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Obese Prader-Willi syndrome patients prone to sudden unexpected death.
Obese Prader-Willi syndrome patients prone to sudden unexpected death. -- Posted by Sweet Zombie Jesus! on 06-07-05 06:42
Patients with Prader-Willi syndrome (PWS) are recognized to have a
tendency of sudden, unexpected death (SED), but its exact cause is
unknown because of paucity of such case reports. Since growth hormone
(GH) treatment was applied to PWS patients worldwide, several cases of
death have been reported. However, whether the therapy is directly
related to their SED remains unknown, too. We collected 13 deceased PWS
patients (Group A, aged 9 months to 34 years) who had never received GH
therapy, and seven deceased patients (Group B, all boys aged 0.7-15
years) having received the therapy from the registration in
PWS-patient-support associations and from the literature, respectively.
We then compared the cause of SED between the two groups.
Irrespective of GH therapy, SED of infants under age 1 year was
associated with milk aspiration or hypothalamic dysregulation of
respiration, while SED of patients in early childhood or adolescence
occurred at sleeping in association with preceding viral infections. In
contrast, SED of four adult (> 20 years of age) patients who never
received GH therapy was associated with complications, such as leg
cellulites and pulmonary embolism, secondary to massive obesity and
diabetes mellitus (DM). Two Group-B patients (aged 14 and 20 years)
without any obesity-related or diabetes-related complications died of
drowning in a bath tub, and their drowning death could be related to
poor respiratory control.
These findings indicated that the cause of SED is not essentially
different between PWS patients with and without GH treatment. Deceased
PWS patients may have had underlying respiratory dysregulation and
hypothalamic dysfunction, and GH therapy might have led to certain
obstructive respiratory disturbances that exacerbated the respiratory
conditions. This will call clinicians' attention when using GH in PWS
patients, for example, careful determination of the dose of GH and
careful monitoring of patient's respiratory conditions, especially in
male obese patients with respiratory problems.
Am J Med Genet A. 2005 Jun 3;Cause of sudden, unexpected death of
Prader-Willi syndrome patients with or without growth hormone
treatment.Nagai T, Obata K, Tonoki H, Temma S, Murakami N, Katada Y,
Yoshino A, Sakazume S, Takahashi E, Sakuta R, Niikawa N.
Department of Pediatrics, Dokkyo University School of Medicine
Koshigaya Hospital, Saitama, Japan.
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