Abstract：
BACKGROUND: Even if understanding of neuronal enteropathies, such as Hirschsprung\'s disease and functional constipation, has been improved, specialized therapies are still missing. Sacral neuromodulation (SNM) has been established in the treatment of defecation disorders in adults. The aim of the study was to investigate effects of SNM in children and adolescents with refractory symptoms of chronic constipation.
METHODS: A two-centered, prospective trial has been conducted between 2019 and 2022. SNM was applied continuously at individually set stimulation intensity. Evaluation of clinical outcomes was conducted at 3, 6, and 12 months after surgery based on the developed questionnaires and quality of life analysis (KINDLR). Primary outcome was assessed based on predefined variables of fecal incontinence and defecation frequency.
RESULTS: Fifteen patients enrolled in the study and underwent SNM (median age 8.0 years (range 4-17 years)): eight patients were diagnosed with Hirschsprung\'s disease (53%). Improvement of defecation frequency was seen in 8/15 participants (53%) and an improvement of fecal incontinence in 9/12 patients (75%). We observed stable outcome after 1 year of treatment. Surgical revision was necessary in one patient after electrode breakage. Urinary incontinence was observed as singular side effect of treatment in two patients (13%), which was manageable with the reduction of stimulation intensity.
CONCLUSIONS: SNM shows promising clinical results in children and adolescents presenting with chronic constipation refractory to conservative therapy. Indications for patients with enteral neuropathies deserve further confirmation.
METHODS: A two-centered, prospective trial has been conducted between 2019 and 2022. SNM was applied continuously at individually set stimulation intensity. Evaluation of clinical outcomes was conducted at 3, 6, and 12 months after surgery based on the developed questionnaires and quality of life analysis (KINDLR). Primary outcome was assessed based on predefined variables of fecal incontinence and defecation frequency.
RESULTS: Fifteen patients enrolled in the study and underwent SNM (median age 8.0 years (range 4-17 years)): eight patients were diagnosed with Hirschsprung\'s disease (53%). Improvement of defecation frequency was seen in 8/15 participants (53%) and an improvement of fecal incontinence in 9/12 patients (75%). We observed stable outcome after 1 year of treatment. Surgical revision was necessary in one patient after electrode breakage. Urinary incontinence was observed as singular side effect of treatment in two patients (13%), which was manageable with the reduction of stimulation intensity.
CONCLUSIONS: SNM shows promising clinical results in children and adolescents presenting with chronic constipation refractory to conservative therapy. Indications for patients with enteral neuropathies deserve further confirmation.
摘要：
背景：即使了解神经元肠病，如先天性巨结肠病和功能性便秘，已经改进了,专业疗法仍然缺失。骶神经调节(SNM)已经在成人排便障碍的治疗中建立。该研究的目的是研究SNM对患有慢性便秘难治性症状的儿童和青少年的影响。
方法：以两个为中心，前瞻性试验在2019年至2022年之间进行。在单独设定的刺激强度下连续施加SNM。基于开发的问卷和生活质量分析（KINDLR），在手术后3、6和12个月对临床结果进行评估。根据粪便失禁和排便频率的预定义变量评估主要结果。
结果：15例患者参加了这项研究并接受了SNM（中位年龄8.0岁（范围4-17岁））：8例患者被诊断为先天性巨结肠（53％）。8/15的参与者（53％）的排便频率得到改善，9/12的患者（75％）的大便失禁得到改善。我们在治疗1年后观察到稳定的结果。一名患者在电极断裂后需要进行手术翻修。在两名患者（13％）中观察到尿失禁是治疗的单一副作用，随着刺激强度的降低，这是可控的。
结论：SNM在保守治疗难治性慢性便秘的儿童和青少年中显示出良好的临床效果。肠内神经病患者的适应症值得进一步确认。
方法：以两个为中心，前瞻性试验在2019年至2022年之间进行。在单独设定的刺激强度下连续施加SNM。基于开发的问卷和生活质量分析（KINDLR），在手术后3、6和12个月对临床结果进行评估。根据粪便失禁和排便频率的预定义变量评估主要结果。
结果：15例患者参加了这项研究并接受了SNM（中位年龄8.0岁（范围4-17岁））：8例患者被诊断为先天性巨结肠（53％）。8/15的参与者（53％）的排便频率得到改善，9/12的患者（75％）的大便失禁得到改善。我们在治疗1年后观察到稳定的结果。一名患者在电极断裂后需要进行手术翻修。在两名患者（13％）中观察到尿失禁是治疗的单一副作用，随着刺激强度的降低，这是可控的。
结论：SNM在保守治疗难治性慢性便秘的儿童和青少年中显示出良好的临床效果。肠内神经病患者的适应症值得进一步确认。
