In the JournalsPerspective

Many children with celiac disease are lost to follow-up

One-quarter of children with celiac disease were lost to follow-up within a year of diagnosis, and some failed to even see a gastroenterologist after their diagnostic biopsy, according to research published in Clinical Gastroenterology and Hepatology.

Jocelyn A. Silvester, MD, PhD, of the Harvard Celiac Disease Program and Boston Children’s Hospital, and colleagues wrote these high rates of loss to specialist follow-up could indicate some failings in how children with celiac disease are managed.

“Although guidelines recommended dietitian education regarding a [gluten-free diet] and annual GI follow-up visits many may not be receiving appropriate treatment as 1 in 6 patients did not receive [gluten-free diet] education, and 9% had no GI follow-up after diagnosis,” they wrote. “It cannot be assumed that patients lost to GI follow-up are doing well nor that they receive specific follow-up in primary care.”

To assess adherence to guidelines for dietitian consultation and follow-up for celiac disease, Silvester and colleagues analyzed data from a retrospective cohort of 241 patients (median age 9.7 years; 63% female) diagnosed with biopsy-confirmed celiac disease at Boston Children’s Hospital between 2010 and 2014. They reviewed records from diagnosis through the end of 2017 to allow for a minimum of a three-year observation period. They considered any patient who did not have a GI visit for more than 18 months lost to follow-up.

Researchers found that one-quarter of patients were lost to follow-up within a year, and 9% (n = 22) had no GI visits after diagnosis. They also found that having a sibling with celiac disease (HR = 1.9), using Medicaid (HR = 2.19), and rescheduling or not attending more than 50% of appointments (HR = 2.43) were all associated with loss to follow-up.

Investigators determined that patients who were lost to follow-up were older at diagnosis compared with patients who stuck with follow-ups for longer (median 11.4 vs. 8.7 years; P = .01), and patients who reached 18 years and continued with follow-up were diagnosed at a younger age than those who did not (median 14.4 vs 16.2 years; P < .01).

Silvester and colleagues wrote that children who are lost to follow-up so quickly after diagnosis might not get the education they need to obtain the skills required to adhere to a gluten-free diet.

“Children who are inadequately adherent to a [gluten-free diet] are subject to gluten exposure and persistent mucosal damage, which is associated with complications of [celiac disease],” they wrote. “Establishing a pattern of regular GI follow-up for [celiac disease] during childhood may establish the habit of continuous, lifelong follow-up and improve long-term outcomes.” – by Alex Young

Disclosures: Silvester has received consulting fees from Takeda and research support from Biomedal SL and Glutenostics LLC. Please see the full study for all other authors’ relevant financial disclosures.

One-quarter of children with celiac disease were lost to follow-up within a year of diagnosis, and some failed to even see a gastroenterologist after their diagnostic biopsy, according to research published in Clinical Gastroenterology and Hepatology.

Jocelyn A. Silvester, MD, PhD, of the Harvard Celiac Disease Program and Boston Children’s Hospital, and colleagues wrote these high rates of loss to specialist follow-up could indicate some failings in how children with celiac disease are managed.

“Although guidelines recommended dietitian education regarding a [gluten-free diet] and annual GI follow-up visits many may not be receiving appropriate treatment as 1 in 6 patients did not receive [gluten-free diet] education, and 9% had no GI follow-up after diagnosis,” they wrote. “It cannot be assumed that patients lost to GI follow-up are doing well nor that they receive specific follow-up in primary care.”

To assess adherence to guidelines for dietitian consultation and follow-up for celiac disease, Silvester and colleagues analyzed data from a retrospective cohort of 241 patients (median age 9.7 years; 63% female) diagnosed with biopsy-confirmed celiac disease at Boston Children’s Hospital between 2010 and 2014. They reviewed records from diagnosis through the end of 2017 to allow for a minimum of a three-year observation period. They considered any patient who did not have a GI visit for more than 18 months lost to follow-up.

Researchers found that one-quarter of patients were lost to follow-up within a year, and 9% (n = 22) had no GI visits after diagnosis. They also found that having a sibling with celiac disease (HR = 1.9), using Medicaid (HR = 2.19), and rescheduling or not attending more than 50% of appointments (HR = 2.43) were all associated with loss to follow-up.

Investigators determined that patients who were lost to follow-up were older at diagnosis compared with patients who stuck with follow-ups for longer (median 11.4 vs. 8.7 years; P = .01), and patients who reached 18 years and continued with follow-up were diagnosed at a younger age than those who did not (median 14.4 vs 16.2 years; P < .01).

Silvester and colleagues wrote that children who are lost to follow-up so quickly after diagnosis might not get the education they need to obtain the skills required to adhere to a gluten-free diet.

“Children who are inadequately adherent to a [gluten-free diet] are subject to gluten exposure and persistent mucosal damage, which is associated with complications of [celiac disease],” they wrote. “Establishing a pattern of regular GI follow-up for [celiac disease] during childhood may establish the habit of continuous, lifelong follow-up and improve long-term outcomes.” – by Alex Young

Disclosures: Silvester has received consulting fees from Takeda and research support from Biomedal SL and Glutenostics LLC. Please see the full study for all other authors’ relevant financial disclosures.

    Perspective
    Benjamin Lebwohl

    Benjamin Lebwohl

    Celiac disease is unique among the autoimmune diseases in that the offending antigen, dietary gluten, is known. This knowledge allows for an effective therapy, the gluten-free diet, which results in clinical, serologic and histologic improvement.

    There is a downside to this solid understanding: the unfortunate misconception that once the diagnosis is made, the patient doesn’t need to follow up. Too often, patients are reassured with a variation of the following: “Just Google the gluten-free diet, and all will be fine.” In fact, the diagnosis is not the final step but rather the beginning of a major change in daily routine for the patient, given the various challenges that a gluten-free diet poses, including concerns regarding hidden sources of gluten and adequate fiber intake. Subsequent long-term sequelae including other autoimmune diseases, low bone density, and a modest but consistently documented increased risk for malignancy are other concerns that weigh on the patient and can be detected and, in some cases, prevented with close follow-up.

    A prior study of adults with celiac disease found that young adults and those with non-classical – ie, non-diarrheal – presentations were less likely to follow up. The study by Blanksy and colleagues, the first such study among children with celiac disease, lends further insight into the too-common phenomenon of patients being lost to follow-up after an initial diagnosis. The finding that this was more common among those with Medicaid insurance underscores that social determinants of health are likely at play in this condition whose treatment requires educational and financial resources.

    There is increasing interest in managing the transition from childhood to adulthood in celiac disease, as this is a time of life in which patients are at risk of falling out of the medical orbit. The study by Blanksy and colleagues underscores the fact that ensuring adequate follow-up should be the focus of ongoing efforts by a caregiver team.

    References:

    Cohen ME, et al. J Clin Gastroenterol. 2018;doi:10.1097/MCG.0000000000000851.

    Lee AR, et al. J Hum Nutr Diet. 2007;doi:10.1111/j.1365-277X.2007.00763.x.

    Ludvigsson JF, et al. Gut. 2016;doi:10.1136/gutjnl-2016-311574.

    • Benjamin Lebwohl, MD, MS
    • Director, clinical research
      The Celiac Disease Center at Columbia University

    Disclosures: Lebwohl reports serving as a consultant for Takeda and serving on a medical advisory board for Innovate Biopharmaceuticals.