Characterization of the intestinal microbiome of Hirschsprung’s disease with and without enterocolitis

Yan, Zhilong; Poroyko, Valeriy; Gu, Song; Zhang, Zheng; Pan, Liya; Wang, Jing; Bao, Nan; Hong, Li

doi:10.1016/j.bbrc.2014.01.104

Cited by 45 publications

(48 citation statements)

References 21 publications

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“…HAEC is characterized by an acute inflammation of the crypts and intestinal epithelium mucosa [21,22], and the intestinal microbiota has been implicated in a variety of inflammatory gut disorders [23], including HAEC [4–9,16,17]. While genetic determinants of HD have also been sought [24], several studies have suggested that bacterial overgrowth or the presence of specific bacterial or viral pathogens, such as C .…”

Section: Discussionmentioning

confidence: 99%

“…While traditional microbiological techniques have been focused on studying individual potential pathogens, the development of high-throughput sequencing and metagenomics approaches has made it possible to analyze the intestinal microbiota comparatively and precisely and allowed the distribution patterns of intestinal microbiomes to be characterized and compared [12–15]. In a small preliminary study, we have previously applied the Illumina-MiSeq high-throughput sequencing platform to study the structure of intestinal microbiota in HD infants with and without enterocolitis [16]. Here, we sought to expand this approach to a larger population, to explore the characteristics of the intestinal microbiome patterns in HAEC remission (HAEC-R) patients, and to investigate the hypothesis that the intestinal microbiome differs significantly among patients with HD, depending on whether they have enterocolitis or not.…”

Section: Introductionmentioning

confidence: 99%

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Characterization of Intestinal Microbiomes of Hirschsprung’s Disease Patients with or without Enterocolitis Using Illumina-MiSeq High-Throughput Sequencing

Poroyko

Yan

et al. 2016

PLoS ONE

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Hirschsprung-associated enterocolitis (HAEC) is a life-threatening complication of Hirschsprung’s disease (HD). Although the pathological mechanisms are still unclear, studies have shown that HAEC has a close relationship with the disturbance of intestinal microbiota. This study aimed to investigate the characteristics of the intestinal microbiome of HD patients with or without enterocolitis. During routine or emergency surgery, we collected 35 intestinal content samples from five patients with HAEC and eight HD patients, including three HD patients with a history of enterocolitis who were in a HAEC remission (HAEC-R) phase. Using Illumina-MiSeq high-throughput sequencing, we sequenced the V4 region of bacterial 16S rRNA, and operational taxonomic units (OTUs) were defined by 97% sequence similarity. Principal coordinate analysis (PCoA) of weighted UniFrac distances was performed to evaluate the diversity of each intestinal microbiome sample. The microbiota differed significantly between the HD patients (characterized by the prevalence of Bacteroidetes) and HAEC patients (characterized by the prevalence of Proteobacteria), while the microbiota of the HAEC-R patients was more similar to that of the HAEC patients. We also observed that the specimens from different intestinal sites of each HD patient differed significantly, while the specimens from different intestinal sites of each HAEC and HAEC-R patient were more similar. In conclusion, the microbiome pattern of the HAEC-R patients was more similar to that of the HAEC patients than to that of the HD patients. The HD patients had a relatively distinct, more stable community than the HAEC and HAEC-R patients, suggesting that enterocolitis may either be caused by or result in a disruption of the patient’s uniquely adapted intestinal flora. The intestinal microbiota associated with enterocolitis may persist following symptom resolution and can be implicated in the symptom recurrence.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Characterization of Intestinal Microbiomes of Hirschsprung’s Disease Patients with or without Enterocolitis Using Illumina-MiSeq High-Throughput Sequencing

Poroyko

Yan

et al. 2016

PLoS ONE

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“…While the majority of clustering they observed was explained by the passage of time, they did note progression of microbiota changes from “pre-enterocolitis”, through acute HAEC, and on to remission phases. Yan, etl.al., applied a similar approach to four patients with HSCR, two with HAEC and two without[54]. They found that the patients with HAEC clustered separately from those without HAEC.…”

Section: Emerging Conceptsmentioning

confidence: 99%

Established and emerging concepts in Hirschsprung’s-associated enterocolitis

Gosain

2016

Pediatr Surg Int

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Hirschsprung’s disease (HSCR) is a common cause of neonatal bowel obstruction and the approach to diagnosis and surgical treatment is well defined and accepted. Hirschsprung’s-associated enterocolitis (HAEC) remains a frequent cause of pre-operative and post-operative morbidity and mortality, with unchanged treatment guidelines over multiple decades. Recent advances in our understanding of the genetics underlying HSCR have allowed the development of animal models, some of which recapitulate the HAEC phenotype. These animal models, along with recent translational studies, have implicated multiple facets of mucosal immunity and microbiome dysbiosis in the development of HAEC. Here, we will review the established epidemiology, modes of diagnosis and treatment of HAEC. Furthermore, we will explore emerging concepts in the pathogenesis of this disease; including animal models, alterations in mucosal immunity, dysbiosis of the intestinal microbiome, specific genetic susceptibility, and novel treatment modalities.

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“…1,2 Conversely, reduced bacterial diversity and altered bacterial composition have been found in children with diseases such as necrotizing enterocolitis (NEC) and Hirschsprung's enterocolitis. [3][4][5] Treatment of these diseases may only require antibiotics, bowel rest, and supportive care, but more severe cases require an operation with or without diversion of luminal flow. The effects of luminal diversion on the intestinal microbiome remain unknown.…”

mentioning

confidence: 99%

“…Small patient sample sizes represent another challenge to comprehensive analyses of microbiota in multiple pediatric operative diseases. 4,5,17,18 To determine the effect of operative disease and treatment on the pediatric intestinal microbiome, we compared mucosal bacteria from 43 samples by age, breastfeeding status, disease, acuity, anatomic location, and presence or absence of luminal flow. We evaluated the alpha diversity, beta diversity, and taxonomic composition of each group.…”

mentioning

confidence: 99%

The pediatric intestinal mucosal microbiome remains altered after clinical resolution of inflammatory and ischemic disease

Wieck

Debelius

Spurrier

et al. 2016

Surgery

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Characterization of the intestinal microbiome of Hirschsprung’s disease with and without enterocolitis

Cited by 45 publications

References 21 publications

Characterization of Intestinal Microbiomes of Hirschsprung’s Disease Patients with or without Enterocolitis Using Illumina-MiSeq High-Throughput Sequencing

Characterization of Intestinal Microbiomes of Hirschsprung’s Disease Patients with or without Enterocolitis Using Illumina-MiSeq High-Throughput Sequencing

Established and emerging concepts in Hirschsprung’s-associated enterocolitis

The pediatric intestinal mucosal microbiome remains altered after clinical resolution of inflammatory and ischemic disease

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