Stimulant intolerance in children with Angelman syndrome with hyperactivity: a case series.
Journal
Psychiatric genetics
ISSN: 1473-5873
Titre abrégé: Psychiatr Genet
Pays: England
ID NLM: 9106748
Informations de publication
Date de publication:
01 04 2022
01 04 2022
Historique:
entrez:
29
3
2022
pubmed:
30
3
2022
medline:
6
5
2022
Statut:
ppublish
Résumé
Angelman syndrome is a neurogenetic disorder resulting from the loss of expression of the ubiquitin-protein ligase E3A gene on chromosome 15. Problematic behaviors including attention-deficit/hyperactivity disorder (ADHD) symptoms of hyperactivity, impulsivity and inattention are highly prevalent in Angelman syndrome. The efficacy, safety and tolerability of stimulant medications in children with Angelman syndrome for the treatment of ADHD symptoms have not been previously reported. We describe three boys with Angelman syndrome who were treated with open-label stimulant medications for ADHD symptoms. Stimulant medications were highly intolerable, and treatment had to be discontinued after limited dosing in all three cases due to marked increases in hyperactivity and impulsivity along with worsened distractibility. The findings of this study suggest that stimulant medications may be ineffective and poorly tolerated in children with Angelman syndrome.
Identifiants
pubmed: 35348128
doi: 10.1097/YPG.0000000000000307
pii: 00041444-202204000-00004
doi:
Substances chimiques
Central Nervous System Stimulants
0
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
80-86Informations de copyright
Copyright © 2021 Wolters Kluwer Health, Inc. All rights reserved.
Références
Albrecht U, Sutcliffe JS, Cattanach BM, Beechey CV, Armstrong D, Eichele G, Beaudet AL (1997). Imprinted expression of the murine Angelman syndrome gene, Ube3a, in hippocampal and Purkinje neurons. Nat Genet 17:75–78.
American Psychiatric Association: Diagnostic and Statistical Manual of Mental Disorders, 5th ed, 2013. American Psychiatric Association, Arlington, VA.
Arnsten AF, Scahill L, Findling RL (2007). Alpha2-Adrenergic receptor agonists for the treatment of attention-deficit/hyperactivity disorder: emerging concepts from new data. J Child Adolesc Psychopharmacol 17:393–406.
Berry RJ, Leitner RP, Clarke AR, Einfeld SL. (2005). Behavioral aspects of Angelman syndrome: a case control study. Am J Med Genet 132A:8–12.
Bird LM (2014). Angelman syndrome: review of clinical and molecular aspects. Appl Clin Genet 7:93–104.
Buiting K, Clayton-Smith J, Driscoll DJ, Gillessen-Kaesbach G, Kanber D, Schwinger E, Williams C, Horsthemke B. (2015). Clinical utility gene card for: Angelman Syndrome. Eur J Hum Genet 23:e1–e3.
Buntinx IM, Hennekam RC, Brouwer OF, Stroink H, Beuten J, Mangelschots K, Fryns JP (1995). Clinical profile of Angelman syndrome at different ages. Am J Med Genet 56:176–183.
Clarke DJ, Marston G (2000). Problem behaviors associated with 15q- Angelman syndrome. Am J Ment Retard 105:25–31.
Clayton-Smith J. (2001). Angelman syndrome: evolution of the phenotype in adolescents and adults. Dev Med Child Neurol 43:476–480.
Falkowitz S, Akbar N, Greenhill L. (2017). Clinical Manual of Child and Adolescent Psychopharmacology, 3rd ed. Arlington, VA.
Feldman ME, Charach A, Bélanger SA (2018). ADHD in children and youth: Part 2-treatment. Paediatr Child Health 23:462–472.
Fernández-Jaén A, Fernández-Mayoralas DM, Calleja Pérez B, Muñoz Jareño N, Campos Díaz MdelR (2010). Atomoxetine for attention deficit hyperactivity disorder in mental retardation. Pediatr Neurol 43:341–347.
Garnock-Jones KP, Keating GM (2009). Atomoxetine: a review of its use in attention-deficit hyperactivity disorder in children and adolescents. Paediatr Drugs 11:203–226.
Harfterkamp M, van de Loo-Neus G., Minderaa RB, van der Gaag R, Escobar R, Schacht A., Pamulapati S., Buitelaar JK, Hoekstra PJ (2012). A randomized double-blind study of atomoxetine versus placebo for attention-deficit/hyperactivitry disorder symptoms in children with autism spectrum disorder. J Am Acad Child Adoelsc Psychiatry 51;733–741.
Keary CJ, Mullett JE, Nowinski L, Wagner K, Walsh B, Saro H, Erhabor G, Thibert RL, McDougle C, Ravichandran CT. (2021). Parent description of anxiety in Angelman syndrome. J. Autism Dev Disord Submitted. https://doi.org/10.1007/s10803-021-05238-8 .
doi: 10.1007/s10803-021-05238-8
Larson AM, Shinnick JE, Shaaya EA, Thiele EA, Thibert RL (2015). Angelman syndrome in adulthood. Am J Med Genet A 167A:331–344.
Margolis SS, Sell GL, Zbinden MA, Bird LM (2015). Angelman Syndrome. Neurotherapeutics 12:641–650.
Michelson D, Adler L, Spencer T, Reimherr FW, West SA, Allen AJ, Kelsey D, Wernicke J, Deitrich A, Milton D (2003). Atomoxetine in adults with ADHD: two randomized placebo-controlled studies. Biol Psychiatry 53:112–120.
Pereira JA, Ravichandran CT, Mullett J, McDougle CJ, Keary CJ (2020). Characterization of sleep habits and medication outcomes for sleep disturbance in children and adults with Angelman syndrome. Am J Med Genet A 182:1913–1922.
Research Units on Pediatric Psychopharmacology Autism Network. (2005). Randomized, controlled, crossover trial of methylphenidate in pervasive developmental disorders with hyperactivity. Arch Gen Psychiatry 62:1266–1274.
Sadhwani A, Willen JM, LaVallee N, Stepanians M, Miller H, Peters SU, et al. (2019). Maladaptive behaviors in individuals with Angelman syndrome. Am J Med Genet A 179:983–992.
Scahill L., McCracken JT., King BH., Rockhill C., Shah B., Politte L., et al., Research Units of Pediatric Psychopharmacology Autism Network. (2015). Extended-release gaufnacine for hyperactivity in children with autism spectrum disorder. Am J Psychiatry 172:1197–1206.
Schachter HM, Pham B, King J, Langford S, Moher D (2001). How efficacious and safe is short-acting methylphenidate for the treatment of attention-deficit disorder in children and adolescents? A meta-analysis. CMAJ 165:1475–1488.
Summers JA, Allison DB, Lynch PS, Sandler L (1995). Behaviour problems in Angelman syndrome. J Intellect Disabil Res 39 (Pt 2):97–106.
Thibert RL, Larson AM, Hsieh DT, Raby AR, Thiele EA (2013). Neurologic manifestations of Angelman syndrome. Pediatr Neurol 48:271–279.
Wheeler AC, Sacco P, Cabo R (2017). Unmet clinical needs and burden in Angelman syndrome: a review of the literature. Orphanet J Rare Dis 12:164.
Wilens TE, Spencer TJ (2000). The stimulants revisited. Child Adolesc Psychiatr Clin N Am 9:573–603, viii.
Williams CA, Beaudet AL, Clayton-Smith J, Knoll JH, Kyllerman M, Laan LA, et al. (2006). Angelman syndrome 2005: updated consensus for diagnostic criteria. Am J Med Genet A 140:413–418.
Yashiro K, Riday TT, Condon KH, Roberts AC, Bernardo DR, Prakash R, et al. (2009). Ube3a is required for experience-dependent maturation of the neocortex. Nat Neurosci 12:777–783.