Sugiyama A, Araki E, Arakawa K, Kikuchi H, Iwaki T, Yamada T, Kira J
Department of Neurology, Faculty of Medicine, Kyushu University.
Rinsho Shinkeigaku. 1998 Oct-Nov;38(10-11):941-4.
A 22-year-old female noted a low grade fever and swelling of the cervical lymph nodes in May 1997, and later developed a dry cough. She was diagnosed to have interstitial pneumonitis, and then administration of corticosteroids alleviated her symptoms. On February 6, 1998, however, a high fever recurred and her swollen cervical lymph node on the right side was biopsied on February 9, 1998. A histological examination revealed an increased number of histiocytes and karyorrhexis of the lymphocytes in the paracortical areas, and she was therefore diagnosed to have histiocytic necrotizing lymphadenitis. She could not fully elevate her arm on February 16, 1998. On admission, her cervical lymph node was swollen on the left side. A neurological examination revealed a marked weakness of the right deltoid muscle, moderate weakness of the right latissimus dorsi, triceps and brachioradialis muscles and also a mild weakness of the serratus anterior, supra- and infra-spinatus, and biceps brachii muscles. The muscle power of the other muscles were normal and no muscle atrophy was evident. Winging of the right scapula was observed. The deep tendon reflexes were normal in all four limbs, and her sensation was also normal. No cerebellar sign was found. The Jackson, Spurling, Allen, Morley and Adson tests were all negative. ESR was mildly elevated to 18 mm/hr, but CRP was negative. RF, ANA and anti-SS-A and SS-B antibodies were positive, whereas LE-test, direct and indirect Coombs tests and other autoantibodies were negative. Needle EMG disclosed fasciculation potentials in the right triceps muscle and polyphasic waves in the right deltoid muscle. MRI showed gadolinium-enhancement of the right brachial plexus. Although an abnormal accumulation of gallium was detected in the right parotid and bilateral submandibular glands, no sicca symptoms were found and the Schirmer test findings were normal. Oral prednisolone (50 mg/day with gradual tapering) alleviated both her symptoms and the gadolinium-enhancement of the right brachial plexus. As a result, her right upper limb paresis was thus considered to have been caused by right brachial plexus neuritis, which was probably associated with histocytic necrotizing lymphadentis. Although acute cerebellar ataxia and meningitis have previously been reported to be complicated with histiocytic necrotizing lymphadenitis, this is the first report to describe the complication of peripheral neuritis with this condition.
一名22岁女性于1997年5月出现低热及颈部淋巴结肿大,随后出现干咳。她被诊断为间质性肺炎,使用皮质类固醇治疗后症状缓解。然而,1998年2月6日,她再次高热,右侧肿大的颈部淋巴结于1998年2月9日进行了活检。组织学检查显示副皮质区组织细胞数量增加,淋巴细胞核溶解,因此她被诊断为组织细胞坏死性淋巴结炎。1998年2月16日,她无法完全抬起手臂。入院时,她左侧颈部淋巴结肿大。神经系统检查发现右侧三角肌明显无力,右侧背阔肌、三头肌和肱桡肌中度无力,前锯肌、冈上肌、冈下肌和肱二头肌轻度无力。其他肌肉力量正常,无明显肌肉萎缩。观察到右侧肩胛骨翼状畸形。四肢的深腱反射正常,感觉也正常。未发现小脑体征。杰克逊、斯珀林、艾伦、莫利和阿德森试验均为阴性。血沉轻度升高至18mm/h,但CRP为阴性。类风湿因子、抗核抗体及抗SS - A和SS - B抗体阳性,而狼疮细胞试验、直接和间接库姆斯试验及其他自身抗体阴性。针电极肌电图显示右侧三头肌有束颤电位,右侧三角肌有多相波。MRI显示右侧臂丛神经钆增强。尽管在右侧腮腺和双侧颌下腺检测到镓异常聚集,但未发现干燥症状,泪液分泌试验结果正常。口服泼尼松龙(50mg/天,逐渐减量)缓解了她的症状及右侧臂丛神经的钆增强。因此,她的右上肢轻瘫被认为是由右侧臂丛神经炎引起的,可能与组织细胞坏死性淋巴结炎有关。尽管先前曾报道急性小脑共济失调和脑膜炎是组织细胞坏死性淋巴结炎的并发症,但这是首次报道该疾病并发周围神经炎。