Yee Ian Yik a,b,c, L.C.Y. Leong b, John M. Hutson a,b,d,, Bridget R. Southwell a

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1 Journal of Pediatric Surgery (2012) 47, The impact of transcutaneous electrical stimulation therapy on appendicostomy operation rates for children with chronic constipation a single-institution experience Yee Ian Yik a,b,c, L.C.Y. Leong b, John M. Hutson a,b,d,, Bridget R. Southwell a a F Douglas Stephens Surgical Research and Gut Motility Laboratories, Murdoch Children's Research Institute, Melbourne, Australia b Department of Paediatrics, University of Melbourne, Australia c Department of General Surgery, Faculty of Medicine, University of Malaya, Kuala Lumpur, Malaysia d Royal Children's Hospital, Melbourne, Australia Received 11 August 2011; revised 13 January 2012; accepted 15 January 2012 Key words: Chronic constipation; Slow-transit constipation (STC); Appendicostomy; Antegrade continence enema (ACE); Transcutaneous electrical stimulation (TES); Nuclear transit scintigraphy (NTS) Abstract Purpose: Appendicostomy for antegrade continence enema is a minimally invasive surgical intervention that has helped many children with chronic constipation. At our institution, since 2006, transcutaneous electrical stimulation (TES) has been trialed to treat slow-transit constipation (STC) in children. This retrospective audit aimed to determine if TES use affected appendicostomy-formation rates and to monitor changes in practice. We hypothesized that appendicostomy rates have decreased for STC but not for other indications. Methods: Appendicostomy-formation rate was determined for the 5 years before and after Children were identified as STC or non-stc from nuclear transit scintigraphy and patient records. Results: Since 1999, 317 children were diagnosed with STC using nuclear transit scintigraphy with 121 during 2001 to 2005 (24.2/year) and 147 during 2006 to 2010 (29.4/year). Seventy-four children had appendicostomy formation. For 2001 to 2005, appendicostomy-formation rates for STC and non-stc children were similar: 5.4 per year (n = 27) and 4.8 per year (n = 24), respectively. For 2006 to 2010, appendicostomy-formation rates were 1.2 per year (n = 6) for STC and 3.2 per year (n = 16) for non- STC (χ 2, P =.04). Conclusion: Since 2006, appendicostomy-formation rates have significantly reduced in STC but not in non-stc children at our institute, coinciding with the introduction of TES as an alternative treatment for STC. Transcutaneous electrical stimulation has not been tested on non-stc children in this period. Crown Copyright 2012 Published by Elsevier Inc. All rights reserved. Corresponding author. Department of Urology, Royal Children's Hospital, Parkville, Victoria 3052, Australia. Tel.: ; fax: address: john.hutson@rch.org.au (J.M. Hutson). Chronic constipation has major psychosocioeconomic impacts on affected families. In children, it accounts for 3% to 5% of visits to pediatricians and 10% to 25% to pediatric gastroenterologists [1,2]. If left untreated, it severely impairs the quality of life, and one third of children will have /$ see front matter. Crown Copyright 2012 Published by Elsevier Inc. All rights reserved. doi: /j.jpedsurg

2 1422 Y.I. Yik et al. persisting symptoms into adulthood [2-5]. There is no standard therapy for chronic constipation despite the varieties of medication currently available. Surgery is the last resort offered to these patients to overcome their intractable symptoms and to improve quality of life. Since its description by Malone et al [6] in 1990, the cecostomy with antegrade continence enema (ACE) has been considered as a less invasive surgical option in the treatment of chronic constipation. Before that, numerous extensive surgical procedures have been described, and most of them were associated with high morbidities and high rates of symptom recurrence [7-12]. Antegrade continence enema is considered in the treatment of patients with severe symptoms of chronic constipation, particularly for soiling and incontinence. Appendicostomy formation with ACE has been described in both adults and children and is well tolerated as a treatment for chronic constipation. At our institute, the appendix is used as a conduit, and the stoma is created laparoscopically [13,14]. Appendicostomy has become our preferred procedure to treat children with symptoms of intractable chronic constipation, both for slow-transit constipation (STC) and non-stc patients (including those with persisting symptoms after surgery for anorectal malformation or Hirschsprung disease). Our review in 2005 showed that ACE provided good management in 81% of 42 patients with STC [15]. Recently we began using transcutaneous electrical stimulation (TES) as an alternative treatment to appendicostomy for children with STC since 2006 [16-20]. Transcutaneous electrical stimulation (a noninvasive form of electrical stimulation) using interferential current has been used by physiotherapists to treat painful musculoskeletal conditions and bladder incontinence. Improved symptoms and quality of life in STC children after TES have been reported by our group [16-20]. We aimed to determine the operation rates for appendicostomy before and after the introduction of TES to audit the changes in practice. We hypothesized that the rates for appendicostomy would decrease for STC children after the introduction of TES, if the symptoms were successfully treated. 1. Methods This is a retrospective review of the treatment options (appendicostomy [13-15] vs TES) taken by children having intractable chronic constipation in a single tertiary institution. From 1999 to 2010, STC was diagnosed by nuclear transit scintigraphy (NTS), as holdup of radioactivity in the transverse colon [21-23], specifically if there was greater than 40% radiotracer retained in the transverse colon at 24 hours and/or greater than 30% at 48 hours or with mean geometric center of less than 2.7 and/or less than 3.7 at 24 and 48 hours, respectively. Children with intractable chronic constipation (N2 years) attending gastroenterology or medical clinics were referred to a surgeon (JMH) with a special interest in this subject. After the diagnosis of STC had been made (clinical presentation and confirmed on NTS), options of treatment were discussed and offered to parents and child. The options offered were appendicostomy and/or TES. The choice of therapy was decided by the parents/child. Detailed discussion on the management plans for the child's chronic constipation was conducted by JMH, and the decision made for ACE formation if this option will help to improve the child's symptoms and with the parent/child committed to the treatment. Moreover, ACE was considered as a surgical option to improve the child's quality of life if medical therapies had failed. These children were on regular followups, at 3 to 6 monthly intervals. Since 2006, TES began being offered as an alternative treatment for children with STC, avoiding surgery in some. However, for those who did not respond to TES, ACE was offered after TES with the aim to improve their symptoms. Appendicostomy-formation rate was determined by retrospective review of operation and medical records for all patients in our institute for the 5 years before and after 2006 in 2 subgroups (STC and non-stc), by YIY. Statistical analysis was performed using χ 2 test, and P b.05 was considered as significant. 2. Results In the non-stc group (n = 40, Table 1), the indications for appendicostomy formation were anorectal retention with fecal incontinence, neurogenic bowel, previous anorectal malformation, and cystic fibrosis. For non-stc children, appendicostomy formation was 4.8 cases per year (n = 24) from 2001 to 2005 and 3.2 cases per year (n = 16) from 2006 to 2010 (Fig. 1 and Table 2). Since 1999, 317 children were diagnosed with STC using NTS (Table 2) with 49 cases from 1999 to 2000, 121 cases from 2001 to 2005 (24.2 cases per year), and 147 cases from 2005 to 2010 (29.4 cases per year). Seventy-three children had appendicostomy formation, with 33 in STC children and 40 in non-stc children. The appendicostomy formation in STC patients was 5.4 cases per year (n = 27) from 2001 to 2005, significantly lower with 1.2 cases per year (n = 6) from 2006 to 2010 (P b.05) (Fig. 1, Table 2). The decrease of appendicostomy-formation rate in STC children was also significantly different from the non-stc Table 1 Indications for appendicostomy in non-stc children from 2001 to 2010 Indication Anorectal retention/functional fecal retention 27 Neurogenic bowel 7 Anorectal malformation 5 Cystic fibrosis 1 Total 40 No. of patients

3 The impact of TES theraphy on appendicostomy operation rates 1423 From 2006 to 2010, 2 of these 134 STC children with continued poor quality of life had appendicostomy formation after using TES for 3 to 6 months, without improvement of their symptom of soiling. In the 33 STC children who had appendicostomy formation from 2001 to 2010, 4 children went on to have colostomy formation, and 2 children had sigmoid colectomy (3 colostomy formation and 1 sigmoid colectomy in and 1 colostomy formation and 1 sigmoid colectomy in ). 3. Discussion Fig. 1 Rate of appendicostomy formation for children with chronic constipation before and after the introduction of TES therapy from 2001 to group (χ 2, P =.04). The recruitment rate for TES therapy for STC children averaged at 27 cases per year (total = 134) from 2006 to At our institute, appendicostomy for ACE has been offered since the 1990s as a surgical treatment in children with symptoms of intractable chronic constipation not responding to medical therapies [13-15]. There was an initial rush in appendicostomy formation in 2000 to 2001 followed by a reduction in number. It was performed almost equally in STC and non-stc children to 2006 (Fig. 1). Our most recent longterm follow-up study has demonstrated the reduction of laxative use and ACE washouts in STC children after TES [24]. Since 2006, the appendicostomy formation rate has fallen significantly, and this coincides with the introduction of TES therapy as an alternative treatment for children with STC. Transcutaneous electrical stimulation was found to be effective in overcoming the symptoms of chronic constipation in STC children, producing improved quality of life [17] as well as increased colonic transit measured objectively by NTS [18], and also improving colonic contractions measured by colonic manometry (unpublished). Because it is noninvasive, TES is offered and accepted as an alternative treatment before considering surgical intervention with 27 patients per year having TES compared with 5 patients per year having appendicostomy formation. Over the most recent 5 years ( ), 2 of 134 patients had appendicostomy formation after TES. With the increasing success of TES as a promising new noninvasive option to treat STC, it is being considered before appendicostomy or other surgery. Surgery is the last resort to treat chronic constipation and should only be considered if medical therapy has failed. Until recently, conventional surgical management of treatmentresistant constipation comprised either a bowel resection (colectomy) and ileorectal/cecorectal/ileosigmoid anastomosis or a segmental colectomy, with or without the formation of a stoma [7-10,25-34]. The incidence of surgical intervention was almost similar to the prevalence of STC [25,27,35,36], which often failed to respond to aggressive medical therapy. These procedures are invasive and carried with them high morbidity and mortality as frequently reported [7-10,32,37,38]. In addition, favorable long-term outcomes have been disappointing in most studies [39-42]. In recent years, however, the use of a continent appendix stoma as first described by Malone et al [6,43] has become

4 1424 Y.I. Yik et al. Table 2 Slow-transit constipation cases diagnosed by NTS and appendicostomy formation for children with chronic constipation from 2001 to 2010 Study period STC cases Total New cases per year (mean ± SEM) Appendicostomy STC Non- STC TOTAL Formation rate per year (mean ± SEM) STC Non-STC ± ± ± 2.0 Introduction of TES therapy ± ± ± 0.73 SEM indicates standard error of mean. widespread for the management of incontinence after surgery for anorectal malformation. It was also found to be effective in treating fecal incontinence and intractable constipation [13-15,31,33,44-59]. Electrical stimulation therapy is a promising new treatment for chronic constipation, both in adults and children [16-20,60-64]. It can be delivered via the direct route (invasive) with electrode implantation in the form of sacral nerve stimulation (SNS) or the indirect route (noninvasive) with pad electrode placement on the skin surface over the abdomen and the paraspinal region [16,19,20]. With the use of noninvasive TES therapy in our institute, surgical resection of bowel is now reserved as the last option for patients not responding to exhaustive conservative measures. Moreover, with less surgery involved in the treatment for chronic constipation, the associated morbidities are expected to reduce, with less cost of providing the care of the related complications. Transcutaneous electrical stimulation therapy is very promising but still in an experimental stage. With more patients responding to treatment and with better understanding on how to monitor treatment response and establishing the long-term effects of TES on follow-up, we hope to establish the effectiveness and the safety profile of this treatment. This will determine whether TES is a realistic alternative to surgery for children with chronic constipation. Transcutaneous electrical stimulation is currently offered to STC children in our hospital, and we are conducting a pilot study to test its effects in treating children with anorectal retention, the more common form of chronic constipation. With other centers showing interest in using TES to treat chronic constipation, we hope that a multicenter study will be possible. This study is based on 10 years' experience of a single institute and has shown that since the introduction of TES, it has reduced our rate of surgical intervention for children with STC, consistent with the early promising results in our trials. For fecal incontinence in children with outlet obstruction after pull-through operations, however, ACE is still required because TES has not been tested formally in this group. We are aware that because we have promising results of using TES to treat children with STC, we will have the tendency to offer this treatment to considering surgical intervention, and hence, there will be selection bias in the choice of therapy. However, if TES was not effective in overcoming the symptoms of children with STC, they would present again with recurrent symptoms and be given appendicostomy. This occurred in only 2 of 134 patients treated by TES ( ) who had persistent symptoms. In conclusion, the operation rates in our institute for appendicostomy formation have reduced significantly in children with STC, coinciding with the introduction of TES as a successful alternative treatment. By contrast, appendicostomy formation rate has not changed in non-stc children during this period, and it is not yet known whether TES is effective in children with outlet obstruction. References [1] Borowitz SM, Cox DJ, Tam A, et al. Precipitants of constipation during early childhood. J Am Board Fam 2003;16: [2] Liem O, Harman J, Benninga M, et al. Health utilization and cost impact of childhood constipation in the United States. J Pediatr 2009;154: [3] Candelli M, Nista EC, Zocco MA, et al. Idiopathic chronic constipation: pathophysiology, diagnosis and treatment. Hepatogastroenterology 2001;48: [4] de Araujo Sant'Anna AM, Calcado AC. Constipation in school-aged children at public schools in Rio de Janeiro, Brazil. J Pediatr Gastroenterol Nutr 1999;29: [5] Inan M, Aydiner CY, Tokuc B, et al. Factors associated with childhood constipation. J Paediatr Child Health 2007;43: [6] Malone PS, Ransley PG, Kiely EM. Preliminary report: the antegrade continence enema. Lancet 1990;336: [7] de Graaf EJ, Gilberts EC, Schouten WR. Role of segmental colonic transit time studies to select patients with slow transit constipation for partial left-sided or subtotal colectomy. Br J Surg 1996;83: [8] Kamm MA, Hawley PR, Lennard-Jones JE. Outcome of colectomy for severe idiopathic constipation. Gut 1988;29: [9] Lubowski DZ, Chen FC, Kennedy ML, et al. Results of colectomy for severe slow transit constipation. Dis Colon Rectum 1996;39: [10] Mollen RM, Kuijpers HC, Claassen AT. Colectomy for slow-transit constipation: preoperative functional evaluation is important but not a guarantee for a successful outcome. Dis Colon Rectum 2001;44:

5 The impact of TES theraphy on appendicostomy operation rates 1425 [11] Sample C, Gupta R, Bamehriz F, et al. Laparoscopic subtotal colectomy for colonic inertia. J Gastrointest Surg 2005;9: [12] Yoshioka K, Keighley MR. Clinical results of colectomy for severe constipation. Br J Surg 1989;76: [13] Marshall J, Hutson JM, Anticich N, et al. Antegrade continence enemas in the treatment of slow-transit constipation. J Pediatr Surg 2001;36: [14] Stanton MP, Shin YM, Hutson JM. Laparoscopic placement of the Chait cecostomy device via appendicostomy. J Pediatr Surg 2002;37: [15] King SK, Sutcliffe JR, Southwell BR, et al. The antegrade continence enema successfully treats idiopathic slow-transit constipation. J Pediatr Surg 2005;40: [16] Chase J, Robertson VJ, Southwell B, et al. Pilot study using transcutaneous electrical stimulation (interferential current) to treat chronic treatment-resistant constipation and soiling in children. J Gastroenterol Hepatol 2005;20: [17] Clarke MC, Chase JW, Gibb S, et al. Improvement of quality of life in children with slow transit constipation after treatment with transcutaneous electrical stimulation. J Pediatr Surg 2009;44: [discussion 1272]. [18] Clarke MC, Chase JW, Gibb S, et al. Decreased colonic transit time after transcutaneous interferential electrical stimulation in children with slow transit constipation. J Pediatr Surg 2009;44: [19] Ismail KA, Chase J, Gibb S, et al. Daily transabdominal electrical stimulation at home increased defecation in children with slow-transit constipation: a pilot study. J Pediatr Surg 2009;44: [20] Yik YI, Clarke MC, Catto-Smith AG, et al. Slow-transit constipation with concurrent upper gastrointestinal dysmotility and its response to transcutaneous electrical stimulation. Pediatr Surg Int [21] Cook BJ, Lim E, Cook D, et al. Radionuclear transit to assess sites of delay in large bowel transit in children with chronic idiopathic constipation. J Pediatr Surg 2005;40: [22] Southwell BR, Clarke MC, Sutcliffe J, et al. Colonic transit studies: normal values for adults and children with comparison of radiological and scintigraphic methods. Pediatric surgery international 2009;25: [23] Sutcliffe JR, King SK, Hutson JM, et al. Gastrointestinal transit in children with chronic idiopathic constipation. Pediatr Surg Int 2009;25: [24] Leong LC, Yik YI, Catto-Smith AG, et al. Long-term effects of transabdominal electrical stimulation in treating children with slowtransit constipation. J Pediatr Surg. 46: [25] Pemberton JH, Rath DM, Ilstrup DM. Evaluation and surgical treatment of severe chronic constipation. Ann Surg 1991;214: [discussion ]. [26] Wexner SD, Daniel N, Jagelman DG. Colectomy for constipation: physiologic investigation is the key to success. Dis Colon Rectum 1991;34: [27] Piccirillo MF, Reissman P, Wexner SD. Colectomy as treatment for constipation in selected patients. Br J Surg 1995;82: [28] Ghosh S, Papachrysostomou M, Batool M, et al. Long-term results of subtotal colectomy and evidence of noncolonic involvement in patients with idiopathic slow-transit constipation. Scand J Gastroenterol 1996;31: [29] Simpson BB, Ryan DP, Schnitzer JJ, et al. Surgical evaluation and management of refractory constipation in older children. J Pediatr Surg 1996;31: [30] Knowles CH, Scott M, Lunniss PJ. Outcome of colectomy for slow transit constipation. Ann Surg 1999;230: [31] Rongen MJ, van der Hoop AG, Baeten CG. Cecal access for antegrade colon enemas in medically refractory slow-transit constipation: a prospective study. Dis Colon Rectum 2001;44: [32] Lundin E, Karlbom U, Pahlman L, et al. Outcome of segmental colonic resection for slow-transit constipation. Br J Surg 2002;89: [33] Lees NP, Hodson P, Hill J, et al. Long-term results of the antegrade continent enema procedure for constipation in adults. Colorectal Dis 2004;6: [34] Marchesi F, Sarli L, Percalli L, et al. Subtotal colectomy with antiperistaltic cecorectal anastomosis in the treatment of slow-transit constipation: long-term impact on quality of life. World J Surg 2007;31: [35] Leon SH, Krishnamurthy S, Schuffler MD. Subtotal colectomy for severe idiopathic constipation. A follow-up study of 13 patients. Dig Dis Sci 1987;32: [36] Glia A, Lindberg G, Nilsson LH, et al. Constipation assessed on the basis of colorectal physiology. Scand J Gastroenterol 1998;33: [37] Gladman MA, Scott SM, Lunniss PJ, et al. Systematic review of surgical options for idiopathic megarectum and megacolon. Ann Surg 2005;241: [38] Platell C, Scache D, Mumme G, et al. A long-term follow-up of patients undergoing colectomy for chronic idiopathic constipation. Aust N Z J Surg 1996;66: [39] FitzHarris GP, Garcia-Aguilar J, Parker SC, et al. Quality of life after subtotal colectomy for slow-transit constipation: both quality and quantity count. Dis Colon Rectum 2003;46: [40] Nylund G, Oresland T, Fasth S, et al. Long-term outcome after colectomy in severe idiopathic constipation. Colorectal Dis 2001;3: [41] Pikarsky AJ, Singh JJ, Weiss EG, et al. Long-term follow-up of patients undergoing colectomy for colonic inertia. Dis Colon Rectum 2001;44: [42] Thaler K, Dinnewitzer A, Oberwalder M, et al. Quality of life after colectomy for colonic inertia. Tech Coloproctol 2005;9: [43] Malone PS, Curry JI, Osborne A. The antegrade continence enema procedure why, when and how? World J Urol 1998;16: [44] Bani-Hani AH, Cain MP, King S, et al. Tap water irrigation and additives to optimize success with the Malone antegrade continence enema: the Indiana University algorithm. J Urol 2008;180: [discussion 1760]. [45] Dick AC, McCallion WA, Brown S, et al. Antegrade colonic enemas. Br J Surg 1996;83: [46] Driver CP, Barrow C, Fishwick J, et al. The Malone antegrade colonic enema procedure: outcome and lessons of 6 years' experience. Pediatr Surg Int 1998;13: [47] Ellsworth PI, Webb HW, Crump JM, et al. The Malone antegrade colonic enema enhances the quality of life in children undergoing urological incontinence procedures. J Urol 1996;155: [48] Griffiths DM, Malone PS. The Malone antegrade continence enema. J Pediatr Surg 1995;30: [49] Hensle TW, Reiley EA, Chang DT. The Malone antegrade continence enema procedure in the management of patients with spina bifida. J Am Coll Surg 1998;186: [50] Hill J, Stott S, MacLennan I. Antegrade enemas for the treatment of severe idiopathic constipation. Br J Surg 1994;81: [51] Koyle MA, Kaji DM, Duque M, et al. The Malone antegrade continence enema for neurogenic and structural fecal incontinence and constipation. J Urol 1995;154: [52] Krogh K, Laurberg S. Malone antegrade continence enema for faecal incontinence and constipation in adults. Br J Surg 1998;85: [53] Lukac M, Krstic Z, Sindjic S, et al. Continent appendicostomy in the treatment of fecal incontinence. Acta Chir Iugosl 2004;51: [54] Pera M, Pares D, Pascual M, et al. [Treatment of severe chronic constipation through the antegrade continent enema procedure]. Cir Esp 2006;80: [55] Redel CA, Motil KJ, Bloss RS, et al. Intestinal button implantation for obstipation and fecal impaction in children. J Pediatr Surg 1992;27: [56] Schell SR, Toogood GJ, Dudley NE. Control of fecal incontinence: continued success with the Malone procedure. Surgery 1997;122: [57] Squire R, Kiely EM, Carr B, et al. The clinical application of the Malone antegrade colonic enema. J Pediatr Surg 1993;28:

6 1426 Y.I. Yik et al. [58] Zerhau P, Husar M, Tuma J. [Malone antegrade continence enema stoma in children with dysfunctions of pelvic organs]. Rozhl Chir 2008;87: [59] Wong AL, Kravarusic D, Wong SL. Impact of cecostomy and antegrade colonic enemas on management of fecal incontinence and constipation: ten years of experience in pediatric population. J Pediatr Surg 2008;43: [60] M. Eleouet, L. Siproudhis, N. Guillou, et al, Chronic posterior tibial nerve transcutaneous electrical nerve stimulation (TENS) to treat fecal incontinence (FI). Int J Colorectal Dis. 25: [61] M.A. Kamm, T.C. Dudding, J. Melenhorst, et al, Sacral nerve stimulation for intractable constipation. Gut. 59: [62] Naldini G, Martellucci J, Moraldi L, et al. Treatment of slow-transit constipation with sacral nerve modulation. Colorectal Dis. 12: [63] S. Norderval, M. Rydningen, R.O. Lindsetmo, et al, Sacral nerve stimulation. Tidsskr Nor Laegeforen. 131: [64] Shi N, Liu S, Xie XP, et al. [Transcutaneous electrical nerve stimulation improves oppilative symptoms and increases colonic transit in patients with slow transit constipation]. Zhonghua Yi Xue Za Zhi 2009;89: