Lu Changgui, Pan Shiwen, Hua Xinyi, Jiang Weiwei, Tang Weibing
Department of Pediatric Surgery, Children's Hospital of Nanjing Medical University, Nanjing, 210008, China.
Department of Anesthesia and Operation, Children's Hospital of Nanjing Medical University, Nanjing, 210008, China.
Eur J Pediatr. 2023 Mar;182(3):1251-1259. doi: 10.1007/s00431-023-04814-7. Epub 2023 Jan 12.
Bowel dysfunction has been proven to be the most typical complication of long-segment Hirschsprung disease (LSHD). Additionally, bowel dysfunction in preschool and early childhood should be properly assessed, because it may result in persistent bowel dysfunction, social issues, and depression in adolescence and adulthood. This study evaluated bowel function during preschool and early childhood in patients with LSHD. A cross-sectional bowel function score (BFS) questionnaire survey was conducted on 270 infants with short-segment Hirschsprung disease (SSHD) and LSHD who underwent pull-through (PT) between January 2014 and December 2019 at the Children's Hospital of Nanjing Medical University. One hundred ninety patients who consented to the survey were asked to complete the questionnaire and were divided into two groups: the LSHD group (n = 42) and the SSHD group (n = 148). Bowel function outcomes were assessed by using a questionnaire. The total BFS score in the LSHD group was significantly lower than that in the SSHD group (15.0 [14.0, 17.0] vs. 18.0 [16.0, 19.0], p < 0.05) and did not improve with age (p > 0.05). Independent BFS items with lower scores were discovered in LSHD than in SSHD, including the capacity to hold back defecation, feeling/reporting the urge to defecate, frequency, soiling, and accidents (all p < 0.05). After subgroup analysis of follow-up age (3-5 years, 5 ~ 7 years, and > 7 years), there was no difference between the score of ability to hold back defecation over 7 years old and feeling/reporting the urge to defecate over 5 years old (all p > 0.05). The frequency and soiling scores were lower in the LSHD group than in the SSHD group over 5 years of age (all p < 0.05). Fortunately, there was no difference in constipation scores and social problems between the LSHD and SSHD groups in every sub-follow-up group (all p > 0.05). Conclusion: Overall, bowel function at preschool and early childhood age was poorer in LSHD than in SSHD and did not improve with age. However, the ability to hold back defecation and feeling/reporting the urge to defecate with age in LSHD may be similar to those in SSHD. Simultaneously, the frequency, soiling, and number of accidents were consistently worse in the LSHD group. Trial registration: This study was retrospectively registered in the ClinicalTrials database.gov (NCT05461924) in August 2022. What is Known: • Bowel function in SSHD is satisfactory or near-normal, and the general consensus is that the longer the aganglionic segment, the worse the bowel function after surgery. • Bowel functional outcomes of LSHD were not defined, and the limited reports of bowel functional outcomes compared with SSHD were extensively varied. Bowel dysfunction at preschool and early childhood may lead to persistent bowel dysfunction, social problems, and depression in adolescence or adulthood. What is New: • Bowel function in preschool and early childhood in patients with LSHD has not been defined and should be thoroughly assessed in a larger group of patients with precise definitions of incontinence or soiling. • The present study was primarily designed to evaluate bowel function at preschool and early childhood age in LSHD compared with SSHD in a relatively large number of HD cases using BFS.
肠功能障碍已被证明是长段型先天性巨结肠(LSHD)最典型的并发症。此外,应适当评估学龄前和幼儿期的肠功能障碍,因为它可能导致持续性肠功能障碍、社会问题以及青少年和成年期的抑郁。本研究评估了LSHD患者在学龄前和幼儿期的肠功能。对2014年1月至2019年12月在南京医科大学附属儿童医院接受拖出术(PT)的270例短段型先天性巨结肠(SSHD)和LSHD婴儿进行了横断面肠功能评分(BFS)问卷调查。190例同意参与调查的患者被要求完成问卷,并分为两组:LSHD组(n = 42)和SSHD组(n = 148)。通过问卷评估肠功能结果。LSHD组的总BFS评分显著低于SSHD组(15.0[14.0,17.0]对18.0[16.0,19.0],p < 0.05),且不随年龄增长而改善(p > 0.05)。发现LSHD组中独立的BFS项目得分低于SSHD组,包括抑制排便的能力、感觉/报告排便冲动、频率、污粪和意外排便(所有p < 0.05)。在对随访年龄(3至5岁、5至7岁和>7岁)进行亚组分析后,7岁以上抑制排便能力得分与5岁以上感觉/报告排便冲动得分之间无差异(所有p > 0.05)。5岁以上LSHD组的频率和污粪得分低于SSHD组(所有p < 0.05)。幸运的是,在每个亚随访组中,LSHD组和SSHD组在便秘得分和社会问题方面无差异(所有p > 0.05)。结论:总体而言,LSHD患者在学龄前和幼儿期的肠功能比SSHD患者差,且不随年龄增长而改善。然而,LSHD患者抑制排便的能力以及随年龄增长感觉/报告排便冲动的情况可能与SSHD患者相似。同时,LSHD组的频率、污粪和意外排便次数一直更差。试验注册:本研究于2022年8月在ClinicalTrials数据库.gov(NCT05461924)进行回顾性注册。已知信息:•SSHD的肠功能良好或接近正常,普遍的共识是无神经节段越长,术后肠功能越差。•LSHD的肠功能结局未明确,与SSHD相比,关于肠功能结局的有限报告差异很大。学龄前和幼儿期的肠功能障碍可能导致持续性肠功能障碍、社会问题以及青少年或成年期的抑郁。新发现:•LSHD患者在学龄前和幼儿期的肠功能未明确,应在更多患者中进行全面评估,并对失禁或污粪进行精确界定。•本研究主要旨在使用BFS在相对大量的HD病例中评估LSHD与SSHD相比在学龄前和幼儿期的肠功能。