Acebutolol and Immediate Release Melatonin in Treatment of Smith-Magenis Syndrome

Abstract

Sleep disturbance occurs in Smith-Magenis syndrome (SMS) as part of the phenotype. It appears that this disturbance lies primarily in an inversion of the circadian rhythm of melatonin. We assessed whether the use of melatonin and acebutolol together would benefit a patient with SMS with regard to sleep and behavior. Our patient entered a four-way crossover, double-blind, double dummy, placebo-controlled n-of-1 trial, using immediate release melatonin, acebutolol and placebo. The duration of the study was 24 consecutive nights. The primary outcome measure was total nocturnal sleep. Placebo/placebo showed the longest total duration of sleep, 10.62 hours (95% CI: 9.88 - 11.36) as compared to melatonin and acebutolol together with 10.25 (95% CI: 9.40 - 11.11). There were no significant differences found between the secondary outcome measures in particular behavior or global satisfaction. Our results suggest there is no benefit to sleep or subsequent behavior from adding acebutolol and/or immediate release melatonin in patients with SMS.




J Med Cases. 2015;6(4):176-178
doi: http://dx.doi.org/10.14740/jmc2102w