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Research ArticlePediatric Neuroimaging
Open Access

White Matter Abnormality Correlates with Developmental and Seizure Outcomes in West Syndrome of Unknown Etiology

J. Natsume, C. Ogawa, T. Fukasawa, H. Yamamoto, N. Ishihara, Y. Sakaguchi, Y. Ito, T. Takeuchi, Y. Azuma, N. Ando, T. Kubota, T. Tsuji, H. Kawai, S. Naganawa and H. Kidokoro
American Journal of Neuroradiology April 2016, 37 (4) 698-705; DOI: https://doi.org/10.3174/ajnr.A4589
J. Natsume
aFrom the Departments of Pediatrics (J.N., C.O., H.Y., N.I., Y.S., Y.I., T. Takeuchi, Y.A., H. Kidokoro)
bDevelopmental Disability Medicine (J.N.)
eBrain and Mind Research Center (J.N., H. Kidokoro), Nagoya University, Nagoya, Japan
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C. Ogawa
aFrom the Departments of Pediatrics (J.N., C.O., H.Y., N.I., Y.S., Y.I., T. Takeuchi, Y.A., H. Kidokoro)
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T. Fukasawa
fDepartment of Pediatrics (T.F., T.K.), Anjo Kosei Hospital, Anjo, Japan
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  • ORCID record for T. Fukasawa
H. Yamamoto
aFrom the Departments of Pediatrics (J.N., C.O., H.Y., N.I., Y.S., Y.I., T. Takeuchi, Y.A., H. Kidokoro)
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N. Ishihara
aFrom the Departments of Pediatrics (J.N., C.O., H.Y., N.I., Y.S., Y.I., T. Takeuchi, Y.A., H. Kidokoro)
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Y. Sakaguchi
aFrom the Departments of Pediatrics (J.N., C.O., H.Y., N.I., Y.S., Y.I., T. Takeuchi, Y.A., H. Kidokoro)
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  • ORCID record for Y. Sakaguchi
Y. Ito
aFrom the Departments of Pediatrics (J.N., C.O., H.Y., N.I., Y.S., Y.I., T. Takeuchi, Y.A., H. Kidokoro)
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T. Takeuchi
aFrom the Departments of Pediatrics (J.N., C.O., H.Y., N.I., Y.S., Y.I., T. Takeuchi, Y.A., H. Kidokoro)
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Y. Azuma
aFrom the Departments of Pediatrics (J.N., C.O., H.Y., N.I., Y.S., Y.I., T. Takeuchi, Y.A., H. Kidokoro)
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N. Ando
dDepartment of Pediatrics and Neonatology (N.A.), Nagoya City University Graduate School of Medical Sciences, Nagoya, Japan
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T. Kubota
fDepartment of Pediatrics (T.F., T.K.), Anjo Kosei Hospital, Anjo, Japan
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T. Tsuji
gDepartment of Pediatrics (T. Tsuji), Okazaki City Hospital, Okazaki, Japan.
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H. Kawai
cRadiology (H. Kawai, S.N.), Nagoya University Graduate School of Medicine, Nagoya, Japan
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S. Naganawa
cRadiology (H. Kawai, S.N.), Nagoya University Graduate School of Medicine, Nagoya, Japan
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H. Kidokoro
aFrom the Departments of Pediatrics (J.N., C.O., H.Y., N.I., Y.S., Y.I., T. Takeuchi, Y.A., H. Kidokoro)
eBrain and Mind Research Center (J.N., H. Kidokoro), Nagoya University, Nagoya, Japan
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    Fig 1.

    Tract-based spatial statistics analysis between patients and controls at 12 and 24 months of age. A, Tract-based spatial statistics at 12 months of age reveals reduced fractional anisotropy values (blue areas) in the corpus callosum, deep white matter, and subcortical white matter in the temporal lobes of patients. The green areas represent white matter skeletons with a threshold FA of ≥0.2. B, Tract-based spatial statistics at 24 months of age shows no significant difference between all patients and controls. C, Tract-based spatial statistics reveals reduced fractional anisotropy values (blue areas) in widespread white matter areas of patients with a developmental quotient of <70 at 24 months of age.

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    Fig 2.

    Correlation of the developmental quotient and diffusion tensor imaging parameters at 12 months of age. The developmental quotient at 12 months of age shows a significant positive correlation with fractional anisotropy and negative correlations with radial ([λ2 + λ3]/2) and mean diffusivity. No correlation is evident between axonal diffusivity (λ1) and the developmental quotient at 12 months of age.

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    Fig 3.

    Abnormal findings on EEG at 12 months of age and diffusion tensor imaging parameters. Decreases in fractional anisotropy and increases in radial diffusivity are observed in patients with abnormalities findings on EEG compared with patients with normal electroencephalography findings at 12 months of age. White dots represent controls, gray dots are patients without seizures after initial treatments, and black dots are patients with relapsing or persistent seizures. Patients with relapsing or persistent seizures have low FA and high radial diffusivity.

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    Fig 4.

    Comparison of tract-based spatial statistics among patients with left temporal hypometabolism on positron-emission tomography at 24 months of age. A, Comparison of tract-based spatial statistics among 7 patients with left temporal hypometabolism and controls shows reduced fractional anisotropy in widespread white matter (blue areas) and more prominent DTI abnormalities in the left temporal lobe (arrows) compared with the right one. The green areas represent white matter skeletons with a threshold FA of ≥0.2. B, Fluorodeoxyglucose positron-emission tomography of 7 patients. Positron-emission tomography shows left temporal or temporofrontal hypometabolism (arrows). Note that the images from 3 patients with right temporal lobe hypometabolism are flipped, so all areas of hypometabolism are shown on the left side. The last patient shows additional hypometabolism in the contralateral frontal lobe (arrowhead).

Tables

  • Figures
  • Mean values of diffusion tensor imaging parameters in FA skeletons with a significant difference on tract-based spatial statistics at 12 months of agea

    Patients (n = 20)Controls (n = 13)P
    FA0.46 ± 0.040.51 ± 0.02<.001
    AD, λ1 (mm2/s)0.0016 ± 0.00010.0016 ± 0.0001NS
    RD, (λ2+ λ3) / 2 (mm2/s)0.00074 ± 0.000070.00066 ± 0.00006.007
    MD (mm2/s)0.0010 ± 0.00010.0010 ± 0.0007NS
    • Note:—AD indicates axial diffusivity; MD, mean diffusivity; NS, not significant; RD, radial diffusivity.

    • ↵a Statistical analysis was performed with a nonparametric Mann-Whitney U test.

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American Journal of Neuroradiology: 37 (4)
American Journal of Neuroradiology
Vol. 37, Issue 4
1 Apr 2016
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J. Natsume, C. Ogawa, T. Fukasawa, H. Yamamoto, N. Ishihara, Y. Sakaguchi, Y. Ito, T. Takeuchi, Y. Azuma, N. Ando, T. Kubota, T. Tsuji, H. Kawai, S. Naganawa, H. Kidokoro
White Matter Abnormality Correlates with Developmental and Seizure Outcomes in West Syndrome of Unknown Etiology
American Journal of Neuroradiology Apr 2016, 37 (4) 698-705; DOI: 10.3174/ajnr.A4589

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White Matter Abnormality Correlates with Developmental and Seizure Outcomes in West Syndrome of Unknown Etiology
J. Natsume, C. Ogawa, T. Fukasawa, H. Yamamoto, N. Ishihara, Y. Sakaguchi, Y. Ito, T. Takeuchi, Y. Azuma, N. Ando, T. Kubota, T. Tsuji, H. Kawai, S. Naganawa, H. Kidokoro
American Journal of Neuroradiology Apr 2016, 37 (4) 698-705; DOI: 10.3174/ajnr.A4589
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