Boumaza Xavier, Lelièvre Lucie, Guenounou Sarah, Borel Cécile, Huynh Anne, Beziat Guillaume, Delavigne Karen, Guinault Damien, Garric Marie, Piel-Julian Marie, Paricaud Kim, Moulis Guillaume, Astudillo Leonardo, Sailler Laurent, Farge Dominique, Pugnet Grégory
Department of Internal Medicine.
Department of infectious and tropical diseases, Toulouse University Hospital.
Medicine (Baltimore). 2020 Jul 31;99(31):e21431. doi: 10.1097/MD.0000000000021431.
The use of autologous hematopoietic stem cell transplantation (AHSCT) for autoimmune diseases has become the first indication for transplant in nonmalignant disease. Mucormycosis is a rare invasive infection with increasing incidence in patients treated with AHSCT. We report the first case of pulmonary mucormycosis following AHSCT for systemic sclerosis (SSc).
A 24-year-old woman with rapidly progressive diffuse cutaneous SSc presented with an acute respiratory distress syndrome 6 days after AHSCT.
The results of clinical and computed tomography scan were consistent with pulmonary mucormycosis and the diagnosis was confirmed by a positive Mucorales Polymerase Chain Reaction on a peripheral blood sample.
Early antifungal therapy by intravenous amphotericin B provided rapid improvement within 4 days and sustained recovery after 2 years of follow-up.
With the progressively increasing use of AHSCT and other stem cell therapy for treatment of severe SSc and other autoimmune diseases, the potential onset of rare post-transplant fungal infections, such as mucormycosis, requires careful patient monitoring and better awareness of early initiation of adequate therapy.
自体造血干细胞移植（AHSCT）用于治疗自身免疫性疾病已成为非恶性疾病移植的首要适应证。毛霉菌病是一种罕见的侵袭性感染，在接受AHSCT治疗的患者中发病率不断上升。我们报告首例系统性硬化症（SSc）患者在接受AHSCT后发生肺毛霉菌病的病例。
一名24岁快速进展性弥漫性皮肤型SSc女性患者在AHSCT后6天出现急性呼吸窘迫综合征。
临床及计算机断层扫描结果符合肺毛霉菌病，外周血样本毛霉目聚合酶链反应呈阳性确诊。
静脉注射两性霉素B进行早期抗真菌治疗，4天内迅速改善，随访2年后持续康复。
随着AHSCT及其他干细胞疗法越来越多地用于治疗重症SSc和其他自身免疫性疾病，移植后罕见真菌感染（如毛霉菌病）的潜在发病需要对患者进行仔细监测，并提高对早期启动充分治疗的认识。
