Drooling outcome measures in paediatric disability: a systematic review

E Sforza; R Onesimo; C Leoni; V Giorgio; F Proli; F Notaro; E M Kuczynska; A Cerchiari; A Selicorni; D Rigante; G Zampino

doi:10.1007/s00431-022-04460-5

Drooling outcome measures in paediatric disability: a systematic review

Eur J Pediatr. 2022 Jul;181(7):2575-2592. doi: 10.1007/s00431-022-04460-5. Epub 2022 Apr 20.

Authors

E Sforza^#¹, R Onesimo^#², C Leoni², V Giorgio², F Proli², F Notaro², E M Kuczynska², A Cerchiari³, A Selicorni⁴, D Rigante^{2

5}, G Zampino^{2

5}

Affiliations

¹ Department of Pediatrics, Faculty of Medicine and Surgery, Catholic University of Rome, Rome, Italy. elisabetta.sforza@unicatt.it.
² 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.
³ Feeding and Swallowing Services Unit, Department of Neuroscience and Neurorehabilitation, Bambino Gesù Children's Hospital IRCCS, Rome, Italy.
⁴ Department of Paediatrics, ASST Lariana, Presidio S. Fermo, Como, Italy.
⁵ Department of Pediatrics, Faculty of Medicine and Surgery, Catholic University of Rome, Rome, Italy.

^# Contributed equally.

Abstract

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.

Keywords: Disability; Drooling; Paediatrics; Personalised medicine; Sialorrhea; Systematic review.

Publication types

Review
Systematic Review

MeSH terms

Cerebral Palsy* / complications
Child
Humans
Outcome Assessment, Health Care
Reproducibility of Results
Sialorrhea* / diagnosis
Sialorrhea* / etiology
Surveys and Questionnaires