A prospective open-label study of aripiprazole in fragile X syndrome

Craig A. Erickson, Kimberly A. Stigler, Logan K. Wink, Jennifer E. Mullett, Arlene Kohn, David J. Posey, Christopher J. McDougle

Research output: Contribution to journalArticle

39 Citations (Scopus)

Abstract

Rationale: Fragile X syndrome (FXS) is the most common inherited form of developmental disability and most common single gene cause of autism. Persons with FXS frequently exhibit irritable behavior marked by aggression, self-injury, and severe tantrums. Despite frequent clinical use of atypical antipsychotic drugs to target this behavioral cluster, no systematic trials to date have assessed the efficacy and safety of these drugs in persons with FXS. Methods: We conducted a prospective open-label 12-week trial of aripiprazole in 12 persons aged 6-25 years (mean age, 14.3 years) with FXS who were free of concomitant psychoactive drugs. Results: Aripiprazole use (mean dose, 9.8 mg/day) was associated with treatment response (defined by a Clinical Global Impressions-Improvement scale score of much improved or very much improved and a ≥25% improvement on the Aberrant Behavior Checklist-Irritability subscale) in 10 of 12 (87%) persons. Two individuals (13%) discontinued aripiprazole prior to study completion due to adverse events. One discontinuation was due to akathisia, mild drooling, and mild tiredness and the other due to moderate tiredness and moderate drooling. No significant changes in vital signs including weight or laboratory measures occurred during treatment with aripiprazole. Conclusions: Aripiprazole was generally safe and well tolerated and was associated with significant improvement in irritable behavior. Given these findings, a double-blind, placebo-controlled study of aripiprazole in FXS is warranted.

Original languageEnglish
Pages (from-to)85-90
Number of pages6
JournalPsychopharmacology
Volume216
Issue number1
DOIs
StatePublished - Jul 2011

Fingerprint

Fragile X Syndrome
Sialorrhea
Psychomotor Agitation
Developmental Disabilities
Vital Signs
Psychotropic Drugs
Autistic Disorder
Checklist
Aggression
Antipsychotic Agents
Aripiprazole
Placebos
Safety
Weights and Measures
Wounds and Injuries
Therapeutics
Pharmaceutical Preparations
Genes

Keywords

  • Aripiprazole
  • Fragile X syndrome
  • Irritability

ASJC Scopus subject areas

  • Pharmacology

Cite this

Erickson, C. A., Stigler, K. A., Wink, L. K., Mullett, J. E., Kohn, A., Posey, D. J., & McDougle, C. J. (2011). A prospective open-label study of aripiprazole in fragile X syndrome. Psychopharmacology, 216(1), 85-90. https://doi.org/10.1007/s00213-011-2194-7

A prospective open-label study of aripiprazole in fragile X syndrome. / Erickson, Craig A.; Stigler, Kimberly A.; Wink, Logan K.; Mullett, Jennifer E.; Kohn, Arlene; Posey, David J.; McDougle, Christopher J.

In: Psychopharmacology, Vol. 216, No. 1, 07.2011, p. 85-90.

Research output: Contribution to journalArticle

Erickson, CA, Stigler, KA, Wink, LK, Mullett, JE, Kohn, A, Posey, DJ & McDougle, CJ 2011, 'A prospective open-label study of aripiprazole in fragile X syndrome', Psychopharmacology, vol. 216, no. 1, pp. 85-90. https://doi.org/10.1007/s00213-011-2194-7
Erickson CA, Stigler KA, Wink LK, Mullett JE, Kohn A, Posey DJ et al. A prospective open-label study of aripiprazole in fragile X syndrome. Psychopharmacology. 2011 Jul;216(1):85-90. https://doi.org/10.1007/s00213-011-2194-7
Erickson, Craig A. ; Stigler, Kimberly A. ; Wink, Logan K. ; Mullett, Jennifer E. ; Kohn, Arlene ; Posey, David J. ; McDougle, Christopher J. / A prospective open-label study of aripiprazole in fragile X syndrome. In: Psychopharmacology. 2011 ; Vol. 216, No. 1. pp. 85-90.
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