Painful ophthalmoplegia with normal cranial imaging

Chih-Hsien Hung,Yao-Liang Chen,Chiou-Lian Lai,Hong-Shiu Chang,Ming-Feng Liao,Chun-Che Chu,Yi-Ming Wu,Long-Sun Ro,Kuo-Hsuan Chang,Rong-Kuo Lyu,Hsiao-Jung Tseng

doi:10.1186/1471-2377-14-7

Abstract

BackgroundPainful ophthalmoplegia with normal cranial imaging is rare and confined to limited etiologies. In this study, we aimed to elucidate these causes by evaluating clinical presentations and treatment responses.MethodsCases of painful ophthalmoplegia with normal cranial MRI at a single center between January 2001 and June 2011 were retrospectively reviewed. Diagnoses of painful ophthalmoplegia were made according to the recommendations of the International Headache Society.ResultsOf the 58 painful ophthalmoplegia cases (53 patients), 26 (44.8%) were diagnosed as ocular diabetic neuropathy, 27 (46.6%) as benign Tolosa-Hunt syndrome (THS), and 5 (8.6%) as ophthalmoplegic migraine (OM). Patients with ocular diabetic neuropathy were significantly older (62.8 ± 7.8 years) than those with benign THS (56.3 ±12.0 years) or OM (45.8 ± 23.0 years) (p < 0.05). Cranial nerve involvement was similar among groups. Pupil sparing was dominant in each group. Patients with benign THS and OM responded exquisitely to glucocorticoid treatment with resolved diplopia, whereas patients with ocular diabetic neuropathy didn’t (p < 0.05). Patients with OM recovered more rapidly than the other groups did (p < 0.05). Overall, most patients (94.8%) recovered completely during the follow-up period.ConclusionsOcular diabetic neuropathy and benign THS accounted for most of the painful ophthalmoplegias in patients with normal cranial imaging. Patient outcomes were generally good.

Highlights

Painful ophthalmoplegia with normal cranial imaging is rare and confined to limited etiologies
53 qualified patients with normal neuroimaging data were recruited for this study
Twenty-five patients (47.2%) were diagnosed as having ocular diabetic neuropathy, and 25 (47.2%) and 3 (5.7%) patients were diagnosed with benign Tolosa-Hunt syndrome (THS) and ophthalmoplegic migraine (OM), respectively (Table 1)

Summary

Introduction

Painful ophthalmoplegia with normal cranial imaging is rare and confined to limited etiologies. We aimed to elucidate these causes by evaluating clinical presentations and treatment responses. Painful ophthalmoplegia consists of periorbital or hemicranial pain with ipsilateral ocular motor nerve palsies [1]. The syndrome involves diverse causes; a comprehensive evaluation is essential. Contrast-enhanced magnetic resonance imaging (MRI) provides sensitive detection of structural abnormalities, such as trauma, infection, malignancy, or vascular anomaly [2,3]. Neuroimaging studies frequently yield negative findings in patients with painful ophthalmoplegia [4]. Cranial neuropathies in diabetic patients due to for patients with THS and normal neuroimaging findings [9]. Glucocorticoids are considered as standard treatment for patients with THS [1,10]

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