Hailemariam Tesfahunegn, Bekele Abenezer Kebede, Manyazewal Tsegahun, Solomon Daniel Zewdneh, Gorfu Yocabel, Shiwarega Zelalem, Getinet Tewodros, Wole Meti, Solomon Samrawit, Hailu Samuel Sisay
Department of Radiology, College of Health Sciences Addis Ababa University Addis Ababa Ethiopia.
Center for Innovative Drug Development and Therapeutic Trials for Africa (CDT-AFRICA), College of Health Sciences Addis Ababa University Addis Ababa Ethiopia.
Health Sci Rep. 2024 Jan 8;7(1):e1798. doi: 10.1002/hsr2.1798. eCollection 2024 Jan.
Hirschsprung's disease (HSD) remains a common cause of pediatric intestinal obstruction. Barium contrast enema (BE) is the primary imaging modality for the evaluation of clinically suspected cases. Here, we aimed to assess the diagnostic accuracy of BE in children with clinically suspected HSD when compared to a gold standard full-thickness rectal biopsy (FTRB).
We recruited and consecutively enrolled children with clinically suspected HSD at two tertiary teaching hospitals. Participants underwent BE imaging and two radiologists interpreted the findings independently. Participants further underwent FTRB by pediatric surgeons as the confirmatory test. Sensitivity, specificity, positive predictive value (PPV), negative predictive value (NPV), and receiver operating characteristics (ROC) with the area under the curve (AUC) were calculated on Stata version 14.2, taking FTRB as the standard.
We enrolled 55 cases, of which 49 completed the evaluation and were included in the final analysis. The median age was 9.4 months (interquartile range: 2-24], with a male-to-female ratio of 4.4:1. The sensitivity, specificity, PPV, and NPV of BE were 0.95 (95% confidence interval [CI] [0.81-0.99]), 0.73 (95% CI [0.39-0.94]), 0.92 (95% CI [0.82-0.97]), and 0.80 (95% CI [0.50-0.94]), respectively. On AUC, the diagnostic accuracy of BE compared to the confirmatory FTRB was 0.84 (95% CI [0.69-0.98]). The diagnostic accuracy was higher in neonates (ROC: 1.00) when compared to infants (ROC: 0.83) or those above 1 year of age (ROC: 0.798). HSD-suggestive BE findings were associated with absence of ganglion cells on FTRB ( = 23.301, < 0.001). Inverted rectosigmoid ratio and transition zone were more sensitive in detecting HSD of 0.92 (95% CI [0.74-0.98]) and 0.81 (95% CI [0.63-0.92]), respectively.
BE is sufficiently accurate in the diagnosis of children with HSD, suggesting BE would likely be used to inform surgical management in settings where confirmatory biopsy is lacking. However, clinical judgment is warranted in interpreting negative BE findings.
先天性巨结肠症（HSD）仍是小儿肠梗阻的常见病因。钡剂灌肠造影（BE）是评估临床疑似病例的主要影像学检查方法。在此，我们旨在评估与金标准全层直肠活检（FTRB）相比，BE对临床疑似HSD患儿的诊断准确性。
我们在两家三级教学医院招募并连续纳入临床疑似HSD的患儿。参与者接受BE成像，两名放射科医生独立解读检查结果。参与者还接受了小儿外科医生进行的FTRB作为确诊检查。以FTRB为标准，在Stata 14.2版本上计算敏感性、特异性、阳性预测值（PPV）、阴性预测值（NPV）以及曲线下面积（AUC）的受试者操作特征（ROC）。
我们纳入了55例病例，其中49例完成评估并纳入最终分析。中位年龄为9.4个月（四分位间距：2 - 24个月），男女比例为4.4:1。BE的敏感性、特异性、PPV和NPV分别为0.95（95%置信区间[CI][0.81 - 0.99]）、0.73（95% CI[0.39 - 0.94]）、0.92（95% CI[0.82 - 0.97]）和0.80（95% CI[0.50 - 0.94]）。在AUC方面，与确诊性FTRB相比，BE的诊断准确性为0.84（95% CI[0.69 - 0.98]）。与婴儿（ROC：0.83）或1岁以上儿童（ROC：0.798）相比，新生儿的诊断准确性更高（ROC：1.00）。提示HSD的BE表现与FTRB上神经节细胞缺失相关（χ² = 23.301，P < 0.001）。直肠乙状结肠反转比例和移行区在检测HSD时更敏感，分别为0.92（95% CI[0.74 - 0.98]）和0.81（95% CI[0.63 - 0.92]）。
BE对HSD患儿的诊断具有足够的准确性，这表明在缺乏确诊性活检的情况下，BE可能用于指导手术治疗。然而，在解读BE阴性结果时需要临床判断。
