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A pilot study characterizing longitudinal changes in fecal microbiota of patients with Hirschsprung-associated enterocolitis

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Abstract

Purpose

Hirschsprung disease is a neurointestinal disease that occurs due to failure of enteric neural crest-derived cells to complete their rostrocaudal migration along the gut mesenchyme, resulting in aganglionosis along variable lengths of the distal bowel. Despite the effective surgery that removes the aganglionic segment, children with Hirschsprung disease remain at high risk for developing a potentially life-threatening enterocolitis (Hirschsprung-associated enterocolitis). Although the etiology of this enterocolitis remains poorly understood, several recent studies in both mouse models and in human subjects suggest potential involvement of gastrointestinal microbiota in the underlying pathogenesis of Hirschsprung-associated enterocolitis.

Methods

We present the first study to exploit the Illumina MiSeq next-generation sequencing platform within a longitudinal framework focused on microbiomes of Hirschsprung-associated enterocolitis in five patients. We analyzed bacterial communities from fecal samples collected at different timepoints starting from active enterocolitis and progressing into remission.

Results

We observed compositional differences between patients largely attributable to variability in age at the time of sample collection. Remission samples across patients exhibited compositional similarity, including enrichment of Blautia, while active enterocolitis samples showed substantial variability in composition.

Conclusions

Overall, our findings provide continued support for the role of GI microbiota in the pathogenesis of Hirschsprung-associated enterocolitis.

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National Institute of General Medical Sciences, DK098696-01A1, Naomi Ward, 2P20GM103432.

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  1. Kristopher D. Parker

    Present address: Department of Natural Sciences, Middle Georgia State University, Cochran, GA, USA

Authors and Affiliations

  1. Department of Botany, University of Wyoming, Laramie, WY, USA

    Kristopher D. Parker & Naomi L. Ward

  2. Department of Pediatric Surgery, Massachusetts General Hospital, Harvard Medical School, Boston, MA, USA

    Jessica L. Mueller, Maggie Westfal & Allan M. Goldstein

  3. Department of Microbiology, Immunology, and Pathology, Colorado State University, Fort Collins, USA

    Naomi L. Ward

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  1. Kristopher D. Parker
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Parker, K.D., Mueller, J.L., Westfal, M. et al. A pilot study characterizing longitudinal changes in fecal microbiota of patients with Hirschsprung-associated enterocolitis. Pediatr Surg Int 38, 1541–1553 (2022). https://doi.org/10.1007/s00383-022-05191-2

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