Division of Pulmonary Medicine, Department of Medicine, Philippine General Hospital, Manila, Philippines.
Department of Dermatology, Philippine General Hospital , Manila, Philippines.
Am J Case Rep. 2022 Aug 10;23:e937061. doi: 10.12659/AJCR.937061.
BACKGROUND Erasmus syndrome is a rare disease entity characterized by the development of systemic sclerosis (SSc) in a background of silica exposure or silicosis. CASE REPORT We report the case of a 40-year-old Filipino man who previously worked in a silica grind mill for 10 years and eventually developed Erasmus syndrome. The patient initially presented with chronic back pain in 2018 associated with findings of pulmonary tuberculosis on chest X-ray, with notable improvement after 6 months of anti-tuberculosis treatment. However, his back pain recurred in 2021; this time with arthralgia, Raynaud's phenomenon, thickening of both hands, skin hypopigmentation on the chest, back, and forehead, and exertional dyspnea. Physical examination revealed salt-and-pepper dermopathy and skin tightening over the back, chest, and extremities. Mobility of his hands was limited, associated with sclerodactyly and digital pitting. Antinuclear antibody-immunofluorescence and anti-scleroderma-70 antibodies were strongly positive, confirming the diagnosis of SSc. Chest computed tomography illustrated multiple subcentimeter nodules and enlarged mediastinal lymph nodes with eggshell calcifications, consistent with silicosis. Spirometry with body plethysmography was normal but diffusing capacity for carbon monoxide was severely reduced. Histopathology of the skin showed markedly thickened collagen bundles in the dermis. CONCLUSIONS Chronic silica exposure is a risk factor for the development of silicosis. The clinical course of patients with silicosis may be complicated by SSc. Maintaining a high index of suspicion is key to the diagnosis of Erasmus syndrome. The present report emphasizes the importance of preventive measures and surveillance among those with occupational exposure to silica. To our knowledge, this is the first documented case of Erasmus syndrome in the Philippines.
Erasmus 综合征是一种罕见的疾病实体,其特征是在暴露于二氧化硅或矽肺的背景下发展为系统性硬化症(SSc)。
我们报告了一例 40 岁的菲律宾男性病例,他曾在二氧化硅研磨厂工作 10 年,最终发展为 Erasmus 综合征。患者最初于 2018 年出现慢性背痛,胸部 X 线检查发现肺结核,抗结核治疗 6 个月后明显改善。然而,他的背痛于 2021 年再次发作;这次伴有关节痛、雷诺现象、双手变厚、胸部、背部和前额皮肤色素减退以及运动性呼吸困难。体格检查显示盐和胡椒样皮肤病变和背部、胸部和四肢皮肤紧绷。手部活动受限,伴有硬皮病和手指凹陷。抗核抗体免疫荧光和抗硬皮病-70 抗体均呈强阳性,确诊为 SSc。胸部 CT 显示多个亚厘米结节和纵隔淋巴结肿大,蛋壳样钙化,符合矽肺。体描法肺量计正常,但一氧化碳弥散量严重降低。皮肤组织病理学显示真皮中胶原束明显增厚。
慢性二氧化硅暴露是矽肺发展的危险因素。矽肺患者的临床病程可能会并发 SSc。保持高度怀疑是诊断 Erasmus 综合征的关键。本报告强调了在职业性接触二氧化硅的人群中采取预防措施和监测的重要性。据我们所知,这是菲律宾首例记录在案的 Erasmus 综合征病例。