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- Springer Journals
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- Copyright © The Author(s) 2023
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- 2090-5394
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- 10.1186/s43159-023-00238-y
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Abstract
Background Transanal endorectal pull-through ( TEPT ) surgery is a new trend in the treatment of Hirschsprung dis- ease, and evaluating its functional outcome is difficult. The purpose of this study is to evaluate the defecation pattern after TEPT surgery in pediatrics. Material and methods In this cross-sectional survey, 40 patients with Hirschsprung disease were studied. They underwent a one-stage transanal pull-through procedure from May 2007 till April 2015 in Namazi hospital by the same surgeon. All the patients had the aganglionic segment in the rectosigmoid, confirmed by pre-operation barium enema and post-operation histopathology. The patients were 40 children with mean operation age of 36.6 months old. The success rate of surgery was evaluated by following the questionnaire form. Clinical evaluation with bowel function score and anorectomanometry were carried out. Result One hundred percent of them had a daily stool. Eighty-five percent of patients never experienced pain during defecation, 10% experienced occasional pain, and the rest felt pain with every defecation. Just one case (5%) reported using a laxative. Regarding improvements after the operation, 75% were completely okay, 20% felt much better, and 5% reported some improvements. The average National Health Service score was 8.375, which seems satisfactory. The mean average anal sphincter in the group with soiling was 39.67 and in the group without soiling was 34.83, which is in the normal range in both groups, and there was no significant difference between these groups (P > 0.05). Conclusion The defecation pattern after TEPT surgery were satisfactory in almost all cases, even in infancy. Most patients had satisfactory manometry and clinical result. Keywords Hirschsprung disease, Transanal endorectal pull-through, Manometry, RAIR *Correspondence: Department of Pediatrics, School of Medicine, Zahedan University Farhad Homapour of Medical Sciences, Zahedan, Iran fhomapoor@gmail.com Student Research Committee, Gonabad University of Medical Sciences, Department of Pediatric Surgery, Namazi Hospital, Shiraz University Gonabad, Iran Laparoscopy Research Center, Shiraz University of Medical Sciences, Shiraz, Iran Oncosurgery Fellowship, Department of Surgery, School of Medicine, Shiraz University of Medical Sciences, PO Box: 9691793718, Shiraz, Iran Departments of Pediatric Gastroenterology, Gastroenterohepatology Research Center, Shiraz University of Medical Sciences, Shiraz, Iran Breast Diseases Research Center, Shiraz University of Medical Sciences, Shiraz, Iran © The Author(s) 2023. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http:// creat iveco mmons. org/ licen ses/ by/4. 0/. Foroutan et al. Annals of Pediatric Surgery (2023) 19:6 Page 2 of 6 nerve supply damage due to anal sphincter stretching, Background and nerve ending damage due to distal anorectal anas- Hirschsprung disease is a congenital intestinal disorder tomosis near the dentate line. Common postoperative characterized by the absence of intrinsic ganglion cells complications of Hirschsprung disease include bowel in the myenteric and submucosal plexuses of the distal obstruction, enterocolitis, and fecal incontinence. Hence, intestine [1]. Since these cells are in charge of normal anorectal manometry have been used to work up patients peristalsis, patients with Hirschsprung disease manifest for these complications [12]. intestinal obstruction at the level of aganglionosis dur- Anorectal manometry has been used to study the phys- ing the newborn period or with constipation in infancy iology of the anorectum. It can evaluate the resting anal [2]. Hirschsprung disease is approximately 1 in 5000 pressure, rectoanal inhibitory reflex, and anorectal sensa - live-born infants. In most cases, patients present with tion [16]. However, its application for post-TEPT opera- obstructive intestinal symptoms such as failure to pass tion assessment has not been established yet. For this meconium within the first 48 h, abdominal distension, or purpose, we conducted a study to validate the use of ano- bilious vomiting [3]. Aganglionosis mainly involves the rectal manometry as a standard tool for assessment after rectum or rectosigmoid, even though the entire colon TEPT operation. With clinical assessment, factors affect - or small intestine may be involved in some cases. The ing the prognosis of this group of patients were analyzed. diagnosis of Hirschsprung disease relies on histological Therefore, this study aimed to evaluate the defecation examination of the rectal biopsy specimen. Also, there pattern after TEPT surgery in pediatrics. are other methods for diagnosing Hirschsprung disease, such as anorectal manometry and water-soluble contrast Materials and methods enema [4–6]. In a cross-sectional study, 182 patients with The recto-anal inhibitory reflex (RAIR) is a noninvasive Hirschsprung disease diagnosis were included in the test to assess anorectal physiology without the require- present study. They underwent a one-stage transanal ment of anesthesia. It is a reflex relaxation of the internal pull-through procedure from May 2007 till April 2015 anal sphincter in response to rectal distension. RAIR is in Namazi hospital by the same surgeon. The exclusion absent in children with Hirschsprung disease because the criteria were (1) patients with severe learning difficul - aganglionic segment impairs the relaxation of the inter- ties (n = 2), (2) patients with an inability to cooperate nal sphincter due to rectal distention [7]. during the study (n = 4), (3) patients with concomitant In most cases, treatment of Hirschsprung disease is anorectal/neurological anomaly (n = 2), (4) patients who surgical. The operative principle for Hirschsprung disease required reoperation or had laparotomy due to failure of is to remove the aganglionic segment and reconstruct the previous procedure (n = 2), and (5) patients with the the intestinal tract by attaching the normally innervated previous colostomy before operation (n = 2). From this bowel to the anus while preserving normal sphincter population, 130 patients were unwilling to participate in function [8–10]. Many types of pull-through procedures the study and we could enroll just 40 patients who had all have been established worldwide, and the most com- of the mentioned criteria. After the operation and during monly performed ones are the Swenson, Duhamel, and outpatient department follow-up, we asked their parents Soave (endorectal pull-through) procedures [11]. De La to sign the enrollment consent and fill out our question - Torre-Mondragon introduced the transanal modified naire forms. The present study was approved by the ethic Soave approach and Transanal endorectal pull-through and research committees of the participating hospital. (TEPT) in 1998 [12]. TEPT procedure protects the The success rate of surgery was evaluated by following patient from colostomy complications and has several the questionnaire form. Clinical evaluation with bowel advantages compared to classical pull-through tech- function score and anorectomanometry were carried out. niques, such as shorter hospital stays, postoperative ileus, Sphincter resting pressure between 30 mmHg and 60 bleeding, and pain. At the same time, no intraperitoneal mmHg was considered normal. adherence or scarring is reported. TEPT has proved Patients received standard postoperative care, includ- to be as beneficial as the older multi-stage procedures ing wound management, and were eventually required since patients benefit from fewer operations and reduced to start enteral feeding according to the individual’s healthcare costs due to shorter hospital stays [13]. bowel recovery. Anal dilatation was started in the early After resectioning, the aganglionic colorectum, sur- postoperative period. Patients’ caregivers were taught gery for Hirschsprung disease, endorectal or retrorectal the technique of anal dilatation and how to perform the pull-through, and coloanal anastomosis surgery consists procedure when patients were discharged from the hos- of colon mobilization [14, 15]. Pelvic nerve injury is one pital. They were followed up regularly at the outpatient of the rare complications of TEPT surgery. However, this clinic. For those who consented to participate in this procedure can cause incontinence by sphincter injuries, F oroutan et al. Annals of Pediatric Surgery (2023) 19:6 Page 3 of 6 study, a standard questionnaire, the modified form of calculated, and it is considered significant if it is lower the questionnaire used in Keshtgar et al.’s study, which than 0.05. was validated in 2000 patients [9, 17], regarding their bowel functions, including frequency of bowel motion, the presence of constipation or soiling and pain during Results defecation, general health improvement was completed The patients were 40 children with a mean operation during the interview and named NHS (National Health age of 36.6 months (max: 11 years old and min: 25 days Service) score (A1), which is provided in the Additional old, neonates: two patients) (Table 1). Thirty of them file 1. In our study, patients underwent the test in Namazi (75%) were male, and 10 of them (25%) were female, hospital, where the primary operation was carried out. with a mean range of 4-year follow-ups (Table 1). With a During the manometry assessment, a well-lubricated, mean range of 4-year follow-up after the operation, 50% water-perfused silicone manometric catheter with side of patients never experienced fecal soiling, 15% of them openings and a distensible balloon at its tip was inserted experienced occasional fecal soiling, 15% had soiling only into the rectum. A change in pressure was detected by a if bowel loaded, 10% had occasional soiling, and the rest transducer, which converted the results into the connect- had continuous fecal soiling day and night (Fig. 1). ing computer. Various parameters of age-appropriated One hundred percent of them had daily stool (Table 1). anorectal physiology, such as sphincteric resting pressure Eighty-five percent never experienced pain during defe - and the presence of rectoanal inhibitory reflex (RAIR), cation, 10% experienced occasional pain, and the rest felt were recorded. No sedation was given to any patient, pain with every defecation. Just two cases (5%) reported and the procedure lasted approximately 5–10 min. All using a laxative. Regarding general health conditions patients were discharged after the procedure without any after the operation, 85% were well, 15% were occasionally complications. ill, and 5% were often ill. There is a lack of standard reference for average RAIR About the behavior related to bowel problems, 80% value in children. In the current study, normal sphinc- were cooperative, and 20% required reminding to use the teric resting pressure is considered between 30 mmHg lavatory or pot. Regarding improvements after the opera- and 60 mmHg, based on two previous studies regarding tion, 75% were completely okay, 20% felt much better, anorectal manometry in a pediatric population [9, 10]. A and 5% reported some improvements. In follow-up DRE value lower or higher than this range would be consid- (digital rectal exam), no fecal impaction was noted in ered sphincter hypotonicity or hypertonicity. For positive any patients. Also, it should be noted that all the studied RAIR, it was defined as a drop-in sphincter pressure for at least 15 mmHg for 5 s when the balloon at the catheter tip was inflated. After collecting data from questionnaire forms and anorectal manometry, patients were divided into two groups regarding if they had any defecation complaints such as soilage, constipation, or incontinence. Then, average anal sphincter resting pressure and RAIR were compared between these two groups. It should be noted that in patients with soling the pseudo- inconti- nence was excluded through clinical evaluation of anal sphincter. Data were collected and analyzed using the software Graphpad Prism Version 6 in a two-tailed t test regarding their average resting pressure and RAIR. P values were Fig. 1 Soilage rate after TEPT procedure Table 1 The descriptive analysis of the studied patients Variables Population: n = 40 Age at operation Mean: 36.6 months/old Min 25 days/old Max 11 years/old Sex Male; N = 30 Female; n = 10 Daily stool 100%; N = 40 Pain during defecation None; N = 34 Occasionally N = 4 Every time; n = 2 Foroutan et al. Annals of Pediatric Surgery (2023) 19:6 Page 4 of 6 patients had adequate length of anal mucosa above den- sexual dysfunction [25]. A very distal anorectal anasto- tate line after the TEPT operation. mosis near the dentate line may also cause nerve-ending The average NHS was 8.375, which seems to be a sat - damage leading to incontinence [26–29]. isfactory score. The mean average anal sphincter in the Although the TEPT procedure has shown clear advan- group with soiling was 39.67 and in the group without tages over other procedures, there is a lack of evidence, soiling was 34.83, which is in the normal range in both particularly concerning the long-term continence out- groups, and there was no significant difference between comes [19]. This study aimed to evaluate the defecation these groups (P > 0.05) (Fig. 2). Since all the patients had pattern after the TEPT surgery. For this purpose, we histopathologic confirmation of aganglionosis, RAIR was conducted a study with a mean range of 4-year follow- absent in the studied patients (Table 1). up after operation. In the present study, the results were gathered from one pediatric surgical center in Shiraz. Discussion In the current study, there were more male than female Swenson and Duhamel are two traditional procedures patients, which was expected due to the modality of for treating Hirschsprung disease, and their outcome Hirschsprung disease (75% male) [30–32]. In the current did not fulfill surgeons’ expectations [18]. Nowadays, study, the frequency of fecal soiling, a common compli- TEPT has become the preferable procedure for treating cation after the TEPT procedure, was 50%. Fortunately, Hirschsprung disease [19]. Like any other surgical proce- only 4 out of 40 patients experienced soiling continu- dure, TEPT has some pros and cons. ously, nearly the same as international statics reported in According to past studies, the TEPT procedure has the study by Cheng Zhang [33]. In a study by Till et al. some advantages, including minimal invasion and a [34], the manometric assessment after TEPT operation shorter duration of surgery. Dissections are being per- was reported to be favorable. They suggested that the formed transanally. Thus, in most cases, no abdomi - functional integrity of the anorectal sphincter complex nal incision is needed. Primary laparotomy is evitable could be preserved. In another study by Soo-Hong et al. in many cases [20–22]. Consequently, it does not leave [35], they evaluated the mid- and long-term outcomes of any abdominal scar. Also, postoperative ileus, abdomi- Transanal single-stage endorectal pull-through (TERPT). nal contamination, and intestinal adhesions are much They demonstrated that the functional outcomes of lesser than in other procedures [23]. TEPT procedure TERPT performed during the infantile period, were simi- can be performed in the early infantile period, and lar to that of the normal population [35]. patients recover shortly after the surgery [24]. Unlike the Duhamel procedure, a lesser residual aganglionic seg- Conclusion ment in the TEPT procedure leads to a lesser functional The current study indicates that the defecation pattern obstruction resulting in better postoperative bowel func- after TEPT surgery were satisfactory in almost all cases, tion. However, the TEPT procedure also comes with even in infancy. Also, most patients had satisfactory some disadvantages, including the possibility of injury to manometry and clinical result after the TEPT surgery. the anal sphincter and its nerve supply and pelvic or gen- In addition, further studies with longer follow-ups and itourinary nerve injury, which leads to incontinence and larger study population are suggested. Fig. 2 Average resting pressure in patients with soilage and without soilage F oroutan et al. Annals of Pediatric Surgery (2023) 19:6 Page 5 of 6 7. Wu J-F, Lu C-H, Yang C-H, Tsai I-J. Diagnostic role of anal sphincter relaxa- tion integral in high-resolution anorectal manometry for hirschsprung disease in infants. J Pediatr. 2018;194:136–41.e2. Abbreviations 8. Pakarinen M. Perioperative complications of transanal pull-through DRE Digital rectal exam surgery for Hirschsprung’s disease. Eur J Pediatr Surg. 2018;28(2):152–5. NHS National Health Service 9. Keshtgar A, Ward H, Clayden G, de Sousa N. Investigations for incon- RAIR Rectoanal inhibitory reflex tinence and constipation after surgery for Hirschsprung’s disease in TEPT Transanal endorectal pull-through children. Pediatr Surg Int. 2003;19(1-2):4–8. 10. Zhang SC, Bai YZ, Wang W, Wang WL. Stooling patterns and colonic motil- Supplementary Information ity after transanal one-stage pull-through operation for Hirschsprung’s disease in children. J Pediatr Surg. 2005;40(11):1766–72. The online version contains supplementary material available at https:// doi. 11. Neuvonen MI, Kyrklund K, Rintala RJ, Pakarinen MP. 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Journal
Annals of Pediatric Surgery – Springer Journals
Published: Jan 10, 2023
Keywords: Hirschsprung disease; Transanal endorectal pull-through; Manometry; RAIR