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Case Reports
. 2011 Sep;3(3):263-7.
doi: 10.1159/000334129. Epub 2011 Oct 21.

Anti-aquaporin-4 antibody-positive neuromyelitis optica presenting with syndrome of inappropriate antidiuretic hormone secretion as an initial manifestation

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Case Reports

Anti-aquaporin-4 antibody-positive neuromyelitis optica presenting with syndrome of inappropriate antidiuretic hormone secretion as an initial manifestation

H Nakajima et al. Case Rep Neurol. 2011 Sep.

Abstract

The distribution of neuromyelitis optica (NMO)-characteristic brain lesions corresponds to sites of high aquaporin-4 (AQP4) expression, and the brainstem and hypothalamus lesions that express high levels of AQP4 protein are relatively characteristic of NMO. The syndrome of inappropriate antidiuretic hormone secretion (SIADH) is one of the important causes of hyponatremia and results from an abnormal production or sustained secretion of antidiuretic hormone (ADH). SIADH has been associated with many clinical states or syndromes, and the hypothalamic-neurohypophyseal system regulates the feedback control system for ADH secretion. We report the case of a 63-year-old man with NMO, whose initial manifestation was hyponatremia caused by SIADH. Retrospective analysis revealed that the serum anti-AQP4 antibody was positive, and an MRI scan showed a unilateral lesion in the hypothalamus. SIADH recovered completely with regression of the hypothalamic lesion. As such, NMO should even be considered in patients who develop SIADH and have no optic nerve or spinal cord lesions but have MRI-documented hypothalamic lesions.

Keywords: Anti-aquaporin-4 antibody; Hypothalamus; Neuromyelitis optica; Syndrome of inappropriate antidiuretic hormone secretion.

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Figures

Fig. 1
Fig. 1
MRI findings of the present case. A hypothalamic lesion (arrow) was observed during the SIADH episode. Axial and coronal sections of a T2-weighted image showed marked hyperintense lesions at the hypothalamus (a, b). The hypothalamic lesion was markedly diminished during a relapse of acute myelitis 3 months after the SIADH episode (c). A sagittal T2-weighted image showed a long spinal cord lesion with high signal intensity during a relapse of acute myelitis (d).

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