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

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.

Figure 1

Pulmonary mucormycosis appearing as a mass with the halo sign on computerized tomography (CT) in 24-year-old woman after autologous hematopoietic stem cell transplantation for rapidly progressive diffuse cutaneous systemic sclerosis. A, CT scan on day 6, showing a bilateral parenchymatous condensation within the right upper lobe the reverse halo sign. B, CT scan on day 20 showing showed a nearly complete regression of bilateral parenchymatous condensation and disappearance of the reverse halo sign.