Fecal Microbiota Transplantation for Recurrent <i>Clostridioides difficile</i> Infections in a Cystic Fibrosis Child Previously Screen Positive, Inconclusive Diagnosis (CFSPID): A Case Report

<i>Clostridioides difficile</i> infection (CDI) is generally treated with vancomycin, metronidazole or fidaxomicin, although fecal microbiota transplantation (FMT) represents a promising therapeutic option for antibiotic-resistant recurrent <i>C. difficile</i> infections (rCD...

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Main Authors: Riccardo Marsiglia, Stefania Pane, Federica Del Chierico, Alessandra Russo, Pamela Vernocchi, Lorenza Romani, Sabrina Cardile, Antonella Diamanti, Luisa Galli, Agnese Tamborino, Vito Terlizzi, Paola De Angelis, Giulia Angelino, Lorenza Putignani
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Language:English
Published: MDPI AG 2024-10-01
Series:Microorganisms
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Online Access:https://www.mdpi.com/2076-2607/12/10/2059
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author Riccardo Marsiglia
Stefania Pane
Federica Del Chierico
Alessandra Russo
Pamela Vernocchi
Lorenza Romani
Sabrina Cardile
Antonella Diamanti
Luisa Galli
Agnese Tamborino
Vito Terlizzi
Paola De Angelis
Giulia Angelino
Lorenza Putignani
author_facet Riccardo Marsiglia
Stefania Pane
Federica Del Chierico
Alessandra Russo
Pamela Vernocchi
Lorenza Romani
Sabrina Cardile
Antonella Diamanti
Luisa Galli
Agnese Tamborino
Vito Terlizzi
Paola De Angelis
Giulia Angelino
Lorenza Putignani
author_sort Riccardo Marsiglia
collection DOAJ
description <i>Clostridioides difficile</i> infection (CDI) is generally treated with vancomycin, metronidazole or fidaxomicin, although fecal microbiota transplantation (FMT) represents a promising therapeutic option for antibiotic-resistant recurrent <i>C. difficile</i> infections (rCDIs) in adults. In pediatric cystic fibrosis (CF) patients, CDIs are generally asymptomatic and respond to treatment. Here, we present the case of an 8-year-old female, initially diagnosed as “CFTR-related metabolic syndrome/cystic fibrosis screen positive, inconclusive diagnosis” (CMRS/CFSPID), who then progressed to CF at 12 months. In the absence of CF-related symptoms, she presented multiple and disabling episodes of bloody diarrhoea with positive tests for <i>C. difficile</i> antigen and A/B toxin. After conventional treatments failed and several CDI relapses, FMT was proposed. Donor screening and GM donor–receiver matching identified her mother as a donor. Metataxonomy and targeted metabolomics provided, through a pre- and post-FMT time course, gut microbiota (GM) profiling to assess GM engraftment. At first, the GM map revealed severe dysbiosis, with a prevalence of Bacteroidetes and Proteobacteria (i.e., <i>Klebsiella</i> spp., <i>Escherichia coli</i>), a reduction in Firmicutes, a GM nearly entirely composed of Enterococcaceae (i.e., <i>Enterococcus</i>) and an almost complete depletion of Verrucomicrobia and Actinobacteria, mostly represented by <i>Veillonella dispar</i>. Post FMT, an increment in <i>Bifidobacterium</i> spp. and <i>Collinsella</i> spp. with a decrease in <i>V. dispar</i> restored intestinal eubiosis. Consistently, four weeks after FMT treatment, the child’s gut symptoms cleared, without CDI recurrence.
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spelling doaj-art-b017e5ba5b5b40d0bba1001ab3ef62902025-08-20T02:11:00ZengMDPI AGMicroorganisms2076-26072024-10-011210205910.3390/microorganisms12102059Fecal Microbiota Transplantation for Recurrent <i>Clostridioides difficile</i> Infections in a Cystic Fibrosis Child Previously Screen Positive, Inconclusive Diagnosis (CFSPID): A Case ReportRiccardo Marsiglia0Stefania Pane1Federica Del Chierico2Alessandra Russo3Pamela Vernocchi4Lorenza Romani5Sabrina Cardile6Antonella Diamanti7Luisa Galli8Agnese Tamborino9Vito Terlizzi10Paola De Angelis11Giulia Angelino12Lorenza Putignani13Immunology, Rheumatology and Infectious Diseases Research Area, Unit of Research Microbiome, Bambino Gesù Children’s Hospital, IRCCS, 00146 Rome, ItalyUnit of Microbiomics, Bambino Gesù Children’s Hospital, IRCCS, 00146 Rome, ItalyImmunology, Rheumatology and Infectious Diseases Research Area, Unit of Research Microbiome, Bambino Gesù Children’s Hospital, IRCCS, 00146 Rome, ItalyUnit of Microbiomics, Bambino Gesù Children’s Hospital, IRCCS, 00146 Rome, ItalyImmunology, Rheumatology and Infectious Diseases Research Area, Unit of Research Microbiome, Bambino Gesù Children’s Hospital, IRCCS, 00146 Rome, ItalyInfectious Diseases Unit, Bambino Gesù Children’s Hospital, IRCCS, 00146 Rome, ItalyUnit of Gastroenterology and Nutrition, Bambino Gesù Children’s Hospital, IRCCS, 00165 Rome, ItalyUnit of Gastroenterology and Nutrition, Bambino Gesù Children’s Hospital, IRCCS, 00165 Rome, ItalyDepartment of Health Sciences, University of Florence, 50121 Florence, ItalyInfectious Disease Unit, Meyer Children’s Hospital IRCCS, 50121 Florence, ItalyDepartment of Pediatric Medicine, Meyer Children’s Hospital IRCCS, Cystic Fibrosis Regional Reference Center, Viale Gaetano Pieraccini 24, 50139 Florence, ItalyUnit of Gastroenterology and Nutrition, Bambino Gesù Children’s Hospital, IRCCS, 00165 Rome, ItalyUnit of Gastroenterology and Nutrition, Bambino Gesù Children’s Hospital, IRCCS, 00165 Rome, ItalyUnit of Microbiomics and Unit of Research of Microbiome, Bambino Gesù Children’s Hospital, IRCCS, 00146 Rome, Italy<i>Clostridioides difficile</i> infection (CDI) is generally treated with vancomycin, metronidazole or fidaxomicin, although fecal microbiota transplantation (FMT) represents a promising therapeutic option for antibiotic-resistant recurrent <i>C. difficile</i> infections (rCDIs) in adults. In pediatric cystic fibrosis (CF) patients, CDIs are generally asymptomatic and respond to treatment. Here, we present the case of an 8-year-old female, initially diagnosed as “CFTR-related metabolic syndrome/cystic fibrosis screen positive, inconclusive diagnosis” (CMRS/CFSPID), who then progressed to CF at 12 months. In the absence of CF-related symptoms, she presented multiple and disabling episodes of bloody diarrhoea with positive tests for <i>C. difficile</i> antigen and A/B toxin. After conventional treatments failed and several CDI relapses, FMT was proposed. Donor screening and GM donor–receiver matching identified her mother as a donor. Metataxonomy and targeted metabolomics provided, through a pre- and post-FMT time course, gut microbiota (GM) profiling to assess GM engraftment. At first, the GM map revealed severe dysbiosis, with a prevalence of Bacteroidetes and Proteobacteria (i.e., <i>Klebsiella</i> spp., <i>Escherichia coli</i>), a reduction in Firmicutes, a GM nearly entirely composed of Enterococcaceae (i.e., <i>Enterococcus</i>) and an almost complete depletion of Verrucomicrobia and Actinobacteria, mostly represented by <i>Veillonella dispar</i>. Post FMT, an increment in <i>Bifidobacterium</i> spp. and <i>Collinsella</i> spp. with a decrease in <i>V. dispar</i> restored intestinal eubiosis. Consistently, four weeks after FMT treatment, the child’s gut symptoms cleared, without CDI recurrence.https://www.mdpi.com/2076-2607/12/10/2059cystic fibrosis (CF)cystic fibrosis screen positiveinconclusive diagnosis (CFSPID)recurrent <i>Clostridioides difficile</i> infections (rCDIs)fecal microbiota transplantation (FMT)gut microbiota (GM)
spellingShingle Riccardo Marsiglia
Stefania Pane
Federica Del Chierico
Alessandra Russo
Pamela Vernocchi
Lorenza Romani
Sabrina Cardile
Antonella Diamanti
Luisa Galli
Agnese Tamborino
Vito Terlizzi
Paola De Angelis
Giulia Angelino
Lorenza Putignani
Fecal Microbiota Transplantation for Recurrent <i>Clostridioides difficile</i> Infections in a Cystic Fibrosis Child Previously Screen Positive, Inconclusive Diagnosis (CFSPID): A Case Report
Microorganisms
cystic fibrosis (CF)
cystic fibrosis screen positive
inconclusive diagnosis (CFSPID)
recurrent <i>Clostridioides difficile</i> infections (rCDIs)
fecal microbiota transplantation (FMT)
gut microbiota (GM)
title Fecal Microbiota Transplantation for Recurrent <i>Clostridioides difficile</i> Infections in a Cystic Fibrosis Child Previously Screen Positive, Inconclusive Diagnosis (CFSPID): A Case Report
title_full Fecal Microbiota Transplantation for Recurrent <i>Clostridioides difficile</i> Infections in a Cystic Fibrosis Child Previously Screen Positive, Inconclusive Diagnosis (CFSPID): A Case Report
title_fullStr Fecal Microbiota Transplantation for Recurrent <i>Clostridioides difficile</i> Infections in a Cystic Fibrosis Child Previously Screen Positive, Inconclusive Diagnosis (CFSPID): A Case Report
title_full_unstemmed Fecal Microbiota Transplantation for Recurrent <i>Clostridioides difficile</i> Infections in a Cystic Fibrosis Child Previously Screen Positive, Inconclusive Diagnosis (CFSPID): A Case Report
title_short Fecal Microbiota Transplantation for Recurrent <i>Clostridioides difficile</i> Infections in a Cystic Fibrosis Child Previously Screen Positive, Inconclusive Diagnosis (CFSPID): A Case Report
title_sort fecal microbiota transplantation for recurrent i clostridioides difficile i infections in a cystic fibrosis child previously screen positive inconclusive diagnosis cfspid a case report
topic cystic fibrosis (CF)
cystic fibrosis screen positive
inconclusive diagnosis (CFSPID)
recurrent <i>Clostridioides difficile</i> infections (rCDIs)
fecal microbiota transplantation (FMT)
gut microbiota (GM)
url https://www.mdpi.com/2076-2607/12/10/2059
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