Prevalence of childhood celiac disease and changes in infant feeding.

OBJECTIVES: Between 1984 and 1996, Sweden experienced an "epidemic" of clinical celiac disease in children <2 years of age, attributed partly to changes in infant feeding. Whether infant feeding affects disease occurrence and/or the clinical presentation remains unknown. We investigated and compared the total prevalence of celiac disease in 2 birth cohorts of 12-year-olds and related the findings to each cohort's ascertained infant feeding. METHODS: A 2-phase cross-sectional screening study was performed in which 13 279 children from 2 birth cohorts participated: children born during the epidemic (1993) and children born after the epidemic (1997). Previously diagnosed cases were reported and confirmed. Blood samples were analyzed for serological markers and children with positive values were referred for small intestinal biopsy. Infant feeding practices in the cohorts were ascertained via questionnaires. Prevalence comparisons were expressed as prevalence ratios. RESULTS: The total prevalence of celiac disease was 29 in 1000 and 22 in 1000 for the 1993 and 1997 cohorts, respectively. Children born in 1997 had a significantly lower risk of having celiac disease compared with those born in 1993 (prevalence ratio: 0.75; 95% confidence interval: 0.60-0.93; P = .01). The cohorts differed in infant feeding (specifically, in the proportion of infants introduced to dietary gluten in small amounts during ongoing breastfeeding). CONCLUSIONS: A significantly reduced prevalence of celiac disease in 12-year-olds indicates an option for disease prevention. Our findings suggest that the present infant feeding recommendation to gradually introduce gluten-containing foods from 4 months of age, preferably during ongoing breastfeeding, is favorable.

Health-related quality-of-life in children with coeliac disease, measured prior to receiving their diagnosis through screening.

OBJECTIVE: To compare the health-related quality-of-life (HRQoL) of children with screening-detected coeliac disease (CD), before they learned of their diagnosis, with that of children without CD and in those previously diagnosed with CD. METHODS: In a cross-sectional CD screening study ('ETICS': Exploring the Iceberg of Coeliacs in Sweden), of 10,041 Swedish 12-year-olds invited, 7567 (75%) consented to participate, and 7208 (72%) children without previously diagnosed CD had serological markers analysed. Before the screening results were known, 7218 children (72%) and 6524 of their parents (65%) answered questionnaires. Questionnaires included the Swedish child-friendly pilot version of the EQ-5D instrument and proxy version of the EQ-5D instrument, which are generic tools used to describe HRQoL. RESULTS: We found no significant difference in HRQoL between the groups of children with screening-detected CD, without CD, and those previously diagnosed with CD. CONCLUSION: The HRQoL reported by 12-year-olds with screening-detected CD, before they learned of their diagnosis, was not worse than that of the children without CD or those previously diagnosed with CD. Thus, mass screening for CD should not be justified on the basis that children with unrecognized CD have a poor HRQoL. However, because these children rated their HRQoL before diagnosis and treatment, they may not have recognized or perceived symptoms as severe enough to seek medical attention which demonstrates how difficult clinical/active case finding can be. Mass screening may still, therefore, be considered if the aim is early detection and prevention of future complications.

Balancing health benefits and social sacrifices: a qualitative study of how screening-detected celiac disease impacts adolescents' quality of life.

BACKGROUND: Celiac disease often goes undiagnosed. Mass screening might be an option to reduce the public health burden of untreated celiac disease. However, mass screening is still controversial since it is uncertain whether the benefits of early detection outweigh the possible negative consequences. Before implementation of screening programs, the experiences of those being identified as cases should be considered. The aim of our study was to explore how screening-detected celiac disease impacts adolescents' quality of life, as perceived by themselves and their parents. METHODS: All adolescents (n = 145) with screening-detected celiac disease found in a Swedish screening study, and their parents, were invited to share their experiences in a qualitative follow-up study. In total, we have information on 117 (81%) of the adolescents, either from the adolescents themselves (n = 101) and/or from their parent/s (n = 125). Written narratives were submitted by 91 adolescents and 105 parents. In addition, 14 focus group discussions involving 31 adolescents and 43 parents were conducted. Data was transcribed verbatim and analyzed based on a Grounded Theory framework. RESULTS: The screening-detected celiac disease diagnosis had varying impact on quality of life that related both to changes in perceived health and to the adolescents' experiences of living with celiac disease in terms of social sacrifices. Changes in perceived health varied from "healthy as anyone else with no positive change" to "something was wrong and then changed to the better", whereas experiences of living with celiac disease ranged from "not a big deal" to "treatment not worth the price". Perceptions about living with celiac disease and related coping strategies were influenced by contextual factors, such as perceived support from significant others and availability of gluten-free products, and were developed without a direct relation to experiencing changes in perceived health. CONCLUSIONS: Screening-detected celiac disease has varying impact on adolescents' quality of life, where their perceived change in health has to be balanced against the social sacrifices the diagnosis may cause. This needs to be taken into account in any future suggestion of celiac disease mass screening and in the management of these patients.

Accuracy in celiac disease diagnostics by controlling the small-bowel biopsy process.

OBJECTIVES: In a Swedish celiac disease screening study (Exploring the Iceberg of Celiacs in Sweden), we systematically reviewed the clinical diagnostic procedures with the aim to evaluate the diagnostic accuracy and to take advantage of lessons learned for improving diagnostic routines. MATERIALS AND METHODS: A school-based celiac disease screening study involving 5 Swedish centers, with 10,041 invited 12-year-olds with 7567 consenting participation. All 192 children with elevated serological markers were recommended to undergo small-bowel biopsy, performed and evaluated according to local clinical routines. All of the mucosal specimens were reevaluated by 1 and, when needed, 2 expert pathologists to reach diagnostic consensus. RESULTS: Small-bowel biopsies were performed in 184 children: 130 by endoscopy and 54 by suction capsule. Endoscopic biopsies were inconclusive in 0.6%, compared with 7.4% of biopsies by suction capsule. A patchy enteropathy was found in 9.1%. Reevaluation by the expert pathologist resulted in 6 additional cases with celiac disease and 1 cleared. Sixteen children with normal or inconclusive biopsies, 4 after endoscopy, and 12 after suction capsule were endoscopically rebiopsied, resulting in another 8 cases. The celiac disease prevalence of 30 of 1000 (95% confidence interval 26-34) was not statistically different from that previously reported. CONCLUSIONS: The present review revealed the importance of controlling each step of the diagnostic procedure. Several cases would have been missed by relying only on local routines. To improve the quality of childhood celiac disease diagnostics, we recommend multiple endoscopic biopsies from both proximal and distal duodenum and standardized evaluation by a pathologist with good knowledge of celiac disease.

Parents' willingness to pay for coeliac disease screening of their child.

OBJECTIVE: The aim of this study is to determine Swedish parents' willingness to pay (WTP) for coeliac disease (CD) screening of their child. SUBJECTS AND METHODS: CD screening was undertaken involving 10,041 12-year-old children, with 7567 (75%) agreeing to participate. Blood samples from the children were analysed for CD serological markers. Parents received a questionnaire including a scenario describing the health-related risks of having CD and screening and diagnostic procedures. Parents were also asked whether they were willing to pay for CD screening, should this not be offered free of charge, and, if so, what their maximum WTP would be. Their WTP was compared with the average cost per child for the screening and case ascertainment procedures. RESULTS: The questionnaire was answered by 6524 parents, and of 6057 valid responses 63% stated that they were willing to pay something. The mean WTP was 79 EUR and the median 10 EUR. The average cost per child for the screening and case ascertainment procedures was 47 EUR, which 23% of the parents stated they were willing to pay. Parents' WTP increased with higher education and income, and with child symptoms that may indicate CD. CONCLUSIONS: Swedish parents' WTP for school-based CD screening of their child was higher than the average cost per child; however, only a minority of the parents were willing to pay that amount.

How do children experience participating in a coeliac disease screening? A qualitative study based on children's written narratives.

AIM: To explore how 12-year-old Swedish children experienced being involved in a coeliac disease (CD) screening. METHODS: A qualitative approach was used to analyse short narratives written by children who had taken part in a school-based CD screening. Narratives were written after blood sampling, but prior to learning of the test results. Through an oscillation between the texts, codes, subcategories and four categories, a theme was generated describing the children's experience. RESULTS: The theme ''A Journey towards Confidence'' captures the overall experience of the screening. It illustrates that, although some children faced fear or anxiety, overall they had or were provided tools allowing them to cope well and experience a journey towards confidence. The categories describe conditions that contributed to the experience. The first, being involved, reflects the importance of involvement in receiving information and deciding to participate. Being a ''good citizen'' refers to feeling a duty to help and a trust to be treated fairly. Being able to cope with the screening was influenced by the children's ability to manage sensations and support received. The last category, being able to balance risk, illustrates that the children were able to balance the risks of screening when they had a realistic understanding of the disease and their vulnerability and had tamed their anxiety. CONCLUSIONS: This study increases the understanding of how 12-year-old Swedish children experienced participating in a CD screening and describes conditions important for a positive experience. We show that, although some children faced anxiety, they had, or were provided with, tools allowing them to cope well and gain confidence.

Celiac disease revealed in 3% of Swedish 12-year-olds born during an epidemic.

OBJECTIVE: Sweden experienced a marked epidemic of celiac disease between 1984 and 1996 in children younger than 2 years of age, partly explained by changes in infant feeding. The objective of this study was to determine the prevalence of celiac disease in 12-year-olds born during the epidemic (1993), including both symptomatic and screening detected cases. PATIENTS AND METHODS: All sixth-grade children in participating schools were invited (n = 10,041). Symptomatic and, therefore, previously diagnosed celiac disease cases were ascertained through the National Swedish Childhood Celiac Disease Register and/or medical records. All serum samples were analyzed for antihuman tissue transglutaminase (tTG)-IgA (Celikey), and serum-IgA, and some for tTG-IgG and endomysial antibodies. A small intestinal biopsy was recommended for all children with suspected undiagnosed celiac disease. RESULTS: Participation was accepted by 7567 families (75%). Previously diagnosed celiac disease was found in 67 children; 8.9/1000 (95% confidence interval [CI] 6.7-11). In another 192 children, a small intestinal biopsy was recommended and was performed in 180. Celiac disease was verified in 145 children, 20/1000 (95% CI 17-23). The total prevalence was 29/1000 (95% CI 25-33). CONCLUSIONS: The celiac disease prevalence of 29/1000 (3%)-with two thirds of cases undiagnosed before screening-is 3-fold higher than the usually suggested prevalence of 1%. When these 12-year-olds were infants, the prevailing feeding practice was to introduce gluten abruptly, often without ongoing breast-feeding, which might have contributed to this unexpectedly high prevalence.

Serum zinc in small children with coeliac disease.

UNLABELLED: In coeliac disease (CD) there is a gluten-induced small bowel enteropathy leading to malabsorption of various nutrients, vitamins and trace elements. Low levels of serum zinc have been reported in adults with untreated CD. In the present study we related the serum concentration of zinc to the morphology of the small bowel mucosa in 58 children, all under 4 years of age and under investigation for coeliac disease. The mean serum concentration of zinc (mean +/- SD; mumol/L) was significantly lower in children with untreated CD (9.7 +/- 2.0) (n = 11) compared to non-coeliac children without enteropathy (15.1 +/- 2.3) (n = 16) (p < 0.001), coeliac children on a gluten-free diet without enteropathy (14.2 +/- 1.6) (n = 14) (p < 0.001), coeliac children on gluten challenge with enteropathy (14.1 +/- 2.1) (n = 12) (p < 0.001) and coeliac children on gluten challenge without enteropathy (13.8 +/- 1.9) (n = 6) (p < 0.005). CONCLUSION: Serum zinc concentration is decreased in untreated coeliac children with enteropathy and normalizes on gluten-free diet. A low serum zinc value in a child being investigated for possible CD on clinical grounds can thus be used as a complementary marker for enteropathy indicating further investigation with small bowel biopsy. The hypothetical role of zinc in the pathogenesis of CD is discussed.

How do Swedish paediatric clinics diagnose coeliac disease? Results of a nationwide questionnaire study.

BACKGROUND AND AIM: Diagnosis of coeliac disease is based on the demonstration of enteropathy in a small bowel biopsy. Correct diagnosis is of utmost importance, since the need for dietary management is life long, and inadequate treatment may lead to potentially serious complications. The Swedish Working Group for Paediatric Coeliac Disease has published guidelines for the diagnosis of childhood coeliac disease. The present questionnaire was designed in order to create the basis for revision of those guidelines. METHODS: In 2004, a nationwide questionnaire concerning current diagnostic routines was sent to all 45 paediatric clinics performing small bowel biopsy. All clinics responded. RESULTS: All clinics base their diagnosis on small bowel biopsy findings at presentation. Furthermore, in 24 (53%) of the clinics, children with suspected coeliac disease are investigated by small bowel biopsy both at presentation and follow-up while on a gluten-free diet. Eighteen (40%) of the clinics employ a different diagnostic routine for children under 2 y of age than for those older than 2 y. All clinics use coeliac serological testing at various stages of the diagnostic procedure. CONCLUSION: All Swedish paediatric clinics perform a small bowel biopsy at presentation in children with suspected coeliac disease, and the majority of clinics perform a second biopsy when the child is on a gluten-free diet. Serological testing is frequently used as a diagnostic aid and in the monitoring of the disease while on a gluten-free diet.

Acute hematogenous osteomyelitis of the neck of the femur in children treated with drilling.

We re-examined 11 children (mean age 6.5 years) who had been treated with surgical drilling and intravenous (i.v.) antibiotics in high doses for acute hematogenous osteomyelitis of the neck of the femur. The mean follow-up time was 3.9 (2.9-6.4) years. 9 children who had been treated with early surgical drilling and i.v. antibiotics were all symptom-free and had normal radiographs at follow-up. The other 2 children had radiographic changes (metaphyseal rarefaction) already when referred to us. They had been treated with antibiotics alone before referral until the infection had spread to the hip joint. At follow-up, the clinical and radiographical findings were abnormal, 1 had a severely deformed hip. In our opinion, treatment with i.v. antibiotics alone may not prevent extension of the infectious process to the hip joint in some cases, even when the medication is given in high doses. Therefore, we believe that the addition of early surgical drilling is justified to prevent this feared complication.