From the AGA Journals

Universal screening doesn't pay in celiac disease

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Model doesn't really test screening issue

Cost-effective screening for any disease

necessitates the ability to detect disease at a stage such that low-cost

intervention could be initiated that avoids undesirable outcomes. In this

carefully constructed decision analysis model from Park et al., two strategies

were studied: screening without symptoms at all in 12-year-old children and

selective screening in those who were symptomatic or at high risk. In general,

evaluation of a symptomatic patient is not usually considered screening

although in this model, the symptoms are of celiac disease and not those of the

study endpoint, complications of osteoporosis.

The inference from this study is that population-based

screening is not cost effective, and a strategy that investigates those at-risk

persons with hereditary diseases such as Down, Turner, and Williams syndromes,

first-degree relatives with celiac disease, or those with symptoms of celiac disease

is cost effective. There are important considerations not taken into account by

this model, for instance those who are tissue transglutaminase (tTG) positive

but biopsy negative are patients who are at higher risk of eventually

developing small bowel abnormalities, though this at most might affect 10% of

these patients.

In addition, the cost of potential complications of

untreated celiac disease other than bone disease, such as lymphoma, seizures,

or peripheral neuropathy, are not accounted for in this analysis due to the

difficulty of quantifying them but would likely increase the cost-effectiveness

of screening. Finally, as the course of osteoporosis in untreated celiac

disease is unknown, it is not possible to model the benefits of earlier

treatment. Ultimately, the only way to really test these strategies is a

carefully constructed clinical trial of these two strategies, but this model

certainly suggests that current practices may end up being the appropriate


Kenneth K. Wang, M.D., is the Van Cleve Professor of

Gastroenterology Research at the Mayo Clinic, Rochester, Minn. He had no

relevant disclosures.



Serologic screening for celiac disease for symptomatic and high-risk children is more cost effective than is universal screening, at least when it comes to future bone disease.

Indeed, the current standard of practice of selective screening is also associated with greater quality-adjusted life year (QALY) gains over universal screening, reported Dr. K.T. Park in the June issue of Clinical Gastroenterology and Hepatology.

In light of "ongoing clinical concern that current practice of celiac disease screening misses a considerable proportion of asymptomatic celiac disease patients," Dr. Park of Stanford (Calif.) University and colleagues developed a decision analytic Markov model of 12-year-old cohorts (1,000 male and 1,000 female) with population-based prevalence of celiac disease in North America (Clin. Gastroenterol. Hepatol. 2013 June [doi:10.1016/j.cgh.2012.12.037]).

They used hip bone and vertebral fractures as clinical endpoints to assess the cost effectiveness of either universal serologic screening for celiac disease or selective screening in only symptomatic or high-risk children.

"Suboptimal bone health in the form of nontraumatic fractures is an established risk factor for celiac disease patients who are nonadherent to a gluten-free diet, or have undiagnosed subclinical disease," they explained.

Selective screening – the current standard of care – included screening of high-risk children, such as those with type 1 diabetes mellitus; Down, Turner, and Williams syndromes; IgA deficiency; systemic lupus; autoimmune thyroiditis; and those with a first-degree and/or second-degree relative with celiac disease.

Selective screening also included screening of symptomatic children exhibiting diarrhea, abdominal pain, bloating, and other irritable bowel–like symptoms, as well as poor growth, wasting, failure to thrive, or anemia.

In the model, "Any positive serologic screens required diagnostic confirmation via endoscopic duodenal mucosal biopsies," wrote the authors.

"Once a celiac disease patient started lifelong therapy by maintaining a strict gluten-free diet, patients were subject to natural adherence and nonadherence rates reported in the literature."

Additionally, the investigators’ model assumed the development of deteriorating bone disease among celiac patients, and calculated this at the same rate as the non–celiac population with comparable bone demineralization found in the literature.

They specifically focused on the development of hip and vertebral fractures, which "carry the highest morbidity rate in terms of progressing to long-term disability."

Cost estimates for procedures were derived from the Centers for Medicare and Medicaid Services for 2012.

Dr. Park found that for males, universal screening accrues a lifetime average cost of $8,532, with an associated QALY-gained of 25.511.

In contrast, selective screening had lower costs of $8,472, as well as a higher QALY-gained of 25.515.

Similarly, for females, universal screening carried lifetime average cost of $11,383 with an associated QALY-gained of 25.74; selective screening was cheaper, at $11,328, and had a slightly higher QALY-gained, of 25.75.

"Adopting the universal serologic screening strategy, where virtually every preadolescent child would be screened for celiac disease as part of his/her routine blood work in the primary care setting, would be more expensive and fails to increase the long-term quality of life of the population as a whole" in terms of bone health, wrote the investigators.

Indeed, "the universal serologic screening strategy introduces potential harm from unnecessary endoscopic evaluations of healthy individuals if serologic screening is falsely positive."

On the other hand, they cautioned that cost and quality of life assessments that use endpoints other than fracture, including anemia, infertility, or malignancy, "could change the cost effectiveness of universal screening for celiac disease."

The authors disclosed that Dr. Park was supported by a grant from the National Institutes of Health. They stated that they have no conflicts of interest.

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