Kamimura Chihiro, Fujitani Junko, Aizawa Isao, Saotome Ikuko, Fujiwara Sayaka, Haga Nobuhiko
Department of Rehabilitation Medicine, National Center for Global Health and Medicine, Tokyo, Japan.
Department of Radiological Physics and Technology, National Center for Global Health and Medicine, Tokyo, Japan.
Intractable Rare Dis Res. 2024 Aug 31;13(3):185-189. doi: 10.5582/irdr.2024.01035.
Individuals with thalidomide embryopathy are now approximately 60 years old. For years, they have been compensating for their hypoplastic limbs in various aspects of daily living, and they face secondary problems such as limb and back pain. Imaging analysis is beneficial for understanding the pathogenesis of these problems. However, previous studies on skeletal imaging were mainly radiographic studies conducted at young ages, and there are few studies on skeletal imaging after aging, with most of them being case reports. In this study, detailed analyses of the skeletons of the upper extremities were performed using three-dimensional computed tomography and multiplanar reconstruction images in five individuals with thalidomide embryopathy aged approximately 60 years. Each individual frequently complained of neck, shoulder, and/or back pain. Dislocation, subluxation, and osteoarthritis were observed in the shoulder joints in some individuals. Hypoplasia of the trochlea and/or capitulum of the humerus, coronoid fossa, olecranon, and coronoid processes was observed in the elbow joints. Fusion and hypoplasia of the carpal bones were frequently observed in wrist joints. Radiocarpal and ulnocarpal synostoses were also observed. The joint instability and osteoarthritis found in this study may have contribute to upper limb pain in individuals with thalidomide embryopathy.
沙利度胺胚胎病患者如今大约60岁。多年来,他们一直在日常生活的各个方面代偿发育不全的肢体,并且面临诸如肢体和背部疼痛等继发问题。影像学分析有助于了解这些问题的发病机制。然而,以往关于骨骼影像学的研究主要是在年轻时进行的放射学研究,关于衰老后骨骼影像学的研究很少,其中大多数是病例报告。在本研究中,对5名年龄约60岁的沙利度胺胚胎病患者的上肢骨骼进行了详细分析,采用三维计算机断层扫描和多平面重建图像。每名患者经常抱怨颈部、肩部和/或背部疼痛。部分患者肩关节出现脱位、半脱位和骨关节炎。肘关节观察到肱骨滑车和/或小头、冠状窝、鹰嘴和冠状突发育不全。腕关节经常观察到腕骨融合和发育不全。还观察到桡腕和尺腕关节融合。本研究中发现的关节不稳定和骨关节炎可能是导致沙利度胺胚胎病患者上肢疼痛的原因。