A case of "pure" progressive autonomic failure in an elderly male]

A 68-year-old man was admitted to our hospital because of postural hypotension in July 1991. He was also suffering from anhydrosis, urinary disturbances, constipation and impotence. He had not developed signs of Parkinsonism, cerebellar or peripheral neuropathy four years from the onset. Various autonomic function tests showed sympathetic and parasympathetic dysfunction of mainly postganglionic origins. Thus we diagnosed this patient as "pure" progressive autonomic failure ("pure" PAF). "Pure" PAF is a new entity described by Bannister and Oppenheimer in 1982. It shows symptoms of autonomic failure without other neurological disturbances which manifest as Shy-Drager syndrome. Treatment with L-DOPS increased his blood pressure level and attenuated his symptoms due to orthostatic hypotension.     

持续正压通气辅助治疗高血压并发睡眠呼吸暂停综合征的临床研究

目的探讨持续正压通气辅助治疗高血压并发睡眠呼吸暂停综合征（SAS）对血压的影响。方法41例高血压并发睡眠呼吸暂停综合征病人,随机分为常规组（21例）和持续正压通气治疗组（20例）均给予常规降压药物治疗4周,治疗在常规治疗基础上加用气道持续正压通气治疗,观察两组治疗前后24 h血压变化。结果治疗组治疗后24 h平均收缩压、舒张压进一步降低,夜间收缩压及舒张压则显著降低（P〈0.01）。结论高血压并发睡眠呼吸暂停综合征病人,在常规应用降压药物同时应用持续正压通气辅助治疗血压下降更理想。     

Chronic lung disease in the sleep apnea syndrome

Several well controlled epidemiologic and hemodynamic studies suggest that about 20% of sleep apnea syndrome (SAS) patients will have chronic obstructive pulmonary disease (COPD), and the majority of these patients (with combined diseases) will have pulmonary hypertension. Indeed it has been suggested that only patients with underlying hypoxemia, such as that from COPD, will develop right heart failure in the OSA setting. Experience shows that apnea/COPD patients will have severe hypersomnolence associated with the OSA, cough and dyspnea with the airways disease, and edema and plethora related to chronic hypoxemia. Many patients present with respiratory failure and are diagnosed at the time of initial intubation and mechanical ventilation. Episodic nocturnal hypoxemia may be worsened by a steeper rate of desaturation due to lower alveolar and blood oxygen stores, and longer apneas perhaps contributed to by depressed chemosensitivity. Daytime hypoxemia may also add to the severe hemodynamic disturbances. Since COPD cannot be cured, aggressive treatment of SAS is critical. Past studies have shown that tracheostomy or nasal CPAP in this setting not only leads to resolution of episodic nocturnal desaturation but may lead to rapid improvement in daytime oxygenation in many patients. Pulmonary hypertension and other measures of cardiopulmonary function improve when apnea is cured. Elimination of the SAS may disclose nonapneic REM related desaturation that could require supplemental oxygen therapy in addition to tracheostomy or nasal CPAP. Pulmonary function testing in SAS patients with smoking histories, followed by aggressive treatment of SAS, is recommended.     

以直立性低血压为主要表现的Shy-Drager综合征42例分析

目的:探讨Shy-Drager综合征的临床表现,以便早期诊断治疗.方法:回顾性分析经临床诊断的42例Shy-Drager综合征患者的相关资料.结果:Shy-Drager综合征患者临床以直立性低血压、睡眠呼吸暂停综合征、鼾症、便秘最常见.结论:Shy-Drager综合征临床以自主神经功能障碍为主要表现,综合性治疗能够改善患者症状,提高生活质量.     

Obstructive sleep apnea syndrome induced by ossification of the anterior longitudinal ligament with ankylosing spondylitis]

A 66-year-old man who had received a diagnosis of ankylosing spondylitis at the age of 50 was referred to our hospital for further evaluation of sleep apnea experienced for the last 3 years. Polysomnography yielded a diagnosis of obstructive sleep apnea syndrome with an apnea index of 41/hr. Computed tomographic scans demonstrated complete closure of the oropharynx during sleep at a site of marked ossification and hypertrophy of the cervical anterior longitudinal ligament. Nasal continuous positive airway pressure (nasal CPAP) with the use of chinstrap, but not nasal CPAP alone, dramatically decreased the patient's apnea index to 2.4/hr. We reported this case because, to the best of our knowledge, it is the first to demonstrate a causative association between ankylosing spondylitis and obstructive sleep apnea syndrome.     

Patient compliance with nasal CPAP therapy for sleep apnea

Continuous positive airway pressure via the nasal route (nasal CPAP) is effective in reducing the frequency of occlusive and mixed patterns of sleep apnea. Little is known about long-term patient compliance with this therapeutic modality, however. In order to evaluate this, questionnaires were mailed to 24 sleep apnea patients who had received a nasal CPAP system for nightly use. Patients were requested not to sign the questionnaire. At the time of mailing, the patients had possessed their nasal CPAP equipment for 10.3 +/- 8 months (mean +/- SD). Twenty patients (83 percent) responded to the questionnaire. Sixteen responders used nasal CPAP during all nightly sleep time, and two used it for all but one and 2.5 hours of nightly sleep time, respectively. One individual used it on alternate nights, and one patient did not use it all. Thus, 17 of 20 (85 percent) responders were compliant. Including information about the four nonresponders, obtained by means other than questionnaire, 18 of 24 (75 percent) patients were compliant with therapy. All but two patients were obese at the time sleep apnea was diagnosed. Only 50 percent of questionnaire responders reported weight loss after receiving nasal CPAP, and these individuals were frequent nasal CPAP users. The most common complaints were mask discomfort (14 responders) and nasal dryness and congestion (13 responders). We conclude that long-term home nasal CPAP is a viable therapy that is conscientiously applied and well-tolerated by most sleep apnea patients.     

正压通气治疗睡眠呼吸暂停综合征并代谢综合征的研究

目的：研究持续气道内正压通气（CPAP）对睡眠呼吸暂停综合征（SAS）并代谢综合征（MS）的疗效。方法：158例SAS并MS患者被随机分成两组，即CPAP治疗组（治疗组）和药物治疗组（对照组），治疗组在药物组治疗基础上加用CPAP治疗。两组患者于治疗前、治疗4周后分别行呼吸睡眠监测：动脉血氧饱和度（SaO2）、呼吸参数，并测空腹血糖（FPG）、空腹胰岛素（FINS）、餐后2h血糖（2hPG）、餐后2h胰岛素（2hPINS）、胰岛素敏感性指数（ISI）、总胆固醇（TC）、甘油三酯（TG）、高密度脂蛋白胆固醇（HDL-C）、体重指数（BMI）、收缩压（SBP）、舒张压（DBP）的变化。结果：与对照组比较，治疗组治疗后呼吸暂停和低通气均明显减少，打鼾消失，低氧发作次数减少，最低SaO2明显提高（P〈0．01）；同时血糖、胰岛素、TC、TG浓度，SBP、DBP均明显降低，HDL-C水平明显升高，与对照组比较有明显差异（P〈0．01）。结论：CPAP治疗SAS并MS除能改善SAS病情外，还能加强药物疗效，改善代谢参数，降低血压，降低心脑血管病危险因素的程度。     

Continuous positive airway pressure intolerance associated with elevated nasal resistance is possible mechanism of complex sleep apnea syndrome

Purpose

Complex sleep apnea syndrome (CompSAS) is diagnosed after an elimination of obstructive events with continuous positive airway pressure (CPAP), when a central apnea index ??5/h or Cheyne?CStokes respiration pattern emerges in patients with obstructive sleep apnea syndrome (OSAS). However, the pathophysiology of CompSAS remains controversial.

Methods

Of the 281 patients with suspected OSAS, all of whom underwent polysomnography conducted at Nagoya University Hospital, we enrolled 52 patients with apnea?Chypopnea index ??15/h (age 51.4?±?13.3?years). The polysomnographic findings, left ventricular ejection fraction (LVEF), and nasal resistance were compared between the CompSAS patients and OSAS patients.

Results

Forty-three patients were diagnosed with OSAS and nine patients with central sleep apnea syndrome by natural sleep PSG. Furthermore, 43 OSAS patients were classified into the OSAS patients (OSAS group, n?=?38) and the CompSAS patients (CompSAS group, n?=?5) by the night on CPAP PSG. The nasal resistance was significantly higher in CompSAS group than in OSAS group (0.30?±?0.10 vs. 0.19?±?0.07?Pa/cm³/s, P?=?0.004). The arousal index, percentage of stage 1 sleep, and oxygen desaturation index were significantly decreased, and the percentage of stage REM sleep was significantly increased in the OSAS group with the initial CPAP treatment, but not in the CompSAS group. In addition, the patients with CompSAS showed normal LVEF.

Conclusion

CPAP intolerance secondary to an elevated nasal resistance might relate to frequent arousals, which could presumably contribute to an increase in central sleep apnea. Further evaluation in a large study is needed to clarify the mechanism of CompSAS.     

Effects of nasal continuous positive airway pressure on soluble cell adhesion molecules in patients with obstructive sleep apnea syndrome

PURPOSE: Obstructive sleep apnea syndrome is common in middle-aged men and may be associated with an increased risk of cardiovascular disease. We investigated the effect of nasal continuous positive airway pressure (CPAP) treatment on levels of soluble cell adhesion molecules-which have been shown to be associated with the development of atherosclerosis-in these patients. SUBJECTS AND METHODS: We studied 23 patients with obstructive sleep apnea syndrome diagnosed by polysomnography who were treated with nasal CPAP. Serum soluble intercellular adhesion molecule-1, E-selectin, and vascular cell adhesion molecule-1 levels were measured before nasal CPAP was started, and after 3 or 4 days (n = 19), 1 month (n = 23), or 6 months (n = 11) of treatment. RESULTS: After 3 to 4 days of nasal CPAP therapy, the mean (+/- SD) soluble E-selectin level had decreased from 89 +/- 44 ng/mL to 69 +/- 28 ng/mL (P = 0.002). After 1 month, the soluble intercellular adhesion molecule-1 level had decreased from 311 +/- 116 ng/mL to 249 +/- 74 ng/mL (P = 0.02). After 6 months, soluble vascular cell adhesion molecule-1 levels had not changed significantly, while the mean soluble intercellular adhesion molecule-1 level (212 +/- 59 ng/mL) had decreased further (P = 0.02). Before treatment, soluble intercellular adhesion molecule-1 levels and the apnea and hypopnea index were correlated (r = 0.43, P = 0.04). CONCLUSIONS: Obstructive sleep apnea and hypopnea have a significant adverse effect on serum soluble cell adhesion molecule-1 levels that may be reduced by nasal CPAP treatment.     

Atrial arrhythmia as a complication of nasal CPAP.

We report a case of supraventricular rhythm disorder occurring as a complication of nasal continuous positive airway pressure (CPAP) in sleep apnea syndrome treatment. The investigations showed a preexisting atrial vulnerability revealed by positive pressure ventilation, hemodynamic changes with decreased cardiac output, and a rise in the right atrial pressure. Such complications may develop further studies to evaluate cardiovascular repercussions of nasal CPAP.     

Improving Heart Rate Variability in Sleep Apnea Patients: Differences in Treatment with Auto-titrating Positive Airway Pressure (APAP) Versus Conventional CPAP

The effect of positive airway pressure treatments in different modalities on the cardiovascular consequences of the disease in sleep apnea patients is still unclear. We aimed to compare auto-titrating positive airway pressure (APAP) and conventional continuous positive airway pressure (CPAP) in terms of improving heart rate variability (HRV) in obstructive sleep apnea patients. This was a prospective study done in a tertiary research hospital. All patients underwent a manual CPAP titration procedure to determine the optimal pressure that abolishes abnormal respiratory events. Then patients underwent two treatment nights, one under APAP mode and one under conventional CPAP mode with a 1-week interval. Forty newly diagnosed obstructive sleep apnea patients were enrolled in the study. We compared heart rate variability analysis parameters between the APAP night and the CPAP night. This final analysis included the data of 28 patients (M/F: 22/6; mean age = 46 ± 10 years). Sleep characteristics were comparable between the two treatment nights, whereas all-night time domains of HRV analysis such as HF, nuLF, and LF/HF were different between APAP and CPAP nights (2.93 ± 0.31 vs. 3.01 ± 0.31; P = 0.041; 0.75 ± 0.13 vs. 0.71 ± 0.14; P = 0.027; and 4.37 ± 3.24 vs. 3.56 ± 2.07; P = 0.023, respectively). HRV analysis for individual sleep stages showed that Stage 2 LF, nuLF, nuHF, LF/HF parameters entirely improved under CPAP treatment whereas APAP treatment resulted in nonsignificant changes. These results suggest that despite comparable improvement in abnormal respiratory events with APAP or CPAP treatments, CPAP may be superior to APAP in terms of correcting cardiovascular alterations in sleep apnea patients.     

Optimal high-frequency oscillatory ventilation settings by nonlinear lung mechanics analysis

Obstructive sleep apnea causes cardiovascular morbidity and premature death. Potential links between sleep apnea and cardiovascular complications are chronically elevated activity of the sympathetic nervous system and abnormal vascular function. To explore vascular function, we determined the reactive hyperemic blood flow (RHBF) responses to 10 minutes of forearm arterial occlusion (plethysmography), blood pressure, and muscle sympathetic nerve activity (MSNA, microneurography) in eight patients with sleep apnea and in nine nonapneic control subjects. Peak RHBF and vascular conductance were markedly attenuated in sleep apnea compared with control subjects (p < 0.05). Seven sleep apnea patients were retested after at least two weeks of continuous positive airway pressure (CPAP) therapy. MSNA decreased after CPAP therapy (p < 0.05, n = 6), whereas blood pressure did not change. After CPAP therapy, peak RHBF and vascular conductance were increased compared with before treatment (p < 0.05; n = 7). Thus, vascular function is abnormal in sleep apnea and is improved by CPAP therapy. Furthermore, effective CPAP therapy decreases sympathetic activity in sleep apnea. Thus, sympathoexcitation and abnormal vascular function in patients with sleep apnea appear to be linked to the repetitive nocturnal apneic events.     

Pneumocephalus associated with nasal continuous positive airway pressure in a patient with sleep apnea syndrome

We report a new potentially dangerous complication of nasal continuous positive airway pressure applied for the treatment of obstructive sleep apnea syndrome. A middle-aged woman had cerebrospinal fluid leak after using nasal CPAP, with generalized seizures and pneumocephalus. She did fine with conservative therapy for the CSF leak and discontinuation of nasal CPAP.     

Circadian and orthostatic blood pressure is abnormal in the carotid sinus syndrome

The authors' objective was to determine circadian blood pressure behavior and the prevalence of orthostatic hypotension in patients with cardioinhibitory carotid sinus syndrome. A prospective consecutive series of 160 patients (mean age, 72 [65–83]) with recurrent syncope attributed to cardioinhibitory carotid sinus syndrome was recruited. Mean maximal slowing of heart rate was 4.5 seconds (3.0–5.5 seconds) during carotid sinus massage. Patients had cardiovascular assessments, including 24-hour ambulatory blood pressure measurements (recordings every half-hour during daytime, hourly at night) and morning orthostatic blood pressures before pacemaker implants. Ambulatory measurements were compared with those of 98 age-and sex-matched controls. Nocturnal systolic blood pressure (130.0±21.0 vs. 122.1±16.7 mm Hg; p< 0.005), diastolic blood pressure (72.8±12.5 vs. 68.7±9.7 mm Hg; p< 0.005), and heart rate (66.5±9.4 vs. 65.2±9.7 bpm; p< 0.005), were significantly higher in patients and independent of cardiovascular medication, cardiovascular diagnoses, and orthostatic hypotension. Almost one half of carotid sinus syndrome patients also had orthostatic hypotension. Blood pressure behavior is abnormal in patients with carotid sinus syndrome as represented by altered nocturnal blood pressures and high prevalence of orthostatic hypotension.     

Sleep apnea syndrome in a patient with Shy-Drager syndrome

A patient with autonomic insufficiency and extrapyramidal signs (Shy-Drager syndrome) and sleep apnea syndrome (SAS) underwent hemodynamic studies. In comparison to patients with SAS and intact autonomic reflexes, systemic hypertension was absent and marked sinus arrhythmia during sleep was blunted. Cyclical pulmonary hypertension associated with frequent apneic episodes during sleep persisted, reflecting a minor role of autonomic reflexes in the generation of this abnormality. Autopsy confirmed the Shy-Drager syndrome and multiple areas of degeneration were observed in areas of the CNS outside the medullary respiratory centers, suggesting their importance in the origin of the respiratory abnormalities in SAS.     

Hypothalamic dysfunction in a case of idiopathic orthostatic hypotension (shy-Drager Syndrome)

Growth Hormone (GH) secretion was studied in a 61-year-old woman with idiopathic orthostatic hypotension (IOH) or Shy-Drager Syndrome. The 24-hour pattern of GH secretion showed multiple daytime peaks and little nighttime activity. The largest daytime peak of 35.0 ng/ml is much greater than the levels observed in seven studies using five age-matched normal women. There were few secretory episodes during sleep in the patient and the control subjects. Paradoxical GH responses occurred during oral glucose tolerance tests, despite high blood glucose. GH was also detected in three studies using TRH. These data suggest that our patient with IOH has abnormal regulation of GH secretion. This is a sign of hypothalamic-pituitary dysfunction in a diffuse neurological disease.     

Treatment of obstructive sleep apnea. A preliminary report comparing nasal CPAP to nasal oxygen in patients with mild OSA

Nasal CPAP is presently accepted as first-line therapy for obstructive sleep apnea, but a significant minority of patients do not tolerate nasal CPAP. The purpose of this study was to compare the benefits of nasal CPAP, nasal oxygen (O2), and placebo (air) using patients as their own controls. We studied eight men, aged 33 to 72 (mean 57 years), who had mild obstructive sleep apnea. To be eligible for study, patients had to have an apnea plus hypopnea index greater than or equal to 5, plus one or more of the following: blood pressure greater than 150/95 mm Hg, multiple sleep latency test mean score less than or equal to 10 minutes, or significant nocturnal cardiac ectopy. After a baseline study, patients received a month each of nocturnal O2 at 4 LPM and air at 4 LPM, presented in random order. The third month of treatment consisted of nasal CPAP (range 2.5 to 12.5 cm H2O). Patients underwent evaluation at baseline and after each month of treatment. It was concluded that oxygen was more effective in improving oxygenation and hypopneas than is nasal CPAP. However, oxygen did not reduce apneas or improve daytime hypersomnolence as well as nasal CPAP in patients with mild OSA. Oxygen might be considered as an alternate form of treatment for patients who are not hypersomnolent, or as an adjunct to nasal CPAP.     

Clinical usefulness of a cardiopulmonary sleep study in patients with sleep apnea syndrome]

Although polysomnography is indispensable in the diagnosis of sleep apnea syndrome (SAS), a common disease in Japan, it is a time-consuming procedure. We therefore assessed the clinical usefulness of a cardiopulmonary sleep study (CPSS), which monitors indices of respiratory and circulatory kinetics during sleep. Of ninety male patients (50 +/- 11 years old, BMI: 27 +/- 4), those with apnea indices (AI) > 5, as determined using an apnomonitor, underwent CPSS with a Night Watch System. Sixty-nine patients with indications for nasal CPAP (NCPAP) therapy underwent overnight CPSS and the optimum pressure was determined. AI was 31 +/- 17, whereas CPSS showed an apnea-hypopnea index of 49 +/- 24, a mean SaO2 of 93 +/- 3%, and a minimum SaO2 of 71 +/- 11%, indicating severe OSAS. The mean optimum pressure was 10 +/- 2 cm H2O, and the compliance was 86%, which was comparatively good. As CPSS can be performed on many patients in a short time, it may be useful for the early diagnosis and initial treatment of SAS.     

Effect of heated humidification on compliance and quality of life in patients with sleep apnea using nasal continuous positive airway pressure

STUDY OBJECTIVE: To determine whether the addition of heated humidification at treatment initiation with nasal continuous positive airway pressure (CPAP) would lead to better CPAP compliance and improve quality of life and subjective sleepiness in patients with sleep apnea. DESIGN: Randomized controlled trial. SETTING: An academic sleep center located at a Veterans Affairs hospital. PATIENTS: Ninety-eight patients with obstructive sleep apnea who had not received nasal CPAP previously. MEASUREMENTS AND RESULTS: Patients received heated humidification at CPAP initiation in the treatment group. In the control group, patients could receive heated humidification only if they had upper airway symptoms that could not be treated successfully with simpler measures. Patients were followed up at 1 month, 3 months, and 12 months. Outcome measures were compliance with nasal CPAP (mean hours per night at effective pressure), quality of life as measured by the Calgary sleep apnea quality of life index, subjective sleepiness measured with the Epworth sleepiness scale, and CPAP side effects. There was no difference in CPAP compliance between groups. Quality of life and subjective sleepiness improved in both groups with nasal CPAP therapy, but there was no difference in the extent of improvement between groups. The overall CPAP side effect score was similar in both groups, but individual symptoms of dry nose and dry mouth and throat were significantly lower in the heated humidification group. CONCLUSIONS: The addition of heated humidification when nasal CPAP was instituted did not lead to better compliance, greater improvement in sleepiness, or improved quality of life, but was associated with fewer symptoms attributable to the upper airway.     

Correction of AV-nodal block in a 27-year-old man with severe obstructive sleep apnea--a case report

A 27-year-old morbidly obese man diagnosed with severe obstructive sleep apnea (OSA) and experiencing significant ventricular asystoles at times exceeding 8 seconds, during polysomnography. The bradyarrhythmias were successfully corrected with the application of a nasal continuous positive airway pressure (CPAP) mask. Follow-up 24-hour ambulatory Holter monitoring without the aid of a nasal CPAP mask and repeat polysomnography with a CPAP mask after several weeks of continuous CPAP therapy during sleep revealed no evidence of ventricular asystole, despite no change in the patient's body mass index. We discuss several mechanisms explaining the findings in this particular patient.