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.
Highlights
Mucormycosis was first described by Furbringer in 1876 [1]
We describe the successful treatment of mucormycosis in a child with acute lymphoblastic leukaemia (ALL), which is the first article describing usage of isavuconazole in a child
Mucormycosis and especially infections caused by Cunninghamella are rare but carry one of the highest mortality rates among all mycoses
Summary
Mucormycosis was first described by Furbringer in 1876 [1]. Almost 100 years later Baker was still characterizing it as most acutely fatal mycosis [2]. Immunostaining of hyphae revealed a strong staining only with monoclonal antibodies (Mab WSSA-RA-1) reacting with genera of the order Mucorales [6] (Fig. 2b). Immunostaining of hyphae revealed a strong staining only with monoclonal antibodies (Mab WSSA-RA-1) reacting with genera of the order Mucorales. Despite continuation of combination antifungal therapy, the follow-up chest CT scan showed progression in the right lung 14 days after surgery. Because of the resistant mucormycosis, combination therapy with ABLC, caspofungin and isavuconazole was implemented. TDM was done on days: 3, 5, 7 and 28 of the isavuconazole therapy reaching trough levels of 2.92, 2.16, 2.1 and 2.45 ug/ml, respectively. When intensive chemotherapy was terminated, the isavuconazole treatment was discontinued, and secondary prophylaxis with posaconazole was started. During a 10-months observation period from completing isavuconazole treatment, the patient has been well, in the first complete haematological remission, during maintenance chemotherapy
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