Cryptococcus gattii infection complicated by immune reconstitution inflammatory syndrome in three apparently immunocompetent children.
Cryptococcus gattii
immune reconstitution inflammatory syndrome
paediatrics
Journal
Journal of paediatrics and child health
ISSN: 1440-1754
Titre abrégé: J Paediatr Child Health
Pays: Australia
ID NLM: 9005421
Informations de publication
Date de publication:
Aug 2019
Aug 2019
Historique:
received:
31
07
2018
revised:
06
11
2018
accepted:
09
11
2018
pubmed:
12
12
2018
medline:
30
1
2021
entrez:
12
12
2018
Statut:
ppublish
Résumé
Paediatric Cryptococcus gattii disease is rare, with only two previous cases recorded in the Northern Territory (NT) over the last 54 years. Immune reconstitution inflammatory syndrome (IRIS) is a recognised complication of C. gattii infection, even in the absence of an identified immunodeficiency syndrome; however, limited paediatric data exist. We present a series of three paediatric patients treated for C. gattii infection in the NT during 2016/2017. All three cases were males aged 8-13 years at the time of presentation. Two were Aboriginal Australians from remote NT communities, and the third was a Timorese child from a remote district in Timor-Leste. All cases had evidence of brain cryptococcomas, and two had associated pulmonary lesions. Each child was treated with a 6-week induction phase of intravenous liposomal amphotericin and flucytosine and then continued on a 2-year course of eradication 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 with IRIS between 5 and 10 weeks after commencement of antifungal treatment and were managed with high-dose corticosteroids, which were weaned slowly (6-20 months post-commencement). Paediatric C. gattii disease is rare, although three recent cases in the NT highlight some of the challenges involved in managing the infection, including persistent raised ICP and complications such as IRIS. There is a need for further collaborative research into paediatric C. gattii disease.
Sections du résumé
BACKGROUND
BACKGROUND
Paediatric Cryptococcus gattii disease is rare, with only two previous cases recorded in the Northern Territory (NT) over the last 54 years. Immune reconstitution inflammatory syndrome (IRIS) is a recognised complication of C. gattii infection, even in the absence of an identified immunodeficiency syndrome; however, limited paediatric data exist. We present a series of three paediatric patients treated for C. gattii infection in the NT during 2016/2017.
CASE DISCUSSIONS
UNASSIGNED
All three cases were males aged 8-13 years at the time of presentation. Two were Aboriginal Australians from remote NT communities, and the third was a Timorese child from a remote district in Timor-Leste. All cases had evidence of brain cryptococcomas, and two had associated pulmonary lesions. Each child was treated with a 6-week induction phase of intravenous liposomal amphotericin and flucytosine and then continued on a 2-year course of eradication 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 with IRIS between 5 and 10 weeks after commencement of antifungal treatment and were managed with high-dose corticosteroids, which were weaned slowly (6-20 months post-commencement).
CONCLUSIONS
CONCLUSIONS
Paediatric C. gattii disease is rare, although three recent cases in the NT highlight some of the challenges involved in managing the infection, including persistent raised ICP and complications such as IRIS. There is a need for further collaborative research into paediatric C. gattii disease.
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
943-947Informations de copyright
© 2018 Paediatrics and Child Health Division (The Royal Australasian College of Physicians).