Bowel function and associated risk factors at preschool and early childhood age in children with anorectal malformation type rectovestibular fistula: An ARM-Net consortium study

被引:14
作者
van der Steeg, Hendrik J. J. [1 ]
van Rooij, Iris A. L. M. [2 ]
Iacobelli, Barbara D. [3 ]
Sloots, Cornelius E. J. [4 ]
Morandi, Anna [5 ]
Broens, Paul M. A. [6 ]
Makedonsky, Igor [7 ]
Leon, Francesco Fascetti [8 ]
Schmiedeke, Eberhard [9 ]
Vazquez, Araceli Garcia [10 ]
Miserez, Marc [11 ]
Lisi, Gabriele [12 ]
Midrio, Paola [13 ]
Amerstorfer, Eva E. [14 ]
Fanjul, Maria [15 ]
Ludwiczek, Johanna [16 ]
Stenstrom, Pernilla [17 ]
van der Steeg, Alida F. W. [18 ,19 ]
de Blaauw, Ivo [1 ]
ARM-Net-Consortium
机构
[1] Radboud Univ Nijmegen, Amalia Childrens Hosp, Dept Surg Pediat Surg, Med Ctr, POB 9101,Geert Grootepl Zuid 10, NL-6500 HB Nijmegen, Netherlands
[2] Radboud UMC, Radboud Inst Hlth Sci, Dept Hlth Evidence, Nijmegen, Netherlands
[3] Bambino Gesu Pediat Hosp, Res Inst, Dept Med & Surg Neonatol, Newborn Surg Unit, Rome, Italy
[4] Sophia Childrens Univ Hosp, Dept Pediat Surg, Erasmus Med Ctr, Rotterdam, Netherlands
[5] Fdn IRCCS CaGranda Osped Maggiore Policlin, Dept Pediat Surg, Milan, Italy
[6] Univ Med Ctr Groningen, Dept Surg, Div Pediat Surg, Groningen, Netherlands
[7] Childrens Hosp Dnepropetrovsk, Dept Pediat Surg, Dnepropetrovsk, Ukraine
[8] Univ Padua, Dept Pediat Surg, Padua, Italy
[9] Klinikum Bremen Mitte, Ctr Child & Youth Hlth, Dept Pediat Surg & Urol, Bremen, Germany
[10] Univ Hosp 12 Octubre, Dept Pediat Surg, Madrid, Spain
[11] Katholieke Univ Leuven, Dept Abdominal Surg, UZ Leuven, Leuven, Belgium
[12] Univ Gabriele Annunzio Chieti Pescara, St Spirito Hosp, Dept Pediat Surg, Pescara, Italy
[13] CaFoncello Hosp, Dept Pediat Surg, Treviso, Italy
[14] Med Univ Graz, Dept Pediat & Adolescent Surg, Graz, Austria
[15] Hosp Gregorio Maranon, Dept Pediat Surg, Madrid, Spain
[16] Kepler Univ Klin GmbH, Dept Pediat Surg, Linz, Austria
[17] Lund Univ, Skane Univ Hosp, Dept Pediat Surg, Lund, Sweden
[18] Univ Amsterdam, Emma Childrens Hosp, Dept Pediat Surg, Med Ctr, Amsterdam, Netherlands
[19] Princess Maxima Ctr Pediat Oncol, Dept Pediat Surg, Utrecht, Netherlands
关键词
Anorectal malformation; Rectovestibular fistula; Bowel function score; Preschool age; Early childhood; ARM-Net; Type of study; Observational cohort-study; QUALITY-OF-LIFE; ANTERIOR SAGITTAL ANORECTOPLASTY; MULTIDISCIPLINARY BEHAVIORAL TREATMENT; FECAL INCONTINENCE; TRANSANAL IRRIGATION; IMPERFORATE ANUS; SACRAL RATIO; CONTINENCE; PERINEAL; REPAIR;
D O I
10.1016/j.jpedsurg.2022.02.015
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Background: Outcome of patients operated for anorectal malformation (ARM) type rectovestibular fistula (RVF) is generally considered to be good. However, large multi-center studies are scarce, mostly describ-ing pooled outcome of different ARM-types, in adult patients. Therefore, counseling parents concerning the bowel function at early age is challenging. Aim of this study was to evaluate bowel function of RVF-patients at preschool/early childhood age and determine risk factors for poor functional outcome.Methods: A multi-center cohort study was performed. Patient characteristics, associated anomalies, sacral ratio, surgical procedures, post-reconstructive complications, one-year constipation, and Bowel Function Score (BFS) at 4-7 years of follow-up were registered. Groups with below normal (BFS < 17; subgroups 'poor' <= 11, and 'fair' 11 < BFS < 17) and good outcome (BFS >= 17) were formed. Univariable analyses were performed to detect risk factors for outcome.Results: The study included 111 RVF-patients. Median BFS was 16 (range 6-20). The 'below normal' group consisted of 61 patients (55.0%). Overall, we reported soiling, fecal accidents, and constipation in 64.9%, 35.1% and 70.3%, respectively. Bowel management was performed in 23.4% of patients. Risk factors for poor outcome were tethered cord and low sacral ratio, while sacral anomalies, low sacral ratio, prior enterostomy, post-reconstructive complications, and one-year constipation were for being on bowel man-agement.Conclusions: Although median BFS at 4-7 year follow-up is nearly normal, the majority of patients suffers from some degree of soiling and constipation, and almost 25% needs bowel management. Several factors were associated with poor bowel function outcome and bowel management. Level of Evidence: Level III.(c) 2022 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY license ( http://creativecommons.org/licenses/by/4.0/ )
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收藏
页码:89 / 96
页数:8
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