Dyck P J, Stevens J C, O'Brien P C, Oviatt K F, Lais A C, Coventry M B, Beabout J W
Neurology. 1983 Mar;33(3):357-67. doi: 10.1212/wnl.33.3.357.
Some patients with radiologic findings of neurogenic arthropathy or multiple fractures do not exhibit overt neurologic signs. Results of nerve conduction velocity, computer-assisted sensory examination, periosteal nociception, and morphometric and graded teased-fiber evaluation of cutaneous nerves allowed us to recognize a mild neuropathic abnormality. Neurogenic arthropathy and subclinical neuropathy were also found in relatives. In three kinships, the underlying disorder was probably hereditary sensory neuropathy type 1 and in several others, it was recessively inherited sensory neuropathy. These arthropathies were often painful, and overt loss of superficial and deep pain sensation was not a prominent or necessary condition. An interplay of multiple factors including insensitivity, trauma, obesity, activity, abuse, personality, mental subnormality, and metabolic joint and bone disease are probably involved in the development of the bony lesions and thus provide further evidence that environmental factors affect expression of human mutant genes for inherited neuropathy.
一些有神经源性关节病或多处骨折影像学表现的患者并未表现出明显的神经学体征。神经传导速度、计算机辅助感觉检查、骨膜伤害感受以及皮肤神经的形态学和分级单纤维评估结果使我们能够识别出轻度的神经病变异常。在亲属中也发现了神经源性关节病和亚临床神经病变。在三个家族中,潜在疾病可能是1型遗传性感觉神经病,在其他几个家族中,则是隐性遗传感觉神经病。这些关节病常常疼痛,而浅表和深部痛觉的明显丧失并非突出或必要条件。包括感觉迟钝、创伤、肥胖、活动、滥用、个性、智力低下以及代谢性关节和骨病在内的多种因素之间的相互作用可能参与了骨病变的发展,从而进一步证明环境因素会影响人类遗传性神经病突变基因的表达。