISSN 0036-5521, Vol. 38, no 7, 751-754 p.Article in journal (Refereed) Published
Abstract [en]

Background: In coeliac disease (CD) there is a permanent gluten intolerance requiring life-long adherence to a strict gluten-free diet (GFD). An inadequate diet increases the risk for long-term complications. Coeliac patients often have great difficulty in maintaining a strictly GFD. We aimed to study whether young adults with CD diagnosed before the age of 4 years have a better dietary compliance than patients diagnosed later in life.

Method: Twenty-nine adults with CD diagnosed in childhood were studied. They had had CD for 17-24 (mean 20) years. Their compliance to GFD was assessed using a questionnaire and serological markers (IgA and IgG anti-endomysium antibodies and IgA anti-tissue transglutaminase antibodies).

Results: At least 80% of the coeliac patients who had been diagnosed before the age of 4 years complied with the GFD compared to 36% of the CD patients older than 4 years at diagnosis ( P &#114 < &#114 0.05).

Conclusion: This is the first study to show that patients with CD diagnosed before 4 years of age keep to a GFD significantly better than patients diagnosed after 4 years. It is thus important to diagnose childhood CD as early as possible in order to minimize the risk for reduced well-being and other potentially serious complications in coeliac individuals on an inadequate diet.

National Category
Medical and Health Sciences
Identifiers
urn:nbn:se:liu:diva-26571 (URN)10.1080/00365520310003318 (DOI)11136 (Local ID)11136 (Archive number)11136 (OAI)
Available from: 2009-10-08 Created: 2009-10-08 Last updated: 2012-10-09Bibliographically approved
6. Oats to children with newly diagnosed coeliac disease: a randomised double blind study
Open this publication in new window or tab >>Oats to children with newly diagnosed coeliac disease: a randomised double blind study
Show others...
2004 (English)In: Gut, ISSN 0017-5749, E-ISSN 1468-3288, Vol. 53, no 5, 649-654 p.Article in journal (Refereed) Published
Abstract [en]

Background: Treatment of coeliac disease (CD) requires lifelong adherence to a strict gluten free diet (GFD) which hitherto has consisted of a diet free of wheat, rye, barley, and oats. Recent studies, mainly in adults, have shown that oats are non-toxic to CD patients. In children, only open studies comprising a small number of patients have been performed.

Aim: To determine if children with CD tolerate oats in their GFD.

Patients and methods: In this double blind multicentre study involving eight paediatric clinics, 116 children with newly diagnosed CD were randomised to one of two groups: one group was given a standard GFD (GFD-std) and one group was given a GFD with additional wheat free oat products (GFD-oats). The study period was one year. Small bowel biopsy was performed at the beginning and end of the study. Serum IgA antigliadin, antiendomysium, and antitissue transglutaminase antibodies were monitored at 0, 3, 6, and 12 months.

Results: Ninety three patients completed the study. Median (range) daily oat intake in the GFD-oats group (n = 42) was 15 (5–40) g at the six month control and 15 (0–43) g at the end of the study. All patients were in clinical remission after the study period. The GFD-oats and GFD-std groups did not differ significantly at the end of the study regarding coeliac serology markers or small bowel mucosal architecture, including numbers of intraepithelial lymphocytes. Significantly more children in the youngest age group withdrew.

Conclusions: This is the first randomised double blind study showing that the addition of moderate amounts of oats to a GFD does not prevent clinical or small bowel mucosal healing, or humoral immunological downregulation in coeliac children. This is in accordance with the findings of studies in adult coeliacs and indicates that oats, added to the otherwise GFD, can be accepted and tolerated by the majority of children with CD.

National Category
Medical and Health Sciences
Identifiers
urn:nbn:se:liu:diva-22329 (URN)10.1136/gut.2003.026948 (DOI)1529 (Local ID)1529 (Archive number)1529 (OAI)
Available from: 2009-10-07 Created: 2009-10-07 Last updated: 2012-10-09Bibliographically approved

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