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Enteric-Release Budesonide May Be Useful in the Management of Non-Responsive Celiac Disease

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Abstract

Background

Non-responsive celiac disease (NRCD) has many aetiologies, including gluten exposure. Budesonide may be used for refractory celiac disease (RCD) and celiac crisis.

Aims

We reviewed the effectiveness of budesonide to induce clinical and histologic response in NRCD with villous atrophy (VA).

Methods

Case series of adult cases with NRCD and VA prescribed budesonide at two celiac centers. Clinical variables and mucosal recovery (i.e., normal villous architecture within 1 year of treatment) were evaluated.

Results

Forty-two cases [77% female, median age 45.0 (IQR 28.3–60.0) years] were included. Most common symptoms were diarrhea (64%) and abdominal pain (62%). Budesonide was initiated at 9 mg (83%) for a median duration of 16.0 weeks (IQR 6.8–25.0 weeks). In total, 57% exhibited a clinical response, positively associated with diarrhea (adjusted OR 6.08 95% CI 1.04–35.47) and negatively with fatigue (adjusted OR 0.18 95% CI 0.03–0.98). Clinical response was higher among those with dietitian counseling prior to budesonide (n = 29, 70 vs. 23%, p < 0.01). Mucosal recovery was observed in 11/24 with follow-up duodenal biopsies. There was no association between clinical response and mucosal recovery, and 79% of clinical responders had a symptomatic relapse. RCD (48%) and chronic gluten exposure (24%) were the main suspected aetiologies of NRCD. Most individuals without a clinical response subsequently received an IBS-related diagnosis.

Conclusions

Budesonide may be effective to induce clinical response in NRCD presenting with diarrhea and VA, but clinical recurrence and lack of mucosal recovery are frequent after tapering. Other diagnoses, including coexisting IBS, may be considered in non-responders to budesonide therapy.

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Funding

Amelie Therrien was supported by Douglas Kinnear Award from Association des Gastroenterologues du Québec and by MSSS/FRQS training Program for specialty medicine residents with an interest in pursuing a research career Phase 2 Award. Jocelyn Silvester was supported by the National Institute of Diabetes and Digestive and Kidney Diseases of the National Institutes of Health under Award Numbers T32 DK 07760 and K23 DK119584. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health.

Author information

Authors and Affiliations

  1. Celiac Research Program, Harvard Medical School, Boston, USA

    Amelie Therrien, Jocelyn A. Silvester, Maureen M. Leonard, Alessio Fasano & Ciaran P. Kelly

  2. Celiac Center, Beth Israel Deaconess Medical Center, 330 Brookline Ave, Boston, MA, 02215, USA

    Amelie Therrien, Daniel A. Leffler & Ciaran P. Kelly

  3. Division of Gastroenterology, Hepatology and Nutrition, Boston Children Hospital, 300 Longwood Ave, Boston, MA, 02215, USA

    Jocelyn A. Silvester

  4. Center for Celiac Research and Treatment, MassGeneral Hospital for Children, 55 Fruit Street, Boston, MA, 02114, USA

    Maureen M. Leonard & Alessio Fasano

  5. Takeda Pharmaceutical International Co, Cambridge, USA

    Daniel A. Leffler

  6. Mucosal Immunology and Biology Research Center, Boston, MA, 02114, USA

    Maureen M. Leonard & Alessio Fasano

Authors
  1. Amelie Therrien
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  2. Jocelyn A. Silvester
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  4. Daniel A. Leffler
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Corresponding author

Correspondence to Amelie Therrien.

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Conflict of interest

Not related to the current work; Daniel Leffler is employed by Takeda Pharmaceuticals International Co. Ciaran Kelly has acted as a scientific advisor to Cour Pharmaceuticals, Glutenostics, Innovate, ImmunogenX, and Takeda Pharmaceuticals. He has received research funding from Aptalis. Alessio Fasano is a stock owner of Alba Therapeutics, has received consulting fees by AbbVie, Innovate Biopharmaceuticals and uBiome, has a speaking agreement with Mead Johnson Nutritional and research agreement with Takeda. Maureen Leonard has received speaker fees from Takeda Pharmaceuticals, research funding from Glutenostics LLC and has received consulting fees from Healthmode and Anokion. Jocelyn A. Silvester has received consulting fees from Takeda Pharmaceuticals International Co, and research funding from Cour Pharmaceuticals, Biomedal SL, and Glutenostics LLC.

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Therrien, A., Silvester, J.A., Leonard, M.M. et al. Enteric-Release Budesonide May Be Useful in the Management of Non-Responsive Celiac Disease. Dig Dis Sci 66, 1989–1997 (2021). https://doi.org/10.1007/s10620-020-06454-5

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  • DOI: https://doi.org/10.1007/s10620-020-06454-5

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