Pulmonary mucormycosis following autologous hematopoietic stem cell transplantation for rapidly progressive diffuse cutaneous systemic sclerosis: A case report

Medicine (Baltimore). 2020 Jul 31;99(31):e21431. doi: 10.1097/MD.0000000000021431.

Abstract

Rationale: 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).

Patient concerns: A 24-year-old woman with rapidly progressive diffuse cutaneous SSc presented with an acute respiratory distress syndrome 6 days after AHSCT.

Diagnoses: 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.

Interventions and outcomes: Early antifungal therapy by intravenous amphotericin B provided rapid improvement within 4 days and sustained recovery after 2 years of follow-up.

Lessons: 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.

Publication types

  • Case Reports

MeSH terms

  • Acute Disease
  • Administration, Intravenous
  • Aftercare
  • Amphotericin B / administration & dosage
  • Amphotericin B / therapeutic use
  • Antifungal Agents / administration & dosage
  • Antifungal Agents / therapeutic use
  • Female
  • Hematopoietic Stem Cell Transplantation / adverse effects*
  • Hematopoietic Stem Cell Transplantation / methods
  • Humans
  • Lung Diseases, Fungal / diagnostic imaging
  • Lung Diseases, Fungal / microbiology
  • Lung Diseases, Fungal / pathology
  • Mucorales / genetics
  • Mucormycosis / etiology*
  • Respiratory Distress Syndrome / etiology
  • Scleroderma, Diffuse / etiology*
  • Scleroderma, Diffuse / pathology
  • Scleroderma, Systemic / therapy*
  • Transplantation, Autologous / adverse effects*
  • Transplantation, Autologous / methods
  • Treatment Outcome
  • Young Adult

Substances

  • Antifungal Agents
  • Amphotericin B