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Assessing and Managing Lung Disease and Sleep Disordered Breathing in Children with Cerebral Palsy Paediatric Respiratory Review

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Assessing and Managing Lung Disease and Sleep Disordered Breathing in Children with Cerebral Palsy Paediatric Respiratory Review Paediatric Respiratory Reviews 10 (2009) 18–24 Contents lists available at ScienceDirect Paediatric Respiratory Reviews CME Article Assessing and managing lung disease and sleep disordered breathing in children with cerebral palsy Dominic A. Fitzgerald 1,3,*, Jennifer Follett 2, Peter P. Van Asperen 1,3 1 Department of Respiratory Medicine, University of Sydney, Sydney, New South Wales, Australia 2 Department of Physiotherapy, The Children’s Hospital at Westmead, University of Sydney, Sydney, New South Wales, Australia 3 The Children’s Hospital at Westmead Clinical School, Discipline of Paediatrics & Child Health, University of Sydney, Sydney, New South Wales, Australia EDUCATIONAL AIMS To appreciate the insidious evolution of suppurative lung disease in children with cerebral palsy (CP). To be familiar with the management of excessive oral secretions in children with CP. To understand the range of sleep problems that are more commonly seen in children with CP. To gain an understanding of the use of non-invasive respiratory support for the management of airway clearance and sleep disordered breathing in children with CP. ARTICLE INFO SUMMARY Keywords: The major morbidity and mortality associated with cerebral palsy (CP) relates to respiratory compromise. Cerebral palsy This manifests through repeated pulmonary aspiration, airway colonization with pathogenic bacteria, Pulmonary aspiration the evolution of bronchiectasis and sleep disordered breathing. An accurate assessment involving a Suppurative lung disease multidisciplinary approach and relatively simple interventions for these conditions can lead to Physiotherapy significant improvements in the quality of life of children with CP as well as their parents and carers. This Airway clearance techniques Obstructive sleep apnoea review highlights the more common problems and potential therapies with regard to suppurative lung Sleep disordered breathing disease and sleep disordered breathing in children with CP. ß 2008 Elsevier Ltd All rights reserved. INTRODUCTION LUNG DISEASE IN CHILDREN WITH CEREBRAL PALSY Cerebral palsy (CP) is a condition commonly seen in paediatric Lung disease is an important cause of morbidity and mortality practice. It is defined as a non-progressive disorder of motor in children with CP. Reddihough et al2 published an audit of child function and occurs in approximately 1 in 500 live births. European deaths from the CP Register in Victoria, Australia. The majority of figures suggest it is associated with comorbidities of severe the 155 children who died between 1970 and 1995 had severe developmental delay/mental retardation in 31%, seizures in 21%, spastic quadriplegia, intellectual disability and epilepsy. Infection severe developmental delay and non-ambulant status in 20% and was felt to be the cause of death in almost half of the children with blindness in 11%.1 Despite the fact that respiratory and sleep pneumonia (including aspiration) in 61 (39.4%) and sepsis in a problems are common in children with CP, there is comparatively further 7 (4.5%). In two publications, Baikie et al3,4 studied 63 little in the literature to guide management of the everyday children with non-ambulant spastic quadriplegic CP to document problems of cough, pulmonary aspiration and sleep disruption. the prevalence of and the best method to detect pulmonary aspiration (Fig. 1). Respiratory symptoms were common in these patients with daily cough or wheeze in 58%, cough with feeding at least 1 day per week in 84%, ‘asthma’ episodes in the last 6 months * Corresponding author. Department of Respiratory Medicine, The Children’s in 34% and snoring in 44% with documented apnoeas in 10%.3 Focal Hospital at Westmead, Locked Bag 4001, Westmead, NSW, Australia, 2145. Tel.: +61 2 9845 3397; Fax: +61 2 9845 3396.. respiratory signs were present in 24% with crackles in 19% and E-mail address: [email protected] (D.A. Fitzgerald). wheeze in 17%, while cough during a milk feed occurred in 44%.3 1526-0542/$ – see front matter ß 2008 Elsevier Ltd All rights reserved. doi:10.1016/j.prrv.2008.10.003 D.A. Fitzgerald et al. / Paediatric Respiratory Reviews 10 (2009) 18–24 19 Figure 2. CT scan demonstrating early bronchiectasis in a 6-year-old child. Figure 1. Chest radiograph of a preterm infant with severe developmental delay, subglottic stenosis and tracheostomy, chronic neonatal lung disease and recurrent pulmonary aspiration at 6 months corrected age. contribute to impaired mucociliary clearance in children with CP, including recurrent pulmonary aspiration, poor nutrition, kyphos- coliosis, motor weakness, upper and lower airway obstruction, These studies serve to highlight the importance of recognizing recurrent infection and bronchiectasis. In turn, impaired muco- potential contributors to lung disease development in children ciliary clearance may lead to the development of airway with CP and ensuring they are addressed in the management of obstruction and atelectasis, recurrent infection, bronchiectasis, these children. ventilation/perfusion inequality, reduced lung compliance and eventually, respiratory failure in these children. Kyphoscoliosis is Potential contributors to lung disease development also common in children with severe CP and occurred in 39% of children in Baikie’s cohort.3 This in turn may contribute to GOR and A number of interrelated factors can potentially contribute to pulmonary aspiration, impaired mucocilary clearance, structural the development of lung disease in children with CP (Table 1). lower airway obstruction and the development of respiratory Lower motor ability, a history of ‘asthma’ or cough and wheeze and failure. In a recent evaluation of 35 patients with severe CP, the the presence of gastro-oesophageal reflux (GOR) have been degree of spinal deformity, neck rotation deformity and severe demonstrated to predict radiological aspiration, and lower asymmetrical deformity posture, and the severity of motor adaptive ability, abnormal voice quality after feeds, lower scoliosis dysfunction were all found to predict respiratory dysfunction.5 angle and history of ‘asthma’, regurgitation or ‘aspiration’ to As highlighted previously, snoring is common in children with predict GOR in children with severe CP.3,4 Baikie et al4 also CP3 and upper airway obstruction can also occur at the supraglottic investigated the use of various tests for aspiration in their cohort of level with redundant aryepiglottic folds causing a clinical picture 63 children and found that the salivagram was most frequently of laryngomalacia. In addition, ‘asthma’ is also commonly positive (56%; CI 43–68%) with barium videofluoroscopy the next diagnosed in these children,3 but is more likely to be due to some most frequently positive test (39% CI 26–53%), while radionuclide of the other contributors to lung disease development, including scan was rarely positive (6%, CI 2–16%). However, there was poor pulmonary aspiration, bronchiectasis, impaired mucociliary clear- agreement between the tests with the best agreement being ance, kyphoscoliosis and structural upper and lower airway between the salivagram and barium videofluoroscopy. The authors obstruction. It is therefore important to be aware of the potential concluded that the salivagram was probably the most useful test at for over-treatment with asthma medications in children with CP. predicting who is at risk of aspiration in this population. An audit of 294 children with CP presenting to the dysphagia clinic at the MANAGEMENT OF LUNG DISEASE IN CHILDREN WITH CEREBRAL Children’s Hospital at Westmead demonstrated evidence of PALSY pulmonary aspiration in 72 (41%) of the 174 videofluoroscopy studies performed, while 42 (67%) of the 62 CT scans performed The management of lung disease in children with CP is demonstrated bronchiectasis (Fig. 2), again highlighting pulmon- summarized in Table 2. While thickened feeds and antireflux ary aspiration as an important cause of lung disease in children therapy should be trialled initially, many children, particularly with CP (unpublished data). A number of factors are likely to those with severe CP, will require gastrostomy feeds combined with fundoplication if there is significant GOR. However, continu- ing aspiration of saliva may be an ongoing problem. Antic- Table 1 holinergic therapy in the form of both benztropine6 and Potential contributors to lung disease development glycopyrrolate7,8 has been documented in open label studies as Pulmonary aspiration being effective in patients with CP and is generally favoured as Impaired mucociliary clearance first-line therapy.9,10 Glycopyrrolate has the theoretical advantage Recurrent infection leading to bronchiectasis of having fewer side effects.10 Botulinum toxin injections into the Kyphoscoliosis Upper airway obstruction salivary glands has been used as alternative treatment for the Lower airway obstruction – ‘asthma’ management of sialorrhea9 but trials have been limited.10,11 Surgery remains an option for patients with persistent problems 20 D.A. Fitzgerald et al. / Paediatric Respiratory Reviews 10 (2009) 18–24 Table 2 Chest physiotherapy Management of lung disease in children with cerebral palsy Chest physiotherapy is now referred to by the more generic Pulmonary aspiration term of ‘airways clearance techniques’ (ACTs).12 There is a dearth Thickened feeds and antireflux treatment of evidence as to the most effective ACT for children with CP. The Gastrostomy and fundoplication Control of saliva degree of disability will influence the most
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