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Paradoxical immune reconstitution inflammatory syndrome due to toxoplasmic encephalitis: two cases and review of initiation of antiretroviral timing in toxoplasmic encephalitis IRIS.

Dinardo AR, Lewis DS, Koo HL, Goodman JC, Chiao E, Andrade R - F1000Res (2013)

Bottom Line: To the best of our knowledge, only two cases of paradoxical TE-IRIS and nine cases of unmasking TE-IRIS have been previously described.We present two additional cases of histopathology-consistent paradoxical TE-IRIS, after early initiation of antiretroviral therapy (ART), and review the literature on TE-IRIS.Three of the four reported cases of paradoxical TE-IRIS were associated with early (within one week) initiation of ART, an issue that was not addressed in the 2009 US Department of Health and Human Services guidelines for the treatment of opportunistic infections.

View Article: PubMed Central - PubMed

Affiliation: Department of Medicine, Baylor College of Medicine, Houston, TX, 77030, USA.

ABSTRACT
Toxoplasma encephalitis immune reconstitution inflammatory syndrome (TE-IRIS) is rare and usually occurs in an unmasking, rather than paradoxical form. To the best of our knowledge, only two cases of paradoxical TE-IRIS and nine cases of unmasking TE-IRIS have been previously described. We present two additional cases of histopathology-consistent paradoxical TE-IRIS, after early initiation of antiretroviral therapy (ART), and review the literature on TE-IRIS. Three of the four reported cases of paradoxical TE-IRIS were associated with early (within one week) initiation of ART, an issue that was not addressed in the 2009 US Department of Health and Human Services guidelines for the treatment of opportunistic infections.

No MeSH data available.


Related in: MedlinePlus

Brain histopathology of patient 1.Significant necrosis with perivascular mixed inflammatory cells, with lymphocyte predominance and concentric vascular wall fibroblastic proliferation. Rare individualToxoplasma gondii bradyzoites and numerous tachyzoites were identified (100X) (A). Immunophenotyping of the inflammatory cells, showing a mixture of mature T- and B-lymphocytes with CD8+ over CD4+ cell predominance. There was no evidence of other infections or CNS lymphoma (100X) (B).
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f2: Brain histopathology of patient 1.Significant necrosis with perivascular mixed inflammatory cells, with lymphocyte predominance and concentric vascular wall fibroblastic proliferation. Rare individualToxoplasma gondii bradyzoites and numerous tachyzoites were identified (100X) (A). Immunophenotyping of the inflammatory cells, showing a mixture of mature T- and B-lymphocytes with CD8+ over CD4+ cell predominance. There was no evidence of other infections or CNS lymphoma (100X) (B).

Mentions: Over the next two weeks, the patient’s function deteriorated: his left deltoid muscle strength weakened to 2/5 (previously 5/5), he developed a left facial droop and left hip flexor and quadriceps weakness were 3/5 (previously 5/5). He consented to a lumbar puncture and his CSF revealed 6 white blood cells (WBC) per mm3 (96% lymphocytes and 4% monocytes), glucose of 41 g/L and protein of 92 g/L. EBV PCR in the CSF was positive. On hospital day ten, he suffered a tonic-clonic seizure. A repeat MRI on day 14 of empiric anti-Toxoplasma therapy, showed enlargement of the two prior lesions and development of a third lesion (Figure 1B). After twenty days of anti-toxoplasmic therapy, a brain biopsy revealed rareToxoplasma gondii tachyzoites and numerous bradyzoites (Figure 2A), as well as CD8+ predominant lymphocytic infiltrates (Figure 2B). A repeat CD4 count and viral load measure was 13 cells/µll (1%) and 10,300 copies/ml respectively. Anti-Toxoplasma treatment and ART were continued and the patient was started on corticosteroids (dexamethasone 4 mg every 4 hours tapered to prednisone 10 mg per day over 2 weeks), with gradual improvement but not complete resolution of his symptoms and signs.


Paradoxical immune reconstitution inflammatory syndrome due to toxoplasmic encephalitis: two cases and review of initiation of antiretroviral timing in toxoplasmic encephalitis IRIS.

Dinardo AR, Lewis DS, Koo HL, Goodman JC, Chiao E, Andrade R - F1000Res (2013)

Brain histopathology of patient 1.Significant necrosis with perivascular mixed inflammatory cells, with lymphocyte predominance and concentric vascular wall fibroblastic proliferation. Rare individualToxoplasma gondii bradyzoites and numerous tachyzoites were identified (100X) (A). Immunophenotyping of the inflammatory cells, showing a mixture of mature T- and B-lymphocytes with CD8+ over CD4+ cell predominance. There was no evidence of other infections or CNS lymphoma (100X) (B).
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC3814919&req=5

f2: Brain histopathology of patient 1.Significant necrosis with perivascular mixed inflammatory cells, with lymphocyte predominance and concentric vascular wall fibroblastic proliferation. Rare individualToxoplasma gondii bradyzoites and numerous tachyzoites were identified (100X) (A). Immunophenotyping of the inflammatory cells, showing a mixture of mature T- and B-lymphocytes with CD8+ over CD4+ cell predominance. There was no evidence of other infections or CNS lymphoma (100X) (B).
Mentions: Over the next two weeks, the patient’s function deteriorated: his left deltoid muscle strength weakened to 2/5 (previously 5/5), he developed a left facial droop and left hip flexor and quadriceps weakness were 3/5 (previously 5/5). He consented to a lumbar puncture and his CSF revealed 6 white blood cells (WBC) per mm3 (96% lymphocytes and 4% monocytes), glucose of 41 g/L and protein of 92 g/L. EBV PCR in the CSF was positive. On hospital day ten, he suffered a tonic-clonic seizure. A repeat MRI on day 14 of empiric anti-Toxoplasma therapy, showed enlargement of the two prior lesions and development of a third lesion (Figure 1B). After twenty days of anti-toxoplasmic therapy, a brain biopsy revealed rareToxoplasma gondii tachyzoites and numerous bradyzoites (Figure 2A), as well as CD8+ predominant lymphocytic infiltrates (Figure 2B). A repeat CD4 count and viral load measure was 13 cells/µll (1%) and 10,300 copies/ml respectively. Anti-Toxoplasma treatment and ART were continued and the patient was started on corticosteroids (dexamethasone 4 mg every 4 hours tapered to prednisone 10 mg per day over 2 weeks), with gradual improvement but not complete resolution of his symptoms and signs.

Bottom Line: To the best of our knowledge, only two cases of paradoxical TE-IRIS and nine cases of unmasking TE-IRIS have been previously described.We present two additional cases of histopathology-consistent paradoxical TE-IRIS, after early initiation of antiretroviral therapy (ART), and review the literature on TE-IRIS.Three of the four reported cases of paradoxical TE-IRIS were associated with early (within one week) initiation of ART, an issue that was not addressed in the 2009 US Department of Health and Human Services guidelines for the treatment of opportunistic infections.

View Article: PubMed Central - PubMed

Affiliation: Department of Medicine, Baylor College of Medicine, Houston, TX, 77030, USA.

ABSTRACT
Toxoplasma encephalitis immune reconstitution inflammatory syndrome (TE-IRIS) is rare and usually occurs in an unmasking, rather than paradoxical form. To the best of our knowledge, only two cases of paradoxical TE-IRIS and nine cases of unmasking TE-IRIS have been previously described. We present two additional cases of histopathology-consistent paradoxical TE-IRIS, after early initiation of antiretroviral therapy (ART), and review the literature on TE-IRIS. Three of the four reported cases of paradoxical TE-IRIS were associated with early (within one week) initiation of ART, an issue that was not addressed in the 2009 US Department of Health and Human Services guidelines for the treatment of opportunistic infections.

No MeSH data available.


Related in: MedlinePlus