Successful isavuconazole salvage therapy for cerebral mucormycosis in a child with relapsed leukemia: A light in the dark tunnel

Abstract

Central nervous system mucormycosis (CNS-M) is a rare, 1 often deadly infection 2,3 with three forms: rhinocerebral mucormycosis, disseminated mucormycosis with CNS involvement, and isolated cerebral mucormycosis (ICM). 4 ICM is the rarest type 5,6 but can be rapidly progressive with high mortality (up to 65%) despite aggressive treatment. 5 We present a case of ICM in a child with relapsed leukemia where isavuconazole (ISAV) monotherapy was successfully used as salvage therapy and the patient was able to resume chemotherapy.A 10-year-old female was diagnosed with bone marrow and CNS relapse of B-cell acute lymphoblastic leukemia (B-ALL) > 3 years after completing chemotherapy for standard risk B-ALL. She was treated per the low-risk control arm of AALL1331. 7 She was in remission at the end of block 1 and continued protocol therapy. Two weeks into block 3, she was admitted with Enterobacter cloacae bacteremia and treated with cefepime. Fevers resolved but then recurred along with leftsided headache, photophobia, emotional lability, and a superficial scalp lesion. Brain magnetic resonance imaging (MRI) showed a 4.3 × 3.7 cm abnormality in the left parieto-occipital area (Figure 1A). Serum fungal biomarkers were negative. Liposomal amphotericin B and voriconazole were initiated. Biopsy of the enhancing scalp lesion was nonconclusive. Open brain biopsy showed no leukemic involvement, and initial fungal cultures were negative. polymerase chain reaction (PCR) testing returned positive for Rhizomucor pucillus, and immunostaining was consistent with mucormycosis (Supporting Information Figure S1).Based on otherwise negative imaging, she was diagnosed with ICM.Liposomal amphotericin B dose was increased. Although micafungin is not effective for Mucormycosis, but it was added for synergism, 2,8 and voriconazole was stopped.

Acute myocardial infarction caused by coronary mucormycotic embolism

“…Once mucormycosis develops, it is often fatal due to its rapid progression [5] . Early diagnosis and treatment are therefore crucial [17] . Definitive diagnosis by invasive techniques is, however, often difficult in patients with hematologic disorders because of the high hemorrhaging tendency [7] .…”

Clinical research advances of isavuconazole in the treatment of invasive fungal diseases

“…After switching from VOR to ISA monotherapy owing to renal damage, the patient was successfully treated. Alali et al (2022) reported the case of a child with CNS-relapsed acute lymphoblastic leukemia who developed solitary cerebral IM. After debridement and L-AmB administration, the infection spread, but the infection resolved after switching to oral ISA single-agent rescue therapy.…”

“…After switching from VOR to ISA monotherapy owing to renal damage, the patient was successfully treated. Alali et al. (2022) reported the case of a child with CNS-relapsed acute lymphoblastic leukemia who developed solitary cerebral IM.…”