Research into the (Cost-) effectiveness of the ketogenic diet among children and adolescents with intractable epilepsy: design of a randomized controlled trial.
BACKGROUND
Epilepsy is a neurological disorder, characterized by recurrent unprovoked seizures which have a high impact on the individual as well as on society as a whole. In addition to the economic burden, epilepsy imposes a substantial burden on the patients and their surroundings. Patients with uncontrolled epilepsy depend heavily on informal care and on health care professionals. About 30% of patients suffer from drug-resistant epilepsy. The ketogenic diet can be a treatment of last resort, especially for children. The beneficial effect of the ketogenic diet has been proven, but information is lacking about its cost-effectiveness. In the current study we will evaluate the (cost-) effectiveness of the ketogenic diet in children and adolescents with intractable epilepsy.


METHODS
In a RCT we will compare the ketogenic diet with usual care. Embedded in this RCT will be a trial-based and model-based economic evaluation, looking from a societal perspective at the cost-effectiveness and cost-utility of the ketogenic diet versus usual care. Fifty children and adolescents (aged 1-18) with intractable epilepsy will be screened for eligibility before randomization into the intervention or the usual care group. The primary outcome measure is the proportion of children with a 50% or more reduction in seizure frequency. Secondary outcomes include seizure severity, side effects/complaints, neurocognitive, socio-emotional functioning, and quality of life. Costs and productivity losses will be assessed continuously by a prospective diary and a retrospective questionnaire. Measurements will take place during consults at baseline, at 6 weeks and at 4 months after the baseline period, and 3, 6, 9 and 12 months follow-up after the 4 months consult.


CONCLUSIONS
The proposed research project will be the first study to provide data about the cost-effectiveness of the ketogenic diet for children and adolescents with intractable epilepsy, in comparison with usual care. It is anticipated that positive results in (cost-) effectiveness of the proposed intervention will contribute to the improvement of treatment for epilepsy in children and adolescents and will lead to a smaller burden to society.
Care and Public Health Research Institute (CAPHRI), Maastricht University, Maastricht, The Netherlands. Reina.dekinderen@maastrichtuniversity.nl
Reina J A
RJ
de Kinderen
Danielle A J E
DA
Lambrechts
Debby
D
Postulart
Alfons G H
AG
Kessels
Jos G M
JG
Hendriksen
Albert P
AP
Aldenkamp
Silvia M A A
SM
Evers
Marian H J M
MH
Majoie
D000293:Adolescent; D002648:Child; D002675:Child, Preschool; D002985:Clinical Protocols; D017281:Cost of Illness; D003362:Cost-Benefit Analysis / Q000379:methods*; D055423:Diet, Ketogenic / Q000191:economics / Q000379:methods*; D004827:Epilepsy / Q000178:diet therapy* / Q000191:economics; D006801:Humans; D007223:Infant; D011569:Psychiatric Status Rating Scales; D011788:Quality of Life; D016032:Randomized Controlled Trials as Topic / Q000191:economics / Q000379:methods*; D012720:Severity of Illness Index
Epilepsia. 2009 Feb;50(2):304-17
18823325
Qual Life Res. 2000;9(8):961-72
11284215
Stat Med. 2000 Dec 15;19(23):3219-36
11113956
Health Econ. 1996 Mar-Apr;5(2):141-54
8733106
Health Econ. 2005 May;14(5):487-96
15497198
Expert Rev Pharmacoecon Outcomes Res. 2005 Feb;5(1):23-8
19807557
Epilepsy Res. 2006 Feb;68(2):145-80
16523530
Epilepsia. 2005 Sep;46(9):1539-44
16146451
Epilepsia. 1996 Jun;37(6):563-71
8641234
Epilepsia. 2005 Feb;46(2):272-9
15679508
Pediatr Neurol. 2006 Jul;35(1):1-5
16814077
J Med Assoc Thai. 2001 Jul;84(7):1027-32
11759962
Epilepsy Res. 2002 Feb;48(3):221-7
11904241
Nutr Clin Pract. 2008 Dec-2009 Jan;23(6):589-96
19033218
Epilepsia. 2005 Oct;46(10):1684-6
16190943
Dev Med Child Neurol. 1997 Aug;39(8):521-6
9295847
Neurologist. 2005 May;11(3):161-70
15860138
Ned Tijdschr Geneeskd. 2005 Jul 9;149(28):1574-8
16038162
Pediatr Neurol. 2001 Sep;25(3):208-12
11587874
J Clin Epidemiol. 2000 Jul;53(7):688-95
10941945
J Behav Ther Exp Psychiatry. 2000 Jun;31(2):73-86
11132119
Epilepsia. 2007 Dec;48(12):2224-33
18088267
Pediatrics. 2000 Apr;105(4):E46
10742367
Epilepsia. 2002;43 Suppl 6:21-5
12190974
Pediatrics. 2002 May;109(5):780-3
11986436
Health Econ. 1997 Jul-Aug;6(4):327-40
9285227
Epilepsia. 2006 Feb;47(2):421-4
16499770
Pharmacoeconomics. 2008;26(6):463-76
18489198
Epilepsia. 1990;31 Suppl 4:S35-40
2279481
Epilepsia. 2005 Apr;46(4):470-2
15816939
Lancet Neurol. 2008 Jun;7(6):500-6
18456557
Arch Neurol. 1998 Nov;55(11):1433-7
9823827
Cochrane Database Syst Rev. 2003;(3):CD001903
12917915
Pediatrics. 1998 Dec;102(6):1358-63
9832569
J Dev Behav Pediatr. 1990 Jun;11(3):116-21
2365832
Health Policy. 1990 Dec;16(3):199-208
10109801
Eur J Neurol. 2005 Apr;12(4):245-53
15804240
Value Health. 2005 Sep-Oct;8(5):521-33
16176491
J Pediatr Psychol. 2009 Jan-Feb;34(1):69-78
18650207