S. Esnault-lavandier et D. Mabin, Effects of clomipramine on excessive daytime sleepiness and respiratory disturbances in a case of Prader-Willi syndrome., NEUROP CLIN, 28(6), 1998, pp. 521-525
The association of Prader-Willi-syndrome with breathing disturbances such a
s sleep apnea syndrome and/or hypoxemia during REM sleep, REM sleep abnorma
lities and excessive daytime sleepiness is well known. We report the case o
f an 11-year-old boy who presented with Prader-Willi syndrome, obesity (bod
y mass index [BMI] = 45.6), severe obstructive sleep apnea syndrome and sig
nificant daytime sleepiness on multiple sleep latency test. Behavioral diso
rders did not allowed the use of continuous positive pressure in this patie
nt. Therefore, clomipramine (20 nag per day) was administered Sleep examina
tion over 8 months showed: slight weight loss (BMI = 44.4), persistence of
severe obstructive sleep apnea syndrome, slight improvement in nocturnal hy
poxemia, and disappearance of excessive daytime drowsiness with mean sleep
latency of IS min 37 s (less than 2 min before treatment) and no diurnal RE
M sleep periods. However, clomipramine had no effect on hyperphagia. (C) 19
98 Elsevier, Paris.