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Therapeutic Management with Airway Clearance in Children with Robin Sequence and Association with Swallowing Outcomes: A Systematic Review and Meta-analysis

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Abstract

Dysphagia in Robin Sequence can be present in varying degrees, requiring multidisciplinary management and specific swallowing assessment by a specialist. Most studies published to date have evaluated only respiratory outcomes, and the available evidence on the improvement of swallowing is questionable. To conduct a systematic review and meta-analysis of studies evaluating swallowing in children with Robin Sequence before and after airway clearance procedures. The research question was developed based on the PICO strategy. The literature search was performed in electronic databases and gray literature. Studies were selected by 3 independent reviewers. The risk of bias and level of evidence of the studies were assessed. A proportion meta-analysis was performed to calculate the prevalence of dysphagia after airway clearance procedures. The search identified 4938 studies, 5 of which were included. All studies had limitations in terms of design and sample size. The prevalence of dysphagia after airway clearance was obtained by analyzing treatment subgroups: mandibular distraction osteogenesis, mandibular distraction osteogenesis + tracheostomy tube, and nasopharyngeal tube. Clinical and/or instrumental assessment was assessed by a swallowing specialist. The meta-analysis was precluded by the limitations of the studies, especially regarding sample size, which affected the accuracy of the findings. Dysphagia remained unresolved in 55% of children (95% CI 1–99%). The methodological quality of the studies indicated a high risk of bias and very low level of evidence. It was not possible to confirm that airway clearance techniques used in Robin Sequence improve dysphagia.

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Acknowledgements

The study was carried out at Universidade Federal do Rio Grande do Sul (UFRGS), Porto Alegre, Rio Grande do Sul, Brazil.

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Authors and Affiliations

  1. Graduate Program in Child and Adolescent Health, School of Medicine, Universidade Federal do Rio Grande do Sul (UFRGS), R. Ramiro Barcelos, 2400, Porto Alegre, RS, 90035-003, Brazil

    Marisa Gasparin, Fabiola Luciane Barth, Luciane Ferreira Pauletti, Miriam Izabel Souza dos Santos Simon, Cláudia Schweiger & Paulo José Cauduro Marostica

  2. Graduate Program in Respiratory Sciences, UFRGS, R. Ramiro Barcelos, 2400, Porto Alegre, RS, 90035-003, Brazil

    Renata Italiano da Nóbrega Figueiredo

  3. Department of Otolaryngology, Pediatric Otolaryngology Unit, Hospital de Clínicas de Porto Alegre (HCPA), Rua Ramiro Barcelos, 2350, Av. Protásio Alves, 211, Porto Alegre, RS, 90035-903, Brazil

    Cláudia Schweiger

  4. School of Speech-Language Pathology and Audiology, UFRGS, Rua Ramiro Barcelos 2492, Porto Alegre, RS, 90035-003, Brazil

    Deborah Salle Levy

  5. Department of Pediatrics, Pediatric Pulmonology Unit, HCPA, Rua Ramiro Barcelos, 2350, Av. Protásio Alves, 211, Porto Alegre, RS, 90035-903, Brazil

    Paulo José Cauduro Marostica

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  1. Marisa Gasparin
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  2. Fabiola Luciane Barth
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  3. Luciane Ferreira Pauletti
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  8. Paulo José Cauduro Marostica
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Contributions

Study conception and design: MG, MIS, DSL, PJCM. Data acquisition: MG, FLB, LFP. Analysis and data interpretation: MG, RINF, CS, DSL, PJCM. Drafting of the manuscript: MG, DSL, PJCM. Critical revision: MG, FLB, DSL, PJCM. Final approval of the article: MG, FLB, LFP, MIS, RINF, CS, DSL, PJCM. Statistical analysis: MG, RINF, CS, DSL, PJCM. Overall responsibility: MG, FLB, LFP, MIS, RINF, CS, DSL, PJCM. All authors have read and approved the final version of the article.

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Correspondence to Marisa Gasparin.

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Gasparin, M., Barth, F.L., Pauletti, L.F. et al. Therapeutic Management with Airway Clearance in Children with Robin Sequence and Association with Swallowing Outcomes: A Systematic Review and Meta-analysis. Dysphagia 38, 1267–1276 (2023). https://doi.org/10.1007/s00455-023-10561-6

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