pii: jc- 00291-15

http://dx.doi.org/10.5664/jcsm.5924

S CI E NT IF IC IN VES TIGATIONS

Pediatric Positive Airway Pressure Adherence in Obstructive Sleep Apnea Enhanced by Family Member Positive Airway Pressure Usage Pooja Puri, MBBS¹; Kristie R. Ross, MD, MS¹; Reena Mehra, MD, MS2; James C. Spilsbury, PhD3; Hong Li, MD, MS4, Carolyn E. Levers-Landis, PhD5; Carol L. Rosen, MD1 ¹Department of Pediatrics, Division of Pulmonology, Allergy and Sleep Medicine, Rainbow Babies and Children’s Hospital - University Hospitals, Case Western Reserve University School of Medicine, Cleveland, OH; ²Sleep Center, Neurologic Institute, Cleveland Clinic, Lerner College of Medicine, Case Western Reserve University School of Medicine, Cleveland, OH; 3Department of Epidemiology and Biostatistics, Case Western Reserve University School of Medicine Cleveland, Cleveland, OH; 4Center for Clinical Investigation, Statistical Science Core, Case Western Reserve University School of Medicine, Cleveland, OH; 5Department of Pediatrics, Division of Behavioral Pediatrics and Psychology, Rainbow Babies and Children’s Hospital - University Hospitals, Case Western Reserve University School of Medicine, Cleveland, OH

Study Objectives: Adherence to positive airway pressure (PAP) therapy for obstructive sleep apnea (OSA) remains a challenge in children. We hypothesized that the presence of another family member on PAP therapy (parent, sibling, other family member) would be associated with better adherence in the child. Methods: We conducted a retrospective chart review to identify children < 18 years of age who had a new diagnosis of OSA between Jan 2011 and May 2013. Outcomes were objective PAP adherence at 1 week, 1 month, and 3 months. Potential predictors included family member on PAP therapy, patient demographics, and clinical characteristics. Group differences between children with and without a family member on PAP therapy were determined using χ2 test and Wilcoxon two-sample test. PAP adherence measures at each time point and patterns of change across time between the two groups were examined using mixed-effects models. Results: The final analytic sample included 56 children: age 13.2 ± 3.7 years, 60% male, 67% African American, 65% obese, and 32% with developmental disabilities. The mean obstructive apnea-hypopnea index was 25.2 ± 28.7, and 19 (33%) had a family member on PAP therapy. Overall PAP adherence was 2.8 ± 2.4 h/night at 3 months. At month 3, the group with a family member on PAP therapy had significantly greater average nightly PAP use on all nights (3.6 ± 0.6 vs. 2.3 ± 0.39) and on nights used (4.8 ± 0.6 vs. 3.8 ± 0.40); (p value = 0.04). Conclusions: Overall PAP adherence was low, but having a family member on PAP therapy as a “role model” was associated with better adherence. Commentary: A commentary on this article appears in this issue on page 941. Keywords: positive airway pressure (PAP), adherence, obstructive sleep apnea, adherence, children Citation: Puri P, Ross KR, Mehra R, Spilsbury JC, Li H, Levers-Landis CE, Rosen CL. Pediatric positive airway pressure adherence in obstructive sleep apnea enhanced by family member positive airway pressure usage. J Clin Sleep Med 2016;12(7):959–963.

I N T RO D U C T I O N

BRIEF SUMMARY

Current Knowledge/Study Rationale: Positive airway pressure (PAP) is long term therapy for children with obstructive sleep apnea (OSA) that persists after adenotonsillectomy or who are not candidates for that surgery, but its efficacy is limited by suboptimal adherence in this age group. The study explored whether having a family member on PAP therapy was associated with better PAP adherence in the child. Study Impact: Having a family member with OSA on PAP therapy was associated with better adherence in the child. We speculate that greater efforts to identify and engage family members on PAP therapy as role models for positive health behaviors may lead to better PAP adherence for children.

Obstructive sleep apnea (OSA) is a serious medical condition characterized by repetitive partial or complete obstruction of the upper airway during sleep.1 Untreated OSA negatively affects daytime behavior, neurocognition, and quality of life.2–5 OSA has been shown to run in families with 50% to 70% higher risk in a subject with first degree relative with OSA.6 While adenotonsillectomy is the first-line treatment in children, positive airway pressure (PAP) is the next line of therapy for children with residual OSA after adenotonsillectomy or for whom surgery is contraindicated.1 PAP is a very effective therapy, but poor adherence to therapy is a limiting factor, with most studies showing a high dropout rates in up to one-third of children and low average nightly use, often less than one-half of their expected sleep period for age.7–9 Usage of the device can be uncomfortable for children, disruptive to the family lifestyle, and requires parental supervision and support. Recent studies have shown better neurobehavioral outcomes in children who are adherent to PAP therapy.10 There are limited data regarding predictors of PAP adherence in pediatric population.

Other than age, no consistent child patient-related factors have been identified for better usage.4 Among family-related factors, higher maternal education,4 better home structure, and communication have been associated with increased usage.11 Identification of factors that operate to enhance PAP adherence in children is key as such knowledge will inform the development of strategies to optimize OSA treatment, thereby translating 959

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into mitigation of OSA-related adverse health outcomes in children. Thus far, no studies have examined whether the presence of family member with PAP therapy who could serve as a potential “role model” would be associated with better PAP adherence in the child. We hypothesized that children with a family member using PAP in the family will predict better PAP adherence.

pediatric sleep nurse and representatives from the durable medical equipment company. Once PAP therapy commenced, phone calls were made by pediatric sleep center staff to support families and trouble-shoot any problems encountered. Face-to-face clinical follow-up with physicians, nurses and/or behavioral specialists was also available. PAP devices (PhilipsRespironics, Murraysville, PA) were equipped with a proprietary algorithm for the collection of adherence data using a data card or modem (SmartCard system or EncoreAnywhere, Philips-Respironics, Murraysville, PA). Adherence data were reviewed through the web-based compliance management system. De-identified patient data from the patient’s medical record and adherence data from web-based management system were abstracted and entered into University Hospitals’ HIPAA-compliant RedCap database for analysis.

METHODS

Sample

The sample was selected retrospectively from the University Hospitals Sleep Center data base of children evaluated at the Pediatric Sleep Center at Rainbow Babies and Children’s Hospital between Jan 2011 and May 2013. Inclusion criteria were children younger than 18 years of age who were started on PAP therapy for OSA with objective PAP adherence data. Patients were excluded if they had previous treatment experience with PAP or other non-OSA indications for PAP therapy (for example, hypoventilation or ventilatory support). Clinical practice for the evaluation and management at this center included laboratory-based polysomnogram (PSG) diagnosis of OSA, prescription of PAP therapy after a laboratory-based titration study, and use of objective monitoring of PAP adherence. PSGs were performed and scored in accordance with the standard American Academy of Sleep Medicine criteria for pediatric studies,12 and OSA was defined using standard pediatric definitions.13 The study was approved by University Hospitals Case Medical Center Institutional Review Board.

Statistical Methods

Patient demographics and sleep parameters were summarized using means ± standard deviations for continuous variables and frequency/percentage for categorical variables. Potential differences between groups with no family member PAP use and family member PAP use were determined using χ2 and Wilcoxon two-sample test. PAP adherence parameters at each time point and pattern of change across week 1, month 1, and month 3 by family member with PAP use were examined using mixed-effects linear models. An interaction between time and group (no family member PAP use vs. family member PAP use) was included in the model to understand changes over time in adherence between the 2 groups, and differences at month 3 were compared between 2 groups using same method. In addition, associations of initial adherence (week 1) with selected factors including family member using PAP, child age, race, gender, BMI percentile, OAHI, and education level of caretaker were examined using linear regression analysis. Associations of adherence change from initial use to 3-month use with selected factors were similarly examined using linear regression models. All p values were two-sided; p < 0.05 was considered as statistical significance. All analyses were performed using Statistical Analysis System version 9.3.

Measures

The primary outcome was adherence to PAP therapy derived from stored electronic data derived from the PAP machines. Adherence data were obtained for 3 time points: 1 week, 1 month, and 3 months after initiation of PAP therapy. Adherence to PAP was assessed in 4 domains: percentage of nights used, percentage of nights used for > 4 h, average nightly hours of use for all nights, and average nightly hours of usage for the nights used. The independent predictor variable was presence of a family member who had PAP therapy for OSA, defined by caregiver report. Objective adherence data on PAP usage was not obtained for this family member. Patient characteristics and baseline PSG results were abstracted from the medical records: age, gender, race, body mass index (BMI), parentreported comorbidities, caretaker education, insurance status, and obstructive apnea-hypopnea index (OAHI). BMI was calculated and converted to an age and gender adjusted BMI z-score based on the Centers for Disease Control and Prevention growth chart. Daytime sleepiness was assessed using a pediatric modification of the Epworth Sleepiness Scale score.14

R ES U LT S

Study Group

Of the 74 patients who met the inclusion criteria for this study, 6 children never received a PAP device: 3 families actively refused PAP therapy and 3 did not return to receive their PAP prescription. Of the 68 patients who received the PAP device, 12 children were “passive refusers” of therapy, that is, they never used the device at home, so no adherence data were recorded. Thus, the final analytic sample consisted of 56 children with PAP devices who had adherence data at the 3 specified time points. Comparing the children included in the analytic sample who accepted PAP therapy with those who refused therapy, there were no significant group differences for demographics or other clinical characteristics. The percentage of children with a family member with PAP therapy was higher (34%) in the analytic sample compared with the

PAP Clinical Practice

Prior to the PAP titration, standard approaches were utilized including: education about OSA and PAP therapy by the medical team; PAP education, mask desensitization as needed, and mask fitting by a sleep technologist with pediatric experience; and additional family training about PAP therapy by the both Journal of Clinical Sleep Medicine, Vol. 12, No. 7, 2016

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group excluded from analysis (16%), but this difference was not statistically different (p = 0.24).

not willing to initiate PAP therapy. Family members with PAP experience may be better equipped with the knowledge to troubleshoot PAP adherence issues based upon their own personal experience and also may have a better understanding of the consequences of OSA and importance of treatment. Since OSA runs in families, we speculate that a more “family-centered” approach to PAP therapy that includes identifying: (1) family members using PAP therapy; (2) family members with prescribed PAP therapy, but who may not be regularly using the therapy, or (3) other family members with OSA symptoms, but who are undiagnosed and encouraging them to be evaluated may be a promising strategy to improve OSA treatment and PAP adherence for the whole family. Consistent with our observations of the potential impact of family

Patient Characteristics

The detailed patient characteristics of the final analytic sample are shown in Table 1. In brief, the age range was 13.3 (10.3–16.3), 60% were male, 65% were obese, 32% had developmental disabilities, and 19 (33%) reported a family member on PAP therapy. There were no statistically significant group differences in the clinical or demographic characteristics of the 19 children who had a family member on PAP therapy compared with the 37 children who did not have a family member on PAP therapy (see Table S1 in the supplemental material).

PAP Adherence

For all children, PAP use was highest at week 1 (used 79% percent of nights), then declined over time: 65% of nights at month 1 and 57% of nights at 3 months, with wide individual variation. Average nightly PAP use also declined over time: 3.5 ± 2.7 h at week 1, 2.9 ± 2.4 h at month 1, and 2.8 ± 2.4 h at month 3. Figure 1 shows the PAP adherence by group status (family member status with PAP vs no family member with PAP therapy) for each of 4 domains of use. Results of the mixed-effects modeling showed that by month 3, having a family member with PAP therapy was associated with higher average nightly hours of PAP use for all nights (p = 0.04) and for nights used (p = 0.04). The month 3 measurements and statistical differences are summarized in Table 2. Adherence was not related to gender, obesity status, or OSA severity. Older age was associated with lower adherence at week 1, but that effect was no longer evident on longer term use.

Table 1—Participant characteristics (n = 56). 13.2 ± 3.7 (1.6–18.0) 34 (60)

Age, y, mean ± SD, (range) Male, n (%) Race, n (%) African American Caucasian Other

38 (67) 16 (28) 2 (4)

Hispanic ethnicity, n (%)

D I SCUS S I O N To our knowledge, this is the first study to show that presence of a family member with PAP was associated with better PAP adherence by the child. This positive effect on PAP adherence was associated with the child’s nightly hours of PAP use rather than the percentage of nights that the PAP device was used. This family member may function as a “role model” for positive health behaviors and help support the child’s use of PAP therapy. Having a family member with experience with PAP therapy may influence the willingness to initiate PAP therapy as demonstrated by the higher proportion of family members using PAP in this study sample compared to those who were

1 (1)

BMI Z-score Obese, n (%)

1.8 ± 1.4 37 (65)

Comorbid conditions, n (%) a Genetic syndrome b Central nervous system abnormality Craniofacial syndrome Neurodevelopmental disability Attention deficit hyperactivity disorder Asthma Allergic rhinitis

14 (25) 2 (3) 3 (5) 18 (32) 19 (33) 22 (39) 25 (44)

Prior adenotonsillectomy

33 (57)

Caretaker education c At least high school

44 (84)

Medicaid insurance Epworth Sleepiness Scale score

42 (79) d

10.7 ± 4.4

Obstructive apnea-hypopnea index, median (IQR) 12.8 (7.9, 24.0) Split night polysomnography for PAP titration

13 (23)

Family member using PAP, n (%)

19 (33)

Data shown as mean ± SD or n (%) unless otherwise specified. a Some patients had more than one diagnosis. b Trisomy 21 (10), Prader-Willi (1) Crouzon (1), Noonan (1). c Caretaker education status unknown in 6 patients. d Pediatric modification.14

Table 2—Adherence at month 3 by family member group status. PAP Adherence at Month 3 Hours of use, all nights Hours of use, nights used Percentage of nights, any use Percentage of nights used > 4 h

Family Member on PAP Therapy in Household 3.6 ± 0.6 4.8 ± 0.6 58.1 ± 6.2 35.8 ± 7.1

No Family Member on PAP Therapy in Household 2.3 ± 0.39 3.8 ± 0.4 54.7 ± 4.4 23.3 ± 5.1

p value (group) 0.04 0.18 0.66 0.11

p value (group × time) 0.04 0.04 0.57 0.13

Data expressed as least square mean ± standard error.

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Figure 1—PAP adherence over time by group status.

(A) Average hours per night on all nights. (B) Average hours per night on nights used. (C) Percentage of nights used. (D) Percentage of nights used more than 4 hours. PAP therapy use was significantly greater at month 3 for hours of nightly use (all nights and nights used, panels A and B, p = 0.04) in the group of children who had a family member with PAP therapy.

member PAP usage on children’s PAP adherence, previous studies have demonstrated that parental behaviors can influence children’s eating behaviors15 and physical activity.16 It is possible that parent training to better support positive health behaviors in their children may be an important measure to improve PAP adherence, especially given that overall adherence was sub-optimal to begin with and worsened over time. Our study sample was typical of a pediatric PAP population: a heterogeneous group of patients with medical comorbidities except we had higher percentage of children with developmental disabilities than shown in recent studies in children with OSA. The proportion of obese and African American children in this study reflects our urban clinical population and the findings are generalizable as such. Similar to other studies, adherence was not related to severity of OSA or race. Although the overall PAP adherence in our sample was very low, it was similar to other pediatric centers.7–9 The observed high risk for poor adherence to PAP therapy confirms why the American Academy of Pediatrics clinical practice guideline for Journal of Clinical Sleep Medicine, Vol. 12, No. 7, 2016

evaluation and management of OSA in children does not recommend PAP therapy as a reliable first-line therapy for OSA in children when adenotonsillectomy is an option.1 In addition, we also observed that adherence for the whole group declined with time. This suggests that the factors which motivate initiation of PAP are different from those which help maintain PAP use. Our study findings have several limitations that makes them less generalizable. First, this was a retrospective chart review at a tertiary care center with a relatively small sample size that included more medically complex children. Second, the family member PAP status was based on caregiver report, and we do not have data about whether the family member was actually adherent with their PAP therapy or whether or not the child was aware of their use or observed their use. It may be important to assess whether objective adherence data from the family member correlates with better PAP use in children. We also do not have data as to how positively or negatively the family member spoke about their own PAP therapy with the child (e.g., 962

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how comfortable it is, if they find it helps them to feel more alert during the day). We did not have the sample size to evaluate whether the positive influence of having a family member on PAP therapy was different depending upon who that family member was in relationship to the child (e.g., mother, father, sibling, or grandparent). Finally, it is possible that efforts to identify a family member who is prescribed PAP therapy, but who does not use it could become a “negative” role model for the child’s PAP adherence. Nevertheless, we do think the results are hypothesis generating and should be explored further. If these relationships are confirmed at other sleep centers, then a family-centered approach to screen and treat other family members of pediatric PAP patients may be warranted, benefitting the child as well as the other family members.

10. Marcus CL, Brooks LJ, Draper KA, et al. Diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics 2012;130:e714–55. 11. Prashad PS, Marcus CL, Maggs J, et al. Investigating reasons for CPAP adherence in adolescents: a qualitative approach. J Clin Sleep Med 2013;9:1303–13. 12. Iber C, Ancoli-Israel S, Chesson A, Quan S; for the American Academy of Sleep Medicine. The AASM manual for scoring of sleep and associated events: rules, terminology and technical specifications, 1st ed. Westchester, IL: American Academy of Sleep Medicine, 2007. 13. American Academy of Sleep Medicine. International classification of sleep disorders, 3rd ed. Darien, IL: American Academy of Sleep Medicine, 2014. 14. Melendres MC, Lutz JM, Rubin ED, Marcus CL. Daytime sleepiness and hyperactivity in children with suspected sleep-disordered breathing. Pediatrics 2004;114:768–75. 15. Brown R, Ogden J. Children’s eating attitudes and behaviour: a study of the modelling and control theories of parental influence. Health Educ Res 2004;19:261–71. 16. Erkelenz N, Kobel S, Kettner S, Drenowatz C, Steinacker JM, Research Group “Join the Healthy Boat - Primary S. Parental activity as influence on children`s BMI percentiles and physical activity. J Sports Sci Med 2014;13:645–50.

A B B R E V I AT I O N S BMI, body mass index OAHI, obstructive apnea-hypopnea index OSA, obstructive sleep apnea PAP, positive airway pressure PSG, polysomnogram

ACK N O W L E D G M E N T S The authors gratefully acknowledge the assistance of Sheree A. Hemphill, Informatics Manager at University Hospitals of Cleveland, Dahms’ Clinical Research Unit, for helping to develop the data abstraction and management tools in the Redcap software.

SUBM I SSI O N & CO R R ESPO NDENCE I NFO R M ATI O N

R E FE R E N CES

Submitted for publication July, 2015 Submitted in final revised form February, 2016 Accepted for publication March, 2016 Address correspondence to: Carol L. Rosen, MD, 11100 Euclid Avenue, RBC 793, Cleveland, OH 44106; Tel: (216) 844-3267; Fax: (216) 844-5916; Email: carol. [email protected]

1. Marcus CL, Brooks LJ, Draper KA, et al. Diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics 2012;130:576–84. 2. Beebe DW, Wells CT, Jeffries J, Chini B, Kalra M, Amin R. Neuropsychological effects of pediatric obstructive sleep apnea. J Int Neuropsychol Soc 2004;10:962–75. 3. Bourke RS, Anderson V, Yang JS, et al. Neurobehavioral function is impaired in children with all severities of sleep disordered breathing. Sleep Med 2011;12:222–9. 4. DiFeo N, Meltzer LJ, Beck SE, et al. Predictors of positive airway pressure therapy adherence in children: a prospective study. J Clin Sleep Med 2012;8:279–86. 5. Marcus CL, Radcliffe J, Konstantinopoulou S, et al. Effects of positive airway pressure therapy on neurobehavioral outcomes in children with obstructive sleep apnea. Am J Respir Crit Care Med 2012;185:998–1003. 6. Redline S, Tishler PV, Tosteson TD, et al. The familial aggregation of obstructive sleep apnea. Am J Respir Crit Care Med 1995;151:682–7. 7. Marcus CL, Rosen G, Ward SL, et al. Adherence to and effectiveness of positive airway pressure therapy in children with obstructive sleep apnea. Pediatrics 2006;117:e442–51. 8. Nixon GM, Mihai R, Verginis N, Davey MJ. Patterns of continuous positive airway pressure adherence during the first 3 months of treatment in children. J Pediatr 2011;159:802–7. 9. O’Donnell AR, Bjornson CL, Bohn SG, Kirk VG. Compliance rates in children using noninvasive continuous positive airway pressure. Sleep 2006;29:651–8.

D I SCLO S U R E S TAT E M E N T This was not an industry supported study. This work was supported in part by the Fellowship Research Award in Pediatrics (FRAP) grant, an internal grant at Rainbow Babies and Children’s Hospitals at University Hospitals. Dr. Rosen has received honoraria from the American Academy of Sleep Medicine and the American College of Chest Physician for educational speaking. She has consulting agreements with Jazz Pharmaceuticals and Advance Medical and grant/research support with Jazz Pharmaceuticals. The article submitted is not related to these relationships. Dr. Mehra reports that she has received NIH funding for which she has served as Principal Investigator (NHLBI RO1 1 R01 HL 109493, R21 HL108226). Her institution has received positive airway pressure machines and equipment from Philips Respironics and Resmed for use in NIH-funded research. She has received honorarium from the American Academy of Sleep Medicine for speaking. She serves as the Associate Editor for the journal CHEST. She has received royalties from Up to Date.The article submitted in not related to these relationships. None of the other authors have financial disclosures or conflicts of interest. The work was performed at University Hospitals-Case Medical Center, Cleveland, OH. There was no investigational or off label use device use.

963

Journal of Clinical Sleep Medicine, Vol. 12, No. 7, 2016

Pediatric Positive Airway Pressure Adherence in Obstructive Sleep Apnea Enhanced by Family Member Positive Airway Pressure Usage.

Adherence to positive airway pressure (PAP) therapy for obstructive sleep apnea (OSA) remains a challenge in children. We hypothesized that the presen...
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