Last December, this blog site discussed a widely-reported, provocative study suggesting that “1 in 5 pediatric celiac disease patients on gluten-free diet sustain persistent intestinal damage” (Are followup biopsies necessary for Celiac disease? Look beyond the headlines).
Apparently a great number of celiac disease (CD) experts took exception to the authors’ conclusion that “neither the presence of symptoms nor positive serology were predictive of a patient’s histology at the time of repeat biopsy. These findings suggest a revisitation of monitoring and management criteria of celiac disease in childhood.”
A recent commentary (S Koletzko et al. JPGN 2017; 65: 267-9) from the CD working group of ESPGHAN critiques the limitations of the study. Limitations included the following:
- Selection bias -70% of patients in this retrospective study had repeat endoscopy were symptomatic
- Retrospective study
- Including children with admitted non-adherence to gluten-free diet (GFD)
- Including ~25% of children who were rebiopsied within 17 months of starting GFD
- Lack of standardized tTG testing
- Lack of blinding of pathologist
- “We do not think that the data..give sufficient support…that routine biopsies should be performed in all children at diagnosis and after the initiation of GFD.”
- “The CD working group of ESPGHAN strongly advises against regular rebiopsy in children on a GFD…Re-endoscopy should be reserved for symptomatic patients–in particular when seronegative.”
The authors of the initial study (MM Leonard, A Fasano. JPGN 2017; 65: 270-1) were given an opportunity to defend their conclusions. Their commentary was much less provocative in my view, and titled: “Zero, One, or Two Endoscopies to Diagnosis and Monitor Pediatric Celiac Disease? The Jury is Still Out”
They note that there is a lack of data to know whether there are “no real clinical consequences” of persistent enteropathy, as stated by S Koletzko et al. They state that until further research is completed a “personalized approach to follow-up care is needed.” It is encouraging that they have started a prospective study to address the limitations of their retrospective study.
- For patients with CD who are strictly-adherent, asymptomatic and with normal serology, repeat endoscopy is of questionable benefit. If there are abnormalities in the histology, what is the appropriate intervention?
- There has been a study (Gastroenterology 2010; 139: 763) which showed that mortality was NOT worsened in undiagnosed CD (identified by review of serology) in Olmstead County. In this population, the main long-term detrimental effect was reduced bone density. My inference is that for CD patients who are asymptomatic, particularly those with normalized serology, they are unlikely to have easily-identifiable adverse effects noted, even if their histology is abnormal.
My take (unchanged from last year): I think it is premature to recommend routine followup biopsies in asymptomatic patients with normal serology.