TY - JOUR
T1 - Methylphenidate use in males with Duchenne muscular dystrophy and a comorbid attention deficit hyperactivity disorder
AU - Lionarons, Judith M.
AU - Hellebrekers, Danique M. J.
AU - Klinkenberg, Sylvia
AU - Faber, Catharina G.
AU - Vies, Johan S. H.
AU - Hendriksen, Jos G. M.
N1 - Funding Information:
The authors have stated that they had no interests which might be perceived as posing a conflict or bias. Dr Hendriksen received personal grants from Spieren voor Spieren Foundation (grant number SvS15- ) and Duchenne Parent Project Netherlands (grant 2016 on non-motor problems in Duchenne), which do not inappropriately influence this work.
Publisher Copyright:
© 2018 European Paediatric Neurology Society
PY - 2019/1
Y1 - 2019/1
N2 - Attention-deficit hyperactivity disorder (ADHD) is a common comorbidity in Duchenne muscular dystrophy (DMD). Until now, treatment with methylphenidate (MPH) has never been systematically assessed and described in this population. Our aim was to evaluate the effectiveness and safety of short acting MPH for learning problems in males with DMD and ADHD. Neuropsychological (cognition and behavior) and medical data of a sample of ten males (mean age = 8.1 years, range 6.3-9.8) with DMD and an ADHD diagnosis was retrospectively analyzed at baseline (TO; without MPH), short-term follow-up (T1; with MPH; mean interval T0-T1 = 8.3 months, range 4.3-15.6), and long-term follow-up (T2; mean interval T1-T2 = 23.1 months, range 2.6-77.7). An initial MPH dose of 5 mg/day was given on school mornings, with an increase of 2.5-5 mg/week depending on individual tolerance and treatment response, until a sufficiently effective dose was reached (range 0.2-0.6 mg/kg/day). At T1, results demonstrated an improvement in attention (i.e. concentration, impulsivity, and distractibility) in four patients. Suboptimal effects were reported in four patients, and no effects in two patients. At T2, seven patients showed considerable improvement in attention. No major side effects were reported. Overall, our data show that short acting MPH can be clinically effective for learning problems in males with DMD and ADHD, with regular cardiac follow-up, and close monitoring of side effects and neuropsychological effects. Furthermore, this underscores the importance of the use of validated cognitive and behavioral measurement tools with adequate sensitivity to objectively evaluate the effect of MPH. (C) 2018 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved.
AB - Attention-deficit hyperactivity disorder (ADHD) is a common comorbidity in Duchenne muscular dystrophy (DMD). Until now, treatment with methylphenidate (MPH) has never been systematically assessed and described in this population. Our aim was to evaluate the effectiveness and safety of short acting MPH for learning problems in males with DMD and ADHD. Neuropsychological (cognition and behavior) and medical data of a sample of ten males (mean age = 8.1 years, range 6.3-9.8) with DMD and an ADHD diagnosis was retrospectively analyzed at baseline (TO; without MPH), short-term follow-up (T1; with MPH; mean interval T0-T1 = 8.3 months, range 4.3-15.6), and long-term follow-up (T2; mean interval T1-T2 = 23.1 months, range 2.6-77.7). An initial MPH dose of 5 mg/day was given on school mornings, with an increase of 2.5-5 mg/week depending on individual tolerance and treatment response, until a sufficiently effective dose was reached (range 0.2-0.6 mg/kg/day). At T1, results demonstrated an improvement in attention (i.e. concentration, impulsivity, and distractibility) in four patients. Suboptimal effects were reported in four patients, and no effects in two patients. At T2, seven patients showed considerable improvement in attention. No major side effects were reported. Overall, our data show that short acting MPH can be clinically effective for learning problems in males with DMD and ADHD, with regular cardiac follow-up, and close monitoring of side effects and neuropsychological effects. Furthermore, this underscores the importance of the use of validated cognitive and behavioral measurement tools with adequate sensitivity to objectively evaluate the effect of MPH. (C) 2018 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved.
KW - Duchenne muscular dystrophy
KW - Attention-deficit hyperactivity disorder
KW - Neuropsychological evaluation
KW - Methylphenidate
KW - ADHD
KW - CHILDREN
KW - ADOLESCENTS
KW - MANAGEMENT
KW - DIAGNOSIS
KW - MELATONIN
KW - STIMULANT
KW - SAFETY
KW - SLEEP
KW - BRAIN
U2 - 10.1016/j.ejpn.2018.09.005
DO - 10.1016/j.ejpn.2018.09.005
M3 - Article
C2 - 30287260
SN - 1090-3798
VL - 23
SP - 152
EP - 157
JO - European Journal of Paediatric Neurology
JF - European Journal of Paediatric Neurology
IS - 1
ER -