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孤立性终丝纤维脂肪瘤患儿视频尿动力学的临床应用及组内一致性

Clinical utility and interrater reliability of video urodynamics in children with isolated fibrolipoma of filum terminale.

机构信息

Division of Pediatric Urology, Children's Medical Center, University of Texas Southwestern Medical Center, Dallas, TX, USA.

Division of Pediatric Urology, Children's Medical Center, University of Texas Southwestern Medical Center, Dallas, TX, USA.

出版信息

J Pediatr Urol. 2023 Oct;19(5):524-531. doi: 10.1016/j.jpurol.2023.05.002. Epub 2023 May 5.

Abstract

INTRODUCTION

Children with an isolated fibrolipoma of filum terminale (IFFT) but otherwise normal spinal cord are often evaluated with video urodynamics (VUDS). VUDS interpretation is subjective and can be difficult in young children. These patients may undergo detethering surgery if there is concern for current or future symptomatic tethered cord.

OBJECTIVE

We hypothesized that VUDS in children with IFFT would have limited clinical utility regarding decision for or against detethering surgery and VUDS interpretation would have poor interrater reliability.

METHODS

Patients with IFFT who underwent VUDS for from 2009 to 2021 were retrospectively reviewed to evaluate clinical utility of VUDS. 6 pediatric urologists who were blinded to patient clinical characteristics reviewed the VUDS. Gwet's first order agreement coefficient (AC) with 95% CI was used to assess interrater reliability.

RESULTS

47 patients (24F:23M) were identified. Median age at initial evaluation was 2.8yrs (IQR:1.5-6.8). 24 (51%) patients underwent detethering surgery (Table). VUDS at initial evaluation were interpreted by treating urologist as normal in 4 (8%), reassuring for normal in 39 (81%), or concerning for abnormal in 4 (9%). Based on neurosurgery clinic and operative notes for the 47 patients, VUDS made no change in management in 37 patients (79%), prompted detethering in 3 (6%), was given as reason for observation in 7 (15%), and was normal or reassuring for normal but not documented as a reason for observation in 16 (34%) (Table). Interrater reliability for VUDS interpretation had fair agreement (AC = 0.27) for overall categorization of VUDS and EMG interpretation (AC = 0.34). Moderate agreement was seen for detrusor overactivity interpretation (AC = 0.54) and bladder neck appearance (AC = 0.46).

DISCUSSION

In our cohort, 90% of patients had a normal or reassuring for normal interpretation of VUDS. VUDS interpretation affected clinical course in a minority of patients. There was fair interrater reliability for overall VUDS interpretation and therefore clinical course regarding detethering surgery could vary depending upon interpreting urologist. This fair interrater variability appeared to be related to variability in EMG, bladder neck appearance, and detrusor overactivity interpretation.

CONCLUSION

VUDS affected clinical management in about 20% of our cohort and supported the choice for observation in around 50% of patients. This suggests VUDS does have clinical utility in pediatric patients with IFFT. The overall VUDS interpretation had fair interrater reliability. This suggest VUDS interpretation has limitations in determining normal versus abnormal bladder function in children with IFFT. Neurosurgeons and urologists should be aware of VUDS limitations in this patient population.

摘要

简介

患有孤立性终丝纤维脂肪瘤(IFFT)但脊髓其他部位正常的儿童通常会接受视频尿动力学检查(VUDS)。VUDS 的解释具有主观性,并且在幼儿中可能会比较困难。如果存在当前或未来症状性脊髓栓系的担忧,这些患者可能会接受松解手术。

目的

我们假设 IFFT 患儿的 VUDS 在决定是否进行松解手术方面的临床应用价值有限,并且 VUDS 的解释具有较差的组内可靠性。

方法

对 2009 年至 2021 年间接受 VUDS 检查的 IFFT 患儿进行回顾性研究,以评估 VUDS 的临床应用价值。6 名小儿泌尿科医生对患者的临床特征进行了盲法评估,对 VUDS 进行了评估。使用 Gwet 的第一级一致性系数(AC)及其 95%置信区间(CI)来评估组内可靠性。

结果

共确定了 47 名患者(24 名女性:23 名男性)。初始评估时的中位年龄为 2.8 岁(IQR:1.5-6.8)。24 名(51%)患者接受了松解手术(表)。初始评估时,治疗泌尿科医生将 VUDS 解释为正常的有 4 例(8%),解释为正常的有 39 例(81%),解释为异常的有 4 例(9%)。根据 47 名患者的神经外科诊所和手术记录,37 名患者(79%)的 VUDS 检查结果对治疗方案没有影响,3 名患者(6%)提示需要松解手术,7 名患者(15%)因观察而给予松解手术,16 名患者(34%)的 VUDS 检查结果为正常或解释为正常,但未作为观察的理由记录(表)。VUDS 解释的组内可靠性对于整体 VUDS 分类和肌电图解释具有中等一致性(AC=0.27),对于逼尿肌过度活动的解释具有良好的一致性(AC=0.54),对于膀胱颈外观的解释具有中度一致性(AC=0.46)。

讨论

在我们的队列中,90%的患者 VUDS 检查结果为正常或解释为正常。只有少数患者的 VUDS 检查结果影响了临床过程。总体 VUDS 解释的组内可靠性为中等,因此,关于松解手术的临床过程可能取决于解释的泌尿科医生。这种中等的组内变异性似乎与肌电图、膀胱颈外观和逼尿肌过度活动的解释有关。

结论

VUDS 检查结果影响了我们队列中约 20%患者的临床管理,并支持约 50%患者选择观察。这表明 VUDS 对患有 IFFT 的儿科患者具有临床应用价值。总体 VUDS 解释的组内可靠性为中等。这表明 VUDS 解释在确定 IFFT 儿童正常与异常膀胱功能方面存在局限性。神经外科医生和泌尿科医生应该意识到 VUDS 在这一患者群体中的局限性。

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