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      Impact and management of drooling in children with neurological disorders: an Italian Delphi consensus

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          Abstract

          Background

          The rate of chronic drooling in children older than 4 years is 0.5%, but it rises to 60% in those with neurological disorders. Physical and psychosocial consequences lead to a reduction in the quality of Life (QoL) of affected patients; however, the problem remains under-recognized and under-treated. We conducted an Italian consensus through a modified Delphi survey to discuss the current treatment paradigm of drooling in pediatric patients with neurological disorders.

          Methods

          After reviewing the literature, a board of 10 experts defined some statements to be administered to a multidisciplinary panel through an online encrypted platform. The answers to the questions were based on a 1–5 Likert scale (1 = strongly disagree; 5 = strongly agree). The scores were grouped into 1–2 (disagreement) and 4–5 (agreement), while 3 was discarded. The consensus was reached when the sum of the disagreement or agreement was ≥75%.

          Results

          Fifteen statements covered three main topics, namely clinical manifestations and QoL, quantification of drooling, and treatment strategies. All statements reached consensus (≥75% agreement). The 55 Italian experts agreed that drooling should be assessed in all children with complex needs, having a major impact on the QoL. Attention should be paid to investigating posterior hypersalivation, which is often neglected but may lead to important clinical consequences. Given that the severity of drooling fluctuates over time, its management should be guided by the patients’ current needs. Furthermore, the relative lack of validated and universal scales for drooling quantification limits the evaluation of the response to treatment. Finally, the shared therapeutic paradigm is progressive, with conservative treatments preceding the pharmacological ones and reserving surgery only for selected cases.

          Conclusion

          This study demonstrates the pivotal importance of a multidisciplinary approach to the management of drooling. National experts agree that progressive treatment can reduce the incidence of complications, improve the QoL of patients and caregivers, and save healthcare resources. Finally, this study highlights how the therapeutic strategy should be reconsidered over time according to the available drugs on the market, the progression of symptoms, and the patients’ needs.

          Supplementary Information

          The online version contains supplementary material available at 10.1186/s13052-022-01312-8.

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          Most cited references24

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          Defining consensus: a systematic review recommends methodologic criteria for reporting of Delphi studies.

          To investigate how consensus is operationalized in Delphi studies and to explore the role of consensus in determining the results of these studies. Systematic review of a random sample of 100 English language Delphi studies, from two large multidisciplinary databases [ISI Web of Science (Thompson Reuters, New York, NY) and Scopus (Elsevier, Amsterdam, NL)], published between 2000 and 2009. About 98 of the Delphi studies purported to assess consensus, although a definition for consensus was only provided in 72 of the studies (64 a priori). The most common definition for consensus was percent agreement (25 studies), with 75% being the median threshold to define consensus. Although the authors concluded in 86 of the studies that consensus was achieved, consensus was only specified a priori (with a threshold value) in 42 of these studies. Achievement of consensus was related to the decision to stop the Delphi study in only 23 studies, with 70 studies terminating after a specified number of rounds. Although consensus generally is felt to be of primary importance to the Delphi process, definitions of consensus vary widely and are poorly reported. Improved criteria for reporting of methods of Delphi studies are required. Copyright © 2014 Elsevier Inc. All rights reserved.
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            Medical assessment by a Delphi group opinion technic.

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              Sialorrhea: a management challenge.

              Sialorrhea (drooling or excessive salivation) is a common problem in neurologically impaired children (i.e., those with mental retardation or cerebral palsy) and in adults who have Parkinson's disease or have had a stroke. It is most commonly caused by poor oral and facial muscle control. Contributing factors may include hypersecretion of saliva, dental malocclusion, postural problems, and an inability to recognize salivary spill. Sialorrhea causes a range of physical and psychosocial complications, including perioral chapping, dehydration, odor, and social stigmatization, that can be devastating for patients and their families. Treatment of sialorrhea is best managed by a clinical team that includes primary health care providers, speech pathologists, occupational therapists, dentists, orthodontists, neurologists, and otolaryngologists. Treatment options range from conservative (i.e., observation, postural changes, biofeedback) to more aggressive measures such as medication, radiation, and surgical therapy. Anticholinergic medications, such as glycopyrrolate and scopolamine, are effective in reducing drooling, but their use may be limited by side effects. The injection of botulinum toxin type A into the parotid and submandibular glands is safe and effective in controlling drooling, but the effects fade in several months, and repeat injections are necessary. Surgical intervention, including salivary gland excision, salivary duct ligation, and duct rerouting, provides the most effective and permanent treatment of significant sialorrhea and can greatly improve the quality of life of patients and their families or caregivers.
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                Author and article information

                Contributors
                pasqualestriano@gaslini.org
                Journal
                Ital J Pediatr
                Ital J Pediatr
                Italian Journal of Pediatrics
                BioMed Central (London )
                1824-7288
                19 July 2022
                19 July 2022
                2022
                : 48
                : 118
                Affiliations
                [1 ]GRID grid.419504.d, ISNI 0000 0004 1760 0109, Pediatric Neurology and Muscular Diseases Unit, IRCCS Istituto Giannina Gaslini, ; Via Gaslini 5, 16148 Genoa, Italy
                [2 ]GRID grid.5606.5, ISNI 0000 0001 2151 3065, Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health, , University of Genova, ; Genoa, Italy
                [3 ]GRID grid.7841.a, Department of Pediatrics, , “Sapienza” University of Rome, ; Rome, Italy
                [4 ]GRID grid.414818.0, ISNI 0000 0004 1757 8749, Neurology Unit, , Maggiore Hospital, ; Lodi, Italy
                [5 ]Department of Child Neuropsychiatry, Hospital Maggiore della Carità, Novara, Italy
                [6 ]GRID grid.7644.1, ISNI 0000 0001 0120 3326, Department of Biomedical Sciences and Human Oncology, Neonatology and Intensive Care Neonatal Unit Section, , University of Bari Aldo Moro, ; Bari, Italy
                [7 ]GRID grid.414759.a, ISNI 0000 0004 1760 170X, Pediatric Neurology Unit and Epilepsy Center, , Fatebenefratelli Hospital, ; ASST Fatebenefratelli Sacco, Milan, Italy
                [8 ]GRID grid.9027.c, ISNI 0000 0004 1757 3630, Department of Pediatrics, , University of Perugia, ; Perugia, Italy
                Author information
                http://orcid.org/0000-0002-6065-1476
                Article
                1312
                10.1186/s13052-022-01312-8
                9297577
                35854335
                86a1d828-6103-4f47-80db-874e569fb44a
                © The Author(s) 2022

                Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.

                History
                : 22 November 2021
                : 24 June 2022
                Funding
                Funded by: Proveca Pharma Ltd
                Categories
                Research
                Custom metadata
                © The Author(s) 2022

                Pediatrics
                drooling,neurological disorders,pediatrics,cerebral palsy,delphi
                Pediatrics
                drooling, neurological disorders, pediatrics, cerebral palsy, delphi

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