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. 2019 Jan;39(1):112-117.
doi: 10.1007/s10875-019-0589-0. Epub 2019 Jan 24.

Outcomes for Nitazoxanide Treatment in a Case Series of Patients with Primary Immunodeficiencies and Rubella Virus-Associated Granuloma

Ludmila Perelygina  1 , David Buchbinder  2 , Morna J Dorsey  3 , Marc Eloit  4 , Fabian Hauck  5 , Timo Hautala  6 , Despina Moshous  7   8 , Ignacio Uriarte  9 , Elena Deripapa  10 , Joseph Icenogle  1 , Kathleen E Sullivan  11
Affiliations

Outcomes for Nitazoxanide Treatment in a Case Series of Patients with Primary Immunodeficiencies and Rubella Virus-Associated Granuloma

Ludmila Perelygina et al. J Clin Immunol. 2019 Jan.

Abstract

Purpose: Nitazoxanide was recently reported as having in vitro effectiveness against the rubella virus. Immunodeficiency-related vaccine-derived rubella occurs in some patients who have an inherited immunodeficiency and who received the MMR vaccine. This study investigated the in vivo effectiveness of nitazoxanide therapy.

Methods: This is a retrospective analysis of seven patients treated with nitazoxanide as salvage therapy for immunodeficiency-related vaccine-derived rubella infection. The patients were recruited from an ongoing rubella detection surveillance project.

Results: Seven patients with persistent rubella were treated with nitazoxanide and one demonstrated significant clinical improvement. Two additional patients exhibited diminished viral capsid production with one patient having transient slowing of progression. The cohort overall generally had low T cell counts and had a high burden of comorbidities. There were three deaths. Two deaths were from PML and one was related to hematopoietic stem cell transplantation.

Conclusions: Nitazoxanide has limited in vivo anti-viral effects for immunodeficiency-related vaccine-derived rubella. Most patients did not exhibit clinical improvement.

Keywords: Granulomas; MMR; chronic inflammation; nitazoxanide; vaccine.

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Conflict of interest statement

Conflict of interest:

The authors declare they have ho conflicts of interest.

Figures

Figure 1.
Figure 1.. Immunodeficiency-related vaccine-derived rubella virus (iVDRV) clinical features.
A) Patient 5 with typical granulomatous inflammation. B) Patient 5 demonstrating relentless progression over one year. C) Patient 6 in 2016 after surgical excision. D) Patient 6 after 2.5 months of nitazoxanide. E) Patient 3 right leg prior to stem cell transplant. F) Legs of Patient 3 legs 20 days after transplantation.
Figure 2.
Figure 2.. Regression of viral capsid expression after nitazoxanide.
A) Patient 5 immunohistochemistry rubella capsid detection (red) in granuloma (CD206 in green is an M2 macrophage marker). DAPI is provided as a nuclear stain. B) Patient 5 after two months of nitazoxanide treatment (NTZ). There is no detectable rubella capsid.
See this image and copyright information in PMC

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