Severe Hypersomnolence After Pituitary/Hypothalamic Surgery in Adolescents: Clinical Characteristics and Potential Mechanisms

Snow, Ayelet B.; Gozal, Evelyne; Malhotra, Atul; Tiosano, Dov; Perlman, Rina; Vega, Carolina; Shahar, Eli; Gozal, David; Hochberg, Zèev; Pillar, Giora

doi:10.1542/peds.110.6.e74

Cited by 89 publications

(69 citation statements)

References 50 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…We have measured orexin in extremely sleepy patients after surgical removal of a craniopharyngioma tumor. Although we expected to find low levels, which would explain the severe sleepiness, we found normal and even high levels, which would explain the constant increase in body weight we observed in our patients (111).…”

Section: Hormonal Changes -It Is Well Established That Sleep Deprivatcontrasting

confidence: 45%

Abdominal Fat and Sleep Apnea

Pillar

Shehadeh²

2008

Diabetes Care

Self Cite

278

158

View full text Add to dashboard Cite

Obstructive sleep apnea (OSA) syndrome is a disorder characterized by repetitive episodes of upper airway obstruction that occur during sleep. Associated features include loud snoring, fragmented sleep, repetitive hypoxemia/hypercapnia, daytime sleepiness, and cardiovascular complications. The prevalence of OSA is 2-3% and 4 -5% in middle-aged women and men, respectively. The prevalence of OSA among obese patients exceeds 30%, reaching as high as 50 -98% in the morbidly obese population. Obesity is probably the most important risk factor for the development of OSA. Some 60 -90% of adults with OSA are overweight, and the relative risk of OSA in obesity (BMI Ͼ29 kg/m 2 ) is Ն10. Numerous studies have shown the development or worsening of OSA with increasing weight, as opposed to substantial improvement with weight reduction. There are several mechanisms responsible for the increased risk of OSA with obesity. These include reduced pharyngeal lumen size due to fatty tissue within the airway or in its lateral walls, decreased upper airway muscle protective force due to fatty deposits in the muscle, and reduced upper airway size secondary to mass effect of the large abdomen on the chest wall and tracheal traction. These mechanisms emphasize the great importance of fat accumulated in the abdomen and neck regions compared with the peripheral one. It is the abdomen much more than the thighs that affect the upper airway size and function. Hence, obesity is associated with increased upper airway collapsibility (even in nonapneic subjects), with dramatic improvement after weight reduction. Conversely, OSA may itself predispose individuals to worsening obesity because of sleep deprivation, daytime somnolence, and disrupted metabolism. OSA is associated with increased sympathetic activation, sleep fragmentation, ineffective sleep, and insulin resistance, potentially leading to diabetes and aggravation of obesity. Furthermore, OSA may be associated with changes in leptin, ghrelin, and orexin levels; increased appetite and caloric intake; and again exacerbating obesity. Thus, it appears that obesity and OSA form a vicious cycle where each results in worsening of the other. Diabetes Care 31 (Suppl. 2):S303-S309, 2008A lthough interest in sleep and dreams has existed since the dawn of history, it has only been in the last 25-35 years that physicians have recognized the importance of sleep disorders, and only in the last 20 -25 years that sleep laboratory services have been commonly available. Over the recent years, several categories of sleep disorders were recognized, with special interest in breathing disorders during sleep. It is quite clear now that quantity and quality of life deteriorates secondary to sleep disordered breathing, with complex associations between sleep-disordered breathing and obesity, cardiac diseases, stroke, and diabetes.Obstructive sleep apnea (OSA) syndrome is characterized by the recurrent collapse of the pharyngeal airway during sleep, which generally requires arousal to reestablish airway patency and...

show abstract

Section: Hormonal Changes -It Is Well Established That Sleep Deprivatcontrasting

confidence: 45%

Abdominal Fat and Sleep Apnea

Pillar

Shehadeh²

2008

Diabetes Care

Self Cite

278

158

View full text Add to dashboard Cite

show abstract

“…Secondary narcolepsy, more correctly called symptomatic narcolepsy, has rarely been reported in humans and can result from brain tumors, stroke, demyelination, traumatic injury, and encephalitis 11, 12, 13, 14, 15, 16. Between 1969 and 2005, 116 human cases of symptomatic narcolepsy‐cataplexy were reported.…”

mentioning

confidence: 99%

Pituitary Macrotumor Causing Narcolepsy‐Cataplexy in a Dachshund

Schmid

Hodshon

Olin

et al. 2017

Veterinary Internal Medicne

View full text Add to dashboard Cite

Familial narcolepsy secondary to breed‐specific mutations in the hypocretin receptor 2 gene and sporadic narcolepsy associated with hypocretin ligand deficiencies occur in dogs. In this report, a pituitary mass is described as a unique cause of narcolepsy‐cataplexy in a dog. A 6‐year‐old male neutered Dachshund had presented for acute onset of feeding‐induced cataplexy and was found to have a pituitary macrotumor on magnetic resonance imaging (MRI). Cerebral spinal fluid hypocretin‐1 levels were normal, indicating that tumor effect on the ventral lateral nucleus of the hypothalamus was not the cause of the dog's narcolepsy‐cataplexy. The dog was also negative for the hypocretin receptor 2 gene mutation associated with narcolepsy in Dachshunds, ruling out familial narcolepsy. The Dachshund underwent stereotactic radiotherapy (SRT), which resulted in reduction in the mass and coincident resolution of the cataplectic attacks. Nine months after SRT, the dog developed clinical hyperadrenocorticism, which was successfully managed with trilostane. These findings suggest that disruptions in downstream signaling of hypocretin secondary to an intracranial mass effect might result in narcolepsy‐cataplexy in dogs and that brain MRI should be strongly considered in sporadic cases of narcolepsy‐cataplexy.

show abstract

“…typical cataplexy clearly triggered by emotion, and the frequent appearance of SOREMP on MSLT) and extremely long sleep episodes during both the nighttime and daytime. Although remarkable long sleep episodes are not frequently observed in narcolepsy cases except for atypical cases (narcolepsy with long sleep time) (8), this case met the ICSD-2 criteria of secondary narcolepsy (6).…”

Section: Discussionmentioning

confidence: 63%

Possible Mechanism of Secondary Narcolepsy with a Long Sleep Time Following Surgery for Craniopharyngioma

et al. 2012

View full text Add to dashboard Cite

A 19-year-old woman suffered from severe excessive daytime sleepiness accompanied with long sleep episodes both in the daytime and nighttime and frequent episodes of cataplexy shortly after the removal of craniopharyngioma in the intrasellar space. Multiple sleep latency test showed a typical finding of narcolepsy, and cerebrospinal fluid orexin concentration was below the narcolepsy cut-off value. MRI-tractography showed a clear lack of neuronal fiber connections from the hypothalamus to the frontal lobe. SPECT using 123 I-IMP showed frontal hypoperfusion. These connection damages could have been responsible for the occurrence of narcolepsy-like symptoms and long daytime sleep episodes in this case.

show abstract

Severe Hypersomnolence After Pituitary/Hypothalamic Surgery in Adolescents: Clinical Characteristics and Potential Mechanisms

Cited by 89 publications

References 50 publications

Abdominal Fat and Sleep Apnea

Abdominal Fat and Sleep Apnea

Pituitary Macrotumor Causing Narcolepsy‐Cataplexy in a Dachshund

Possible Mechanism of Secondary Narcolepsy with a Long Sleep Time Following Surgery for Craniopharyngioma

Contact Info

Product

Resources

About