Direct and indirect costs and cost-driving factors in adults with tuberous sclerosis complex: a multicenter cohort study and a review of the literature

Zöllner, Johann Philipp; Grau, Janina; Rosenow, Felix; Sauter, Matthias; Knuf, Markus; Kurlemann, Gerhard; Mayer, Thomas; Hertzberg, Christoph; Bertsche, Astrid; Immisch, Ilka; Klein, Karl Martin; Knake, Susanne; Marquard, Klaus; Meyer, Sascha; Noda, Anna H.; von Podewils, Felix; Schäfer, Hannah; Thiels, Charlotte; Willems, Laurent M.; Zukunft, Bianca; Schubert-Bast, Susanne; Strzelczyk, Adam

doi:10.1186/s13023-021-01838-w

Orphanet Journal of Rare Diseases

Table 6 Univariate and multivariate analysis of cost-driving factors for total direct, total indirect, and nursing care-level costs (3-month period; in 2019 Euro)

From: Direct and indirect costs and cost-driving factors in adults with tuberous sclerosis complex: a multicenter cohort study and a review of the literature

	n	Total direct costs in €	Median	SD	p-value^§	Total indirect costs in €**	Median	SD	p-value^§	Nursing care level costs in €	Median	SD	p-value^§
Gender					0.321				0.088				0.082
Male	93	6173	1527	8156		2796	0	4687		1079	948	1102
Female	99	6714	2722	7036		3529	538	4714		788	0	1016
Age					0.012*				0.062*^#				0.003*
18–29 years	90	8067	3460	8419		2171	0	4117		1084	948	1081
30–39 years	54	5849	1646	6700		3978	323	5081		1072	948	1072
40 years and above	48	4102	1350	6154		4192	430	5011		477	0	911
Number of antiseizure drugs					< 0.001				0.057				< 0.001
≥ 2	77	9237	9743	8639		4054	430	5131		1461	1635	1004
0—1	115	4587	1224	6155		2574	0	4308		573	0	955
mTOR inhibitors intake					< 0.001^#				0.510				0.250
Yes	71	14,473	13,100	5641		3335	0	4729		1041	948	1085
No	121	1746	712	3548		3077	0	4705		863	0	1053
Seizures					< 0.001^#				0.105				< 0.001
Recurrent seizures	75	9082	5440	8930		3859	323	5061		1530	1635	1055
Seizure free > 1 year or no seizures	117	4729	1401	5997		2717	0	4412		535	0	875
Epilepsy					0.001				0.904				< 0.001^#
Yes (72.9%)	140	7195	2184	8106		3284	0	4866		1249	1635	1072
No (27.1%)	52	4451	1257	5545		2866	0	4242		68	0	288
Structural brain disorders					0.003				0.889				0.014
Yes (67.2%)	129	7353	2343	8041		3106	0	4681		1059	948	1099
No (32.8%)	63	4607	1307	6211		3318	0	4784		663	0	947
Psychiatric disorders					< 0.001				0.260				< 0.001^#
Yes (50.5%)	97	8034	2982	8083		3741	0	5110		1521	1635	1037
No (49.5%)	95	4837	1199	6703		2583	0	4182		325	0	693
Heart and circulatory manifestations					< 0.001				0.558				0.170
Yes (50.5%)	97	8180	6568	7764		3229	0	4719		1032	948	1113
No (49.5%)	95	4687	1307	7007		3118	0	4710		823	0	1010
Kidney and urinary tract manifestations					< 0.001				0.248				0.317
Yes (74.0%)	142	7311	3357	7419		3381	0	4768		973	474	1078
No (26.0%)	50	4013	614	7591		2594	0	4511		805	0	1030
Skin manifestations					0.009				0.296				0.004
Yes (94.8%)	182	6704	2112	7669		3207	0	4702		980	948	1072
No (5.2%)	10	1873	380	3666		2498	0	4957		0	0	0
Lung manifestation					0.030				0.014				0.139
Yes (11.5%)	22	8894	7646	7928		5249	2588	5220		598	0	912
No (88.5%)	170	6136	1667	7505		2902	0	4578		972	0	1079
Other disorders					0.013				0.116				0.959
Yes (39.1%)	75	7856	7315	7352		3711	430	4982		938	0	1070
No (60.9%)	117	5552	1460	7625		2823	0	4499		923	0	1067
Total Disorders					< 0.001*				0.031*				< 0.001*
1–3 Manifestations (27.1%)	52	2652	331	5369		1982	0	3890		361	0	786
4 Manifestations (20.3%)	39	6488	1980	7796		2676	0	4299		652	0	922
5 Manifestations (19.8%)	38	6353	1739	7158		4816	1075	5345		1212	1292	1065
6 Manifestations (19.8%)	38	8080	6861	8022		3169	215	4770		1193	948	1070
7–8 Manifestations (13.0%)	25	11,977	15,151	7402		3844	0	5176		1709	2184	1083
Level of disability					< 0.001				0.001^#				< 0.001^#
None or ≤ 60%	68	4395	496	6823		1483	0	3085		110	0	470
70–100%	124	7580	2641	7768		4074	323	5158		1378	1635	1034

^§Mann–Whitney-U-test; *Kruskal–Wallis-test; **for indirect costs only indivuals of woking age (n = 190) were considered; SD = standard deviation; ^#significant predictor in multivariate analysis

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ISSN: 1750-1172

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