Merriam-Webster: Double Whammy Definition: “a combination of two usually adverse forces, circumstances, or effects”

M Bramuzzo et al. Inflamm Bowel Dis 2021; 27: 1881-1888. Open Access: Phenotype and Natural History of Children With Coexistent Inflammatory Bowel Disease and Celiac Disease

Methods: This was a retrospective case-control study based on a national registry. Cases included children diagnosed with both IBD and Celiac Disease (CeD). Two matched IBD controls without CeD, and 2 matched CeD controls were selected for each case.

Key findings:

• Forty-nine (1.75%) patients with IBD and CeD were identified out of 2800 patients with IBD. CeD was diagnosed before IBD in 37 (75.5%)
• Compared with patients with IBD alone, patients with IBD and CeD presented more frequently with autoimmune diseases (odds ratio, 2.81; 95% CI, 0.97–8.37; P = 0.04) (mainly thyroiditis 6.1% vs 0%)
• Children with ulcerative colitis and CeD had an increased risk of colectomy despite similar medical treatments compared with patients with ulcerative colitis alone (13.0% vs 0%); however, this was based on a small number (3 surgeries out of 23 patients)
• Anti-TNF biologics (46.2% vs 69.2%) were less commonly administered in patients with Crohn’s disease and CeD than in patients with Crohn’s disease alone
• Pubertal delay was more common in patients with IBD and CeD compared with patients with IBD alone (14.9% vs 3.2%; odds artio, 5.24)

The discussion emphasizes the need to consider the risk of developing IBD in children with CeD and to recognize the increased risk of autoimmune diseases. Children with both UC and CeD may have a more severe phenotype. The authors recognize the possibility of misdiagnosis of CeD as patients with IBD could present with similar upper GI findings; however, this is likely infrequent as most cases of CeD preceded the diagnosis of IBD.

My take: One point that the authors neglect is the need to consider an underlying monogenetic disorder (eg. CTLA4B) in children with multiple immune-mediated diseases. The main message for children with this double whammy, though, is to consider the need for more aggressive treatment (especially with UC) and the need to screen for other autoimmune conditions (especially thyroiditis).

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