Department of Pediatrics, St. Luke's International Hospital, 9-1, Akashi-Cho, Chuo-Ku, Tokyo, Japan.
Department of Pediatric Surgery, St. Luke's International Hospital, Tokyo, Japan.
BMC Infect Dis. 2022 Oct 23;22(1):797. doi: 10.1186/s12879-022-07773-w.
Exophiala dermatitidis is a dematiaceous fungus isolated from various environmental sources. Systemic E. dermatitidis infections can lead to fatal outcomes, and treatment has not yet been standardized. Although E. dermatitidis is also known to cause cutaneous infection, it has not been previously reported to appear as ecthyma gangrenosum (EG), an uncommon cutaneous lesion in neutropenic patients that is mainly caused by Pseudomonas aeruginosa.
A 2-month-old male infant with mixed-phenotype acute leukemia presented with prolonged fever unresponsive to antibacterial and antifungal agents during myelosuppression due to remission induction therapy. He also presented with skin lesions on the left wrist and left lower quadrant of the abdomen. The abdominal lesion gradually turned black and necrotic, which was consistent with the findings of the EG. E. dermatitidis was isolated from the blood, stool, wrist skin, and endotracheal aspirate. During hematopoietic recovery, consolidation in both lungs was evident. Multiagent antifungal treatment failed to eliminate E. dermatitidis from blood. In order to salvage the central venous catheter, ethanol lock therapy (ELT) was adopted, following which the blood culture became negative. The abdominal lesion that evolved as a necrotic mass connecting the small intestine and subcutaneous tissue adjacent to the skin was surgically resected. After these interventions, the general condition improved.
Disseminated E. dermatitidis mycosis in the neutropenic infant was successfully managed with a multidisciplinary treatment consisting of multiagent antifungal treatment, ELT, and surgery.
皮炎外瓶霉是一种从各种环境来源中分离出来的暗色真菌。系统性皮炎外瓶霉感染可导致致命后果，且治疗尚未标准化。虽然皮炎外瓶霉也已知可引起皮肤感染，但它以前从未被报道过会出现坏疽性脓皮病（EG），这是一种中性粒细胞减少症患者中罕见的皮肤病变，主要由铜绿假单胞菌引起。
一名 2 个月大的男性婴儿，患有混合表型急性白血病，在缓解诱导治疗导致骨髓抑制期间，因长时间发热对抗菌和抗真菌药物无反应而就诊。他还在手腕左侧和腹部左下象限出现皮肤损伤。腹部病变逐渐变黑坏死，与 EG 的表现一致。皮炎外瓶霉从血液、粪便、手腕皮肤和气管内吸出物中分离出来。在造血恢复期间，双肺均有明显的巩固。多药抗真菌治疗未能从血液中消除皮炎外瓶霉。为了挽救中心静脉导管，采用乙醇锁治疗（ELT），随后血培养转为阴性。腹部病变演变为与皮肤相邻的皮下组织和小肠相连的坏死肿块，进行了手术切除。经过这些干预，一般情况得到改善。
采用多学科治疗，包括多药抗真菌治疗、ELT 和手术，成功治疗了中性粒细胞减少症婴儿的播散性皮炎外瓶霉真菌感染。
