Cutaneous Granulomatous Disease With Presence of Rubella Virus in Lesions

Dhossche, Julie; Johnson, Luke R.; White, Kevin P.; Funk, Tracy; Leitenberger, Sabra; Perelygina, Ludmila; Krol, Alfons

doi:10.1001/jamadermatol.2019.0814

Cited by 10 publications

(16 citation statements)

References 7 publications

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“…Rubella-associated granulomas are a known complication in IEIs (10)(11)(12)(28)(29)(30)(31)(32). In a series of 66 patients, 17 suffered from AT, 16 from atypical SCID, 25 from various forms of combined immunodeficiencies, 6 patients from CVID and 2 patients from Xlinked agammaglobulinemia (13).…”

Section: Discussionmentioning

confidence: 99%

Rubella vaccine–induced granulomas are a novel phenotype with incomplete penetrance of genetic defects in cytotoxicity

Groß

Speckmann

May

et al. 2022

Journal of Allergy and Clinical Immunology

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Section: Discussionmentioning

confidence: 99%

Rubella vaccine–induced granulomas are a novel phenotype with incomplete penetrance of genetic defects in cytotoxicity

Groß

Speckmann

May

et al. 2022

Journal of Allergy and Clinical Immunology

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“…Detailed case descriptions have been published separately [69, 70]. Briefly, all four patients received MMR vaccination at about one year of age and one patient received the second dose at age 5 (the OR case).…”

Section: Methodsmentioning

confidence: 99%

Infectious vaccine-derived rubella viruses emerge, persist, and evolve in cutaneous granulomas of children with primary immunodeficiencies

et al. 2019

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Rubella viruses (RV) have been found in an association with granulomas in children with primary immune deficiencies (PID). Here, we report the recovery and characterization of infectious immunodeficiency-related vaccine-derived rubella viruses (iVDRV) from diagnostic skin biopsies of four patients. Sequence evolution within PID hosts was studied by comparison of the complete genomic sequences of the iVDRVs with the genome of the vaccine virus RA27/3. The degree of divergence of each iVDRV correlated with the duration of persistence indicating continuous intrahost evolution. The evolution rates for synonymous and nonsynonymous substitutions were estimated to be 5.7 x 10−3 subs/site/year and 8.9 x 10−4 subs/site/year, respectively. Mutational spectra and signatures indicated a major role for APOBEC cytidine deaminases and a secondary role for ADAR adenosine deaminases in generating diversity of iVDRVs. The distributions of mutations across the genes and 3D hotspots for amino acid substitutions in the E1 glycoprotein identified regions that may be under positive selective pressure. Quasispecies diversity was higher in granulomas than in recovered infectious iVDRVs. Growth properties of iVDRVs were assessed in WI-38 fibroblast cultures. None of the iVDRV isolates showed complete reversion to wild type phenotype but the replicative and persistence characteristics of iVDRVs were different from those of the RA27/3 vaccine strain, making predictions of iVDRV transmissibility and teratogenicity difficult. However, detection of iVDRV RNA in nasopharyngeal specimen and poor neutralization of some iVDRV strains by sera from vaccinated persons suggests possible public health risks associated with iVDRV carriers. Detection of IgM antibody to RV in sera of two out of three patients may be a marker of virus persistence, potentially useful for identifying patients with iVDRV before development of lesions. Studies of the evolutionary dynamics of iVDRV during persistence will contribute to development of infection control strategies and antiviral therapies.

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“…The majority of these patients have an identifiable underlying immunodeficiency 6 . Classically, the disease presents a few months to several years after vaccination with live‐virus RuV vaccine, as in our case 1,2 . Four immunocompetent adults with RuV in cutaneous granulomas have recently been described 7 .…”

Section: Discussionmentioning

confidence: 82%

“…Granulomatous dermatitis associated with rubella virus (RuV) is rare, and most commonly seen in children with primary immunodeficiencies, largely DNA repair disorders affecting T cells 1–4 . Skin lesions are most commonly found on the face and limbs 1–3 . When facial involvement has been reported, the lesions were reported to involve the cheeks and nose 1,2 .…”

Section: Introductionmentioning

confidence: 99%

Rubella virus‐associated necrotizing granulomatous inflammation with extensive eyelid, ocular, and orbital involvement

Pimentel

Kim

Walker

et al. 2023

Pediatric Dermatology

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Cutaneous Granulomatous Disease With Presence of Rubella Virus in Lesions

Cited by 10 publications

References 7 publications

Rubella vaccine–induced granulomas are a novel phenotype with incomplete penetrance of genetic defects in cytotoxicity

Rubella vaccine–induced granulomas are a novel phenotype with incomplete penetrance of genetic defects in cytotoxicity

Infectious vaccine-derived rubella viruses emerge, persist, and evolve in cutaneous granulomas of children with primary immunodeficiencies

Rubella virus‐associated necrotizing granulomatous inflammation with extensive eyelid, ocular, and orbital involvement

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