Department of Pediatrics, Faculty of Medicine and Surgery, Catholic University of Rome, Rome, Italy.
Centre for Rare Diseases and Birth Defects, Department of Woman and Child Health and Public Health, Fondazione Policlinico Universitario A. Gemelli IRCCS, Largo A. Gemelli 8, 00168, Rome, Italy.
Eur J Pediatr. 2022 Jul;181(7):2575-2592. doi: 10.1007/s00431-022-04460-5. Epub 2022 Apr 20.
Drooling, or sialorrhea, is a common condition in patients with cerebral palsy, rare diseases, and neurodevelopmental disorders. The goal of this review was to identify the different properties of sialorrhea outcome measures in children. Four databases were analysed in search of sialorrhea measurement tools, and the review was performed according to the Preferred Reporting Items for Systematic reviews and Meta-Analyses (PRISMA) statement. The COnsensus-based Standards for the selection of health status Measurement INstruments (COSMIN) checklist was used for quality appraisal of the outcome measures. The initial search yielded 891 articles, 430 of which were duplicates. Thus, 461 full-text articles were evaluated. Among these, 21 met the inclusion criteria, reporting 19 different outcome measures that encompassed both quantitative measures and parent/proxy questionnaires. Conclusions: Among the outcome measures found through this review, the 5-min Drooling Quotient can objectively discriminate sialorrhea frequency in patients with developmental disabilities. The Drooling Impact Scale can be used to evaluate changes after treatment. The modified drooling questionnaire can measure sialorrhea severity and its social acceptability. To date, the tests proposed in this review are the only tools displaying adequate measurement properties. The acquisition of new data about reliability, validity, and responsiveness of these tests will confirm our findings. What is Known: • Although sialorrhea is a recognized problem in children with disabilities, especially those with cerebral palsy (CP), there is a lack of confidence among physicians in measuring sialorrhea. What is New: • Few sialorrhea measures are available for clinicians that may guide decision-making and at the same time have strong evidence to provide confidence in the results. • A combination of both quantitative measures and parent/proxy questionnaires might provide an adequate measurement of sialorrhea in children.
流涎,又称涎液过多,是脑瘫、罕见病和神经发育障碍患者的常见病症。本综述旨在确定儿童流涎症结果测量的不同特性。分析了四个数据库以寻找流涎症测量工具,并根据系统评价和荟萃分析的首选报告项目 (PRISMA) 声明进行了综述。使用共识基础健康状况测量工具选择标准 (COSMIN) 清单对结果测量进行质量评估。最初的搜索产生了 891 篇文章,其中 430 篇是重复的。因此,评估了 461 篇全文文章。其中,21 篇符合纳入标准,报告了 19 种不同的结果测量方法,包括定量测量和家长/代理人问卷。结论:通过本次综述发现的结果测量中,5 分钟流涎量可以客观地区分发育障碍患者的流涎频率。流涎影响量表可用于评估治疗后的变化。改良流涎问卷可测量流涎的严重程度及其社会可接受性。迄今为止,本综述中提出的测试是唯一显示出充分测量特性的工具。获得这些测试的可靠性、有效性和响应性的新数据将证实我们的发现。已知:•尽管流涎症是残疾儿童(尤其是脑瘫儿童)公认的问题,但医生在测量流涎症方面缺乏信心。新发现:• 可供临床医生使用的流涎症测量方法很少,这些方法可能有助于指导决策,同时具有强有力的证据,使结果更可信。•定量测量和家长/代理人问卷的组合可能会为儿童流涎症提供充分的测量。