Right-sided Colonic Juvenile Polyp [Polyps] in Paediatric Primary Sclerosing Cholangitis Patients

Wyatt, Allyson; Kellermayer, Richárd

doi:10.1093/ecco-jcc/jjy090

Cited by 2 publications

(1 citation statement)

References 4 publications

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“… 1 This may be one reason for the significantly increased colorectal cancer rate in PSC-UC over non–PSC-UC. 10 Because of these clinical characteristics, novel modes of colitis control, such as vancomycin therapy, are important to investigate.…”

Section: Discussionmentioning

confidence: 99%

Microbiome Responses to Vancomycin Treatment in a Child With Primary Sclerosing Cholangitis and Ulcerative Colitis

et al. 2021

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The therapeutic effects of off-label oral vancomycin in pediatric and adult primary sclerosing cholangitis (PSC)-inflammatory bowel disease, more commonly PSC-ulcerative colitis (UC), indicate the translational relevance of disease-associated microbiome findings. This is the first report on longitudinal salivary and fecal microbiome changes in a pediatric PSC-UC patient over the first 90 days of vancomycin therapy. Increase in bacterial diversity and abundance changes in Fusobacterium , Haemophilus , and Neisseria were observed. Our findings highlight the importance of longitudinal microbiome sampling in PSC-UC and serve as a nidus for larger-scale observations toward advancing microbial therapeutics for PSC.

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Section: Discussionmentioning

confidence: 99%

Microbiome Responses to Vancomycin Treatment in a Child With Primary Sclerosing Cholangitis and Ulcerative Colitis

et al. 2021

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Identifying a therapeutic window of opportunity for people living with primary sclerosing cholangitis: Embryology and the overlap of inflammatory bowel disease with immune-mediated liver injury

Kellermayer,

Carbone,

Horvath

et al. 2024

Hepatology

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Primary sclerosing cholangitis (PSC) is a variably progressive, fibrosis-causing autoimmune disorder of the intra- and extra-hepatic bile ducts of unclear etiology. PSC is commonly (in 60-90% of cases) associated with an inflammatory bowel disease (IBD) like PSC-IBD, and less commonly with an autoimmune hepatitis (AIH) like PSC-AIH or AIH-overlap disorder. Hepatologists and Gastroenterologists often consider these combined conditions as distinctly different from the classical forms in isolation. Here, we review recent epidemiologic observations and highlight that PSC-IBD and PSC-AIH overlap appear to represent aspects of a common PSC clinico-pathological pathway and manifest in an age-of-presentation dependent manner. Particularly from the pediatric experience, we hypothesize that all cases of PSC likely originate from a complex “Early PSC”-“IBD”-“AIH” overlap in which PSC defines the uniquely and variably associated “AIH” and “IBD” components along an individualized lifetime continuum. We speculate that a distinctly unique, ‘diverticular autoimmunity’ against the embryonic cecal- and hepatic diverticulum derived tissues may be the origin of this combined syndrome, where “AIH” and “IBD” variably commence then variably fade while PSC progresses with age. Our hypothesis provides an explanation for the age-dependent variation in the presentation and progression of PSC. This is critical for the optimal targeting of studies into PSC etiopathogenesis and emphasizes the concept of a “developmental window of opportunity for therapeutic mitigation” in what is currently recognized as an irreversible disease process. The discovery of such a window would be critically important for the targeting of interventions, both administration of current therapies and therapeutic trial planning.

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Right-sided Colonic Juvenile Polyp [Polyps] in Paediatric Primary Sclerosing Cholangitis Patients

Cited by 2 publications

References 4 publications

Microbiome Responses to Vancomycin Treatment in a Child With Primary Sclerosing Cholangitis and Ulcerative Colitis

Microbiome Responses to Vancomycin Treatment in a Child With Primary Sclerosing Cholangitis and Ulcerative Colitis

Identifying a therapeutic window of opportunity for people living with primary sclerosing cholangitis: Embryology and the overlap of inflammatory bowel disease with immune-mediated liver injury

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