Elevated Morbidity and Health Care Use in Children withObstructive Sleep Apnea SyndromeAriel Tarasiuk, Sari Greenberg-Dotan, Tzahit Simon-Tuval, Bruria Freidman, Aviv D. Goldbart, Asher Tal, andHaim Reuveni

Sleep-Wake Disorders Unit, and Department of Pediatrics B, Faculty of Health Sciences, Soroka Medical Center, and Departments of Physiology,Epidemiology, and Health Systems Management, Faculty of Health Sciences, and Department of Economics, Faculty of Humanities andSocial Sciences, Ben-Gurion University of the Negev, Beer-Sheva, Israel

Rationale: Health care use, a reliable measure of morbidity, is notice-ably higher 1 yr before obstructive sleep apnea syndrome (OSAS)diagnosis in preschool children. It is not clear at what age OSAS-related morbidity becomes expressed.Objective: To explore morbidity and health care use among childrenwith OSAS starting from first year of life.Methods: Case-control study, starting from the first year of life todate of OSAS diagnosis, among 156 patients (age range, 3–5 yr)and their pair-matched healthy control subjects, by age, sex, pri-mary care physician, and geographic location.Measurements: Patients with OSAS underwent nocturnal polysom-nography studies. Medical records during hospital visits were re-viewed for diagnosis. Variables of health care use were obtainedfrom computerized databases of Clalit Health Care Services, thelargest health maintenance organization in Israel.Main Results: From the first year of life to date of OSAS diagnosis,children with OSAS had 40% more (p � 0.048) hospital visits, 20%more repeated (two or more) visits (p � 0.0001), and higher con-sumption of antiinfective and respiratory system drugs (p �

0.0001). Referrals of children with OSAS to otolaryngology surgeonsand pediatric pulmonologists were higher from Year 1 (p � 0.0001)to date of OSAS diagnosis, especially in Year 4 (odds ratio, 9.4; 95%confidence interval, 4.2–21.1). The 215% elevation (p � 0.0001)in health care use of the OSAS group was due mainly to higheroccurrence of respiratory tract morbidity (p � 0.0001).Conclusions: Practitioners should be aware that starting in Year 1until date of diagnosis, children with OSAS have higher health careuse, mostly related to respiratory diseases.

Keywords: children; respiratory tract morbidity; heath care use; obstruc-tive sleep apnea syndrome

Childhood obstructive sleep apnea syndrome (OSAS) is abreathing disorder during sleep characterized by prolongedpartial upper airway obstruction and/or intermittent completeobstruction that disrupts normal ventilation during sleep andnormal sleep patterns (1). OSAS has an estimated prevalenceof 1 to 2% among young children (2, 3) and can lead to morbidity(4–7).

Adult patients with untreated OSAS have higher health careuse than matched control subjects many years before diagnosis(8–11). Health care use is a powerful index for morbidity inboth adults (11, 12) and children with OSAS (13, 14). We havepreviously shown that children with OSAS are heavy consumersof health care resources 1 yr before diagnosis (14). However,it is not clear at what age OSAS-related morbidity becomes

(Received in original form April 30, 2006; accepted in final form October 11, 2006 )

Correspondence and requests for reprints should be addressed to Ariel Tarasiuk,Ph.D., Department of Physiology, Faculty of Health Sciences, P.O. Box 653, Ben-Gurion University of the Negev, Beer-Sheva, Israel 84105. E-mail: tarasiuk@bgu.ac.il

Am J Respir Crit Care Med Vol 175. pp 55–61, 2007Originally Published in Press as DOI: 10.1164/rccm.200604-577OC on October 12, 2006Internet address: www.atsjournals.org

AT A GLANCE COMMENTARY

Scientific Knowledge on the Subject

Little is known regarding health care consumption andmorbidity among children with obstructive sleep apnea syn-drome (OSAS) from the first year of life.

What This Study Adds to the Field

Children with OSAS have higher morbidity and health careuse starting from the first year of life. This study suggeststhe need for early diagnosis and intervention.

expressed. Therefore, we explored morbidities such as respira-tory tract diseases (13), as well as health care use (hospital visits,consultations, and drugs) and costs, for each year of life startingfrom time of diagnosis back to the first year of life.

We hypothesized that morbidity in children with OSAS be-gins at an early age, resulting in higher health care use yearsbefore diagnosis. Therefore, we analyzed heath care use amongpatients with OSAS and pair-matched control subjects, fromtime of diagnosis back to the first year of life. In addition, weexplored the morbidity that led to increased health careconsumption.

METHODS

Study Design

The study design was that of a case-control study. Patients were diag-nosed as having OSAS after laboratory-proven polysomnographic(PSG) evaluation and were recruited according to their date of birth.Healthy control subjects were not assessed by PSG.

The Institutional Ethics Committee of the Soroka University Medi-cal Center (Beer-Sheva, Israel) approved this study.

Setting

The study took place at the Sleep-Wake Disorders Center at SorokaMedical Center (affiliated with Ben-Gurion University of the Negev).Children had been residents of the region for at least 4 yr before thePSG study.

Subjects

All children (a total of 161 children, ages 3.11 to 5.09 yr), enrollees ofClalit Health Care Services (CHS), the largest health maintenanceorganization in the region, with clinically suspected OSAS were re-cruited consecutively during the period of January 2001 through Decem-ber 2004. In our setting, these children are first referred by their familyphysician to otolaryngology (ear, nose, and throat [ENT]) surgeons orpulmonologists (OSAS-related consultants), who then refer the patientsfor a PSG study. The control group, selected randomly, was matched

56 AMERICAN JOURNAL OF RESPIRATORY AND CRITICAL CARE MEDICINE VOL 175 2007

1:1 to the OSAS group by age, sex, and primary care physician to adjustfor socioeconomic factors and differences in physician daily practice.We explored “typical” patients with OSAS (14) by excluding childrenwho exhibited extreme consumption of health care services, that is,more than 10 times the mean values of their group. Control subjectswere otherwise healthy (i.e., were not included in the databases ofseverely ill children, based on a review of medical records during hospi-tal visits and supplied drugs in the community clinics). It is possiblethat 1–2% of the control group might have had undiagnosed OSAS(14). Five children with OSAS and one control subject who had knowncomorbidity were excluded from the original participants. The finaldatabase included 156 otherwise healthy children with OSAS and theirpaired control subjects.

Data resources for the patients with OSAS included PSG data (15),sleep questionnaires, and hospital records. Diagnoses were classified(International Classification of Diseases, 9th revision) into three catego-ries (13): upper respiratory diseases, lower respiratory diseases, andother. We were not permitted to contact either group to obtain addi-tional information because of patient confidentiality (12–14). Data onhealth care use for both groups were obtained from the CHS EconomicsDepartment.

Health care use and costs were analyzed by going back from thedate of OSAS diagnosis to the child’s day of birth. Total annual cost(12–14) includes the following: number and duration of admissions,number of emergency department visits, and number of new and re-peated visits for consultations. Number and type of prescriptions sup-plied were described according to anatomic and therapeutic criteria ofthe World Health Organization (16). Cost of the PSG study was notincluded.

Data Analysis

Health care use was analyzed in two modes: for the 12-mo period beforePSG diagnosis (n � 156) (12–14) and in full years from the first to thefourth year of life (Years 1–4). The latter was defined according to theinformation available in the CHS computerized databases, starting inJanuary 1998.

A case-control data analysis (17) was performed with SPSS (version12.0; SPSS, Chicago, IL) software. Cost elements were performed aspreviously described (12–14). Data were presented as means � SD forall PSG parameters and as means � SEM and median and range forcosts. The null hypothesis was rejected at the 5% level.

RESULTS

Group Characteristics

One hundred and fifty-six typical children with OSAS with amean age of 4.02 � 0.7 yr (95 males and 61 females) at the timeof PSG diagnosis were included in the study. The control groupincluded 156 children matched 1:1 by age, sex, geographic loca-tion, and pediatrician or family physician. Among patients withOSAS, 43 (29.9%) of the parents reported smoking at least fivecigarettes/d; of those, 32 (78%) were fathers.

The OSAS group (Table 1) had an average apnea–hypopneaindex (AHI) of 8.1 � 7.8 events/h. Relative to the average AHI,REM AHI was higher by 10 events/h (p � 0.0001), that is,compatible with OSAS of moderate severity (18) (i.e., in oursettings we define mild, moderate, and severe OSAS as havingan AHI of � 1 to � 4.9, 5 to � 9.9, and � 10 events/h, respec-tively). AHI severity was comparable in boys and girls. As agroup, children with OSAS did not present evidence of consider-able nocturnal hypoxemia, that is, as a group, mean T90 � 2.1 �4.6%, and total desaturation index was 3.4 � 4.9 events/h (Table 1).After PSG, 103 children (66%) underwent surgery.

Health Care Use 1 Year before PSG Diagnosis

Analysis of health care use was performed per complete 12-moperiod to minimize seasonal effects. Annual health care useamong the OSAS group was $275 � 54 compared with $128 �22 in the control group—that is, 215% higher compared with

TABLE 1. RESPIRATORY AND SLEEP PARAMETERS INCHILDREN WITH OBSTRUCTIVE SLEEP APNEA SYNDROME

Total(n � 156)

Age, yrMean � SD 4.02 � 0.7Range 3.11 to 5.09

Sex, M/F 95/61AHI (events/h), mean � SD 8.1 � 7.8REM AHI (events/h), mean � SD 18.1 � 18.2T90 (%), mean � SD 2.1 � 4.6Total DI (events/h), mean � SD 3.4 � 4.9Non-REM DI (events/h), mean � SD 2.2 � 3.1REM DI (events/h), mean � SD 7.0 � 12.2*Sleep efficiency (%), mean � SD 85.8 � 8.7TST (min), mean � SD 416.5 � 33.7

Definition of abbreviations: AHI � apnea–hypopnea index; DI � desaturationindex; T90 � percent sleep time with saturation below 90%; TST � total sleeptime.

* p � 0.0001, comparing REM with non-REM DI by paired t-test.

the control group (p � 0.0001, Wilcoxon test). Cost elementsper patient per year before the PSG study are summarized inTable 2. Patients with OSAS had an average of 0.14 new admis-sions per year compared with 0.07 new admissions in the controlgroup (p � 0.8). Children with OSAS had a similar number ofvisits to the emergency room compared with the control group,0.4 versus 0.3 visits (p � 0.2), respectively. In our CHS region,when ENT consultation is required, more than 85% of childrenare referred to the 10 ENT surgeons rotating among 11 clinics.Patients with OSAS and control subjects were referred to thesesurgeons in equal proportions (p � 0.6). Children with OSAShad more consultations (ENT consultation before PSG referralwas not included in the analysis) compared with the controlgroup, 1.2 versus 0.4 visits, respectively (p � 0.0001). The OSASgroup needed more recurrent consultations (at least two visits)than the control group, 29.6 versus 7.7% (p � 0.0001, McNemartest). Common consultations (specialist referrals) in the OSASgroup included ENT surgeons for 58 children (40%), pediatricpulmonologists for 16 children (10.3%), and ophthalmologistsfor 22 children (14.1%). Other consultations included neurolo-gists, cardiologists, dermatologists, and orthopedic surgeons (lessthan 10% for each specialty). The control group had significantlylower visit rates to specialists: 20 children (12.8%) to ENT sur-geons and 7 children (4.5%) to ophthalmologists; all other spe-cialists had 1–5% referrals per specialty.

Costs for drugs for patients with OSAS were six times higher(p � 0.0001, Wilcoxon test) than for the control group (Table2). The prevalence and number of supplied drugs 1 yr beforePSG diagnosis are summarized in Table 3. More subjects withOSAS (odds were up to 6.1 times higher) were supplied withthe medications presented in Table 3, which includes only thepharmacologic groups in which differences were found. The av-erage cost of drugs supplied per patient per year to patientswith OSAS was up to 140% more than to the control subjects.Specifically, more drugs in the general antiinfectives (J) categoryand respiratory system (R) category were supplied (p � 0.0001)to patients with OSAS, that is, 236 and 275%, respectively.Ninety percent of supplied system N group drugs were acetamin-ophen. Ninety-nine percent of the musculoskeletal drugs wereibuprofen. These drugs can also be purchased over the counter.

When the OSAS group is arbitrarily divided by cost, theupper 25% (n � 39) of patients, defined as the most costly, hada mean consumption per person per year of $864 � 187, whichwas significantly higher than the lower 75% of patients ($79 � 7;

Tarasiuk, Greenberg-Dotan, Simon-Tuval, et al.: Health Care Use in Children with OSAS 57

TABLE 2. COST ELEMENTS FOR ALL SUBJECTS 1 YEAR BEFORE DIAGNOSIS

Parameter Control (n � 156) OSAS (n � 156) p Value

Hospitalization 58.3 � 18.9 (0.0–1864) 89.3 � 33 (0.0–4475) 0.79Emergency room 36.0 � 5.9 (0.0–359) 48.2 � 7.2 (0.0–478) 0.22Consultations 18.8 � 3.2 (0.0–275) 44.9 � 6.8 (39.0–604) � 0.0001Drugs 15.4 � 1.9 (8.0–154) 91.7 � 38.5 (22.0–1380) � 0.0001Total annual costs 128 � 22 (17.0–1919) 275 � 54 (106.0–4718) � 0.0001

Definition of abbreviations: ENT � ear, nose, and throat; OSAS � obstructive sleep apnea syndrome; PSG � polysomnographic.Values represent costs of the services per patient per year in U.S. dollars (4.5 new Israeli sheqalim equal to $1.00). Values are

presented as means � SEM (median, range).Cost for OSAS consultations did not include the last ENT surgeon visits before the PSG study. Statistical significance was

determined by Wilcoxon signed rank test.

p � 0.0001, Mann-Whitney test). These upper 25% of costlypatients consumed 78.4% of all annual OSAS group costs, thatis, 11-fold more health care resources than the lower 75% ofpatients. The most costly subgroup had similar age, sex distribu-tion, AHI, and sleep characteristics compared with the lower75% of patients. For example, the AHI was 8.2 � 6.9 versus8.1 � 8.2 events/h, respectively.

AHI correlated (Spearman correlation) only with cost formedical consultations (r � 0.16, p � 0.05) and J category drugs(r � 0.19, p � 0.02). Total annual costs did not correlate withAHI. Children with AHI � 5 events/h, versus AHI � 5 events/h,were supplied with 10% more drugs from group J (p � 0.0001),had 22% more repeated (two or more) consultations (p �0.0001), and two times more hospital visits (p � 0.0001). Similarresults regarding J group drugs and consultations were obtainedin children with � 2 events/h versus AHI � 2. The percentageof time with SaO2 � 90% in our children with OSAS did notcorrelate with any of the health care use indices. Children ex-posed to passive smoking showed similar health care use com-pared with children who were not exposed to passive smoke($209 � 58 and $310 � 79, respectively; p � 0.54, Mann-Whitneytest).

Costs and Health Care Use during Years 1–4 (Inclusive)

During the fourth year of life, 44 children underwent adenoton-sillectomy and did not complete the 12-mo period; they wereexcluded from that year’s data. Compared with the remaining112 (72%) children with OSAS, the 44 children (28%) who wereexcluded from data analysis because of adenotonsillectomy areyounger (3.4 � 0.3 vs. 4.3 � 0.7 yr, p � 0.0001) and have moresevere OSAS (AHI � 10.7 � 8.3 vs. 7.1 � 7.4 events/h, p �

TABLE 3. PREVALENCE OF SUPPLIED DRUGS AND MEAN NUMBER OF PRESCRIPTIONS 1 YEARBEFORE POLYSOMNOGRAPHIC DIAGNOSIS

OSAS Group MeanControl OSAS Number of Supplied Difference

Pharmacologic Group [% (n )] [% (n )] OR (CI) Drugs (95% CI)

A: Alimentary tract and metabolism 20.5 (32) 36.5 (57) 2.2 (1.3–3.7) 0.64 � 0.1 (0.0–8) 0.32 (0.1–0.6)*D: Dermatologic 34.0 (53) 48.1 (75) 1.8 (1.1–2.8) 0.94 � 0.15 (0.0–10) 0.31 (0.02–0.6)*H: Systemic hormonal 10.3 (16) 21.8 (34) 2.4 (1.3–4.6) 0.46 � 0.09 (0.0–6) 0.29 (0.1–0.5)*J: General antiinfectives 59.0 (92) 89.1 (139) 5.7 (3.1–0.3) 3.64 � 0.25 (3.0–17) 2.1 (1.6–2.7)†

M: Musculoskeletal 6.4 (10) 29.5 (46) 6.1 (2.9–12.6) 0.51 � 0.07 (0.0–4) 0.44 (0.3–0.6)†

N: Nervous system 41.7 (65) 55.1 (86) 1.7 (1.1–2.7) 1.55 � 0.19 (1.0–15) 0.7 (0.3–1.13)*R: Respiratory system 47.4 (74) 81.4 (127) 4.9 (2.9–8.09) 4.40 � 0.42 (3.0–31) 2.8 (1.9–3.7)†

Definition of abbreviations: CI � confidence interval; OR � odds ratio; OSAS � obstructive sleep apnea syndrome.Supplied medications are presented as the mean � SEM (median, range) number of drugs per patient per year. Difference �

mean difference (95% CI) between cases and control subjects in number of times per year the drug was given.* p � 0.05 by Wilcoxon signed rank test.† p � 0.0001 by Wilcoxon signed rank test.

0.0001). No significant differences were found regarding totalannual cost, number of consultations per child per year, andpercentage of children requiring two or more consultations ($258� 146, 1.2 � 0.18, and 48% vs. $242 � 49, 1.1 � 0.15, and 42%,respectively) and sex distribution.

In comparison with the control group, total annual cost (Fig-ure 1) was 160 to 190% higher (p � 0.0001, two-way analysisof variance [ANOVA]) for each year of life in the OSAS group.For each 1-yr increase in age, total annual cost decreased by35% in the OSAS group and by 50% in the control group (p �0.0001, two-way ANOVA). The total number of hospital visitsthroughout Years 1–4 was 40% higher in the OSAS group com-pared with the control subjects (3.2 � 3.5 visits per child vs. 2.3 �2.6 visits per child, respectively; p � 0.048). Patients with OSASrequired more (about 20%) repeated (two or more) hospitalvisits compared with control subjects (p � 0.0001, McNemartest). Compared with control subjects, the number of OSAS-related consultations (Table 4) was in the range of 50 to 600%higher (p � 0.0001). The rate of referral of patients with OSASto general consultants (not including ENT or pediatric pulmonol-ogists) was in the range of 30 to 50% higher (p � 0.0001), exceptfor Year 1. Odds for referral of patients with OSAS to OSAS-related consultants (not including the last ENT surgeon visitbefore PSG study) is especially higher in Year 4 only: oddsratio, 9.4; 95% confidence interval, 4.2–21.2. Patients with OSASrequired two to four times more (p � 0.0001, McNemar test)repeated (two or more) consultations with specialists.

The annual cost of drugs for patients with OSAS was 70 to200% higher through the first 4 yr of life (p � 0.0001). Signifi-cantly more (p � 0.0001, two-way ANOVA) patients with OSASwere supplied with J and R categories of drugs, beginning in

58 AMERICAN JOURNAL OF RESPIRATORY AND CRITICAL CARE MEDICINE VOL 175 2007

Figure 1. Total annual costs from first year of life, including Year 4.Values were obtained for 156 children with obstructive sleep apneasyndrome (OSAS) and pair-matched control subjects (n � 112 for Year4). Columns represent average annual costs (� SEM) per subject peryear in U.S. dollars. #p � 0.0001, two-way analysis of variance.

Year 2 of life. The odds (95% CI) of supplying these drugs topatients with OSAS are presented in Table 5. The most suppliedrespiratory subcategory was nasal preparations, which was pro-vided more (17 vs. 25%) to patients with OSAS (p � 0.0001).Interestingly, drugs for obstructive airway diseases were suppliedmore (15%) to subjects with OSAS in Years 3 and 4 (odds ratio,2.0; 95% confidence interval, 1.1–3.9). Other pharmacologic cate-gories were not consistently supplied more to patients withOSAS.

Review of Medical Records

Medical diagnoses made during hospital visits in all 4 yr of lifeare summarized in Table 6. Compared with the control group,children with OSAS had higher rates of lower respiratory tractdiseases (i.e., pneumonia, bronchiolitis, and asthma) and “other”diseases (e.g., gastrointestinal or orthopedic) (p � 0.0001). Nodifferences were found in upper respiratory tract diseases (i.e.,otitis media, tonsillitis, laryngitis, and croup).

TABLE 4. NUMBER OF CHILDREN REFERRED TO CONSULTANTS AND ODDS FOR EACHYEAR OF LIFE

Year 1 Year 2 Year 3 Year 4

OSAS-related consultants*Control, % (n) 9 (14) 12.2 (19) 10.9 (17) 7.1 (8)OSAS, % (n) 21.2 (33)† 19.9 (31)‡ 25.0 (39)‡ 42.0 (47)†

OR (95% CI) 2.7 (1.4–5.3) 1.8 (0.9–3.3) 2.7 (1.5–5.1) 9.4 (4.2–21.2)General consultants*

Control, % (n) 23.7 (37) 18.6 (29) 21.2 (33) 19.6 (22)OSAS, % (n) 29.5 (46) 31.4 (49)† 37.2 (58)† 38.4 (43)†

OR (95% CI) 1.3 (0.8–2.2) 2.0 (1.2–3.4) 2.2 (1.3–3.6) 2.6 (1.4–4.7)

Definition of abbreviations: CI � confidence interval; n � 156 children for Years 1–3; n � 112 for Year 4; OR � odds ratio; OSAS� obstructive sleep apnea syndrome.

* OSAS-related consultants included pediatric pulmonologists and otolaryngology surgeons. OSAS consultations did not includethe last ENT surgeon visits before the PSG study. General consultants included ophthalmologists, neurologists, allergologists,cardiologists, dermatologists, and orthopedic surgeons.

† p � 0.0001, by McNemar test.‡ p � 0.01, by McNemar test.

DISCUSSION

Main Findings

The major new finding in the current report is that health careuse and morbidity are increased for several years before OSASdiagnosis and treatment. Increased morbidity among childrenwith OSAS was related to lower respiratory airway diseases.The total number of hospital visits from the time of diagnosis,starting from the first year of life, was 40% higher in the OSASgroup, and these children required 20% more repeated hospitalvisits. Referral of children with OSAS to ENT and pediatricpulmonologists was significantly higher beginning in Year 1, andespecially in Year 4. Antiinfective and respiratory categories ofdrugs were supplied to patients with OSAS significantly morefrom Year 2 and up.

Studied Group

We present results of “typical” otherwise healthy children withOSAS compared with their healthy control subjects (19, 20).The study group were less than 5 yr of age; children 5 yr of ageor younger have more severe OSAS than do children older than5 yr (14). In this age category, the occurrence of adenotonsillarhypertrophy peaks during the fifth year of life (7, 21) and healthcare use is maximal (14). To better understand the influence ofOSAS per se on health care use in a physician’s daily practice,we had to exclude children who either exhibited extreme con-sumption of health care services or had additional serious mor-bidities. Therefore, our result presents the minimum annual ex-penditures of children with OSAS because none of the childreninvestigated had concomitant diseases. It may be postulated thatcontrol subjects should be “normal” subjects (regarding AHIand oxygen saturation) referred for PSG evaluation from thesame doctor. However, most subjects referred to OSAS evalua-tion are symptomatic, mainly reporting snoring. It was suggestedthat snoring is not just an innocent noise during sleep in infantsbut may represent the lower end of the disease spectrum associ-ated with sleep-disordered breathing (22). Therefore, labora-tory-proven “normal” children are not an adequate controlgroup.

We analyzed data in two modes: 1 yr before diagnosis andfrom the first year of life including Year 4. These analyses arecomplementary and minimize uncertainties regarding healthcare use. At the time of diagnosis, most children with OSAShad not completed the full last year of life. Forty-four childrenwho underwent surgery were excluded from data analysis of

Tarasiuk, Greenberg-Dotan, Simon-Tuval, et al.: Health Care Use in Children with OSAS 59

TABLE 5. NUMBER OF CHILDREN AND ODDS FOR DRUGS SUPPLIED FOR EACH YEAR OF LIFE

Pharmacologic Group Year 1 Year 2 Year 3 Year 4

J: General antiinfectives (systemic use) 1.06 (0.6–1.8) 1.9 (1.02–3.4) 2.5 (1.4–4.4) 3.1 (1.7–5.7)n (control/OSAS) 114/116 121/135 109/133 65/91

R: Respiratory system 1.0 (0.6–1.7) 1.9 (1.04–3.3) 3.3 (1.9–5.6) 2.9 (1.6–5)n (control/OSAS) 121/121 118/133 92/129 59/85

Definition of abbreviations: ANOVA � analysis of variance; n � number of children supplied with at least one drug from groupJ or R; OSAS � obstructive sleep apnea syndrome.

Drugs supplied in group R include the following: R1, nasal preparations; R3, drugs for obstructive airway diseases; R5, coughand cold preparations; and R6, antihistamines for systemic use. Both the general antiinfectives group and the respiratory groupof the OSAS drug group were significantly different from their pair-matched control, using two-way ANOVA (p � 0.01).

Year 4, because adenotonsillectomy reduces health care use (13).These children were significantly younger and had more severeAHI, but their total annual costs 1 yr before diagnosis did notdiffer from those of the 112 children with OSAS included in thefinal analysis of this year.

Associated Morbidity

Medical diagnoses made during hospital visits and the type ofsupplied drugs in the community settings revealed that childrenwith OSAS had significantly higher rates of lower respiratorytract diseases. This finding does not corroborate our previousreport (13) that increased morbidity among children with OSASis related to upper respiratory tract infections. This may bepartially explained by the possibility that in this study we ex-plored morbidity in younger children from day of birth to dateof diagnosis. In this age category, the upper respiratory soundsmay be wrongly interpreted as small airway disease becauseclinical and pulmonary function testing of small airway diseasesis difficult and not routinely performed in children who areyounger than 1 yr (23). Both upper and lower airway diseases inyoung children may present similar symptoms preceding OSAS,signs that become more obvious after infancy (24). Inflammationoccurs in lymphadenoid tissue in older children (25, 26), leadingphysicians to the diagnosis at a later age. This information maydeviate from or mask a physician’s decision to refer patients forPSG study.

There is a true link between lower respiratory tract illnessand OSA that has not yet been well elucidated in children.This implies that children who have had lower respiratory tractinfections should be watched more carefully for OSAS.

Health Care Use

Elevation of health care consumption among children withOSAS has already been reported (14). The main factors in ele-vated total annual costs in children with OSAS and not exceeding

TABLE 6. MEDICAL DIAGNOSES MADE INHOSPITAL SETTING

Diagnosis [% (n)]

OSAS (n � 228) Control (n � 162)

Upper respiratory infection 22 (50) 28 (46)Lower respiratory disease 35 (80) 9 (14)*Other disease 43 (98) 63 (102)*

Definition of abbreviations: n � all hospital visits during the study period; OSAS� obstructive sleep apnea syndrome.

Upper respiratory tract disease included otitis media, tonsillitis, laryngitis, andcroup. Lower respiratory disease included pneumonia, bronchiolitis, and asthma.Other disease included, for example, gastrointestinal and orthopedic diseases.

* p � 0.0001, �2 test.

5 yr of age are in-patient admissions and visits to the emergencyroom, representing more than 50% of total annual costs. Thesecost elements reflect severe morbidity.

Objective variables such as AHI have little predictive valueof health care use in patients with OSAS. Our findings supportthe evidence that in children (27) and adults (11–14) with OSAS,PSG parameters weakly correlate with outcome measures suchas the Epworth Sleepiness Scale (27). The information that PSGfindings did not predict health care consumption may resultfrom the fact that AHI in these patients is probably above thethreshold for elevated health care use. The increased numberof referrals to specialists was the only significant cost elementamong children with OSAS during the first year of life. It is notclear why the odds for referral to OSAS-related consultantsand diagnosis were considerably higher in Year 4, even afterexcluding ENT consultation before PSG referral. The answer isnot straightforward. In fact, as in adults (28), awareness of sleep-disordered breathing symptoms in children by parents and physi-cians is low (29, 30). Among children with a significant historyof snoring, only 8% of parents mentioned this symptom duringthe concurrent clinical evaluation, and only 15% had done sopreviously (29).

Health care use in adults with OSAS is related to obesity,alcohol use, caffeine and tobacco consumption, and cardiovascu-lar comorbidity (8, 9, 11), among other risk factors, and probablyto low socioeconomic status (31). It is possible that multiplemodifiers, including passive smoking, socioeconomic status, andsnoring (22, 31), together with predisposing genetic risk factors(32–37) and evolving airway inflammation (26), may contributeto the occurrence of sleep-disordered breathing starting in thefirst year of life.

Our data on health care use may be difficult to compare withthose from other health care systems that have more than onepayer, as in the United States. However, our data represent ahealth care system similar to that in Canada (8–11). The informa-tion presented reflects the “true” consumption of health careresources (12–14, 31) of children with OSAS: all PSGs and therelevant medical information regarding patients with OSAS arestored in the only sleep center in the region. CHS uses one billingsystem to include community and hospital services. According tothe National Health Care Law, equal access to medical servicesis provided to all enrollees and there is no economic incentiveto increase consumption of sleep laboratory services due to reim-bursement policies. Physicians are paid a capitation fee onceevery 3 mo per patient and do not have any economic incentiveto increase consumption of services (12–14).

The elevated health care costs attributed to lower respiratorytract diseases may represent missed diagnoses of OSAS, sug-gesting the need for early diagnosis and intervention. This wouldpresumably be the case if an effective treatment is available, asin our system in which equal access to treatment is available

60 AMERICAN JOURNAL OF RESPIRATORY AND CRITICAL CARE MEDICINE VOL 175 2007

and performed (13, 14, 31). Two-thirds of our children underwentsurgery. This group of children probably benefited from treat-ment by improved sleep characteristics, behavior, and psycholog-ical and neurocognitive functions (15, 19, 23). In addition, totalannual cost savings are maximal among children with AHI � 8events/h (13). One-third of children were not treated surgically,probably because of a low level of awareness among patientsand physicians to the potential benefits of surgery or to OSAS-associated morbidity (13).

Study Limitations

The main challenge with these results is to derive a causal infer-ence from the correlative data at hand. One cannot necessarilyattribute increased health care use to OSAS per se. It could bethat some parents assertively seek health care services for theirchildren, including PSG, consultations, and medications, eventu-ally being diagnosed as having OSAS. However, this possibilityis unlikely because all enrollees have equal access to all medicalservices with no economic barrier (12–14). The ability of patientsto pursue medical help may be influenced by socioeconomicstatus; however, we minimized this effect by selecting controlsubjects from the same geographic location (31). The ability ofparents to pursue medical help may be related to the fact thatchildren with OSAS have nonspecific symptoms including a dele-terious effect on cognition, inattention, hyperactivity, and reduc-tion of quality of life (27, 38–42) that can lead parents to engagehealth care services. Further studies are needed to explore thisimportant issue. Moreover, passive cigarette smoke is a recog-nized environmental risk factor for snoring-related sleep frag-mentation among infants (22). In our study, passive smoking didnot increase health care use. This result should be interpretedwith caution because the number of smoking parents was toosmall to enable us to reach a conclusion; therefore, this requiresfurther study. It is unlikely that a practice pattern of individualENT surgeons affects the “risk for sleep laboratory referral,”because in our region all children with OSAS and control sub-jects are referred to the same limited group of ENT consultingsurgeons.

Conclusions

Children with OSAS present significantly greater morbidity andhigher health care use, starting from the first year of life. Mostconsumed health care resources are related to respiratory dis-eases. Practitioners should be aware that an increase in airwaydiseases and health care costs, starting from the first year oflife, may be associated with the presence of sleep-disorderedbreathing. This study suggests the need for early diagnosis andintervention in children with OSAS.

Conflict of Interest Statement : None of the authors has a financial relationshipwith a commercial entity that has an interest in the subject of this manuscript.

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