Cryptococcus gattii infection complicated by immune reconstitution inflammatory syndrome in three apparently immunocompetent children

Matthew P. O'Brien, Timothy J. Ford, Bart J. Currie, Joshua R. Francis

    Research output: Contribution to journalArticleResearchpeer-review

    Abstract

    Background: Paediatric Cryptococcus gattii disease is rare, with only twoprevious cases recorded in the Northern Territory (NT) over the last 54 years.Immune reconstitution inflammatory syndrome (IRIS) is a recognised complicationof C. gattii infection, even in the absence of an identified immunodeficiencysyndrome; however, limited paediatric data exist. We present a series of threepaediatric patients treated for C. gattii infection in the NT during 2016/2017.


    CaseDiscussions: All three cases were males aged 8–13 years at the timeof presentation. Two were Aboriginal Australians from remote NT communities,and the third was a Timorese child from a remote district in Timor‐Leste. Allcases had evidence of brain cryptococcomas, and two had associated pulmonarylesions. Each child was treated with a 6‐week induction phase of intravenousliposomal amphotericin and flucytosine and then continued on a 2‐year course oferadication oral fluconazole. Persistent high intracranial pressure (ICP)complicated each case, requiring serial lumbar punctures and, in two cases,insertion of ventriculoperitoneal shunts. All three cases were diagnosed withIRIS between 5 and 10 weeks after commencement of antifungal treatment and weremanaged with high‐dose corticosteroids, which were weaned slowly (6–20 monthspost‐commencement).


    Conclusions: Paediatric C. gattii disease is rare, although threerecent cases in the NT highlight some of the challenges involved in managing theinfection, including persistent raised ICP and complications such as IRIS.There is a need for further collaborative research into paediatric C. gattiidisease.

    Original languageEnglish
    Article numberJPC14321
    Pages (from-to)943-947
    Number of pages5
    JournalJournal of Paediatrics and Child Health
    Volume55
    Issue number8
    Early online date8 Dec 2018
    DOIs
    Publication statusPublished - 1 Aug 2019

    Fingerprint

    Cryptococcus gattii
    Immune Reconstitution Inflammatory Syndrome
    Northern Territory
    Pediatrics
    Intracranial Pressure
    Rare Diseases
    Infection
    Flucytosine
    Ventriculoperitoneal Shunt
    Spinal Puncture
    Fluconazole
    Amphotericin B
    Adrenal Cortex Hormones
    Brain
    Research

    Cite this

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    title = "Cryptococcus gattii infection complicated by immune reconstitution inflammatory syndrome in three apparently immunocompetent children",
    abstract = "Background: Paediatric Cryptococcus gattii disease is rare, with only twoprevious cases recorded in the Northern Territory (NT) over the last 54 years.Immune reconstitution inflammatory syndrome (IRIS) is a recognised complicationof C. gattii infection, even in the absence of an identified immunodeficiencysyndrome; however, limited paediatric data exist. We present a series of threepaediatric patients treated for C. gattii infection in the NT during 2016/2017.CaseDiscussions: All three cases were males aged 8–13 years at the timeof presentation. Two were Aboriginal Australians from remote NT communities,and the third was a Timorese child from a remote district in Timor‐Leste. Allcases had evidence of brain cryptococcomas, and two had associated pulmonarylesions. Each child was treated with a 6‐week induction phase of intravenousliposomal amphotericin and flucytosine and then continued on a 2‐year course oferadication oral fluconazole. Persistent high intracranial pressure (ICP)complicated each case, requiring serial lumbar punctures and, in two cases,insertion of ventriculoperitoneal shunts. All three cases were diagnosed withIRIS between 5 and 10 weeks after commencement of antifungal treatment and weremanaged with high‐dose corticosteroids, which were weaned slowly (6–20 monthspost‐commencement).Conclusions: Paediatric C. gattii disease is rare, although threerecent cases in the NT highlight some of the challenges involved in managing theinfection, including persistent raised ICP and complications such as IRIS.There is a need for further collaborative research into paediatric C. gattiidisease.",
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    Cryptococcus gattii infection complicated by immune reconstitution inflammatory syndrome in three apparently immunocompetent children. / O'Brien, Matthew P.; Ford, Timothy J.; Currie, Bart J.; Francis, Joshua R.

    In: Journal of Paediatrics and Child Health, Vol. 55, No. 8, JPC14321, 01.08.2019, p. 943-947.

    Research output: Contribution to journalArticleResearchpeer-review

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    T1 - Cryptococcus gattii infection complicated by immune reconstitution inflammatory syndrome in three apparently immunocompetent children

    AU - O'Brien, Matthew P.

    AU - Ford, Timothy J.

    AU - Currie, Bart J.

    AU - Francis, Joshua R.

    PY - 2019/8/1

    Y1 - 2019/8/1

    N2 - Background: Paediatric Cryptococcus gattii disease is rare, with only twoprevious cases recorded in the Northern Territory (NT) over the last 54 years.Immune reconstitution inflammatory syndrome (IRIS) is a recognised complicationof C. gattii infection, even in the absence of an identified immunodeficiencysyndrome; however, limited paediatric data exist. We present a series of threepaediatric patients treated for C. gattii infection in the NT during 2016/2017.CaseDiscussions: All three cases were males aged 8–13 years at the timeof presentation. Two were Aboriginal Australians from remote NT communities,and the third was a Timorese child from a remote district in Timor‐Leste. Allcases had evidence of brain cryptococcomas, and two had associated pulmonarylesions. Each child was treated with a 6‐week induction phase of intravenousliposomal amphotericin and flucytosine and then continued on a 2‐year course oferadication oral fluconazole. Persistent high intracranial pressure (ICP)complicated each case, requiring serial lumbar punctures and, in two cases,insertion of ventriculoperitoneal shunts. All three cases were diagnosed withIRIS between 5 and 10 weeks after commencement of antifungal treatment and weremanaged with high‐dose corticosteroids, which were weaned slowly (6–20 monthspost‐commencement).Conclusions: Paediatric C. gattii disease is rare, although threerecent cases in the NT highlight some of the challenges involved in managing theinfection, including persistent raised ICP and complications such as IRIS.There is a need for further collaborative research into paediatric C. gattiidisease.

    AB - Background: Paediatric Cryptococcus gattii disease is rare, with only twoprevious cases recorded in the Northern Territory (NT) over the last 54 years.Immune reconstitution inflammatory syndrome (IRIS) is a recognised complicationof C. gattii infection, even in the absence of an identified immunodeficiencysyndrome; however, limited paediatric data exist. We present a series of threepaediatric patients treated for C. gattii infection in the NT during 2016/2017.CaseDiscussions: All three cases were males aged 8–13 years at the timeof presentation. Two were Aboriginal Australians from remote NT communities,and the third was a Timorese child from a remote district in Timor‐Leste. Allcases had evidence of brain cryptococcomas, and two had associated pulmonarylesions. Each child was treated with a 6‐week induction phase of intravenousliposomal amphotericin and flucytosine and then continued on a 2‐year course oferadication oral fluconazole. Persistent high intracranial pressure (ICP)complicated each case, requiring serial lumbar punctures and, in two cases,insertion of ventriculoperitoneal shunts. All three cases were diagnosed withIRIS between 5 and 10 weeks after commencement of antifungal treatment and weremanaged with high‐dose corticosteroids, which were weaned slowly (6–20 monthspost‐commencement).Conclusions: Paediatric C. gattii disease is rare, although threerecent cases in the NT highlight some of the challenges involved in managing theinfection, including persistent raised ICP and complications such as IRIS.There is a need for further collaborative research into paediatric C. gattiidisease.

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