Exp Neurobiol 2016; 25(3): 139-142
Published online June 30, 2016
© The Korean Society for Brain and Neural Sciences
Kee Hong Park1, Young-Soo Kim1, Soo-Kyung Kim1, Nack-Cheon Choi1,2, Oh-Young Kwon1,2, ByeongHoon Lim1,2 and Ki-Jong Park2,3*
1Department of Neurology, Gyeongsang National University Hospital, Jinju 52727, 2Department of Neurology, Gyeongsang National University, School of Medicine, Gyeongsang Institue of Health Science, Jinju 52727, 3Department of Neurology, Gyeongsagn National University Changwon Hospital, Changwon 51472, Korea
Correspondence to: *To whom correspondence should be addressed.
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The existence of
Keywords: Myelitis, Toxocara canis, Eosionophilia, Pulmonary eosinophilia
Atopic myelitis refers to myelitis characterized by hyperimmunoglobulin E (IgE)-emia and the presence of mite antigen-specific IgE . Its clinical features include a young age of onset, a subacute or chronic nature, and sensory symptoms rather than weakness as the chief complaint . Currently,
A 35-year-old male patient was admitted with a chief complaint of hypoesthesia in the right leg lasting for 1 month. At first, the progress of the hypoesthesia was only monitored, as the symptoms were mild and there was no discomfort reported. One week later, the hypoesthesia progressed to a degree that the patient was unable to feel hot water. The patient did not complain of weakness, pain, paresthesia, dysuria, or erectile dysfunction. He had experienced pain in his left chest 3 months prior to his visit, and he had been admitted to the department of pulmonary medicine. At this point, the patient's eosinophil count was elevated to 13.3% (normal range is 1~5%) and a chest computed tomography (CT) scan revealed consolidation with ground-glass opacification in both lungs, which led to a diagnosis of eosinophilic pneumonia (Fig. 1). Chest pain was naturally improved after 2 weeks, and no other respiratory symptoms, such as cough, sputum, or dyspnea, were observed. Furthermore, the eosinophil count decreased to 7.7% and a chest radiograph revealed slight improvements in the patient's lungs. Thus, the patient's progress was monitored without any further treatment. No other disease history, such as asthma, atopic dermatitis, sinusitis, or arthritis was reported, and no symptoms such as photosensitivity, oral ulcers, or genital ulcers were observed. Moreover, the patient did not consume any raw meat other than raw fish, and reported no exposure to animals or pets. A neurological examination revealed that pain, touch, and vibration sensations were all decreased to approximately 25% of normal levels on the right side below the T8 level. Motor power was intact as revealed by a medical research council (MRC) grade of V, and the deep tendon reflex was found to be normal. The Babinski reflex and ankle clonus examinations revealed no pathologic reflexes.
Spinal magnetic resonance imaging (MRI) revealed a high T2 signal intensity lesion with focal enhancement at the C5-T3 level (Fig. 2). Brain MRI was normal. Furthermore, although a somatosensory evoked potential central conduction defect was observed at the thoracic level, motor evoked potential, brainstem auditory evoked potential, and visual evoked potential test results were all normal. The blood eosinophil count was 7.5%, which was elevated to a similar level as the patient's previous visit. The patient was negative for the presence of antinuclear, anti-Ro/La, antineutrophil cytoplasm (P-ANCA, C-ANCA), antiphospholipid, and aquaporin-4 antibodies. The level of angiotensin converting enzyme was 21 IU/L (normal range 20~70 IU/L), the level of vitamin B12 was 893.4 pg/ml (normal range 243~894 pg/ml), and the level of folate was 6.33 ng/ml (normal range 3.1~17.5 pg/ml). Test results for human immunodeficiency virus and human T-lymphotrophic virus were both negative. No white blood cells were detected in the cerebrospinal fluid (CSF), and the CSF protein level was 23.8 mg/dL (normal range 15~45 mg/dL). No oligoclonal bands were observed and the IgG index was 0.67. Serum IgE levels were elevated to 1,216 IU/ml (normal range 1.0~183.0 IU/ml), with antigen-specific IgE against
Atopic myelitis was first described in 4 patients with acute myelitis accompanied by hyper-IgE-emia and atopic dermatitis . Later in 1998, Kira et al. suggested the use of the term atopic myelitis to describe patients diagnosed with the condition, which commonly occurs in the cervical cord and often accompanies atopic dermatitis with normal CSF findings . Yoon et al. compared 14 atopic myelitis patients with 15 non-atopic myelitis patients in 2009 and reported an earlier age of onset and a non-acute and slowly progressive disease course in atopic myelitis patients . They also observed that motor symptoms were relatively mild, while sensory symptoms were predominant in atopic myelitis patients . MRI findings revealed a high percentage of swelling, the predominance of large eccentric vs. central lesions in axial images, and focal lesions with gadolinium enhancement . The specific IgE to
Here, we present a case of myelitis accompanied by hyper-IgE-emia in a patient with a history of eosinophilic pneumonia. We confirmed the presence of