Recurrent alternating ophthalmoplegia with ipsilateral headache: unusual but possible manifestation of recurrent painful
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LETTER TO THE EDITOR
Recurrent alternating ophthalmoplegia with ipsilateral headache: unusual but possible manifestation of recurrent painful ophthalmoplegic neuropathy Jae-Myung Kim 1 & You-Ri Kang 1 & Kyung Wook Kang 1 & Seung-Han Lee 1 Received: 27 February 2020 / Accepted: 30 May 2020 # Fondazione Società Italiana di Neurologia 2020
Dear Editor, Recurrent painful ophthalmoplegia is typically associated with headache or ocular pain and can be caused by several diseases such as Tolosa-Hunt syndrome, recurrent painful ophthalmoplegic neuropathy (RPON; previously known as ophthalmoplegic migraine (OM)), and anti-GQ1b antibody syndrome [1]. Similarities in clinical manifestation often make differentiation between etiologies difficult and may lead to poor prognoses. RPON is a rare disorder characterized by recurrent bouts of ophthalmoplegia associated with ipsilateral headache [1]. Ipsilateral headache usually precedes RPON by up to 14 days [2, 3]. Although typical RPON presents with ipsilateral migrainous headache with past history of migraine, various clinical presentations with recurrent ophthalmoplegia and spontaneous resolution within a few days to weeks have been reported, which may indicate that RPON is a heterogenous disorder [1, 3]. To our knowledge, RPON that subsequently developed in different ocular motor nerves at either side with preceding ipsilateral headache has not been reported. Here, we report a case of recurrent alternating ophthalmoplegia with ipsilateral headache, which is a possible manifestation of RPON. A previously healthy 30-year-old woman visited our clinic presenting with diplopia and right ptosis for 4 days. The patient had no history of diplopia, migraine, or head trauma. The diplopia was binocular, horizontal, and vertical. Her symptoms were constant without diurnal variation. Throbbing Electronic supplementary material The online version of this article (https://doi.org/10.1007/s10072-020-04502-6) contains supplementary material, which is available to authorized users. * Seung-Han Lee [email protected] 1
Department of Neurology, Chonnam National University Hospital and Chonnam National University Medical School, 42 Jebong-ro, Dong-gu, Gwangju 61469, South Korea
headache, with moderate to severe intensity, occurred in the ipsilateral hemicranium, with maximal intensity in the periorbital region, 8 days before the onset of ophthalmoplegia. Migraine auras and other migraine-associated symptoms, such as nausea/vomiting and photophobia/phonophobia, were all absent. Initial neurological examination revealed deficits in adduction and vertical gaze with incomplete ptosis in the right eye; however, light reflex was normal. These findings were compatible with the diagnosis of partial right-side oculomotor nerve palsy (Online figure 1). Other neurological and cranial nerve examinations, including those for visual acuity, facial sense, and deep tendon reflexes (DTR), were normal. Brain magnetic resonance imaging (MRI) with gadolinium enhancement did not show abnormal findings. For differe
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