Compliance Rates in Children Using Noninvasive Continuous Positive Airway Pressure

Transcription

1 PEDIATRICS Compliance Rates in Children Using Noninvasive Continuous Positive Airway Pressure Anne R. O Donnell, FRACP, PhD1; Candice L. Bjornson, MD, MSc1; Shelly G. Bohn, BSc, RPSGT; Valerie G. Kirk, MD1 University of Calgary, Alberta Children s Hospital, Calgary, Canada nea-hypopnea index was 11.3 (interquartile range, ). Follow-up ranged from 8 to 7 days. Forty-eight percent of children used ncpap immediately. Seventy-six percent of children used ncpap for at least half the days, with use defined as 1 or more hours of recording during a 24hour period. Mean daily use was 4.7 hours (interquartile range, ), and mean daily use on days ncpap was used was 6.3 hours (interquartile range, ) Conclusions: With patience, a behavioral modification approach, and parental commitment, children will be accepting of ncpap and reasonably compliant with treatment. Keywords: Obstructive sleep apnea, sleep-disordered breathing, noninvasive ventilation Citation: O Donnell AR; Bjornson CL; Bohn SG et al. Compliance rates in children using noninvasive continuous positive airway pressure. SLEEP 2006;2(5): Study Objectives: For a subpopulation of children with obstructive sleep apnea, the mainstay of treatment is nasal continuous positive airway pressure (ncpap). Accurate measures of time in use have not been used to assess compliance with ncpap in large numbers of children. Data from a comprehensive ncpap program are used to describe ncpap use among children aged 6 months to 18 years and provide time-in-use compliance rates. Design: Retrospective cohort study Setting: University Pediatric Teaching Hospital Interventions: Initiation of ncpap treatment Measurements and Results: Seventy-nine children were identified as requiring treatment with ncpap, with 65 (82%) successfully established on ncpap during the 46-month study period. Objective compliance data were available on 50 children: 66% were boys, 78% had a complicating medical disorder, the mean age was 10 ± 5.1 years, and the median ap- Pediatric Sleep Service in Calgary, Alberta, Canada, between October 1 and July Funding for ncpap devices was provided by the government agency Alberta Aids to Daily Living, allowing all eligible children access to ncpap machines. Children were considered eligible for ncpap treatment if they had OSA, as defined by an apnea-hypopnea index (AHI) greater than 1 on a polysomnogram (PSG) 12 and had either previously undergone adenotonsillectomy or were felt by the pediatric sleep physicians to be a poor candidate for adenotonsillectomy, for example, very high anesthetic risk or minimal adenotonsillar tissue on nasendoscopy. Children with an AHI between 1 and 5 were required to have associated symptoms (e.g., daytime behavioral or learning problems, sleep disruption) or gas-exchange abnormalities before ncpap treatment was prescribed. Children shown on PSG to need bilevel positive airway pressure (BiPAP ) rather than CPAP to treat underlying airway obstruction were not included in the study. Because of local funding regulations, BiPAP is reserved for use in those children in whom ncpap is not effective and is therefore uncommonly used for the treatment of OSA. The study was approved by the institutional review board. Parents gave written informed consent for their child s data to be compiled and analyzed for research purposes. INTRODUCTION OBSTRUCTIVE SLEEP APNEA (OSA) IS A COMMON CONDITION THAT AFFECTS 2% TO 4% OF CHILDREN.2,3 UNTREATED, OSA MAY ADVERSELY AFFECT school performance, behavior, and growth, as well as lead to pulmonary hypertension and cor pulmonale.4-13 Although adenotonsillectomy is first-line therapy for OSA in the pediatric population, surgery is not always indicated or effective.8,14 For these children, nasal continuous positive airway pressure (ncpap) is the preferred treatment. Since it was first used in children in 180,15 ncpap has been shown to be a safe and effective therapy for infants and children, with success rates for use of over 80% Nevertheless, none of these studies have been prospective, only a few have objectively assessed compliance with therapy, and none have accurately measured time in use to assess ncpap compliance in large numbers of children This report uses data from a comprehensive pediatric ncpap program in Alberta, Canada, to describe ncpap use among children aged 6 months to 18 years and provide time-in-use information on ncpap compliance during childhood. MATERIALS AND METHODS Patient Population Polysomnography The study was conducted at the Alberta Children s Hospital Overnight PSG studies were performed at the Alberta Children s Hospital Pediatric Sleep Laboratory. Computerized laboratory PSGs were performed according to American Thoracic Society guidelines using the Sandman NT (Nellcor Puritan Bennett, Ottawa, ON, Canada).2 PSG monitoring included electroencephalogram (C4-A1, C3-A2, O2-A1, O1-A2), electrooculogram (right and left), electromyogram (chin, intercostal, diaphragm and bilateral tibial), electrocardiogram, oxygen saturation (SaO2), pulse-wave monitoring (N200, Nellcor Puritan Bennett, Ottawa, ON, Canada), respiratory inductance plethysmography (chest, Disclosure Statement This was not an industry supported study. Drs. Kirk, Bohn, Bjornson, and O Donnell have indicated no financial conflicts of interest. Submitted for publication August 2005 Accepted for publication January 2006 Address correspondence to: Dr V. Kirk, 1820 Richmond Rd SW, Calgary, AB, Canada T2T 5C7; Tel: (403) ; Fax: (403) ; val. kirk@calgaryhealthregion.ca SLEEP, Vol. 2, No. 5, 2006 Downloaded from 651

2 abdominal wall and sum channel movements)(respitrace Plu s, SensorMedics Corporation, Yorba Linda, CA), end-tidal CO 2 (Novametrix Medical Systems Inc, Wallinfgord, CT), nasal/oral airflow using a thermistor device (Edentec Model 3170 Sleep Lab Airflow Cable, Edentec Corporation, Eden Prairie, MN), nasal pressure (Ultima Airflow Pressure Sensor Model 0580, Braebon Medical Corporation, Ottawa, ON, Canada) and transcutaneous CO 2 (Radiometer Compact Combined PCO2/PO2 Monitoring System TCM3, Radiometer Medical, Copenhagen, Denmark). PSGs were manually scored by trained sleep technicians using standard criteria. 30 Specialists in pediatric sleep medicine provided interpretation of scored PSG studies. Total AHI was calculated as the number of respiratory events per hour of sleep. OSA was defined as an AHI of greater than 1 per hour. 31,32 A titration PSG was carried out to confirm ncpap as the correct treatment and to determine the optimal pressure required to overcome obstructive apnea, arterial oxygen desaturation, and hypercapnia. If ncpap did not correct these parameters, then the child was treated with BiPAP and was not included in the study. Laboratory ncpap titration was performed using the Knightstar 335 Respiratory Support System (Nellcor Puritan Bennett), with ncpap pressures titrated according to standardized laboratory titration protocols (available upon request). Nasal CPAP ncpap devices used for patient treatment were the Aria LX and REMstar Pro models (Respironics). A selection of nasal masks (ResMed Infant System and Ultra Mirage, Respironics Child Profile Lite, Profile Lite, Simplicity and Comfort Series, Fisher and Paykel Flexifit 407) and full face masks (ResMed Ultra Mirage, Respironics Spectrum and Comfort) were used with choice depending on facial structure, individual patient preference and mask availability. Heated air humidifiers (Fisher and Paykel Healthcare, Auckland, New Zealand; REMstar Pro, Respironics, Murrysville, PA) were routinely used in all patients. ncpap pressure was ramped when possible. Before being given a ncpap machine, children and at least 1 parent or caregiver underwent a 1-on-1 educational consultation with a qualified sleep technologist. 25,32,33 The sleep technologist spent approximately 1 hour with the parent or parents and child, during which time families were given verbal and written information on OSA, the role of ncpap therapy, the use and care of the ncpap machine, and how to comfortably and appropriately apply the child s mask. The child s acceptance of the ncpap mask was assessed during the initial session. If the child readily accepted temporary mask placement, a mask fitting was performed, and ncpap therapy commenced immediately. If the child was fearful of or uncooperative with mask placement, no attempt was made to adjust fitting, and, instead, the family was given a practice mask of approximate size for the child to take home, and a progressive introduction to ncpap therapy was used. This involved placement of the mask alone, initially during the day and then gradually at bedtime. When the child was able to fall asleep with the mask in place, then air was introduced, slowly increasing from lower pressures to the prescribed pressure. Incentive contingencies, parent or sibling modeling, and distraction techniques were also used. Formal mask fitting was carried out once the child was comfortable with the practice mask. Children immediately accepting of the ncpap mask had a titration PSG shortly after commencing ncpap. Children not immediately accepting of the mask were started on an interim best-estimate pressure as ordered by the sleep physician based on clinical estimate of requirement and had a titration PSG when they were reported by the parents to be wearing ncpap for at least 2 hours a night. For the purpose of this report, children were considered to be accepting of ncpap therapy the day they had a titration PSG. Compliance Data Collected ncpap machines contained a built-in monitoring card for collection and storage of ncpap compliance data. The compliance card recorded the duration of time when the set pressure was maintained by actual patient use of the machine (time in use), rather than the duration of time the ncpap unit was turned on. Parents returned compliance cards personally at each visit or by mail every few months. The monitoring card provided information on the number of days ncpap was used and the hours of daily use. These values were used to calculate compliance parameters 35 (Table 1). Follow up Technologists phoned parents daily then every 1 to 2 weeks until ncpap was in use and then every 1 to 2 months. Technologists were available for phone or personal consultation during work hours, with home care company support available at night and on weekends. Ongoing concerns were brought to the attention of the pediatric sleep service physicians, who made personal contact with the families to address difficulties. Children considered, by the pediatric sleep physicians, to be having persistent difficulties tolerating ncpap were referred to the pediatric sleep service psychologist for more intensive, ongoing therapy. In particular, the psychologist assisted the parent or parents in implementing the systematic desensitization approach by providing ongoing support to persist with ncpap treatment and by recommending creative ideas for distracting activities and incentives to moderate the child s distress and resistance. PSGs were carried out at regular intervals to monitor ongoing ncpap requirements; typically, every 6 months for children less than 5 years old, every year for children 5 to 40 years old, and every 2 years for children 10 years or older. Difficulties with CPAP treatment, in particular side effects such as abrasions or local irritation, were documented and addressed as they arose. The sleep service reviewed the children at regular intervals as directed by its medical staff. Table 1 Definitions for ncpap Compliance Parameters Parameter Days ncpap used Percentage of days ncpap used Mean daily use (hours) Mean daily use (hours) on days ncpap used Definition Number of days when 1 hour or more of ncpap use was recorded Number of days when 1 hour or more of use was recorded / total number of days with machine Total hours of ncpap use / total number of days with machine Total hours of ncpap use / total number of days when 1 hour or more of use was recorded ncpap refers to nasal continuous positive airway pressure. SLEEP, Vol. 2, No. 5,

3 Table 2 Characteristics of Children Involved in Study Parameter Study Successfully Compliance population used ncpap data available Subjects, no Age, y a.7 ± ± ± 5.1 Boys, % Caucasian, % Single parent family, % Adenotonsillectomy, % Complicating medical disorder, % AHI, no./h b,c SaO 2, % b,c Mean Minimum PetCO 2, mm Hg a,b Maximum 55.8 ± ± ± 5.0 Mean 47.5 ± ± ± 4.0 Full face mask, % ncpap refers to nasal continuous positive airway pressure; AHI, apnea-hypopnea index; SaO 2, oxygen saturation; PetCO 2, end tidal CO 2. a Data are presented as mean ± SD. b Data from diagnostic PSG. c Data are presented as median. Statistical Analysis OSA severity was graded according to AHI as mild, 1 to 5; moderate, 5.1 to 10; and severe, > 10. Because of small numbers, children less than 1 year old were combined with children aged 1 to 5 years for analyses. Ordinal and binary variables were compared using a χ 2 test. Mean and SD of continuous variables were compared using Student 2-tailed t test or 1-way analysis of variance. Nonnormally distributed variables were analyzed as median values and compared using the Mann-Whitney test. Data were managed using Microsoft Excel software and analyses performed using SPSS Version 12.0 (SPSS Inc.). Statistical significance was defined at the 5% level. 36 RESULTS Study Population During the study period, 74 children underwent a diagnostic PSG for a suspected sleep disorder. Three children were eligible for the study but were not included because they required Bi- PAP rather than ncpap to adequately manage their OSA. Seventy-nine children (10%) were identified as eligible for the study and were commenced on ncpap therapy. Characteristics of all children studied are shown in Table 2. At the time of commencing ncpap treatment, 2 (2.5%) children were less than 1 year, 1 (24.1%) children were aged 1 to 5 years, 31 (3.2%) children were aged 6 to 12 years, and 27 (34.2%) children were aged 13 to 18 years. Sixty-six children (84%) had complicating medical disorders (Table 3). Forty-two children (53%) had undergone previous adenotonsillectomy (n = 3) or adenoidectomy only (n = 3). Of the 37 children who had not undergone adenotonsillectomy or adenoidectomy, 32 children (85%) had a complicating medical diagnosis (7, Down syndrome; 5, cerebral palsy; 3, Table 3 Complicating Medical Disorders in 66 Children Identified as Requiring ncpap Treatment Medical disorder No. Down syndrome 22 Developmental delay 15 Unknown 5 Noonan syndrome 2 Fragile X 1 Medulloblastoma 1 William syndrome 1 Dandy-Walker malformation 1 Rett syndrome 1 Rubenstein-Taybi 1 Seizures 1 Autism 1 Obesity a 10 Cerebral palsy 6 Pierre-Robin syndrome 3 Tumor 3 Seizure disorder 1 Neuromuscular disease 2 Tetralogy of Fallot 1 Klippel-Feil syndrome 1 Marfan syndrome 1 Bipolar disorder 1 Severe asthma 1 ncpap refers to nasal continuous positive airway pressure. a One child also has Pierre-robin syndrome obese; 3, Pierre robin syndrome; 1, myelomeningocele; 8, other syndrome; 2, oncology; and 1, cardiac). For the other 5 children, risk of surgery was deemed greater than perceived benefit. Fifteen children received a full face mask (11, Down syndrome; 1, neuromuscular; 1, obese; 1, seizure disorder; and 1, Klippel-Feil syndrome). Fourteen children were referred to the psychologist for assistance, of which 13 children had a complicating medical diagnosis (6, Down syndrome; 1, cerebral palsy; 2, obese; and 4, developmental delay). Acceptance of ncpap Sixty-five children (82%) accepted CPAP over the 46-month study period. Eight of these families (12%) had required psychology support. Reasons for loss of follow-up are shown in Figure 1. Children aged 6 to 12 years were more likely to accept nc- PAP treatment (4%, p =.045) than children aged 13 to 18 years (82%) or children less than 6 years old (67%). There were no other significant differences in characteristics between children who did and did not accept ncpap (Table 2). Mean ncpap pressure required was 6.8 ± 1. cm H 2 O, with ncpap pressure higher in children aged 13 to 18 years compared with children younger than 13 years old (7.7 ± 1. vs 6.3 ± 1.8 cm H 2 O, p =.006). There was no correlation between AHI and CPAP pressure requirement. Of the children who accepted ncpap, 31 children (48%) accepted it immediately. The remaining 34 children took between and 25 days (median, 68.5 days; interquartile range [IQR) days) to accept ncpap. All children without a complicating medical disorder accepted ncpap, with 3% using it immediately. Seventy-nine percent of children with a complicat- SLEEP, Vol. 2, No. 5,

4 Eligible for ncpap therapy (n=7) Accepted ncpap (n=65) Compliance data available Still accepting ncpap (n=7) Declined ncpap (n=6) ncpap ceased by doctor (n=1) Failed to return compliance cards (n=8) Declined ncpap (n=5) Death from medulloblastoma (n=1) Continuing ncpap treatment (n =31) ing medical disorder accepted ncpap, with 50% doing so immediately. Children were more likely to use ncpap immediately if they were boys (5% vs 21%; p =.006), older (11.6 years vs 8.85 years; p =.027) and had a higher maximum PetCO 2 (58±4.8 vs 54.2±4.2 mm Hg; p =.003). There was no difference in time until acceptance of ncpap between children with and without a complicating medical disorder or between children who had and had not previously undergone adenotonsillectomy or adenoidectomy. Children using a full face mask tended to take longer to establish use (median 58 days vs 11 days). Compliance Data ncpap ceased by patient (n=4) ncpap ceased by family (n=2) Change to dental device (n=2) OSA resolved (n=4) Figure 1 Progress of children prescribed nasal continuous positive airway pressure (ncpap) therapy. Seventy-nine children were eligible for ncpap therapy, 65 children (82%) accepted ncpap therapy, and 50 children (77%) provided compliance data. Overall, 11 children (14%) declined to use ncpap, and 7 children are still trying to establish regular ncpap use. OSA refers to obstructive sleep apnea. Of the 65 children who accepted ncpap, compliance data was available on 50 (77%). Reasons for loss of follow-up are shown in Figure 1. There were no significant differences in characteristics between children who did and did not return compliance data (Table 2). Follow-up ranged from 8 to 7 days (median 207 days; IQR days). Compliance data are shown in Figure 2 and Figure 3. Seventy-six percent of children used ncpap for at least half the days. Fifty-two percent of children used ncpap for at least 75% of days. Mean daily use was 4.7 hours (IQR ), and mean daily use on days ncpap used was 6.3 hours (IQR ). Compliance parameters were compared (1) between children with and without impaired cognition, with the presence of cognitive impairment based on coexisting medical diagnosis; (2) across the age groups; (3) by sex; (4) by OSA severity; (5) between children who had and had not undergone previous adenotonsillectomy or adenoidectomy; (6) between children wearing full versus nasal masks; and (7) between children who did and did not receive psychologist support. There was no difference in compliance parameters between children who had normal cognition and children who did not; children with mild, moderate, or severe OSA; children who had and had not undergone previous children of number Figure 2 Distribution of days nasal continuous positive airway pressure (ncpap) used (n = 50). Most children used ncpap at least half of the nights hours Figure 3 Compliance with nasal continuous positive airway pressure (ncpap) (n = 50). Dark bars represent mean daily use, in hours; shaded bars represent mean ncpap daily use on days ncpap used, in hours. adenotonsillectomy or adenoidectomy; and boys compared with girls. Compliance decreased with increasing age, which was statistically significant for percentage of days ncpap used and mean daily use on days ncpap used (Table 4). Compliance tended to be reduced in children wearing full face masks compared with nasal masks for percentage of days used (median 82% vs 52%), mean daily use (median 5.2 hours vs 1.7 hours), and mean daily use on days ncpap used (median 6. hours vs 3.7 hours). Children requiring psychology support achieved compliance rates similar to those of children who were not referred for psychology support (mean daily ncpap use, median and IQR 4.7, vs 4.7, ). Compliance was not influenced by duration to acceptance of ncpap. Thirteen children (20%) took longer than 0 days to use ncpap (median 115 days, IQR days). Ten of these children had impaired cognition (6, Down syndrome; 4, developmental delay). Of the other 3 children, 2 had difficulty tolerating ncpap, but all SLEEP, Vol. 2, No. 5,

5 Table 4 Compliance Parameters According to Age 5 y 6-12 y y p value No Days 4.6 ( ) 74.2 ( ) 57. ( ).023 used, % a Mean daily 7.2 ± ± ± use, h Mean daily 7.7 ± ± ± use on days ncpap used, h ncpap refers to nasal continuous positive airway pressure. a Data are presented as median (interquartile range). b Data are presented as mean ± SD. 3 experienced delay (1 to 4 months) between parental report of ncpap acceptance and date of titration PSG. This was because of either the PSG waiting list or parental preference for study date. Compliance data were available for 7 of these 13 children, with mean daily ncpap use 4.7 hours (IQR ) and mean daily ncpap use on days used 4.8 hours (IQR ). Minor side effects, such as local irritation, were seen in a small percentage of children; however, these were easily addressed with adjustment of the mask interface and were not reported to be responsible for noncompliance with ncpap use. Side effects were considered uncommon because of the close and continued long-term follow-up support provided by the CPAP coordinator. Of the 50 children returning compliance data, 31 (67%) continued to wear ncpap, 8 (16%) ceased ncpap treatment at patient or families request, 6 (12%) ceased ncpap for medical reasons, and 5 (10%) changed hospitals and were therefore lost to follow-up (Figure 1). DISCUSSION This study of children prescribed ncpap shows that 3% of the children accepted ncpap immediately, 66% accepted ncpap within 3 months, and 82% of children accepted ncpap within 46 months, not only confirming previous reports, 18,1,26 but also suggesting that such rates are possible in children with a complicating medical disorder. In addition, this is the first study to analyze objective time-in use measures of ncpap compliance in a large number of children to show ncpap use rates between 4.7 and 6.3 hours a night. The excellent implementation rate and reasonable compliance rate support the use of a progressive behavior modification program in conjunction with ongoing support and close follow-up as a successful protocol for ncpap use in children. In our experience, several factors are integral to successful use of ncpap therapy in children. (1) The presence of a dedicated staff comfortable with managing ncpap in children, as well as a structured program that is intensive, involves education of families and children, and provides ongoing support and followup. Several studies have shown that compliance with ncpap is significantly improved by behavioral techniques, particularly in the pediatric age group, with more-intensive support equating with higher rates of compliance. 25,33-35,37,38 (2) Obtaining a comfortable fit of the mask, which is regularly reassessed. This is an important factor in children, as they have growing facial structures. Improvement in pediatric mask design over recent years has produced more mask options and facilitated improvement in mask fit. For some children, particularly those with Down syndrome, we have found that changing from a nasal to a full face mask results in greater success. A comfortable mask fit increases tolerance of the mask interface and minimizes complications such as pressure sores and midfacial hypoplasia. 3 Routine use of pressure ramping and heated humidification has been found to be important in optimizing satisfaction with, and adherence to, treatment in our patients. (3) Removal of the financial burden of equipment. (4) Finally, but in our practice most importantly, the involvement, resolve, and competency of the parent or parents. This is in agreement with other experience in children. 1,25 Nevertheless, it is challenging to gain a child s acceptance of the nc- PAP mask and, despite all our efforts, it is inevitable that a small number of children will never reach a point of consistent wearing of ncpap. ncpap treatment declined by 24% of subjects during the study period: 6 prior to being established on ncpap and 13 once established on ncpap. Reasons given for not using ncpap included parental factors (feeling overwhelmed, not convinced of need for treatment, discomfort enforcing an undesirable treatment on their child) and patient factors (children intolerant; neurologic condition causing such excess head motion that the mask could not be stabilized in position; and occasionally, child removing the mask during sleep). Side effects were not responsible for failure to wear ncpap. Rates are comparable with those from previous studies. 26,40 Reliable information on ncpap compliance in children is limited, with previous studies relying on report or equipment measures of ncpap use. Although personal and parental report have been shown to overestimate compliance rates, as compared with measured compliance, the majority of previous studies have reported subjective measures of compliance obtained from questionnaires, with very few studies reporting objective measures of compliance. 17,1,20,22-26,28,40-43 The largest series to date describes ncpap use in 4 children with OSA from academic pediatric sleep disorder centers. 1 Adequate compliance was defined as use for more than 50% of prescribed hours based on parental report or equipment hour meter (reflects time machine is on) and varied dramatically between centers from 50% to 100%. The inherent variability in patient selection and follow-up among so many centers, although yielding important general information about ncpap use in children, is likely to result in suboptimal compliance data. Koontz et al showed mean compliance rates between 7.5 and 8.6 hours a night in 20 children undergoing a behavioral intervention program. 24 Children were being treated with Bi- PAP not ncpap, were difficult cases referred by physicians for assistance, and were likely to be motivated to wear ncpap. In addition, compliance was measured using equipment meters. Unfortunately, built-in counters on equipment devices are likely to overestimate actual compliance as measured by pressure-actuated devices, particularly if the machine is allowed to remain on without being attached to the patient. 22,43-45 Brooks et al is the only published study to measure actual time-in-use ncpap compliance rates in children. 22 Using internal piezoelectric transducers to measure compliance, a ncpap compliance rate of 6.0 ± 2.7 hours was reported in children. However, there were only 4 subjects in each group, subjects were teenagers (mean age 13 ± 1.1 years), and results have only been published in abstract form. Our findings, which were obtained from a large number of children and derived from recording only hours when set ncpap pressure was maintained rather than just time the machine was turned on, SLEEP, Vol. 2, No. 5,

6 are therefore likely to provide increased accuracy regarding hours of ncpap use in children, compared with previously published studies. In this study, a difference was shown between mean daily use and mean daily use on days ncpap used, suggesting that, although most children wore ncpap for at least half the days, there were days when ncpap use was minimal. This reflects the reality of ncpap use in children, who typically require time and patience to establish adequate hours of ncpap use and, in the majority of cases, are reliant on adult intervention to apply ncpap. This again suggests the importance of parental involvement and persistence in achieving therapeutic levels of ncpap use in children. Successful use of ncpap, particularly in children, hinges upon 2 important stages: first of all, acceptance and tolerance of the ncpap interface and, secondly, ongoing compliance with ncpap itself. This study looked at these 2 components in detail and investigated some of the factors influencing success. Although median time to acceptance of ncpap was 68.5 days, 80% of children who accepted ncpap did so within 3 months. Only 3 children took longer than 6 months to accept ncpap treatment (duration 23, 27, and 25 days). Two of these children had Down syndrome and a relatively low AHI (2.8 and 7.8), and 1 child had developmental delay and a relatively high AHI (34.0). Although this duration of time may seem clinically unacceptable, in these children, the benefit of persevering with establishment of ncpap therapy was considered preferable to the alternative treatment of tracheostomy, particularly for the children with relatively mild OSA. For the child with an AHI of 34, the absence of right ventricular strain or significant disruption of gas exchange was supportive of the more conservative approach. Had there been any suggestion of cardiac compromise, significant hypoxia or hypercarbia, or worsening clinical condition, then perseverance with establishing ncpap may not have been appropriate. In our study, acceptance of CPAP was greatest in children aged 6 to 12 years and was not influenced by other factors, in particular, sex, coexisting diagnoses, and AHI. Findings are consistent with previous work that showed children aged 1 to 5 years to be least accepting of CPAP and sex and associated diagnosis not playing a role in ncpap acceptance. 26 Compliance with ncpap lessened with increasing age but, perhaps surprisingly, was not influenced by cognitive status, severity of OSA, or previous adenotonsillectomy or adenoidectomy. Use of a full face mask was associated with lower compliance; however, this may reflect the small numbers studied (n = 7) and the type of child given a full face mask, that is, those deemed more likely to have difficulties with tolerance of a nasal mask. Unfortunately, the impact of socioeconomic status and parental education on compliance were not assessed in this study, and the role of the psychologist was unable to be fully investigated because this intervention was not randomized. There is little published information on factors influencing ncpap compliance in children; therefore, further research is needed to clarify the issues identified in this study as well as the hours of ncpap use needed to prevent medical and neurocognitive sequelae of OSA. This study has certain limitations. Success rates may have been altered because titration PSG date was used to define successful establishment of ncpap use. Time until acceptance of ncpap therapy may have been overestimated because of waiting time for a titration PSG after parents reported the child to be successfully wearing ncpap. Alternatively, number of children recorded as successfully establishing ncpap may have been overestimated because readiness for titration PSG was based on parental report. Compliance rates may have been influenced by the subject s or parent s awareness of being monitored and the fact that ncpap machines were free of charge. Type II error may have occurred as a result of relatively small sample size and, therefore, lack of power or from multiple analyses. Because children typically sleep longer than adults and sleep time varies with age of the child, clinical interpretation of compliance based on absolute hours of ncpap use is difficult. Describing ncpap use as a percentage of time asleep would have aided in interpretation and comparison of compliance results across the age groups. This study was focused on the initiation and compliance with a specific treatment and not on treatment effects other than improvement in PSG parameters. Therefore, it was not possible to assess clinical outcomes. This is clearly an area that needs to be investigated and reported in future work. A large proportion of this study population was comprised of children with complicating medical disorders. These children, in particular those with Down syndrome and cerebral palsy, are thought to be more difficult to establish on ncpap and less compliant with therapy. Results from this study showed that 77% of children with cerebral palsy and 72% of children with Down syndrome accepted ncpap, with ncpap worn for a similar time irrespective of medical diagnosis. In fact, many children with a complicating medical disorder wore ncpap for more hours than did children without a complicating medical disorder. These results are encouraging and may well reflect the extent of intervention in these children and the impressive degree of motivation that parents of these children often demonstrate. Higher ncpap pressures were required in the 13- to 18-yearolds compared with the younger children. This is at variance with results of previous studies, which have shown no effect of age on ncpap pressure. 18,1,26 Age and type of population studied may be responsible for the difference in findings, particularly as the mean ncpap pressure was lower in this population compared with those of other studies. In this study, higher maximum PetCO 2 level but not AHI, mean PetCO 2 level, or SaO 2 appeared to be an important predictor of immediate ncpap use. This suggests that symptoms from CO 2 retention may underlie acceptance of ncpap and that maximum PetCO 2 may be a more sensitive indicator of symptomatic CO 2 retention than mean PetCO 2 level. Studies disagree on the role of OSA severity in acceptance of ncpap treatment in adults, 35,45-50 and literature is lacking in children. Further studies in large numbers of children are required to expand on these findings and exclude the possibility of type I and type II errors. Nevertheless, results contribute significantly to the available knowledge on the use of ncpap in children and may aid in allocation of resources and direct focus of follow-up for children commenced on ncpap therapy. In summary, this study shows that, irrespective of associated medical disorders, the vast majority of children are able to be established on ncpap, with most children wearing ncpap for a substantial part of the night. Therefore, with patience, a behavioral modification approach, and parental commitment, children will use ncpap and will be reasonably compliant with treatment. ACKNOWLEDGEMENTS The authors would like to thank the parents and children who took part in this study, Ms. Nancy Quennell (sleep technologist) for her ongoing commitment to commencing these children on SLEEP, Vol. 2, No. 5,

7 ncpap, and Mr. James Bannard (pediatric sleep psychologist) for his dedication in assisting these children and their families in their use of ncpap, as well as Ms. Carolyn Langen for the many hours she spent completing the database for this study. Financial Support: Alberta Aids to Daily Living Provincial Program supplied ncpap machines. REFERENCES 1. O Donnell AR, Bohn SG, Bjornson CL, Kirk VG. Compliance rates in children using nasal continuous positive airway pressure. Am J Respir Crit Care Med 2003;167:A Ali NJ, Pitson DJ, Stradling JR. Snoring, sleep disturbance, and behaviour in 4-5 year olds. Arch Dis Child 13;68: Redline S, Tishler PV, Schluchter M, Aylor J, Clark K, Graham G. Risk factors for sleep-disordered breathing in children. Associations with obesity, race, and respiratory problems. Am J Respir Crit Care Med 1;15: Gozal D. Sleep-disordered breathing and school performance in children. Pediatrics 18;102: Gozal D, O Brien L, Row BW. 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