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Journal of Nippon Medical School

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-Short Communication-

Central Diabetes Insipidus Associated with Orbital Apex Syndrome

Jung Hwan Yoon1, Su Hyeon Cho1, Seung-Han Lee2, Seong Kwon Ma1, Soo Wan Kim1 and Eun Hui Bae1

1Department of Internal Medicine, Chonnam National University Medical School, Gwangju, Korea
2Department of Neurology, Chonnam National University Medical School, Gwangju, Korea


Orbital apex syndrome (OAS) manifests as multiple cranial nerve palsies caused by an abnormal nerve response to inflammation or other processes. Central diabetes insipidus (CDI) is characterized by deficient synthesis or secretion of antidiuretic hormone. A 62-year-old woman underwent myringotomy for otitis media with effusion. Two months after the procedure, symptoms of hearing loss had not improved, and she underwent left tympanoplasty and mastoidectomy. After surgery, she presented with left ocular pain and visual loss. Neurologic examination revealed ptosis, total ophthalmoplegia, and a relative afferent pupillary defect on the left eye. Magnetic resonance imaging showed an asymmetric contrast-enhancing lesion in the left orbital apex and left cavernous sinus, with adjacent dural thickening and enhancement. OAS was diagnosed, and steroid treatment was started. During the regular follow-up period, she reported polyuria, and CDI was diagnosed. Treatment with intranasal desmopressin 10 μg twice daily was started, and symptoms greatly improved. The mechanism underlying the association of CDI with OAS is unclear, and further research is needed. The present case suggests that polyuria in OAS should alert neurologists and ophthalmologists to possible CDI.

J Nippon Med Sch 2019; 86: 254-257

Keywords
orbital apex syndrome, central diabetes insipidus, malignant otitis externa, ophthalmoplegia

Correspondence to
Eun Hui Bae, MD, PhD, Department of Internal Medicine, Chonnam National University Medical School, 42 Jebongro, Gwangju 61469, Korea
baedak@hanmail.net

Received, January 4, 2019
Accepted, April 17, 2019