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一例巨细胞动脉炎合并风湿性多肌痛伴头皮坏死及难治性颌部间歇性运动障碍。

A case of giant cell arteritis complicated by polymyalgia rheumatica with scalp necrosis and refractory jaw claudication.

作者信息

Kosaka Shumpei, Nawata Masao, Yamazumi Kenji, Nawata Aya, Nakamura Katsumi, Saito Kazuyoshi, Tanaka Yoshiya

机构信息

Department of Clinical Immunology and Rheumatology, Tobata General Hospital, Kitakyushu, Japan.

The First Department of Internal Medicine, School of Medicine, University of Occupational and Environmental Health, Kitakyushu, Japan.

出版信息

Mod Rheumatol Case Rep. 2023 Jan 3;7(1):177-181. doi: 10.1093/mrcr/rxac084.

Abstract

Scalp necrosis is a rare complication of giant cell arteritis (GCA); however, it is a predictor of severe disease. In this case study, a patient presented with GCA complicated by polymyalgia rheumatica with scalp necrosis. An 86-year-old woman was admitted to the hospital for pulsating headache, scalp pain, jaw claudication, and generalised pain. Bilateral temporal arteries were found to be distended and pulseless, and scalp necrosis was observed in the parietal region. Simultaneous high-resolution contrast-enhanced magnetic resonance imaging (MRI) sequences of the head, shoulder, and hip showed staining around the bilateral shallow temporal arteries, shoulder, and hip joints, which was confirmed as GCA with polymyalgia rheumatica using other examination findings. After treatment with early induction remission therapy, scalp necrosis healed, but jaw claudication persisted. Six months after the start of treatment, scalp necrosis was cured to full hair growth. Despite remission induction therapy combined with tocilizumab, the patient had persistent jaw claudication for several months. At that time, a high-resolution contrast-enhanced MRI re-examination was useful in assessing disease activity. GCA with scalp necrosis may cause prolonged jaw claudication reflecting the progression of ischaemic lesions, whereas the disease activity can be accurately assessed by combining MRI studies.

摘要

头皮坏死是巨细胞动脉炎(GCA)的一种罕见并发症;然而,它是严重疾病的一个预测指标。在本病例研究中,一名患者表现为GCA并发风湿性多肌痛伴头皮坏死。一名86岁女性因搏动性头痛、头皮疼痛、颌部间歇性运动障碍和全身疼痛入院。发现双侧颞动脉扩张且无搏动,并在顶叶区域观察到头皮坏死。头部、肩部和髋部的同步高分辨率对比增强磁共振成像(MRI)序列显示双侧颞浅动脉、肩部和髋关节周围有染色,结合其他检查结果确诊为GCA并发风湿性多肌痛。经过早期诱导缓解治疗后,头皮坏死愈合,但颌部间歇性运动障碍持续存在。治疗开始6个月后,头皮坏死治愈,头发完全重新生长。尽管使用托珠单抗进行了缓解诱导治疗,但患者的颌部间歇性运动障碍持续了数月。此时,高分辨率对比增强MRI复查有助于评估疾病活动度。伴有头皮坏死的GCA可能导致反映缺血性病变进展的持续性颌部间歇性运动障碍,而通过结合MRI研究可以准确评估疾病活动度。

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