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* From the Sleep Disorders Centre (Drs. Berg and Kryger and Mr. Delaive), Section of Respiratory Diseases and Department of Medicine, St. Boniface General Hospital Research Centre, Winnipeg, Manitoba; and the Department of Community Health Sciences (Dr. Manfreda), Centre for Health Policy and Evaluation, University of Manitoba (Mr. Walld), Winnipeg, Manitoba. Supported in part by National Institutes of Health grant R01 HL6334201A1.
Correspondence to: Meir H. Kryger, MD, FCCP, Director, Sleep Disorder Centre, St. Boniface General Hospital, Room R2034, 351 Taché Ave, Winnipeg, Manitoba R2H 2A6; e-mail: kryger{at}sleep.umanitoba.ca
| Abstract |
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Design: Retrospective observational cohort study.
Setting: University-based sleep disorders center in Manitoba, Canada.
Patients and control subjects: Twenty OHS patients (mean [± SD] age, 52.7 ± 9.5 years; body mass index [BMI], 47.3 ± 11.0 kg/m2; PaCO2, 59.7 ± 13.8 mm Hg; PaO2, 51.6 ± 12.4 mm Hg) were matched to two sets of control subjects. First, each case was matched to 15 general population control subjects (GPCs) by age, gender, and geographic location, and, second, each case was matched to a single obese control subject (OBC) who was matched using the same criteria as for the GPCs, plus the measurement of BMI.
Measurements and results: In the 5 years before diagnosis, the 20 OHS patients had (mean ± SE) 11.2 ± 1.8 physician visits per patient per year vs 5.7 ± 0.8 (p < 0.01) visits for OBCs and 4.5 ± 0.4 (p < 0.001) visits for GPCs. OHS patients generated higher fees, $623 ± 96 per patient per year for the 5 years prior to diagnosis compared to $252 ± 34 (p < 0.001) for OBCs and $236 ± 25 (p < 0.001) for GPCs. OHS patients were much more likely to be hospitalized than were subjects in either control group in the 5 years prior to diagnosis (odds ratio [OR] vs GPCs, 8.6) (95% confidence interval [CI], 5.9 to 12.7); OR vs OBCs, 4.9 (95% CI, 2.3 to 10.1). In the 2 years after diagnosis and the initiation of treatment (usually continuous positive airway pressure or bilevel positive airway pressure), there was a significant linear reduction in physician fees. In the 2 years after the initiation of treatment, there was a 68.4% decrease in days hospitalized per year (5 years before treatment, 7.9 days per patient per year; after 2 years of treatment, 2.5 days per patient per year [p = 0.01]).
Conclusions: OHS patients are heavy users of health care for several years prior to evaluation and treatment of their sleep breathing disorder; there is a substantial reduction in days hospitalized once the diagnosis is made and treatment is instituted.
Key Words: apnea hypoventilation obesity sleep utilization
| Introduction |
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Obesity, which can impair the function of the respiratory system, is a significant contributor to obstructive sleep apnea-hypopnea syndrome (OSAHS). Before OSAHS was described, it was known that morbid obesity might result in hypoventilation.7 8 9 10 Several terms have been used to describe the combination of obesity and hypoventilation, including pickwickian syndrome and, more recently, obesity-hypoventilation syndrome (OHS). Although both OSAHS and OHS patients have excessive daytime hypersomnolence, there are fundamental differences between the two conditions. In OSAHS, disordered breathing is present typically only during sleep and obesity is not always present. In OHS, hypoventilation is present during sleep and wakefulness, and by definition obesity is always present. Manifestations frequently associated with OHS include polycythemia, pulmonary hypertension, right-sided heart failure, and awake respiratory failure as documented by daytime hypercapnia.8 11
MacGregor et al12 reported a high level of mortality, including sudden death, in patients with pickwickian syndrome and a subsequent reduction in mortality with the institution of mechanical ventilation. It is now generally agreed that the severe and often life-threatening cardiopulmonary sequelae resulting from hypoventilation often demand instituting therapy, frequently on an emergency basis.13 Although we have shown that OSAHS patients are heavy users of health-care resources,14 15 the long-term health-care utilization of OHS patients has not been rigorously reviewed. It is reasonable to hypothesize that OHS patients are heavy users of health-care resources in a manner that is similar to that by OSAHS patients and that the institution of treatment also will result in a reduction in health-care utilization. To test our hypothesis, we compared the physician claims for payment and hospitalizations in patients with OHS before and after diagnosis, to obese control subjects (OBCs) and to non-OBCs.
| Materials and Methods |
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Manitoba Health protects patient confidentiality by encrypting the health insurance numbers of individuals, thereby creating a unique subject number. This procedure was performed by Manitoba Health before submitting their data to us. We then constructed a final working database that included, for example, complete health-care utilization and diagnosis for each selected patient and the randomly matched selected control subjects (see below). The Human Ethics Committee of the University of Manitoba and the Access and Confidentiality Committee of Manitoba Health approved the study.
Patient Selection and Evaluation
We selected all patients who had received a diagnosis of OHS and
were studied at our sleep disorders center from 1990 to 1995.
This disorder was defined as the combination of obesity (body mass
index [BMI] of > 30 kg/m2) and awake
hypoventilation (PaCO2 of > 45 mm
Hg) during a medically stable period. All patients were residents of
Manitoba for the duration of this study. They all were assessed with
formal polysomnography and were evaluated by a sleep physician
specialist. Data were available in the database for the 5 years before
polysomnography and for the 2 years after the institution of treatment.
We confirmed that the patients were alive at the end of the 2 years
they received treatment, since the health database was linked to the
population registry that tracks deaths in the province. Data were
available on 26 patients who had received a diagnosis of OHS. Six
patients were excluded from the data analysis (see "Exclusion
Criteria" section).
Exclusion Criteria
Patients were excluded if their age was < 18 years (n = 1)
or > 70 years (n = 3), or if they did not have utilization data for
the period of 5 years before and 2 years after diagnosis (n = 2; one
patient died and one left the province). Age limits were set to avoid
the influence of other significant diseases skewing the limited patient
data and to allow for the presence of OBCs, who also had an age limit
in the Manitoba Heart Health Study (see below).
Polysomnography
The study included multichannel monitoring, including the
following: oxygen saturation; electrocardiography; electro-oculography;
electromyography; and electroencephalography.
PCO2 in the nares was monitored using
a capnometer (Normocap 200; Datex; Madison, WI), which was used
as an indicator of airflow. Because entrained air could dilute the
sample, the measured PCO2 values were
lower than actual end-tidal PCO2
levels. However, when the measurement of nasal
PCO2 was elevated, it provided
evidence of hypoventilation. Two plethysmograph belts were used to
monitor thoracic and abdominal movements. The apnea-hypopnea index
(AHI) was used as an indication of the severity of the apnea. A
hypopnea was defined as a visual reduction by 50% in flow or movement
combined with an arousal or a desaturation of arterial oxygen
saturation (SaO2) of > 3%. The
severity of desaturation during sleep was measured by evaluating the
SaO2 time < 90%. Based on
polysomnography findings, patients were started on a regimen of home
ventilatory assistance (either continuous positive airway pressure
[CPAP] or bilevel positive airway pressure).
Control Subjects
Two sets of control subjects were established for each OHS
patient. First, an OBC, who was matched according to the closest BMI,
age, gender, and postal code (to control for geographic and
socioeconomic variables), was selected from the Manitoba Heart Health
Study database. This database, which has been described
elsewhere,18
19
20
was part of a Canadian population-based
investigation of cardiovascular disease risk factor prevalence. Adults
aged 18 to 70 years (n = 2,792) were randomly selected from the
Manitoba population, and 2,212 individuals (79.2%) were interviewed
and examined. BMI and other data were collected on each individual.
Second, each OHS patient also was matched to 15 control subjects selected from the entire MHDB (general population control subjects [GPCs]). These control subjects again were matched to patients for age, gender, and postal code but not for BMI, because BMI data are not stored in the MHDB. Patients and control subjects had been residents of Manitoba continuously for the 7-year period. Exclusion criteria for control subjects were the same as for the case patients, with the additional requirements of being noninstitutionalized and having not received a diagnosis for a chronic psychiatric condition.
Data Analysis
The data were compiled on a yearly basis, with the 5 years prior
to the date of initial sleep laboratory evaluation being labeled years
-5 to -1, and the 2 years after diagnosis being labeled years + 1
and + 2. In order to facilitate trend analysis and cost utilization
prior to and after evaluation, year -1 was included in the two
analyses, years -5 to -1 and years -1 to + 2.
Data were analyzed utilizing a three-way analysis of variance model
with repeated measurements on subjects.21
Linear contrasts
also were utilized to compare differences between OHS patients and both
the OBCs and GPCs. The two periods in which this was performed included
the following: the first year of the study (year -5) to the year prior
to diagnosis and treatment (year -1); and the period year -1 to the
second year after diagnosis (year + 2). A
2
statistic was used to analyze hospitalization data and to calculate the
odds ratios (ORs) for admissions and comorbidity analysis. A Fishers
Exact Test also was utilized where appropriate.
| Results |
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Polysomnography revealed an average AHI of 20.7 ± 23.9 and marked hypoxemia during sleep, with patients spending 63.0 ± 33.8% of sleep time with SaO2 levels of < 90%. Patients were assessed (usually on the same night using a split-night protocol) during treatment (usually with CPAP or bilevel pressure). AHI decreased to 9.7 ± 21.2, and the time spent with SaO2 levels of < 90% decreased to 37.1 ± 28.1% of sleep time. In this very ill group of patients, we were not always able to completely correct hypoxemia with ventilatory assistance. Patients were sent home to use the equipment on settings determined in the sleep laboratory.
Of the two control groups, the OBCs numbered 20 and the GPCs totaled 296. We attempted to match 15 control subjects per OHS patient, which was not achieved in all instances, since we excluded subjects who had received a diagnosis of a chronic psychiatric condition or were institutionalized. In 4 of the patients, we were only able to match 14 instead of 15 GPCs.
The OHS group had an average BMI of 47.3 ± 11.0 kg/m2, and the OBC group had an average BMI of 43.4 ± 5.1 kg/m2. The two groups were matched for other criteria (ie, age, location, and gender) in addition to BMI. Despite matching to the closest BMI, because some of our case patients were so morbidly obese (eg, BMI, 72.86; 64.14 kg/m2), the average BMI for the two groups differed statistically (p = 0.04) by paired t test.
Physician Visits
Each physician encounter resulted in the creation of the following
three variables: the number of physician visits; the number of
physician claims; and physician fees. Each physician visit could
possibly result in several services being used (eg,
investigations), therefore, there are more claims than
visits. Fees are generated from each physician visit claim and
are recorded in Canadian dollars. Overall, OHS patients utilized
significantly more physician visits and claims, and they generated
higher fees than either the OBCs or GPCs. There was no difference
between the two control groups in any category. Only the physician fees
resulted in a significant linear relationship increasing for the 5
years prior to diagnosis and decreasing over years -1 to + 2.
Over the 5-year period prior to diagnosis and treatment, the OHS patients had an average (± SE) of 11.2 ± 1.8 physician visits per patient per year vs 5.7 ± 0.8 physician visits for the OBCs (p < 0.007) and 4.5 ± 0.4 physician visits for the GPCs (p < 0.0001; Fig 1 ). For the same 5-year period for OHS patients, physician claims averaged (± SE) 32.4 ± 5.2 claims per patient per year vs 14.4 ± 1.6 for OBCs (p < 0.002) and 13.4 ± 1.6 for GPCs (p < 0.0005).
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What Patients Were Being Treated for Prior to the Diagnosis of OHS: The MHDB was analyzed for years -1 to -5 for all groups to obtain various diagnoses submitted at any time by a physician. A diagnosis must accompany every physician claim (Table 1 ). Compared to the OBCs, OHS patients were more likely to have received a diagnosis of congestive heart failure, angina, and chronic pulmonary heart disease. Although half the patients in the OHS group had a claim for diabetes, which was twice the rate of the OBC group, this did not quite achieve statistical significance. In all the diagnoses listed in Table 1 , OHS patients had significantly higher claims than did the GPCs (Table 1) .
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| Discussion |
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Up to 8 to 12.5% of morbidly obese patients undergoing gastric surgery in previous studies were found to have OHS.24 25 Why some obese patients develop OHS is not fully understood. Contributing factors may include the restrictive pulmonary function defect, increased work of breathing and CO2 production, and altered central respiratory control.26 Although in most series about three quarters or more of OSAHS patients are men,27 we were surprised that most of our OHS patients were women. This may explain, in part, the relatively low AHI of our OHS patient group since women with sleep breathing abnormalities tend to have lower AHI values.27
We found that compared to GPC, OHS patients were much more likely to have cardiovascular disease (eg, congestive heart failure, angina pectoris, or cor pulmonale), a history of metabolic disease (eg, diabetes or hypothyroidism), and osteoarthritis. Interestingly, osteoarthritis was not more common in OHS patients than in their matched OBCs. OSAHS patients also have increased comorbidity, as we have previously documented.15 Fletcher and colleagues13 have reported previously that OSAHS patients requiring ICU admission often have associated COPD and are more likely to present with progressive respiratory failure.
We have shown in this report that in OHS patients there is increased medical resource utilization, which decreased with treatment. Previously, we have shown that obese OSAHS patients without hypoventilation are heavy users of health-care resources for a decade prior to diagnosis15 and that with the institution of treatment there was a significant reduction within a 2-year period.14
In OHS patients, only physician fees showed a significant linear trend, increasing to the year prior to diagnosis and decreasing for the 2 years after diagnosis. The number of physician visits and claims reflected the same trend but did not achieve statistical significance. Funds paid to physicians are actually underestimated by our data, as salaries are paid to physicians for work in ICUs, for some hospital services, and if they are trainees (house staff). These salaries are not tracked by the database. In addition, the database does not track home-care costs (including mechanical ventilation equipment and nursing supervision), medications, and in-hospital expenditures. Since none of the latter is tracked in a single database, such costs can only be estimated.
OHS patients used significantly more total hospital days than both control groups prior to diagnosis, and this was reduced significantly by treatment. The cost savings due to the reduction in days hospitalized alone (5.4 days per year per patient) can be estimated to be about $5,400 per patient per year, assuming that 1 day spent in the hospital costs about $1,000. The cost of the sleep evaluation per patient in our center was about $410. If we conservatively estimate that the provision of a CPAP or a bilevel system is about $1,000 per patient per year, then the net savings to the health-care system is estimated to be about $3,900 per patient in the first year, and $4,400 in the second year and each additional year. These numbers are conservative because the capital cost of a CPAP system is about $700, and the annual cost of the mask and the disposables is about $250. Clearly, in other locations the costs and savings would be different.
| Conclusions |
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| Footnotes |
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Received for publication October 24, 2000. Accepted for publication March 1, 2001.
| References |
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