一例患1型复杂性区域疼痛综合征的父子在功能磁共振成像上表现出不同静息态功能连接的病例。

A Case of a Father and Son With Complex Regional Pain Syndrome Type 1 Exhibiting Different Resting-State Functional Connectivity on Functional MRI.

作者信息

Moriwaki Katsuyuki, Yoshino Atsuo, Ikejiri Yumi, Nakamura Ryuji, Tsutsumi Yasuo

机构信息

Department of Anesthesiology, Hiroshima University Hospital, Hiroshima, JPN.

Department of Anesthesiology, Hiroshima Hiramatsu Hospital, Hiroshima, JPN.

出版信息

Cureus. 2024 Jan 19;16(1):e52589. doi: 10.7759/cureus.52589. eCollection 2024 Jan.

Abstract

Complex regional pain syndrome (CRPS) type 1 is a chronic pain condition whose pathogenesis involves changes in the central and peripheral nervous systems, with potential genetic contributions. Functional magnetic resonance imaging (fMRI) studies report that alterations in resting-state functional connectivity (rsFC) may reflect central nervous system anomalies in CRPS type 1. Herein, we describe the case of a father and son with CRPS type 1 who exhibited different rsFC patterns in fMRI analyses correlating with their individual CRPS phenotypes. A 39-year-old male and his 61-year-old father presented with severe pain and mobility limitations in their right upper limbs following a vehicle accident and a fall, respectively, and were diagnosed with CRPS type 1. Despite receiving treatment, they experienced severe pain and limited mobility. The son exhibited dystonia and musculoskeletal atrophy while the father experienced extensive sensory disturbances. Bone scintigraphy revealed increased uptake in affected regions. The patients' resting-state fMRI data were compared with those of 48 healthy adults using the CONN software, with the false discovery rate set at p<0.05. Distinct brain regions for the father and son exhibited decreased rsFC (between the rostral prefrontal cortex and orbitofrontal cortex in the father and between the supplementary motor area and pallidum in the son; all in the right hemisphere). These changes corresponded to pain sensation and cognitive-emotional alterations in the father and limb movement disorders (dystonia) in the son. Our findings strongly support the idea that abnormalities in rsFC are closely linked to CRPS type 1 phenotypes.

摘要

1型复杂性区域疼痛综合征(CRPS)是一种慢性疼痛病症,其发病机制涉及中枢和周围神经系统的变化,并可能存在遗传因素。功能磁共振成像(fMRI)研究报告称,静息态功能连接(rsFC)的改变可能反映1型CRPS中枢神经系统异常。在此,我们描述了一对患有1型CRPS的父子病例,他们在fMRI分析中表现出不同的rsFC模式,且与各自的CRPS表型相关。一名39岁男性及其61岁的父亲分别在经历车祸和跌倒后,出现右上肢严重疼痛和活动受限,并被诊断为1型CRPS。尽管接受了治疗,但他们仍经历着严重疼痛和活动受限。儿子表现出肌张力障碍和肌肉骨骼萎缩,而父亲则有广泛的感觉障碍。骨闪烁显像显示受累区域摄取增加。使用CONN软件将患者的静息态fMRI数据与48名健康成年人的数据进行比较,错误发现率设定为p<0.05。父子各自不同的脑区显示rsFC降低(父亲为额前皮质和眶额皮质之间,儿子为辅助运动区和苍白球之间;均在右半球)。这些变化分别对应于父亲的疼痛感觉和认知 - 情绪改变以及儿子的肢体运动障碍(肌张力障碍)。我们的研究结果有力地支持了rsFC异常与1型CRPS表型密切相关这一观点。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c38f/10874683/f2e0aaae2556/cureus-0016-00000052589-i01.jpg

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