Fingolimod in children with Rett syndrome: the FINGORETT study

Naegelin, Yvonne; Kuhle, Jens; Schädelin, Sabine; Datta, Alexandre N.; Magon, Stefano; Amann, Michael; Barro, Christian; Ramelli, Gian Paolo; Heesom, Kate; Barde, Yves-Alain; Weber, Peter; Kappos, Ludwig

doi:10.1186/s13023-020-01655-7

Orphanet Journal of Rare Diseases

Table 4 Association between BDNF/NfL and cerebral blood flow at the same visit, adjusted for age

From: Fingolimod in children with Rett syndrome: the FINGORETT study

Brain region	Source	Estimate	SE	CI	p
BDNF
Cortex*	Serum	0.10	0.55	[− 0.984; 1.185]	0.856
Cortex*	CSF	1.42	0.94	[− 0.415; 3.261]	0.129
Insula	Serum	0.35	0.59	[− 0.800; 1.505]	0.549
Insula	CSF	1.19	1.00	[− 0.761; 3.145]	0.232
Occipital	Serum	− 0.20	0.74	[− 1.652; 1.260]	0.792
Occipital	CSF	2.27	1.44	[− 0.547; 5.091]	0.114
Parietal	Serum	0.06	0.49	[− 0.904; 1.033]	0.896
Parietal	CSF	1.59	0.87	[− 0.119; 3.309]	0.068
Temporal	Serum	− 0.25	0.55	[− 1.327; 0.823]	0.646
Temporal	CSF	1.21	1.02	[− 0.791; 3.219]	0.235
Basal ganglia	Serum	− 0.08	0.26	[− 0.589; 0.432]	0.763
Basal ganglia	CSF	0.94	0.38	[0.199; 1.673]	0.013
Thalamus	Serum	0.02	0.77	[− 1.487; 1.535]	0.975
Thalamus	CSF	3.11	1.14	[0.867; 5.351]	< 0.01
Hippo-campus	Serum	− 0.02	0.47	[− 0.950; 0.912]	0.967
Hippo-campus	CSF	1.12	0.69	[− 0.224; 2.464]	0.102
NfL
Cortex*	Serum	− 0.58	0.11	[− 0.796; − 0.368]	< 0.01
Cortex*	CSF	− 0.01	0.01	[− 0.023; − 0.002]	0.018
Insula	Serum	− 0.40	0.11	[− 0.610; − 0.196]	< 0.01
Insula	CSF	− 0.01	0.00	[− 0.017; − 0.005]	< 0.01
Occipital	Serum	− 0.91	0.14	[− 1.185; − 0.637]	< 0.01
Occipital	CSF	− 0.02	0.01	[− 0.034; 0.000]	0.049
Parietal	Serum	− 0.70	0.14	[− 0.970; − 0.434]	< 0.01
Parietal	CSF	− 0.10	0.01	[− 0.025; − 0.003]	0.011
Temporal	Serum	− 0.65	0.11	[− 0.861; − 0.449]	< 0.01
Temporal	CSF	− 0.01	0.01	[− 0.025; − 0.004]	< 0.01
Basal ganglia	Serum	− 0.38	0.07	[− 0.519; − 0.237]	< 0.01
Basal ganglia	CSF	− 0.01	0.00	[− 0.014; − 0.003]	< 0.01
Thalamus	Serum	− 0.53	0.14	[− 0.810; − 0.251]	< 0.01
Thalamus	CSF	− 0.01	0.01	[− 0.026; − 0.002]	0.019
Hippo-campus	Serum	− 0.47	0.10	[− 0.661; − 0.269]	< 0.01
Hippo-campus	CSF	− 0.02	0.00	[− 0.023; − 0.008]	< 0.01

*Cortex: includes cortex of Insula, occipital, parietal, and temporal lobes. Frontal cortex evaluation was not possible due to susceptibility artefacts

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ISSN: 1750-1172

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