U.S. children with celiac disease are not being adequately followed up for adherence to a gluten-free diet, a crucial component for their treatment and protection against intestinal damage, researchers report.
Discomfort aside, the villous atrophy associated with chronic celiac poses serious risks of nutritional deficits, fractures, and lymphoma, noted Jocelyn A. Silvester, MD, PhD, of Boston Children’s Hospital and Harvard Medical School, and colleagues, writing online in Clinical Gastroenterology and Hepatology. And since it requires time to master and skills to maintain a gluten-free diet, initial consultation with an expert registered dietitian and annual assessment are recommended for successful management, the team emphasized.
“Celiac disease is a chronic condition that requires lifelong treatment and follow-up,” Silvester told MedPage Today. “More studies are needed to understand why patients are not followed up. Just because patients are not coming back doesn’t mean that they’re doing well. More studies are needed to understand why patients are not following up.”
For the study, the investigators randomly selected participants from a database of children (<18 years) diagnosed with biopsy-confirmed celiac disease at Boston Children's Hospital from January 1, 2010 to December 31, 2014, who were studied at a rate of 50 per year. Medical records were reviewed from diagnosis through December 31, 2017, to allow for a minimum of 3 years of observation. Patients who did not have a gastrointestinal (GI) visit for 18 months were deemed lost to follow-up.
Of the 250 patients selected, nine were excluded because they were seen for a second opinion, yielding 241 eligible subjects, of whom 63% were female. Median age at celiac diagnosis was 9.7 years (interquartile range 6.20-13.3). Abdominal pain (24%) and constipation (14%) were common symptoms at diagnosis, and only 2% of children were asymptomatic.
The majority (83%) of patients consulted a dietitian, with 31% attending both a dietitian-led class and an individual consultation. Within a year of diagnosis, 25% were lost to follow-up and 9% had no GI visits after their diagnostic biopsy.
Variables associated with loss to follow-up included having a sibling with celiac disease (hazard ratio 1.90), using Medicaid (HR 2.19), and rescheduling or not attending >50% appointments (HR 2.43). “The association of reliance on Medicaid with loss to follow-up suggests that socioeconomic disparities may further compromise the health outcomes of children with [celiac disease] beyond decreased resources to obtain gluten-free foods,” Silvester and co-authors wrote.
In addition, patients lost to follow-up within the first year were older at diagnosis than those who had longer follow-up: median 11.4 vs 8.7 years (P=0.01), while patients who continued with follow-up after age 18 were diagnosed at a younger age than those who did not: median age of 14.4 vs 16.2 years (P<0.01).
Overall, 73% visited another department at the hospital during the observation period, with 47% of those lost to GI follow-up maintaining a care relationship with the hospital more than 12 months after their last GI visit.
More than half the cohort (n=155) achieved normal serum tissue transglutaminase immunoglobulin A (TTG IgA), with a median time to normalization of 17.0 months (IQR 7.0-2.0). Of 141 patients who underwent celiac serology at last GI follow-up, 25% exhibited an elevated TTG IgA.
Eighteen participants had celiac serology ordered by non-GI providers after loss to GI follow-up. Seven patients (39%) had abnormal serology at a median of 43.6 months (IQR 38.6-72.3) after diagnosis and 13.9 months (IQR 12.5-21.5) after their last GI visit.
Previous research has shown that loss to follow-up is associated with non-adherence to a gluten-free diet, positive serology, and poorly controlled disease.
Asked for her perspective, Norelle R. Reilly, MD, of Columbia University Medical Center in New York City, who was not involved in the research, told MedPage Today that the results provide valuable information and are consistent with the experience of many clinicians caring for children with celiac disease.
“Fortunately, many children with celiac disease who are well treated do quite well following diagnosis, though I agree with the authors’ conclusions that ‘no-news is not always good news’ for those who do not return for follow-up care,” she said.
“High rates of loss to specialist follow-up indicate significant shortcomings in the management of children with [celiac disease],” the authors wrote. “Many may not be receiving appropriate treatment as one in six patients did not receive [gluten-free diet] education and 9% had no GI follow-up after diagnosis.”
The team called for research into why having a sibling with celiac disease is a risk for loss to follow-up and how loss to follow-up occurs across the lifespan. Silvester and co-authors also recommended establishing a pattern of regular GI assessment during childhood and educating patients, families, and care providers about the importance of continuity of follow-up in order to ensure the best long-term outcomes.
“Even in the case of families given the recommended nutritional education and medical advice, some do not return regularly for follow-up, which may be a function of lack of symptoms, lack of pharmacotherapy for celiac disease, or the family’s confidence in their own ability to navigate the diet,” Reilly said. “Things in this field are continually changing, and the disease impacts the mind and body of children at various ages differently – all of which make regular follow-up worthwhile.”
Silvester reported research support from the National Institutes of Health, Biomedal SL, and Glutenostics LLC, as well as consulting fees from Takeda Pharmaceuticals, Inc.
Reilly reported having no conflicts of interest relevant to her comments.