[CIS PIDD] [cis-pidd] 53yoM with cryptococcal meningitis

[Dewton Vasconcelos]

Dear Lauren

We have seen several HIV negative patients with neurocryptococcosis here 
in Brazil in the last years.
Among these there were more cases of C. gattii than among HIV or other 
immunosuppressed patients (most of them presented some type of 
neoplasia, transplantation, autoimmune diseases treated with 
immunosuppressors or anti-TNF drugs etc.).
Two patients received anti-TNF and developed neurocryptococcosis (one 
infliximab and other adalimumab).
Moreover, three patient presented "idiopathic" CD4 lymphocytopenias, one 
IL_12p40 deficiency and two IL-12 Rb1 deficiency.
It is interesting to look at the histopathological aspect of the 
granulomas, if they are loose or normal, the number of fungal forms, the 
palisade and the presence of central necrosis.
Immunohistochemistry to cytokines can help in the treatment. If the 
presence of IFN-gamma is low in the granulomas of the tissue, you can 
use IFN-gamma at the dose of 50 mcg/m2/3 times a week, usually with a 
brilliant response in these cases.
An other important feature is the presence of intraparenchymatous 
lesions of the CNS, that requires more prolonged treatment. The addition 
of 5-fluorocytosine to liposomal amphotericin is very important, leading 
to a better prognosis for the patient.

Hope it helps,

Best regards,

Dewton

Dewton de Moraes Vasconcelos, MD, PhD
Primary Immunodeficiencies Outpatient Unit ADEE3003
Lab. of Medical Investigation Unit 56
University of São Paulo School of Medicine

Temino, Viviana M. wrote:
> I am looking for help with the workup of a patient with cryptococcal meningitis which is either recurrent or poorly responsive to treatment.  I have included a brief clinical summary and labs below.  Any advice on further testing that would be appropriate would be appreciated.  IgG and IgM to cryptococcal antigen has not been done yet to my knowledge.
>
> 53 year old man who has no known immunodeficiency and no prior significant infections (no sinusitis, GI infections/giardiasis, meningitis/encephalitis, sinusitis, pneumonia or skin infections/abscesses) who presented in November 2013 with altered mental status and seizures.  Diagnosed with cryptococcal meningitis at that time and was treated with amphotericin, fluconazole and voriconazole.  He may have had some lapses in treatement/poor compliance due to intolerance to medications and since then has had further imaging with signs that the cryptococcal meningitis has been progressing/worsening and has now developed disseminated disease (hematologic and possibly skin).
>
> Lab workup includes:
> HIV- negative
> Immunoglobulin profile- normal
> Response to pneumovax- normal
> Tetanus titer undetectable (no response checked as of yet)
> ANCA negative
> Complement (C3, C4, CH50, AH50) normal
> Lymphocytes (B cells, NK cells and immunophenotypically normal DC4+ and CD8+ T cells) in normal proportions
> Granulocytes immunophenotypically mature
>
>
> Thank You,
>
> Lauren Fine
> Assistant Professor of Allergy, Asthma & Immunology
> Division of Pulmonary, Allergy, Critical Care, and Sleep Medicine
> University of Miami Miller School of Medicine
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The CIS-PIDD listserv is supported by the Clinical Immunology Society
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