Abstract
Objectives: Since 2010, several researches demonstrated that microbiota dynamics correlate and can even predispose to Hirschsprung (HSCR) associated enterocolitis (HAEC). This study aims at assessing the structure of the microbiota of HSCR patients in relation to extent of aganglionosis and HAEC status.Methods: All consecutive HSCR patients admitted to Gaslini Institute (Genova, Italy) between May 2012 and November 2014 were enrolled. Institutional review board (IRB) approval was obtained. Stools were sampled and 16S rDNA V3-V4 regions were sequenced using the Illumina-MiSeq. Taxonomy assignments were performed using QIIME RDP. Alpha diversity indexes were analyzed by Shannon and Simpson Indexes, and Phylogenetic Diversity.Results: We enrolled 20 patients. Male to female ratio was 4:1. Six patients suffered from Total Colonic Aganglionosis (TCSA). Considering sample site (i.e., extent of aganglionosis), we confirmed the known relationship between sample site and both biodiversity and composition of intestinal microbiota. Patients with TCSA showed lower biodiversity and increased Proteobacteria/Bacteroidetes relative abundance ratio. When addressing biodiversity, composition and dynamics of TCSA patients we could not find any significant relationship with regard to HAEC occurrences.Conclusions: The composition of HAEC predisposing microbiota is specific to each patient. We could confirm that total colon resections can change the composition of intestinal microbiota and to dramatically reduce microbial diversity. The subsequent reduction of system robustness could expose TCSA patients to environmental microbes that might not be part of the normal microbiota. Future long-term studies should investigate both patients and their family environment, as well as their disease history.
Highlights
Enterocolitis (HAEC) is an extremely serious, life-threatening complication that can occur in children with Hirschsprung disease (HSCR) pre- and even post-operatively
We report results of a metagenomics study on fecal microbiota performed on HSCR patients, addressing limitations, drawbacks and potential benefits of such approach, delineating future perspectives in this field of research
All pediatric patients with HSCR consecutively admitted to Giannina Gaslini Institute (Genova, Italy) between May 2012 and November 2014 were eligible for this study
Summary
Enterocolitis (HAEC) is an extremely serious, life-threatening complication that can occur in children with Hirschsprung disease (HSCR) pre- and even post-operatively. The intestinal microbiota associated with enterocolitis may persist following symptom resolution and can be implicated in symptom recurrence In another recent study on a mouse model of HSCR, the metagenomics analysis of Ednrb−/− and wild type mice showed that mutants had a distinct microbiota with respect to wild type (WT) and that the HAEC group had lower alpha diversity by Chao index compared with WT. Another study on Finnish patients showed how those with HD and HAEC had a significantly altered intestinal microbiome compared to healthy individuals, characterized by a lack of richness and pathologic expansions of taxa, Enterobacteria and Bacilli [17]. We report results of a metagenomics study on fecal microbiota performed on HSCR patients, addressing limitations, drawbacks and potential benefits of such approach, delineating future perspectives in this field of research
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