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DOI: 10.1055/s-0045-1809676
Sacral Neuromodulation in Child with Anorectal Malformation: A Case Report
Funding The author(s) received no financial support for the research.
Abstract
Anorectal malformations (ARMs) are congenital anomalies that can be related to poor functional prognoses for the patient, with issues such as fecal incontinence and obstructed defecation, despite the performing of corrective surgery in early childhood. Given the complexity of treating this type of disease, the aim of this study is to present the feasibility of sacral neuromodulation as a minimally invasive technique that can be used for managing pelvic floor dysfunctions in children with ARM, refractory to conventional therapies, and show outcomes improvement in the bowel function, and especially psychosocial well-being of the child more than one year of follow-up.
Introduction
Anorectal malformations (ARMs) are congenital anomalies that affect both sexes and can involve the distal rectum, anal canal, anus, and even the urinary and genital tracts.[1] [2] [3] The severity of these defects ranges from minor abnormalities, which can be treated with excellent functional outcomes, to more complex and severe forms, often associated with other anomalies and poor functional prognoses.[2] [3] Corrective surgeries for ARMs are typically performed in early childhood; however, even after successful procedures, functional issues such as fecal incontinence and obstructed defecation can persist, significantly impacting the patient's quality of life. Bowel dysfunctions, including constipation and fecal incontinence, affect approximately 50% of patients' post-surgery for ARMs during childhood. The lack of effective treatment options can have lifelong consequences, affecting social, emotional, athletic, professional, and sexual development, potentially leading to psychiatric disorders and loss of independence.[4] [5] [6]
Sacral neuromodulation (SN) is a well-established, safe, and minimally invasive advanced therapy for managing lower urinary tract and bowel dysfunctions.[4] [5] [6] However, there are few case reports in the literature concerning the use of sacral neuromodulation in children with anorectal malformations.[7] [8] The aim of this study is to present the feasibility of sacral neuromodulation in children with anorectal malformation and pelvic floor dysfunctions (symptoms of both fecal incontinence and obstructed defecation), refractory to conventional therapies, and the long-term outcomes after more than one year of follow-up. The present study has been developed with the standards established by the CARE criteria in the documentation of case reports and appropriate consent from the patients obtained per institutional protocol and guidelines.
Presentation of Case
A 12-year-old female child presented to our clinic with complaints of worsening fecal incontinence, a history of fecal impaction, and obstructed defecation. She had a history of isolated anorectal malformation, specifically a low imperforate anus, which had been treated during infancy with a pull-through procedure. Despite multiple clinical treatments throughout her childhood, she continued to suffer from severe fecal incontinence, with a Cleveland Clinic – CCF Incontinence Score (CCIS) of 17/20 and a Renzi obstructed defecation score of 16/20.[9] [10] A visual analog scale from 100 (worst) to 0 (best) to evaluate the satisfaction rate showed 100.[11] The primary issue was the negative impact on her quality of life, particularly in her relationships with friends and family. She refused to attend school due to social isolation and concerns about needing diapers and the associated smell.
On physical examination, she had a patulous anus with hypotonia and poor resting/squeezing tone. Anal manometry revealed low resting and squeeze pressures, and the patient was unable to maintain squeezing. Endoanal ultrasound showed intact puborectal muscles, but the external anal sphincter was visualized in the right and left circumference, while the internal anal sphincter was not identified ([Fig. 1]). Given these findings, sphincteroplasty was not indicated, and sacral neuromodulation was offered.


Technique
The technique for sacral neuromodulation performed in this case has been previously described using fluoroscopy-guided.[4] [5] Under sedation and local anesthesia, the patient was placed in a prone position with a reduction of the lumbar lordosis. The fluoroscopy guidance under antero-posterior and latero-lateral X-ray helps locate the landmarks, and the sacral foramina and shows the needles in the correct position. The two needles were inserted bilaterally into the S3 foramina ([Figs. 2] and [3]), and an external pulse generator was used for stimulation. Following a motor and sensorial positive response (bellows and toe flex response), an electrode a permanent MRI (magnetic resonance imaging) tined lead (with four electrodes) was implanted with a temporary stimulation wire ([Figs. 4] and [5]). After 20 days, the patient was evaluated through a clinical interview, reporting more than 70% improvement in both symptoms. Consequently, a permanent implant was placed (recharge-free InterStim™ SureScan™). This model allows full-body magnetic resonance imaging. The patient did not experience any complications and was followed regularly, with visits scheduled at 1, 3, and 6 months post-implantation, and every 6 months thereafter. The total follow-up period was 22 months. Throughout the follow-up, the patient reported sustained improvement in fecal incontinence, with the CCIS decreasing from 17 pre-SN to 1 post-SN. Constipation also improved, with the Renzi score dropping from 16 pre-SN to 4 post-SN, and the VAS decreasing from 100 pre-SN to 1 post-SN after 22 months.








Commentary
The indications for sacral neuromodulation have expanded in recent years [12-15].[12] [13] [14] [15] This report further supports its use in patients with a history of congenital anorectal malformation and symptoms of both fecal incontinence and obstructed defecation, demonstrating excellent therapeutic response. In this case, the patient experienced significant improvements in both symptoms, leading to a remarkable enhancement in her quality of life. She successfully reintegrated into her social environment and reported feeling happy, highlighting the positive impact of this treatment. To our knowledge, this is the first case report of sacral neuromodulation for pelvic floor dysfunctions (fecal incontinence associated with obstructed defecation) in a child with a history of congenital anal malformation in Brazil. Despite the case report of a child, the following learning points: the anatomy and technique were similar as adults; the outcomes demonstrate improvement in the bowel function, and especially psychosocial well-being of the child and it will keep away from psychiatric problems in the future.
Conflicts of Interest
The authors have no conflicts of interest.
Authors Contributions
Sthela Murad Regadas, Lavier Kelvin Holanda Vidal, Carlos Magno Queiroz da Cunha: Conception and design; Sthela Murad Regadas, Lavier Kelvin Holanda Vidal, Carlos Magno Queiroz da Cunha, Isabela Porto Pinheiro Marques: Acquisition of data, Analysis and interpretation of data, Drafting the article and Final approval; Sthela Murad-Regadas: Revising it critically for important intellectual content.
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References
- 1 Wood RJ, Levitt MA. Anorectal Malformations. Clin Colon Rectal Surg 2018; 31 (02) 61-70
- 2 Levitt MA, Peña A. Anorectal malformations. Orphanet J Rare Dis 2007; 2 (26) 33
- 3 Thomas GP, Nicholls RJ, Vaizey CJ. Sacral nerve stimulation for faecal incontinence secondary to congenital imperforate anus. Tech Coloproctol 2013; 17 (02) 227-229
- 4 Siegel SW, Catanzaro F, Dijkema HE. et al. Long-term results of a multicenter study on sacral nerve stimulation for treatment of urinary urge incontinence, urgency-frequency, and retention. Urology 2000; 56 (6, Suppl 1) 87-91
- 5 Murad-Regadas SM, Holanda Ede C, Regadas CM. et al. Functional outcomes and satisfaction rates of sacral nerve stimulation in the treatment of one and multiples pelvic floor dysfunctions. J Coloproctol 2023; 43 (02) 104-109
- 6 Bordeianou L, Paquette IM, Johnson E. et al. The American Society of Colon and Rectal Surgeons Clinical Practice Guidelines for the Management of Fecal Incontinence. Dis Colon Rectum 2023; 66: 647-661
- 7 Grossi U, Carrington EV, Scott SM, Knowles CH. Sacral neuromodulation for anorectal dysfunction secondary to congenital imperforate anus: report of two cases. Int J Colorectal Dis 2014; 29 (07) 889-890
- 8 Castillo J, Cristóbal L, Alonso J. et al. Sacral nerve stimulation lead implantation in partial sacral agenesis using intra-operative computerized tomography. Colorectal Dis 2016; 18 (09) O330-O333
- 9 Jorge JM, Wexner SD. Etiology and management of fecal incontinence. Dis Colon Rectum 1993; 36 (01) 77-97
- 10 Caetano AC, Dias S, Santa-Cruz A, Rolanda C. Score for obstructed defecation syndrome – validation of the Portuguese version according to the COSMIN checklist. Arq Gastroenterol 2018; 55 (01) 55-60
- 11 Klimek L, Bergmann KC, Biedermann T. et al. Visual analogue scales (VAS): Measuring instruments for the documentation of symptoms and therapy monitoring in cases of allergic rhinitis in everyday health care: Position Paper of the German Society of Allergology (AeDA) and the German Society of Allergy and Clinical Immunology (DGAKI), ENT Section, in collaboration with the working group on Clinical Immunology, Allergology and Environmental Medicine of the German Society of Otorhinolaryngology, Head and Neck Surgery (DGHNOKHC). Allergo J Int 2017; 26 (01) 16-24
- 12 Bougie A, McFadden N, Mayer S, Lebel M, Devroede G. Neurostimulation for fecal incontinence after correction of repair of imperforate anus. World J Clin Cases 2017; 5 (03) 124-127
- 13 Danielson J, Karlbom U, Graf W, Wester T. Persistent fecal incontinence into adulthood after repair of anorectal malformations. Int J Colorectal Dis 2019; 34 (03) 551-554
- 14 Mass-Lindenbaum M, Calderón-Pollak D, Goldman HB, Pizarro-Berdichevsky J. Sacral neuromodulation - when and for who. Int Braz J Urol 2021; 47 (03) 647-656
- 15 Goldman HB, Lloyd JC, Noblett KL. et al. International Continence Society best practice statement for use of sacral neuromodulation. Neurourol Urodyn 2018; 37 (05) 1823-1848 https://www.ics.org/education/icsstandards
Address for correspondence
Publication History
Received: 15 December 2024
Accepted: 21 March 2025
Article published online:
20 June 2025
© 2025. The Author(s). This is an open access article published by Thieme under the terms of the Creative Commons Attribution 4.0 International License, permitting copying and reproduction so long as the original work is given appropriate credit (https://creativecommons.org/licenses/by/4.0/)
Thieme Revinter Publicações Ltda.
Rua Rego Freitas, 175, loja 1, República, São Paulo, SP, CEP 01220-010, Brazil
Sthela Murad-Regadas, Lavier Kelvin Holanda Vidal, Carlos Magno Queiroz da Cunha, Isabela Porto Pinheiro Marques. Sacral Neuromodulation in Child with Anorectal Malformation: A Case Report. Journal of Coloproctology 2025; 45: s00451809676.
DOI: 10.1055/s-0045-1809676
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References
- 1 Wood RJ, Levitt MA. Anorectal Malformations. Clin Colon Rectal Surg 2018; 31 (02) 61-70
- 2 Levitt MA, Peña A. Anorectal malformations. Orphanet J Rare Dis 2007; 2 (26) 33
- 3 Thomas GP, Nicholls RJ, Vaizey CJ. Sacral nerve stimulation for faecal incontinence secondary to congenital imperforate anus. Tech Coloproctol 2013; 17 (02) 227-229
- 4 Siegel SW, Catanzaro F, Dijkema HE. et al. Long-term results of a multicenter study on sacral nerve stimulation for treatment of urinary urge incontinence, urgency-frequency, and retention. Urology 2000; 56 (6, Suppl 1) 87-91
- 5 Murad-Regadas SM, Holanda Ede C, Regadas CM. et al. Functional outcomes and satisfaction rates of sacral nerve stimulation in the treatment of one and multiples pelvic floor dysfunctions. J Coloproctol 2023; 43 (02) 104-109
- 6 Bordeianou L, Paquette IM, Johnson E. et al. The American Society of Colon and Rectal Surgeons Clinical Practice Guidelines for the Management of Fecal Incontinence. Dis Colon Rectum 2023; 66: 647-661
- 7 Grossi U, Carrington EV, Scott SM, Knowles CH. Sacral neuromodulation for anorectal dysfunction secondary to congenital imperforate anus: report of two cases. Int J Colorectal Dis 2014; 29 (07) 889-890
- 8 Castillo J, Cristóbal L, Alonso J. et al. Sacral nerve stimulation lead implantation in partial sacral agenesis using intra-operative computerized tomography. Colorectal Dis 2016; 18 (09) O330-O333
- 9 Jorge JM, Wexner SD. Etiology and management of fecal incontinence. Dis Colon Rectum 1993; 36 (01) 77-97
- 10 Caetano AC, Dias S, Santa-Cruz A, Rolanda C. Score for obstructed defecation syndrome – validation of the Portuguese version according to the COSMIN checklist. Arq Gastroenterol 2018; 55 (01) 55-60
- 11 Klimek L, Bergmann KC, Biedermann T. et al. Visual analogue scales (VAS): Measuring instruments for the documentation of symptoms and therapy monitoring in cases of allergic rhinitis in everyday health care: Position Paper of the German Society of Allergology (AeDA) and the German Society of Allergy and Clinical Immunology (DGAKI), ENT Section, in collaboration with the working group on Clinical Immunology, Allergology and Environmental Medicine of the German Society of Otorhinolaryngology, Head and Neck Surgery (DGHNOKHC). Allergo J Int 2017; 26 (01) 16-24
- 12 Bougie A, McFadden N, Mayer S, Lebel M, Devroede G. Neurostimulation for fecal incontinence after correction of repair of imperforate anus. World J Clin Cases 2017; 5 (03) 124-127
- 13 Danielson J, Karlbom U, Graf W, Wester T. Persistent fecal incontinence into adulthood after repair of anorectal malformations. Int J Colorectal Dis 2019; 34 (03) 551-554
- 14 Mass-Lindenbaum M, Calderón-Pollak D, Goldman HB, Pizarro-Berdichevsky J. Sacral neuromodulation - when and for who. Int Braz J Urol 2021; 47 (03) 647-656
- 15 Goldman HB, Lloyd JC, Noblett KL. et al. International Continence Society best practice statement for use of sacral neuromodulation. Neurourol Urodyn 2018; 37 (05) 1823-1848 https://www.ics.org/education/icsstandards









