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CC BY 4.0 · Journal of Coloproctology 2025; 45(02): s00451809676
DOI: 10.1055/s-0045-1809676
Case Report

Sacral Neuromodulation in Child with Anorectal Malformation: A Case Report

Sthela Murad-Regadas 
1   Department of Surgery, School of Medicine, Universidade Federal do Ceará, Fortaleza, CE, Brazil
,
Lavier Kelvin Holanda Vidal 
1   Department of Surgery, School of Medicine, Universidade Federal do Ceará, Fortaleza, CE, Brazil
,
Carlos Magno Queiroz da Cunha 
1   Department of Surgery, School of Medicine, Universidade Federal do Ceará, Fortaleza, CE, Brazil
2   School of Medicine, University of Fortaleza, Fortaleza, CE, Brazil
,
Isabela Porto Pinheiro Marques 
3   School of Medicine, Christus University Center
› Author Affiliations

Funding The author(s) received no financial support for the research.
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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.


 

Keywords

sacral neuromodulation - fecal incontinence - anorectal malformation - bowel dysfunction

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.

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Fig. 1 3D Ultrasound: The external anal sphincter was visualized in the right and left circumference, while the internal anal sphincter was not identified. EAS – external anal sphincter. a. Axial plane. b. Coronal plane.

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.

Zoom
Fig. 2 Patient is placed in prone position with reduction of the lumbar lordosi. Needles were positioned into the S3 foramina bilaterally. The fluoroscopy guidance under latero-lateral X-ray shows the two needles in the correct position.
Zoom
Fig. 3 The needle is then replaced by a guidewire in the left S3 (the best response).
Zoom
Fig. 4 Placement of a permanent tined lead (with four electrodes). The fluoroscopy guidance under an antero-posterior X-ray shows the correct position.
Zoom
Fig. 5 The optimal placement of the permanent tined lead is attached to a temporary wire that plugs temporary external pulse generator.

 

 

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.



Address for correspondence

Sthela Murad-Regadas, MD, PhD
Department of Surgery, School of Medicine, Universidade Federal do Ceará
Fortaleza, CE
Brazil   

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/)

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Bibliographical Record
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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Zoom
Fig. 1 3D Ultrasound: The external anal sphincter was visualized in the right and left circumference, while the internal anal sphincter was not identified. EAS – external anal sphincter. a. Axial plane. b. Coronal plane.
Zoom
Fig. 2 Patient is placed in prone position with reduction of the lumbar lordosi. Needles were positioned into the S3 foramina bilaterally. The fluoroscopy guidance under latero-lateral X-ray shows the two needles in the correct position.
Zoom
Fig. 3 The needle is then replaced by a guidewire in the left S3 (the best response).
Zoom
Fig. 4 Placement of a permanent tined lead (with four electrodes). The fluoroscopy guidance under an antero-posterior X-ray shows the correct position.
Zoom
Fig. 5 The optimal placement of the permanent tined lead is attached to a temporary wire that plugs temporary external pulse generator.