Sukarelawanto Afnandito Valeno Risky, Ritana Azmi, Balela Naisya, Putri Wayan Julita Krisnanti, Sirait Dian Nirmala, Paramita Vincentia Meta Widya, Sasmita Andika Purba, Dwihantoro Andi, Makhmudi Akhmad
Pediatric Surgery Division, Department of Surgery, Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada/Dr. Sardjito Hospital, Jl. Kesehatan No. 1, Yogyakarta, 55281, Indonesia.
BMC Pediatr. 2020 Oct 2;20(1):457. doi: 10.1186/s12887-020-02360-x.
Hirschsprung-associated enterocolitis (HAEC) is the most severe and potentially lethal complication of Hirschsprung disease (HSCR) which might occur following definitive surgery. Our objectives were: 1) to compare the incidence of HAEC after Duhamel and Soave procedures using different cut-off values of the HAEC scoring method; and 2) to associate them with the risk factors, including sex, aganglionosis type, mothers' age at childbirth, gestational age, and mothers' educational level.
Medical records of patients with HSCR who underwent Soave and Duhamel procedures in our institution, Indonesia (January 2012 - December 2016) were reviewed retrospectively. Two cut-off values of the HAEC scoring system (i.e., ≥10 and ≥ 4) were utilized.
Eighty-three patients with HSCR were recruited in this study (Soave: 37 males and 7 females vs. Duhamel: 28 males and 11 females; p = 0.18). The incidence of HAEC after surgery was 14/83 (16.9%) and 38/83 (45.8%) for cut-off values of ≥10 and ≥ 4, respectively (p = 0.00012), and tended to have an association with sex (p = 0.09). Although it was not statistically significant (p = 0.07), the frequency of HAEC after Soave procedure tended to be higher in patients with their mother's age of ≤35 years at childbirth than those with their mother's age of > 35 years (OR = 7.9; 95% CI = 0.9-72.1). Multivariate analysis indicated none of the risk factors were associated with the frequency of HAEC after definitive surgery.
The lower cut-off value of ≥4 might increase the possibility to diagnose HAEC, particularly the mild cases. The incidence of HAEC after definitive surgery was not associated with any risk factors in our cohort patients. Further multicenter studies with a larger sample size are necessary to confirm our findings.
先天性巨结肠相关小肠结肠炎(HAEC)是先天性巨结肠症(HSCR)最严重且可能致命的并发症,可能在根治性手术后发生。我们的目标是:1)使用HAEC评分方法的不同临界值比较Duhamel手术和Soave手术后HAEC的发生率;2)将它们与危险因素相关联,包括性别、无神经节细胞症类型、母亲分娩年龄、孕周和母亲教育水平。
回顾性分析2012年1月至2016年12月在印度尼西亚我们机构接受Soave和Duhamel手术的HSCR患者的病历。使用HAEC评分系统的两个临界值(即≥10和≥4)。
本研究纳入了83例HSCR患者(Soave手术组:37例男性和7例女性,Duhamel手术组:28例男性和11例女性;p = 0.18)。对于临界值≥10和≥4,术后HAEC的发生率分别为14/83(16.9%)和38/83(45.8%)(p = 0.00012),并且倾向于与性别相关(p = 0.09)。虽然无统计学意义(p = 0.07),但分娩时母亲年龄≤35岁的患者Soave手术后HAEC的发生率往往高于母亲年龄>35岁的患者(OR = 7.9;95%CI = 0.9 - 72.1)。多因素分析表明,没有危险因素与根治性手术后HAEC的发生频率相关。
较低的临界值≥4可能增加诊断HAEC的可能性,尤其是轻度病例。在我们的队列患者中,根治性手术后HAEC的发生率与任何危险因素均无关联。需要进一步进行更大样本量的多中心研究来证实我们的发现。