ESPE Yearbook of Paediatric Endocrinology

Brief Summary: This retrospective study evaluated fracture risk among 2372 children and adolescents (59% females, aged 1-16) with biopsy-proven celiac disease (CD) compared to 11,860 children without CD matched by age, sex, socioeconomic status, and population sector (general Jewish population, ultra-orthodox Jews and Arabs). The overall fracture incidence rate was higher in the CD group (256 vs 165 per 10,000 patient-years).

Median age at the end of the follow up was 12.8 years; median follow-up was 5.5 years. Patients with chronic diseases that could impact bone health were excluded. The hazard ratio (HR) for fracture was 1.57 for the CD group compared to the matched group (1.47 for boys and 1.67 for girls). The CD group also showed higher HR for multiple fractures (1.67), fractures in the pre-diagnosis period (1.64), and fractures after CD diagnosis (1.52). For both groups, the most common site of fracture was the radius/ulna. There were no differences in the mean Z-scores for height, weight and BMI between children with or without fractures.

According to this interesting and well-designed Israeli study, children with CD showed an increased fracture risk both preceding and following CD diagnosis and treatment, corroborating previous data from a large population-based cohort study reporting that the risk of fracture remained elevated up to 20 years after CD diagnosis. The persistence of increased fracture risk years after CD diagnosis could be explained by an incomplete intestinal healing due to incomplete adherence to gluten free diet (GFD) or by the lower quality of micronutrients in the GFD.

Strengths of this study are the large, homogeneous and well-matched population and the long follow-up. Limitations include the retrospective design, which entailed the extraction of information from an electronic database. Moreover, blood tests and anthropometric measurements at diagnosis were performed in a non-experimental setting according to the clinician’s discretion; therefore, some of the data during the follow-up were missing. Lastly, there were no measurements of bone mineral density and other bone parameters at the time of the fracture. Prospective studies are needed to evaluate changes in bone quantity and quality after initiation of GFD in children with CD, in order to identify those at risk for persistent metabolic bone disease and prevent fractures.

Reference: 1. Ludvigsson, J. F., Michaelsson, K., Ekbom, A. & Montgomery, S. M. Coeliac disease and the risk of fractures – a general population-based cohort study. Aliment Pharm. Ther. 25, 273–285 (2007).