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hirschsprung's enterocolitis

M I Romo Muñoz, A Martínez de Aragón, V Núñez Cerezo, C Udaondo, M Sellers, S Barrena, M De Ceano, M López Santamaría, L Martínez Martínez
OBJECTIVES: Hirschsprung's-associated enterocolitis (HAEC) is a live-threatening complication that remains badly understood. Our objective is to identify the risk factors related to the development of HAEC in the cohort of patients with Hirschsprung's disease (HD) treated in our center. METHODS: We reviewed the patients treated for HD between 2000 and 2016. Ninety four patients were included, and the clinical details related to the disease were evaluated. Our primary outcome measure was the development of HAEC...
February 1, 2018: Cirugía Pediátrica: Organo Oficial de la Sociedad Española de Cirugía Pediátrica
Hiroki Nakamura, Christian Tomuschat, David Coyle, Anne-Marie O'Donnel, Tristan Lim, Prem Puri
AIMS AND OBJECTIVES: Hirschsprung's disease-associated enterocolitis (HAEC) is the most serious complication of Hirschsprung's disease (HSCR). HAEC occurs in 17-50% of patients with HSCR and may occur before or after a properly performed pull-through operation. The pathogenesis of HAEC is poorly understood. It is well recognized that a complex mucosal barrier protects, as the first line of defense, the surface of healthy intestinal tract from adhesion and invasion by luminal micro-organisms...
February 2018: Pediatric Surgery International
Robert O Heuckeroth
Hirschsprung disease is defined by the absence of enteric neurons at the end of the bowel. The enteric nervous system (ENS) is the intrinsic nervous system of the bowel and regulates most aspects of bowel function. When the ENS is missing, there are no neurally mediated propulsive motility patterns, and the bowel remains contracted, causing functional obstruction. Symptoms of Hirschsprung disease include constipation, vomiting, abdominal distension and growth failure. Untreated disease usually causes death in childhood because bloodstream bacterial infections occur in the context of bowel inflammation (enterocolitis) or bowel perforation...
January 4, 2018: Nature Reviews. Gastroenterology & Hepatology
Carmina Wanden-Berghe Lozano, Nuria Virgili Casas, Esther Ramos Boluda, Cristina Cuerda Compés, José Manuel Moreno Villares, José Luis Pereira Cunill, Carmen Gómez Candela, Rosa Burgos Peláez, Mª Ángeles Penacho Lázaro, Antonio Pérez de la Cruz, Julia Álvarez Hernández, Montserrat Gonzalo Marín, Pilar Matía Martín, Ceferino Martínez Faedo, Eva Ángeles Sánchez Martos, Alejandro Sanz Paris, Cristina Campos Martín, Tomás Martín Folgueras, M Ángela Martín Palmero, María de Los Ángeles Martín Fontalba, Luis Miguel Luengo Pérez, Ana Zugasti Murillo, María José Martínez Ramírez, Fátima Carabaña Pérez, Cecilia Martínez Costa, Patricia Díaz Guardiola, Cristina Tejera Pérez, Rosa Mª Parés Marimón, José Antonio Irles Rocamora, Carmen Garde Orbaiz, Miguel Ángel Ponce González, María Victoria García Zafra, Rebeca Sánchez Sánchez, Juan Ramón Urgeles Planella, Antxón Apezetxea Celaya, Olga Sánchez-Vilar Burdiel, Clara Joaquín Ortiz, José Pablo Suárez Llanos, Begoña Pintor de la Maza, Pere Leyes García, Mª Carmen Gil Martínez, Silvia Mauri Roca, María José Carrera Santaliestra
OBJECTIVE: To communicate HPN data obtained from the HPN registry of the NADYA-SENPE group ( for the year 2016. MATERIAL AND METHODS: Descriptive analysis of the data collected from adult and pediatric patients with HPN in the NADYA-SENPE group registry from January 1st, 2016 to December 31st, 2016. RESULTS: There were 286 patients from 42 Spanish hospitals (54.2% women), 34 children and 252 adults, with 294 episodes, which represent a prevalence rate of 6...
November 24, 2017: Nutrición Hospitalaria: Organo Oficial de la Sociedad Española de Nutrición Parenteral y Enteral
Yong-Zhong Mao, Shao-Tao Tang, Shuai Li
OBJECTIVE: To compare treatment outcomes in children with Hirschsprung's disease who underwent treatment using the Duhamel or TERPT surgical procedures. METHODS: Medline, Cochrane, EMBASE, and Google Scholar databases were searched through December 26, 2016. Search strings included Hirschsprung's disease, fecal incontinence, transanal endorectal pull-through, and Duhamel operation. Randomized controlled studies (RCTs) and retrospective studies that compared the treatment of Hirschsprung's disease in with TERPT or Duhamel surgical procedures in neonates, infants, or children were included...
October 16, 2017: Journal of Pediatric Surgery
Gakuto Tani, Christian Tomuschat, Anne Marie O'Donnell, David Coyle, Prem Puri
BACKGROUND: Enteric glial cells are essential for normal gastrointestinal function. Abnormalities in glial structure, development, or function lead to disturbances in gastrointestinal physiology. Fatty acid-binding protein 7 (FABP7) is a marker of immature enteric glial cells, whereas S100 is expressed only by mature glial cells. Patients with Hirschsprung's disease (HSCR) often suffer from dysmotility and enterocolitis despite proper surgery. We designed this study to determine the distribution and expression of glial cells in patients with HSCR compared to normal controls...
October 2017: Journal of Surgical Research
H Nakamura, T Lim, P Puri
BACKGROUND/PURPOSE: Hirschsprung-associated enterocolitis (HAEC) is a life-threatening complication of Hirschsprung's disease. HAEC is reported to occur in 6-50% of patients preoperatively and in 2-35% postoperatively. The exact cause of HAEC is not fully understood, but disturbances of intestinal microbiota have recently been reported in patients with HAEC. In recent years, the administration of probiotics has been proposed to reduce the incidence of HAEC. We conducted a systematic review and meta-analysis to determine the effect of probiotics on postoperative HAEC...
February 2018: Pediatric Surgery International
H Nakamura, T Lim, P Puri
AIM AND OBJECTIVES: Hirschsprung-associated enterocolitis (HAEC) continues to be an important cause of morbidity in patients with Hirschsprung's disease (HSCR). HAEC can occur at any time during the course of the disease. The reported incidence of HAEC before surgery ranges from 6 to 50%, and after surgery, it ranges from 2 to 35%. HAEC and inflammatory bowel disease (IBD) have similar clinical presentation including diarrhea, hematochezia, and abdominal pain. In recent years, isolated cases of IBD have been reported in patients who had surgical treatment for HSCR...
February 2018: Pediatric Surgery International
Wen-Kai Huang, Xue-Li Li, Jin Zhang, Shu-Cheng Zhang
BACKGROUND: Although most of patients do well after surgery for Hirschsprung disease (HSCR), there are complications in some instances that impact social aspects and quality of life. The aim of this study was to explore the prevalence, risk factors, and prognosis of these complications, providing guidance for surgeons and healthcare personnel. METHODS: A cohort of patients (N = 229) was retrospectively reviewed in the aftermath of surgery for HSCR. All medical data and operative notes were assessed...
September 27, 2017: Journal of Gastrointestinal Surgery: Official Journal of the Society for Surgery of the Alimentary Tract
Paul K H Tam, Patrick H Y Chung, Shawn D St Peter, Christopher P Gayer, Henri R Ford, Greta C H Tam, Kenneth K Y Wong, Mikko P Pakarinen, Mark Davenport
Recent developments in paediatric gastrointestinal surgery have focused on minimally invasive surgery, the accumulation of high-quality clinical evidence, and scientific research. The benefits of minimally invasive surgery for common disorders like appendicitis and hypertrophic pyloric stenosis are all supported by good clinical evidence. Although minimally invasive surgery has been extended to neonatal surgery, it is difficult to establish its role for neonatal disorders such as oesophageal atresia and biliary atresia through clinical trials because of the rarity of these disorders...
September 9, 2017: Lancet
Margarita Sellers, Clara Udaondo, Bárbara Moreno, Gonzalo Martínez-Alés, Jesús Díez, Leopoldo Martínez, María de Ceano-Vivas
INTRODUCTION: Hirschsprung-associated enterocolitis is a significant cause of morbidity and mortality in infants with Hirschsprung's disease. The fact that the symptoms are so variable and unspecific leads to a slow or incorrect diagnosis. The purpose of this study is to identify clinical factors associated with the diagnosis, as well as to evaluate the subsequent management of children with suspected Hirschsprung-associated enterocolitis in a paediatric emergency department. MATERIAL AND METHODS: A retrospective descriptive study was conducted on patients with Hirschsprung's disease who were seen in a paediatric emergency department between April 2011 and November 2015 due to clinical symptoms compatible with enterocolitis...
August 9, 2017: Anales de Pediatría: Publicación Oficial de la Asociación Española de Pediatría (A.E.P.)
Philip K Frykman, Sungjin Kim, Tomas Wester, Agneta Nordenskjöld, Akemi Kawaguchi, Thomas T Hui, Daniel H Teitelbaum, Anna L Granström, Andre Rogatko
OBJECTIVE: To identify the optimal clinical criteria to diagnose Hirschsprung-associated enterocolitis (HAEC) in children with Hirschsprung disease (HSCR). BACKGROUND: HAEC is the most common life-threatening complication in HSCR patients, yet the diagnostic criteria for HAEC remain unclear. The consensus-based HAEC scoring system was not validated using patient data, thereby making its diagnostic accuracy uncertain. METHODS: From 2009 to 2015, consecutive children with HSCR underwent retrospective evaluation of their medical records, and questionnaire-directed parent interviews to identify treatment of suspected HAEC episodes and the 16 clinical criteria in the HAEC score...
July 14, 2017: Journal of Pediatric Surgery
Isidora Galuh Parahita, Akhmad Makhmudi, Gunadi
BACKGROUND: Hirschsprung-associated enterocolitis (HAEC) represents the primary cause of high morbidity and mortality in Hirschsprung disease (HSCR) patients. The most common surgical methods for HSCR are the Soave and Duhamel procedures. Therefore, we aimed to compare the HAEC frequency following the Soave and Duhamel procedures. METHODS: Medical records were retrospectively analyzed for patients who underwent the Soave and Duhamel pull-through at Dr. Sardjito Hospital, Indonesia from 2010 to 2015...
July 17, 2017: Journal of Pediatric Surgery
Xi Zhang, Li Yang, Shao-Tao Tang, Guo-Qing Cao, Shuai Li, Meng Jiang, Meng Xiong, De-Hua Yang, Xiao-Pan Chang, Kang Li, Ya-Zhen Ma
BACKGROUND: Increased defecation frequency and soiling are common complications of surgery for right-sided Hirschsprung's disease (HD). Though the laparoscopic Duhamel procedure is a favorable option in right-sided HD, the conventional laparoscopic technique is time consuming and has complications that are associated with the reservoir. In this study, we described a modified laparoscopic Duhamel technique with ex-anal rectal transection combined with the Deloyer's procedure for right-sided HD...
September 2017: Journal of Laparoendoscopic & Advanced Surgical Techniques. Part A
Xi Zhang, Guo-Qing Cao, Shao-Tao Tang, Xiao-Pan Chang, Shuai Li, Li Yang, Kang Li, Ying Zhou, De-Hua Yang
PURPOSE: Laparoscopic-assisted Duhamel procedure has a larger anastomosis and a reservoir which allows early recovery of defecation frequency, but concerns have been raised regarding the long operative time, high incidence of pouchitis and Hirschsprung associated enterocolitis (HAEC). The purpose of this study was to evaluate the postoperative complications and functional outcomes for patients with TCA undergoing modified laparoscopic-assisted Duhamel procedure (MLDP) with ex-abdominal partial colectomy and ex-anal rectal transection...
June 27, 2017: Journal of Pediatric Surgery
Christian Tomuschat, Anne Marie O'Donnell, David Coyle, Prem Puri
PURPOSE: Hirschsprung's associated enterocolitis (HAEC) is the most common cause of morbidity and mortality in Hirschsprung's Disease (HSCR). The pathogenesis of HAEC is poorly understood. In recent years, there is increasing evidence that a compromised intestinal barrier function plays a major role in the pathogenesis of HAEC. Hydrogen sulfide, synthesized from L-cysteine by two key enzymes, cystathionine-β-synthase (CBS) and cystathionine-γ-lysase (CSE) is reported to play a key role in regulating gastrointestinal motility and promoting resolution of inflammation...
June 23, 2017: Journal of Pediatric Surgery
Satoshi Obata, Suguru Fukahori, Minoru Yagi, Makoto Suzuki, Shigeru Ueno, Kosuke Ushijima, Tomoaki Taguchi
PURPOSE: To investigate the incidence and treatment of internal anal sphincter achalasia (IASA) in Japan based on an analysis of data from a nationwide retrospective cohort study of the allied disorders of Hirschsprung's disease. METHODS: Five cases of definitive IASA were collected from a nationwide retrospective cohort study conducted from 2001 to 2010 and a search of the Japanese literature. RESULTS: Symptoms developed during the neonatal period in two patients, during early childhood in two, and at school age in one...
December 2017: Surgery Today
Siyang Cheng, Jun Wang, Weihua Pan, Wenbo Yan, Jia Shi, Wenbin Guan, Yang Wang, Wei Cai
PURPOSE: The aim of this study was to investigate the relationship between the grade of enterocolitis on pathological assessment of resected colon and postoperative bowel function in children with Hirschsprung's disease (HD). METHODS: Children with HD who were seen at a large tertiary center from January 2012 to December 2013 were enrolled into this study. Resected colon was assessed using the histopathologic grade scoring system for Hirschsprung-associated enterocolitis (HAEC), and the relationship of these scores to postoperative bowel function was assessed...
November 2017: Journal of Pediatric Surgery
Alexander W D Guillaume, Andrew C Miller, Michael C Nguyen
Hirschsprung disease is a congenital abnormality that can be surgically corrected. However, Hirschsprung-associated enterocolitis can be a life-threatening sequela. Very little has been published in the emergency medicine literature about the risk of enterocolitis and shock in patients with a history of Hirschsprung disease. We describe the case of a 6-month-old male infant with a history of multiple surgeries for Hirschsprung disease who presented to the emergency department with a seemingly benign viral gastrointestinal illness...
March 21, 2017: Pediatric Emergency Care
Kyoko Mochizuki, Masato Shinkai, Norihiko Kitagawa, Hiroshi Take, Hidehito Usui, Takashi Hosokawa, Kaori Yamoto
BACKGROUND: The purpose of this study is to assess the usefulness of continuous bowel decompression using an indwelling transanal tube (ITT) for preoperative management in infants with long-segment (L)- or total (T)-type Hirschsprung's disease (HD). CASE PRESENTATION: Between 2012 and 2015, seven patients with L- or T-type HD underwent preoperative bowel management by continuous bowel decompression using an ITT during waiting period for curative surgery. Continuous bowel decompression was done using an ITT, a 10-12F flexible dual lumen tube placed through the rectum up to the dilated colon under fluoroscopic guidance and secured to the bilateral buttocks...
December 2017: Surgical Case Reports
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