Literature DB >> 22711495

Lung function, diagnosis, and treatment of sleep-disordered breathing in children with achondroplasia.

Sébastien Julliand1, Michèle Boulé, Geneviève Baujat, Adriana Ramirez, Vincent Couloigner, Nicole Beydon, Michel Zerah, Federico di Rocco, Martine Lemerrer, Valérie Cormier-Daire, Brigitte Fauroux.   

Abstract

Children with achondroplasia are at risk of sleep-disordered breathing. The aim of the study was to evaluate lung function and sleep-disordered breathing in children with achondroplasia. An interview, clinical examination, lung function tests with blood gases, and a polygraphic sleep study were obtained as part of routine annual evaluation in consecutive children with achondroplasia. We included 30 children (median age 3.0 years, range: 0.4-17.1) over a period of 21 months. Habitual snoring and witnessed apneas were observed in 77% and 33% of the patients, respectively. Prior to the sleep study, 10/29 (34%) patients had undergone upper airway surgery and 5/29 (17%) craniocervical decompression operation. Arterial blood gases were abnormal in two (7%) patients. Sleep findings were abnormal in 28/30 (93%) patients. Eleven (37%) patients had an apnea index≥1 event/hr and 26 (87%) had an apnea-hypopnea index≥5 events/hr. The ≥3% desaturation index was >5/hr in 22 (73%) patients. Sixteen (53%) patients had a minimal pulse oximetry<90% but only two (7%) patients had a maximal transcutaneous carbon dioxide pressure>50 mmHg during sleep. As a consequence, the following therapeutic interventions were performed: upper airway surgery in four patients and noninvasive positive pressure ventilation (NPPV) in five other patients, resulting in an improvement in sleep studies in all nine patients. Systematic sleep studies are recommended in children with achondroplasia because of the high prevalence of sleep-disordered breathing. Upper airway surgery and NPPV are effective treatments of sleep-disordered breathing.
Copyright © 2012 Wiley Periodicals, Inc.

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Year:  2012        PMID: 22711495     DOI: 10.1002/ajmg.a.35441

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  11 in total

1.  A three-month-old achondroplastic baby with both obstructive apneas and central apneas.

Authors:  Lourdes M DelRosso; Eduardo Gonzalez-Toledo; Romy Hoque
Journal:  J Clin Sleep Med       Date:  2013-03-15       Impact factor: 4.062

2.  Sleep-Disordered Breathing in Children with Rare Skeletal Disorders: A Survey of Clinical Records.

Authors:  Marco Zaffanello; Giorgio Piacentini; Luca Sacchetto; Angelo Pietrobelli; Emma Gasperi; Marco Barillari; Nicolò Cardobi; Luana Nosetti; Diego Ramaroli; Franco Antoniazzi
Journal:  Med Princ Pract       Date:  2018-06-21       Impact factor: 1.927

Review 3.  Sleep disordered breathing in children with achondroplasia.

Authors:  Marco Zaffanello; Gaetano Cantalupo; Giorgio Piacentini; Emma Gasperi; Luana Nosetti; Paolo Cavarzere; Diego Alberto Ramaroli; Aliza Mittal; Franco Antoniazzi
Journal:  World J Pediatr       Date:  2016-10-15       Impact factor: 2.764

Review 4.  International Consensus Statement on the diagnosis, multidisciplinary management and lifelong care of individuals with achondroplasia.

Authors:  Ravi Savarirayan; Penny Ireland; Melita Irving; Dominic Thompson; Inês Alves; Wagner A R Baratela; James Betts; Michael B Bober; Silvio Boero; Jenna Briddell; Jeffrey Campbell; Philippe M Campeau; Patricia Carl-Innig; Moira S Cheung; Martyn Cobourne; Valérie Cormier-Daire; Muriel Deladure-Molla; Mariana Del Pino; Heather Elphick; Virginia Fano; Brigitte Fauroux; Jonathan Gibbins; Mari L Groves; Lars Hagenäs; Therese Hannon; Julie Hoover-Fong; Morrys Kaisermann; Antonio Leiva-Gea; Juan Llerena; William Mackenzie; Kenneth Martin; Fabio Mazzoleni; Sharon McDonnell; Maria Costanza Meazzini; Josef Milerad; Klaus Mohnike; Geert R Mortier; Amaka Offiah; Keiichi Ozono; John A Phillips; Steven Powell; Yosha Prasad; Cathleen Raggio; Pablo Rosselli; Judith Rossiter; Angelo Selicorni; Marco Sessa; Mary Theroux; Matthew Thomas; Laura Trespedi; David Tunkel; Colin Wallis; Michael Wright; Natsuo Yasui; Svein Otto Fredwall
Journal:  Nat Rev Endocrinol       Date:  2021-11-26       Impact factor: 47.564

5.  Tyrosine kinase inhibitor NVP-BGJ398 functionally improves FGFR3-related dwarfism in mouse model.

Authors:  Davide Komla-Ebri; Emilie Dambroise; Ina Kramer; Catherine Benoist-Lasselin; Nabil Kaci; Cindy Le Gall; Ludovic Martin; Patricia Busca; Florent Barbault; Diana Graus-Porta; Arnold Munnich; Michaela Kneissel; Federico Di Rocco; Martin Biosse-Duplan; Laurence Legeai-Mallet
Journal:  J Clin Invest       Date:  2016-04-11       Impact factor: 14.808

Review 6.  Sleep-disordered breathing in paediatric setting: existing and upcoming of the genetic disorders.

Authors:  Marco Zaffanello; Franco Antoniazzi; Laura Tenero; Luana Nosetti; Michele Piazza; Giorgio Piacentini
Journal:  Ann Transl Med       Date:  2018-09

Review 7.  Current Care and Investigational Therapies in Achondroplasia.

Authors:  Sheila Unger; Luisa Bonafé; Elvire Gouze
Journal:  Curr Osteoporos Rep       Date:  2017-04       Impact factor: 5.096

8.  Clinical Practice Guidelines for Achondroplasia.

Authors:  Takuo Kubota; Masanori Adachi; Taichi Kitaoka; Kosei Hasegawa; Yasuhisa Ohata; Makoto Fujiwara; Toshimi Michigami; Hiroshi Mochizuki; Keiichi Ozono
Journal:  Clin Pediatr Endocrinol       Date:  2020-01-09

Review 9.  Sleep and sleep disorders in rare hereditary diseases: a reminder for the pediatrician, pediatric and adult neurologist, general practitioner, and sleep specialist.

Authors:  Natan Gadoth; Arie Oksenberg
Journal:  Front Neurol       Date:  2014-07-17       Impact factor: 4.003

10.  Evaluation of neurocognitive abilities in children affected by obstructive sleep apnea syndrome before and after adenotonsillectomy.

Authors:  Domenico Testa; Marco Carotenuto; Francesco Precenzano; Alessia Russo; Anna Donadio; Giuseppina Marcuccio; Gaetano Motta
Journal:  Acta Otorhinolaryngol Ital       Date:  2020-04       Impact factor: 2.124

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