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      Comparison of Frequency and Severity of Sleep-Related Breathing Disorders in Children with Simple Obesity and Paediatric Patients with Prader–Willi Syndrome

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          Abstract

          Sleep-related breathing disorders (SRBDs) can be present in children with simple obesity and with Prader–Willi syndrome (PWS) and influence an individual diagnostic and treatment approach. We compared frequency and severity of SRBDs in children with simple obesity and with PWS, both without and on recombinant human growth hormone (rhGH) treatment, and correlation of SRBDs with insulin resistance tests. A screening polysomnography-polygraphy (PSG), the oral glucose tolerance test (OGTT) and homeostasis model assessment of insulin resistance (HOMA-IR) were analysed in three groups of patients—with simple obesity (group 1, n = 30, mean age 14.2 years), patients with PWS without the rhGH therapy (group 2, n = 8, mean age 13.0 years) and during the rhGH treatment (group 3, n = 17, mean age 8.9 years). The oxygen desaturation index (ODI) was significantly higher in groups 2 and 3, compared to group 1 ( p = 0.00), and hypopnea index (HI) was higher in group 1 ( p = 0.03). Apnea–hypopnea index (AHI) and apnea index (AI) results positively correlated with the insulin resistance parameters in groups 1 and 3. The PSG values worsened along with the increasing insulin resistance in children with simple obesity and patients with PWS treated with rhGH that may lead to a change in the patients’ care.

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          Diagnosis and management of childhood obstructive sleep apnea syndrome.

          C Marcus, L. Brooks, K. Draper … (2012)
          This revised clinical practice guideline, intended for use by primary care clinicians, provides recommendations for the diagnosis and management of the obstructive sleep apnea syndrome (OSAS) in children and adolescents. This practice guideline focuses on uncomplicated childhood OSAS, that is, OSAS associated with adenotonsillar hypertrophy and/or obesity in an otherwise healthy child who is being treated in the primary care setting. Of 3166 articles from 1999-2010, 350 provided relevant data. Most articles were level II-IV. The resulting evidence report was used to formulate recommendations. The following recommendations are made. (1) All children/adolescents should be screened for snoring. (2) Polysomnography should be performed in children/adolescents with snoring and symptoms/signs of OSAS; if polysomnography is not available, then alternative diagnostic tests or referral to a specialist for more extensive evaluation may be considered. (3) Adenotonsillectomy is recommended as the first-line treatment of patients with adenotonsillar hypertrophy. (4) High-risk patients should be monitored as inpatients postoperatively. (5) Patients should be reevaluated postoperatively to determine whether further treatment is required. Objective testing should be performed in patients who are high risk or have persistent symptoms/signs of OSAS after therapy. (6) Continuous positive airway pressure is recommended as treatment if adenotonsillectomy is not performed or if OSAS persists postoperatively. (7) Weight loss is recommended in addition to other therapy in patients who are overweight or obese. (8) Intranasal corticosteroids are an option for children with mild OSAS in whom adenotonsillectomy is contraindicated or for mild postoperative OSAS.
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            Growth Hormone Research Society Workshop Summary: Consensus Guidelines for Recombinant Human Growth Hormone Therapy in Prader-Willi Syndrome

            Cheri Deal, Michèle Tony, Charlotte Höybye … (2013)
            Context: Recombinant human GH (rhGH) therapy in Prader-Willi syndrome (PWS) has been used by the medical community and advocated by parental support groups since its approval in the United States in 2000 and in Europe in 2001. Its use in PWS represents a unique therapeutic challenge that includes treating individuals with cognitive disability, varied therapeutic goals that are not focused exclusively on increased height, and concerns about potential life-threatening adverse events. Objective: The aim of the study was to formulate recommendations for the use of rhGH in children and adult patients with PWS. Evidence: We performed a systematic review of the clinical evidence in the pediatric population, including randomized controlled trials, comparative observational studies, and long-term studies (>3.5 y). Adult studies included randomized controlled trials of rhGH treatment for ≥ 6 months and uncontrolled trials. Safety data were obtained from case reports, clinical trials, and pharmaceutical registries. Methodology: Forty-three international experts and stakeholders followed clinical practice guideline development recommendations outlined by the AGREE Collaboration (www.agreetrust.org). Evidence was synthesized and graded using a comprehensive multicriteria methodology (EVIDEM) (http://bit.ly.PWGHIN). Conclusions: Following a multidisciplinary evaluation, preferably by experts, rhGH treatment should be considered for patients with genetically confirmed PWS in conjunction with dietary, environmental, and lifestyle interventions. Cognitive impairment should not be a barrier to treatment, and informed consent/assent should include benefit/risk information. Exclusion criteria should include severe obesity, uncontrolled diabetes mellitus, untreated severe obstructive sleep apnea, active cancer, or psychosis. Clinical outcome priorities should vary depending upon age and the presence of physical, mental, and social disability, and treatment should be continued for as long as demonstrated benefits outweigh the risks.
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              Recommendations for the diagnosis and management of Prader-Willi syndrome.

              John M. Tauber, B Hauffa, … (2008)
              The objective of the study was to provide recommendations for the diagnosis and management of Prader-Willi syndrome throughout the life span to guide clinical practice. An open international multidisciplinary expert meeting was held in October 2006 in Toulouse, France, with 37 invited speakers and session chairs (see Acknowledgments) and 85 additional registered participants. The meeting was supported by an unrestricted educational grant from Pfizer. Invited participants with particular expertise reviewed the published evidence base for their specialist topic and unpublished data from personal experience, previous national and international PWS conferences, and PWS Association clinical advisory groups. Sessions covered epidemiology, psychiatric, and behavioral disorders; breathing and sleep abnormalities; genetics; endocrinology; and management in infancy, childhood, transition, and adulthood. This included group meetings including open discussion after each session. The guidelines were written by the Scientific Committee (authors), using the conclusions provided by the sessions chairs and summary provided by each speaker, including incorporation of changes suggested after review by selected meeting participants (see Acknowledgments). The diagnosis and management of this complex disorder requires a multidisciplinary approach with particular emphasis on the importance of early diagnosis using accredited genetic testing, use and monitoring of GH therapy from early childhood, control of the food environment and regular exercise, appropriate management of transition, consideration of group home placement in adulthood, and distinction of behavioral problems from psychiatric illness.
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                Author and article information

                Contributors
                Stephen B. Liggett: Role: Academic Editor
                Journal
                Journal ID (nlm-ta): J Pers Med
                Journal ID (iso-abbrev): J Pers Med
                Journal ID (publisher-id): jpm
                Title: Journal of Personalized Medicine
                Publisher: MDPI
                ISSN (Electronic): 2075-4426
                Publication date (Electronic): 18 February 2021
                Publication date Collection: February 2021
                Volume: 11
                Issue: 2
                Electronic Location Identifier: 141
                Affiliations
                [1 ]Department of Endocrinology and Diabetology, The Children’s Memorial Health Institute, Al. Dzieci Polskich 20, 04-730 Warsaw, Poland; m.wysocka@ 123456ipczd.pl (M.W.-M.); a.swiercz@ 123456ipczd.pl (A.Ś.); m.szalecki@ 123456ipczd.pl (M.S.)
                [2 ]Centre of Diagnosis and Therapy of Respiratory Diseases Pul-Med, 25-322 Kielce, Poland; jedrzejczak1958@ 123456tlen.pl
                [3 ]Collegium Medicum, Jan Kochanowski University, 25-369 Kielce, Poland
                Author notes
                [* ]Correspondence: aleckaambroziak@ 123456gmail.com ; Tel.: +48-22-815-75-80
                Article
                Publisher ID: jpm-11-00141
                DOI: 10.3390/jpm11020141
                PMC ID: 7923084
                PubMed ID: 33670584
                SO-VID: 5893bf21-fb0f-437f-ac7f-29ee1870e476
                Copyright © © 2021 by the authors.
                License:

                Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license ( http://creativecommons.org/licenses/by/4.0/).

                History
                Date received : 17 January 2021
                Date accepted : 15 February 2021
                Categories
                Subject: Article

                Keywords: simple obesity,prader–willi syndrome,sleep-related breathing disorders,obstructive sleep apnea,central sleep apnea
                Data availability:
                Keywords: simple obesity, prader–willi syndrome, sleep-related breathing disorders, obstructive sleep apnea, central sleep apnea

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