The goal of this study was to assess the effectiveness of risperidone monoantipsychotic therapy targeting irritability in patients with Fragile X syndrome (FXS) in a naturalistic outpatient clinical setting.
Methods:
We examined the use of risperidone, predominantly in combination with other nonantipsychotic psychotropic agents, targeting irritability in 21 male patients with FXS with a retrospective analysis of a prospectively collected large developmental disabilities-specific treatment database. Mean age at start of treatment, treatment duration, final dose, body mass index (BMI), and Clinical Global Impressions-Improvement (CGI-I) Scale score at final visit were determined, and changes with treatment were analyzed using paired t-tests.
Results:
Mean age at start of treatment was 14.0 years. The final mean dose of risperidone was 2.5 mg/day. The mean duration of treatment was 22 months. Seven (33.33%) participants were considered treatment responders based on the CGI-I. Change in BMI between initiation and cessation of treatment episode was not significant, however, these data were only available for a subset (n = 11) of patients.
Conclusions:
Risperidone may be effective in the treatment of irritability in males with FXS. The overall effectiveness of monoantipsychotic treatment with risperidone was limited in this study compared with previous published reports; however, this may be the result of differences in outcome measures as well as a reflection of the level of functioning and severity of irritability in this sample.
Get full access to this article
View all access options for this article.
References
1.
AdlerBA, WinkLK, EarlyM, ShafferR, MinshawiN, McDougleCJ, EricksonCA: Drug-refractory aggression, self-injurious behavior, and severe tantrums in autism spectrum disorders: A chart review study. Autism, 19:102–106, 2015.
2.
AmanMG, SinghNN, StewartAW, FieldCJ: The aberrant behavior checklist: A behavior rating scale for the assessment of treatment effects. Am J Ment Defic, 89:485–491, 1985a.
3.
AmanMG, SinghNN, StewartAW, FieldCJ: Psychometric characteristics of the aberrant behavior checklist. Am J Ment Defic, 89:492–502, 1985b.
4.
AmariaRN, BilleisenLL, HagermanR: Medication use in fragile X syndrome. Ment Health Aspects Dev Disabil, 4:143–147, 2001.
5.
American Psychiatric Association: Diagnostic and Statistical Manual of Mental Disorders (4th ed.) text revision ed. Washington, DC, American Psychiatric Association, 2000.
6.
BaileyDBJr., HattonDD, TassoneF, SkinnerM, TaylorAK: Variability in FMRP and early development in males with fragile X syndrome. Am J Ment Retard, 106:16–27, 2001.
7.
BaileyDBJr., MesibovGB, HattonDD, ClarkRD, RobertsJE, MayhewL: Autistic behavior in young boys with fragile X syndrome. J Autism Dev Disord, 28:499–508, 1998.
8.
BaileyDBJr., RaspaM, OlmstedM, HolidayDB: Co-occurring conditions associated with FMR1 gene variations: Findings from a national parent survey. Am J Med Genet A, 146A:2060–2069, 2008.
9.
BaileyDB, RaspaM, OlmstedMG: Using a parent survey to advance knowledge about the nature and consequences of fragile X syndrome. Am J Intellect Dev Disabil, 115:447–460, 2010.
10.
BaileyDB, SkinnerD, HattonD, RobertsJ: Family experiences and factors associated with the diagnosis of fragile X syndrome. J Dev Behav Pediatr, 21:315–321, 2000.
11.
BaranekGT, RobertsJE, DavidFJ, SiderisJ, MirrettPL, HattonDD, BaileyDBJr.: Developmental trajectories and correlates of sensory processing in young boys with fragile X syndrome. Phys Occup Ther Pediatr, 28:79–98, 2008.
12.
BaumgardnerTL, ReissAL, FreundLS, AbramsMT: Specification of the neurobehavioral phenotype in males with fragile X syndrome. Pediatrics, 95:744–752, 1995.
Berry-KravisE, PotanosK: Psychopharmacology in fragile X syndrome—present and future. Ment Retard Dev Disabil Res Rev, 10:42–48, 2004.
15.
Berry-KravisE, SumisA, HerveyC, MathurS: Clinic-based retrospective analysis of psychopharmacology for behavior in fragile x syndrome. Int J Pediatr, 2012:843016, 2012.
16.
BudimirovicDB, KaufmannWE: What can we learn about autism from studying fragile X syndrome?. Dev Neurosci, 33:379–394, 2011.
CornishK, SudhalterV, TurkJ: Attention and language in fragile X. Ment Retard Dev Disabil Res Rev, 10:11–16, 2004.
19.
DavenportMH, SchaeferTL, FriedmannKJ, FitzpatrickSE, EricksonCA: Pharmacotherapy for fragile X syndrome: Progress to date. Drugs, 76:431–445, 2016.
20.
EricksonCA, StiglerKA, PoseyDJ, McDougleCJ: Managing maladaptive behaviors in fragile X patients. Curr Psychiatry, 5:80, 2006.
21.
EricksonCA, StiglerKA, WinkLK, MullettJE, KohnA, PoseyDJ, McDougleCJ: A prospective open-label study of aripiprazole in fragile X syndrome. Psychopharmacology (Berl), 216:85–90, 2011.
22.
GuyW, National Institute of Mental H, Psychopharmacology Research B, Early Clinical Drug Evaluation P: ECDEU assessment manual for psychopharmacology. Rockville, Md.: U.S. Dept. of Health, Education, and Welfare, Public Health Service, Alcohol, Drug Abuse, and Mental Health Administration, National Institute of Mental Health, Psychopharmacology Research Branch, Division of Extramural Research Programs, 1976.
23.
HagermanRJ, Berry-KravisE, KaufmannWE, OnoMY, TartagliaN, LachiewiczA, KronkR, DelahuntyC, HesslD, VisootsakJ, PickerJ, GaneL, TranfagliaM: Advances in the treatment of fragile X syndrome. Pediatrics, 123:378–390, 2009.
24.
HagermanRJ, JacksonAW3rd, LevitasA, RimlandB, BradenM: An analysis of autism in fifty males with the fragile X syndrome. Am J Med Genet, 23:359–374, 1986.
25.
HattonDD, BaileyDB, RobertsJP, SkinnerM, MayhewL, ClarkRD, WaringE, RobertsJE: Early intervention services for young boys with fragile X syndrome. J Early Intervent, 23:235–251, 2000.
26.
HattonDD, HooperSR, BaileyDB, SkinnerML, SullivanKM, WheelerA: Problem behavior in boys with fragile X syndrome. Am J Med Genet, 108:105–116, 2002.
27.
HesslD, TassoneF, CordeiroL, KoldewynK, McCormickC, GreenC, WegelinJ, YuhasJ, HagermanRJ: Brief report: Aggression and stereotypic behavior in males with fragile X syndrome—moderating secondary genes in a “single gene” disorder. J Autism Dev Disord, 38:184–189, 2008.
NewmanI, LeaderG, ChenJL, MannionA: An analysis of challenging behavior, comorbid psychopathology, and attention-deficit/hyperactivity disorder in fragile X syndrome. Res Dev Disabil, 38:7–17, 2015.
31.
SansoneSM, WidamanKF, HallSS, ReissAL, LightbodyA, KaufmannWE, Berry-KravisE, LachiewiczA, BrownEC, HesslD: Psychometric study of the aberrant behavior checklist in fragile X syndrome and implications for targeted treatment. J Autism Dev Disord, 42:1377–1392, 2012.
32.
SchaeferTL, DavenportMH, EricksonCA: Emerging pharmacologic treatment options for fragile X syndrome. Appl Clin Genet, 8:75–93, 2015.
33.
SchneiderA, HagermanRJ, HesslD: Fragile X syndrome—from genes to cognition. Dev Disabil Res Rev, 15:333–342, 2009.
34.
SymonsFJ, ByiersBJ, RaspaM, BishopE, BaileyDB: Self-injurious behavior and fragile X syndrome: Findings from the national fragile X survey. Am J Intellect Dev Disabil, 115:473–481, 2010.
35.
SymonsFJ, ClarkRD, HattonDD, SkinnerM, BaileyDBJr.: Self-injurious behavior in young boys with fragile X syndrome. Am J Med Genet A, 118A:115–121, 2003.
36.
WheelerAC, RaspaM, BishopE, BaileyDBJr.: Aggression in fragile X syndrome. J Intellect Disabil Res, 60:113–125, 2016.
