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1/8. Hypersomnia with periodic breathing (an acromegalic Pickwickian).

    The syndromes of Pickwickian, Ondine's curse, and primary alveolar hypoventilation are respiratory disorders manifesting increased sleepiness and irregular respiratory rhythms. These disorders are currently grouped as hypersomnia with periodic breathing (HPB). Polygraphic techniques have lead to a reasonable hypothesis as to the pathophysiology of the multiple variants of HPB. Discernible causes of HPB have been attributed to both central and peripheral factors. Peripheral factors encompass those conditions relating to upper airway obstruction. An acromegalic person suffering the HPB syndrome secondary to laryngeal stenosis is described.
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2/8. Pickwickian syndrome, 20 years later.

    The Pickwickian syndrome stimulated new pathophysiological concepts in regard to control of ventilation. With the advent of sleep laboratories, the peculiar sleep apnea occurring in some of these patients has been explained on the basis of intermittent upper airway obstruction. Two patients with different manifestations of the Pickwickian syndrome are presented. The suggestion is made that these two subsyndromes should have unique designations. The Auchincloss syndrome is manifested by right heart failure and respiratory acidosis in obese patients who are alert and have no major abnormality of breathing pattern. The fundamental cause of this abnormality is the increased work of breathing caused by the obesity. The cost of breathing is so high that the ventilatory regulation is compromised and respiratory acidosis results. The Gastaut syndrome is characterized principally by hypersomnia and sleep apnea. The fundamental defect is upper airway obstruction during sleep, resulting in increased work of breathing, which together with the increased work caused by obesity leads to respiratory acidosis and right ventricular failure. Hypersomnia, rather than heart failure or respiratory acidosis, is the major manifestation of this syndrome, and is the result of sleep loss.
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keywords = breathing, upper
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3/8. Spontaneous rupture of the stomach in an adult.

    We have reported a case of spontaneous rupture of the stomach in an adult. Immediate onset of severe upper abdominal pain after overindulgence in food and drink along with radiographic evidence of pneumoperitoneum and the clinical findings of massive abdominal distention, epigastric tenderness, shock, and occasionally subcutaneous emphysema should suggest the possibility of gastric rupture. The treatment is simple, but mortality is high when surgical intervention is not rapid.
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keywords = upper
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4/8. Endoscopic findings in sleep apnea syndrome.

    Sleep apnea syndrome is a constellation of symptoms resulting from recurrent episodes of apnea during sleep. Often the upper airway becomes obstructed during slumber in this disorder. Symptoms relate to sleep deprivation and include morning headaches, daytime somnolence, personality changes with deteriorating intellectual capacity, nocturnal enuresis, and sexual dysfunction. diagnosis is assisted by polysomnographic recordings. Therapy is directed at the cause of obstruction when one can be found, weight loss in massively obese patients, tracheostomy in the symptomatic patient. Four patients with documented sleep apnea syndrome are discussed. One patient, a thin adolescent female underwent adenoidectomy without improvement. Two massively obese adult males required tracheostomy with marked amelioration of symptoms. One additional adult male was found to have sleep apnea due to severe, acquired micrognathia; he was significantly improved by tracheostomy. All three adult patients were found by endoscopic visualization to have marked pharyngeal soft tissue collapse with inspiration during apneic episodes. Possible causes of pharyngeal collapse are discussed.
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keywords = upper
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5/8. Hypersomnia and periodic breathing. Report of a case and review of the literature.

    A patient with hypersomnia and periodic breathing has been studied. Both airflow obstruction and an abnormally of the respiratory control mechanism were implicated in the pathogenesis of the ventilatory arrhythmia. It is suggested that the older terms "Pickwick" syndrome and primary alveolar hypoventilation are abandoned for more descriptive terms, e.g. "hypersomnia with periodic breathing".
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ranking = 1.1998505882669
keywords = breathing
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6/8. Remission of severe obesity-hypoventilation syndrome after short-term treatment during sleep with nasal continuous positive airway pressure.

    Two patients with the Pickwickian syndrome and with life-threatening sleep hypoxemia were treated with continuous positive airway pressure (CPAP) applied through the nares only during sleep periods. Each patient presented with severe daytime somnolence, disturbed sleep, nocturnal confusion, and daytime awake cardiorespiratory failure (PaCO2, 63 and 55 mmHg). Both patients demonstrated grossly abnormal breathing during sleep with severe sleep hypoxemia, the arterial oxyhemoglobin saturation (SaO2%) falling repetitively to levels below 50%. One patient had a hypoxemic convulsion during the initial sleep evaluation. Low levels (3.5 and 8.0 cm H2O) of continuous positive airway pressure, when applied via a comfortable nose mask, prevented occlusive apnea and obstructive hypopnea during sleep in both patients and maintained steady levels of arterial oxyhemoglobin saturation. There was rapid recovery of mental function and loss of cardiorespiratory failure within 3 days of treatment. After short-term treatment with nocturnal CPAP therapy (23 days and 35 days) both patients were able to sleep, unaided, without sleep-induced upper airway occlusion with arterial oxyhemoglobin levels sustained above 80%. We conclude that nasal CPAP therapy during sleep is an effective noninvasive therapy for patients with the Pickwickian syndrome, and may lead to a stable remission of the underlying severe disordered breathing in sleep.
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7/8. Therapeutic effects of tracheostomy in two cases of hypersomnia with respiratory disturbance during sleep.

    Recently the association of hypersomnia and respiratory insufficiency without lesion in the respiratory organ has attracted attention of many investigators. Obese patients with such a condition have been called the Pickwickian syndrome. In this report, two non-obese patients with a similar condition were presented, one with micrognathia and frequent apneic episodes during sleep, and the other with laryngeal stenosis due to paralysis of the bilateral laryngeal nerves and chronic laryngitis. tracheostomy had a prompt and long-lasting therapeutic effect to make their sleep stable and also to relieve their excessive daytime sleepiness. These findings suggest that the obstruction or stenosis of the upper airway during sleep disturbed their nocturnal sleep, and that their excessive daytime sleepiness was a phenomenon compensating for their disturbed nocturnal sleep.
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keywords = upper
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8/8. Polygraphic study of periodic breathing and hypersomnolence in a patient with severe hypothyroidism.

    A 67-year-old woman with primary hypothyroidism, who had Pickwickian syndrome as well, was studied electrophysiologically. A polygraphic study with simultaneous recordings of electroencephalography, actograms of the abdominal walls, the mentum and the anterior neck, and electromyogram of the intercostal muscles revealed a cyclic appearance of apnea in the sleep phase and gasping preceding arousal, which, together with macroglossia and sleep in the sitting position, suggested a cyclic obstruction of the upper airway. The obstruction appeared responsible for both alveolar hypoventilation and disturbance of consciousness.
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keywords = breathing, upper
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