Yibrehu Betel, Georgakopoulos Bianca, Mudd Pamela A, Rana Md Sohel, Bauman Nancy M
The George Washington University School of Medicine and Health Sciences, Washington, DC, USA.
Children's National Hospital, Washington, DC, USA.
Ann Otol Rhinol Laryngol. 2020 Dec;129(12):1195-1209. doi: 10.1177/0003489420931894. Epub 2020 Jun 12.
To explore long-term patient reported outcome (PRO) measures of pediatric paradoxical vocal cord motion (PVCM) including ease of diagnosis, management, symptom duration and effect on quality of life.
All children >8 years of age diagnosed with PVCM at a tertiary pediatric hospital between 2006 and 2017 were invited to complete a survey addressing study objectives.
21/47 eligible participants could be contacted and 18/21 (86%) participated. 78% were female with a mean age at diagnosis of 11.6 and 15.0 years at survey completion. Common PVCM symptoms reported were dyspnea (89%), globus sensation (56%), and stridor (50%). The median time to diagnosis was 3 months (IQR 2-5 months). Nearly all reported being misdiagnosed with another condition, usually asthma, until being correctly diagnosed usually by an otolaryngologist. Participants reported undergoing 3.7 diagnostic studies (range 0-8); pulmonary function testing was most common. Of numerous treatments acknowledged, breathing exercises were common (89%) but only reported helpful by 56%. Use of biofeedback was recalled in 1/3 of subjects but reported helpful in only 14% of them. Anti-reflux, allergy, anticholinergics, inhalers and steroids were each used in >50%, but rarely reported effective. PVCM was reportedly a significant stressor when initially diagnosed but despite 2/3 of participants still reporting ongoing PVCM symptoms, the perceived stress significantly decreased over time (Z = 3.26, = 0.001).
This first PVCM PRO study endorses that diagnosis is often delayed and prescribed treatments often viewed as ineffective. While biofeedback and breathing exercises may be critical for short-term control of PVCM episodes, lifestyle changes and stress reduction are likely necessary for long-term management. Increased awareness and improvements in management are needed for this condition.
探讨儿童反常性声带运动障碍（PVCM）患者长期的患者报告结局（PRO）指标，包括诊断的难易程度、治疗情况、症状持续时间及对生活质量的影响。
邀请2006年至2017年间在一家三级儿科医院被诊断为PVCM的所有8岁以上儿童完成一项针对研究目标的调查。
47名符合条件的参与者中有21名能够被联系上，其中18名（86%）参与了调查。78%为女性，诊断时的平均年龄为11.6岁，完成调查时的平均年龄为15.0岁。报告的常见PVCM症状有呼吸困难（89%）、咽部异物感（56%）和喘鸣（50%）。诊断的中位时间为3个月（四分位间距2 - 5个月）。几乎所有人都报告在被正确诊断（通常由耳鼻喉科医生诊断）之前被误诊为其他疾病，通常是哮喘。参与者报告接受了3.7项诊断性检查（范围0 - 8）；肺功能测试最为常见。在承认的众多治疗方法中，呼吸练习很常见（89%），但只有56%的人报告有帮助。三分之一的受试者回忆使用过生物反馈，但只有14%的人报告有帮助。抗反流、抗过敏、抗胆碱能药物、吸入器和类固醇的使用比例均超过50%，但很少有人报告有效。据报道，PVCM在最初诊断时是一个重大的压力源，但尽管三分之二的参与者仍报告有持续的PVCM症状，但随着时间的推移，感知到的压力显著降低（Z = 3.26，P = 0.001）。
这项首次关于PVCM的PRO研究证实，诊断往往延迟，且所开的治疗方法通常被认为无效。虽然生物反馈和呼吸练习可能对PVCM发作的短期控制至关重要，但生活方式的改变和压力的减轻可能是长期管理所必需的。对于这种疾病，需要提高认识并改善管理。
