Multidisciplinary management of disseminated Exophiala dermatitidis mycosis in an infant with mixed phenotype acute leukemia: a case report

Ryo Nakatani, Miho Ashiarai, Hiroki Yoshihara, Keigo Yada, Taiki Nozaki, Takeshi Ushigusa, Nobuyoshi Mori, Daisuke Hasegawa

Research output: Contribution to journalArticlepeer-review

1 Citation (Scopus)

Abstract

Background: 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. Case presentation: 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. Conclusion: Disseminated E. dermatitidis mycosis in the neutropenic infant was successfully managed with a multidisciplinary treatment consisting of multiagent antifungal treatment, ELT, and surgery.

Original languageEnglish
Article number797
JournalBMC infectious diseases
Volume22
Issue number1
DOIs
Publication statusPublished - 2022 Dec
Externally publishedYes

Keywords

  • Acute leukemia
  • Ecthyma gangrenosum
  • Ethanol lock therapy
  • Exophiala dermatitidis
  • Infant

ASJC Scopus subject areas

  • Infectious Diseases

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