Isavuconazole in a Successful Combination Treatment of Disseminated Mucormycosis in a Child with Acute Lymphoblastic Leukaemia and Generalized Haemochromatosis: A Case Report and Review of the Literature

Mycopathologia. 2019 Feb;184(1):81-88. doi: 10.1007/s11046-018-0287-0. Epub 2018 Jul 23.

Abstract

Invasive mucormycosis in immunocompromised children is a life-threatening fungal infection. We report a case of a 7-year-old girl treated for acute lymphoblastic leukaemia complicated by disseminated mucormycosis during induction therapy. Microscopic examination of surgically removed lung tissue revealed wide, pauci-septate hyphae suggesting a Mucorales infection. This diagnosis was confirmed immunohistochemically and by PCR analysis followed by a final identification of Cunninghamella sp. The patient was treated successfully with surgical debridement and antifungal combination therapy with amphotericin B, caspofungin and isavuconazole. The use of isavuconazole in a child was not previously reported. Additionally, case reports concerning pulmonary mucormycoses in paediatric population published after 2010 were reviewed. Nineteen out of 26 identified patients suffered from haematological diseases. Reported mortality reached 38.5%. By the fact of rising morbidity, unsatisfactory results of treatment and remaining high mortality of mucormycoses in immunocompromised patients, new therapeutic options are warrant. Isavuconazole, with its broad-spectrum activity, good safety profile and favourable pharmacokinetics, is a promising drug. However, further studies are necessary to confirm positive impact of isavuconazole on mucormycosis treatment in children.

Keywords: Children; Cunninghamella; Isavuconazole; Mucormycosis.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Amphotericin B / administration & dosage
  • Antifungal Agents / administration & dosage*
  • Caspofungin / administration & dosage
  • Child
  • Cunninghamella / isolation & purification*
  • Debridement
  • Drug Therapy, Combination / methods
  • Drug-Related Side Effects and Adverse Reactions / epidemiology
  • Drug-Related Side Effects and Adverse Reactions / pathology
  • Female
  • Hemochromatosis / complications*
  • Humans
  • Invasive Fungal Infections / diagnosis*
  • Invasive Fungal Infections / therapy
  • Mucormycosis / diagnosis*
  • Mucormycosis / therapy
  • Nitriles / administration & dosage*
  • Precursor Cell Lymphoblastic Leukemia-Lymphoma / complications*
  • Pyridines / administration & dosage*
  • Treatment Outcome
  • Triazoles / administration & dosage*

Substances

  • Antifungal Agents
  • Nitriles
  • Pyridines
  • Triazoles
  • isavuconazole
  • Amphotericin B
  • Caspofungin