Subcutaneous entomophthoromycosis mimicking soft-tissue sarcoma in children

J Pediatr Surg. 2015 Jul;50(7):1150-5. doi: 10.1016/j.jpedsurg.2014.11.031. Epub 2014 Nov 20.

Abstract

Aim: Subcutaneous entomophthoromycosis (EM) is an uncommon fungal infection of childhood. This article is intended to draw the attention of pediatric surgeons to the fact that EM can mimic soft-tissue tumor.

Methods: It is a retrospective review of 16 children treated for subcutaneous EM between 2000 and 2013.

Results: The median age of patients was 3.5 years. The typical lesion was a discoid subcutaneous mass that can be easily lifted from deeper tissues (the doughnut lifting sign). Lesions were mostly distributed in the lower half of body. All the patients were immunocompetent. Correct clinical diagnosis was made only in 4 cases while others were mistaken for a tumor. All the 8 children who underwent wide excision of the pseudotumor had local recurrence. Supersaturated solution of potassium iodide was curative in 11 cases while addition of itraconazole was needed in one case. One child died of multi-drug resistant infection. The mean treatment duration was 4.7 months (range 2-8 months).

Conclusion: Subcutaneous EM can mimic soft-tissue tumor. High index of suspicion is essential to avoid misdiagnosis and inappropriate treatment. A newly described "doughnut-lifting sign' may be helpful in clinical diagnosis. Emergence of multi-drug resistant infection is a source of concern.

Keywords: Basidiobolus; Conidiobolus; Differential diagnosis; Fungal infection; Phycomycosis; Potassium iodide; Pseudo-tumor; Soft-tissue sarcoma; Zygomycosis.

MeSH terms

  • Adolescent
  • Child
  • Child, Preschool
  • Dermatomycoses / pathology*
  • Dermatomycoses / therapy
  • Diagnosis, Differential
  • Diagnostic Errors
  • Female
  • Humans
  • Itraconazole / therapeutic use
  • Male
  • Physical Examination
  • Potassium Iodide / administration & dosage
  • Retrospective Studies
  • Sarcoma / pathology*
  • Soft Tissue Neoplasms / pathology*
  • Zygomycosis / pathology*
  • Zygomycosis / therapy

Substances

  • Potassium Iodide
  • Itraconazole