Ho T W, Hsieh S T, Nachamkin I, Willison H J, Sheikh K, Kiehlbauch J, Flanigan K, McArthur J C, Cornblath D R, McKhann G M, Griffin J W
Department of Neurology, Johns Hopkins University School of Medicine, Baltimore, MD, USA.
Neurology. 1997 Mar;48(3):717-24. doi: 10.1212/wnl.48.3.717.
We investigated the possible mechanisms of paralysis and recovery in a patient with the acute motor axonal neuropathy (AMAN) pattern of the Guillain-Barré syndrome. The AMAN pattern of GBS is characterized clinically by acute paralysis without sensory involvement and electrodiagnostically by low compound motor action potential amplitudes, suggesting axonal damage, without evidence of demyelination. Many AMAN patients have serologic or culture evidence of recent Campylobacter jejuni infection. Pathologically, the most severe cases are characterized by wallerian-like degeneration of motor axons affecting the ventral roots as well as peripheral nerves, but some fatal cases have only minor changes in the roots and peripheral nerves, and some paralyzed patients with the characteristic electrodiagnostic findings of AMAN recover rapidly. The mechanism of paralysis and recovery in such cases has been uncertain. A 64-year-old woman with culture-proven Campylobacter upsaliensis diarrhea developed typical features of AMAN. She improved quickly following plasmapheresis. Her serum contained IgG anti-GM1 antibodies. The lipopolysaccharide of the organism bound peanut agglutinin. This binding was blocked by cholera toxin, suggesting that the organism contained the Gal(beta1-3)GalNAc epitope of GM1 in its lipopolysaccharide. Motor-point biopsy showed denervated neuromuscular junctions and reduced fiber numbers in intramuscular nerves. In contrast, the sural nerve biopsy was normal and skin biopsy showed normal dermal and epidermal innervation. In AMAN the paralysis may reflect degeneration of motor nerve terminals and intramuscular axons. In addition, the anti-GM1 antibodies, which can bind at nodes of Ranvier, might produce failure of conduction. These processes are potentially reversible and likely to underlie the capacity for rapid recovery that characterizes some cases of AMAN.
我们研究了一名患有吉兰 - 巴雷综合征急性运动轴索性神经病(AMAN)型患者的麻痹及恢复的可能机制。GBS的AMAN型在临床上的特征为急性麻痹且无感觉受累,电诊断表现为复合运动动作电位波幅降低,提示存在轴突损伤,而无脱髓鞘证据。许多AMAN患者有近期空肠弯曲菌感染的血清学或培养证据。病理上,最严重的病例表现为运动轴突的瓦勒氏样变性,累及腹根以及周围神经,但一些致命病例在神经根和周围神经仅有轻微改变,并且一些具有AMAN特征性电诊断表现的麻痹患者恢复迅速。此类病例中麻痹和恢复的机制尚不确定。一名64岁经培养证实有乌普萨拉弯曲菌腹泻的女性出现了AMAN的典型特征。她在进行血浆置换后迅速好转。她的血清中含有IgG抗GM1抗体。该微生物的脂多糖与花生凝集素结合。这种结合被霍乱毒素阻断,提示该微生物在其脂多糖中含有GM1的Gal(β1-3)GalNAc表位。运动点活检显示神经肌肉接头失神经支配且肌内神经纤维数量减少。相比之下,腓肠神经活检正常,皮肤活检显示真皮和表皮神经支配正常。在AMAN中,麻痹可能反映运动神经末梢和肌内轴突的变性。此外,可在郎飞结处结合的抗GM1抗体可能导致传导障碍。这些过程可能是可逆的,并且可能是一些AMAN病例具有快速恢复能力的基础。