Jahngir Muhammad Umair, Govindarajan Raghav
Neurology, University of Missouri Healthcare, Columbia, USA.
Neurology, University of Missouri, Columbia, USA.
Cureus. 2018 Aug 6;10(8):e3105. doi: 10.7759/cureus.3105.
The association of symmetrical distal sensorimotor polyneuropathy with anti-myelin-associated glycoprotein antibodies (MAG) has been well established. Although autonomic symptoms are uncommon with anti-MAG antibody neuropathy (MAN). We are presenting an unusual case, who developed acute onset urinary retention, orthostatic hypotension, bradycardia and was found to have MAN. She was a 68-year-old lady, presented with progressive ascending numbness, weakness of her extremities and balance problems. On neurological examination, she had generalized muscle weakness, reduced perception to all modalities of sensation with marked impairment of vibration and position sense in the lower limbs. Reflexes were diminished throughout and Romberg sign was positive. Initial lab work including thyroid-stimulating hormone (TSH), vitamin B12, Hb1c, and routine lab reports was normal. The patient then developed acute urinary retention, severe orthostatic hypotension, and symptomatic bradycardia. Workup during this time revealed M spike on serum electrophoresis with IgM kappa on immunofixation. IgM titers were higher than normal. Initially, she was thought to have monoclonal gammopathy of undetermined significance (MGUS) related neuropathy but further workup showed very high levels of anti-MAG antibody titer. Further workup including a bone marrow biopsy revealed a small B cell lymphoma. Only a few cases have reported a small B cell lymphoma presenting with MAN-associated autonomic symptoms. She is currently being treated with rituximab with significant improvement in her neuropathic symptoms. Further case studies are needed to show whether autonomic symptoms are the feature of MAN or this atypical presentation is the paraneoplastic manifestation of the lymphoma.
对称性远端感觉运动性多发性神经病与抗髓鞘相关糖蛋白抗体(MAG)之间的关联已得到充分证实。尽管抗MAG抗体神经病(MAN)很少出现自主神经症状,但我们报告了一例不寻常的病例,该患者急性起病,出现尿潴留、体位性低血压、心动过缓,经检查发现患有MAN。患者为一名68岁女性,表现为进行性上升性麻木、肢体无力和平衡问题。神经系统检查发现,她全身肌肉无力,所有感觉方式的感知均减退,下肢振动觉和位置觉明显受损。全身反射减弱,闭目难立征阳性。包括促甲状腺激素(TSH)、维生素B12、糖化血红蛋白(Hb1c)和常规实验室报告在内的初始实验室检查结果均正常。随后,该患者出现急性尿潴留、严重体位性低血压和症状性心动过缓。在此期间的检查发现血清电泳有M峰,免疫固定显示为IgM κ型。IgM滴度高于正常。最初,她被认为患有意义未明的单克隆丙种球蛋白病(MGUS)相关神经病,但进一步检查显示抗MAG抗体滴度非常高。包括骨髓活检在内的进一步检查发现了小B细胞淋巴瘤。仅有少数病例报告过小B细胞淋巴瘤伴有MAN相关的自主神经症状。她目前正在接受利妥昔单抗治疗,神经病变症状有显著改善。需要进一步的病例研究来表明自主神经症状是MAN的特征,还是这种非典型表现是淋巴瘤的副肿瘤表现。