Aripiprazole treatment for temper outbursts in Prader-Willi syndrome

Orphanet J Rare Dis. 2022 Aug 26;17(1):324. doi: 10.1186/s13023-022-02470-y.

Abstract

Background: Prader-Willi syndrome (PWS) is a rare neurodevelopmental disorder based on a loss of paternally expressed genes in chromosome segment 15q11-13. Behavioral traits such as temper outbursts, stereotypic, and ritualistic behavior, as well as an increased risk of psychosis accompany the syndrome, representing a major issue in the treatment of adults with PWS. Up to now, no treatment guideline for these conditions in PWS exist. This study aimed to retrospectively analyze the effect and adverse effects of treatment with aripiprazole for temper outbursts in 10 adults with PWS.

Results: Aripiprazole was prescribed for temper outbursts (n = 10). Treatment outcome was assessed using the Clinical Global Impression-Severity (CGI-S) and -Improvement Scale (CGI-I). Treatment success (CGI-I < 3) was observed in 70% of cases, with adverse effects from mild to partly serious extent in 60% of cases. The major adverse effect observed was increased daytime sleepiness. In total, 50% of the individuals were treated successfully for temper outbursts. The BMI did not change significantly in the successfully treated group after 6 months of treatment.

Conclusions: Aripiprazole can be a treatment option for temper outbursts in people with PWS. Although a high rate of side effects was detected, their severity led to discontinuation in only 20% of the cases. Furthermore, the absence of weight gain makes aripiprazole interesting especially for the PWS population.

Keywords: Aripiprazole; Mental health; Pharmacotherapy; Prader–Willi syndrome; Temper outbursts.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aripiprazole
  • Humans
  • Prader-Willi Syndrome*
  • Retrospective Studies
  • Treatment Outcome

Substances

  • Aripiprazole