Successful Treatment of Lomentospora Prolificans Infection Following Allogeneic Hematopoietic Stem Cell Transplantation: A Case Report and Literature Review

Abstract

Lomentospora prolificans is an opportunistic fungal pathogen known for its intrinsic multidrug resistance. This pathogen poses a significant challenge in immunocompromised individuals, particularly patients with hematologic malignancies. We present a case of a 27-year-old male diagnosed with adverse-risk acute myeloid leukemia (AML) who developed pulmonary Lomentospora prolificans infection following therapy for positive measurable residual disease (MRD) and severe chronic graft-versus-host disease (cGVHD) after allogeneic hematopoietic stem cell transplantation (allo-HSCT). Despite the pathogen's multidrug resistance and the typically poor prognosis associated with invasive infections in immunocompromised hosts, the patient achieved sustained remission and favorable outcome. This study systematically reviews Lomentospora prolificans infections following HSCT by analyzing 37 reported cases from 24 studies identified through a PubMed search. The majority of cases had acute myeloid leukemia as the most common underlying disease. Disseminated infections were predominant (83.8%), with frequent pulmonary and central nervous system involvement. Antifungal treatment strategies largely involved combination therapy, yet outcomes remained poor, with an overall survival rate of only 13.5%. These findings highlight the critical need for novel therapeutic approaches and early intervention strategies to improve patient outcomes.

Keywords: Lomentospora prolificans; hematopoietic stem cell transplantation; terbinafine; voriconazole.

Figure 1

Timeline of the episodes of Lomentospora prolificans infection involving therapeutic interventions and diagnostic hallmarks.

Figure 2

Representative CT scans of the patient’s lungs at different time points post-HSCT. (A) Day +151; (B) Day +161; (C) Day +171, first sputum culture identified as Lomentospora prolificans; (D) Day +178; (E) Day +189; (F) Day +197, (G) Day +203, last sputum culture identified as Lomentospora prolificans; (H) Day +210, (I) Day +231, (J) Day +274, outpatient, (K) Day +318, (L) Day +367.

Figure 3

Morphological characteristics of Lomentospora prolificans. (A) colonies on SCDA medium; (B) On SDA medium; (C and D) Lactophenol cotton blue staining.