Abstract | IMPORTANCE: Immunodeficiency-related, vaccine-derived rubella virus (RuV) as an antigenic trigger of cutaneous and visceral granulomas is a rare, recently described phenomenon in children and young adults treated with immunosuppressant agents. OBJECTIVE: To perform a comprehensive clinical, histologic, immunologic, molecular, and genomic evaluation to elucidate the potential cause of an adult patient's atypical cutaneous granulomas. DESIGN, SETTING, AND PARTICIPANTS: A prospective evaluation of skin biopsies, nasopharyngeal swabs, and serum samples submitted to the Centers for Disease Control and Prevention was conducted to assess for RuV using real-time reverse-transcriptase polymerase chain reaction (RT-PCR) and viral genomic sequencing. The samples were obtained from a man in his 70s with extensive cutaneous granulomas mimicking both cutaneous sarcoidosis (clinically) and CD8+ granulomatous cutaneous T-cell lymphoma (histopathologically). The study was conducted from September 2019 to February 2021. MAIN OUTCOMES AND MEASURES: Identification and genotyping of a novel immunodeficiency-related RuV-associated granulomatous dermatitis. RESULTS: Immunohistochemistry for RuV capsid protein and RT-PCR testing for RuV RNA revealed RuV in 4 discrete skin biopsies from different body sites. In addition, RuV RNA was detected in the patient's nasopharyngeal swabs by RT-PCR. The full viral genome was sequenced from the patient's skin biopsy (RVs/Philadelphia.PA.USA/46.19/GR, GenBank Accession #MT249313). The patient was ultimately diagnosed with a novel RuV-associated granulomatous dermatitis. CONCLUSIONS AND RELEVANCE: The findings of this study suggest that clinicians and pathologists may consider RuV-associated granulomatous dermatitis during evaluation of a patient because it might have implications for the diagnosis of cutaneous sarcoidosis, with RuV serving as a potential antigenic trigger, and for the diagnosis of granulomatous cutaneous T-cell lymphoma, with histopathologic features that may prompt an evaluation for immunodeficiency and/or RuV.
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Authors | Bridget E Shields, Ludmila Perelygina, Sara Samimi, Paul Haun, Thomas Leung, Emily Abernathy, Min-Hsin Chen, LiJuan Hao, Joseph Icenogle, Beth Drolet, Barbara Wilson, Joshua S Bryer, Ross England, Emily Blumberg, Karolyn A Wanat, Kathleen Sullivan, Misha Rosenbach |
Journal | JAMA dermatology
(JAMA Dermatol)
Vol. 157
Issue 7
Pg. 842-847
(Jul 01 2021)
ISSN: 2168-6084 [Electronic] United States |
PMID | 34037685
(Publication Type: Journal Article)
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Topics |
- Adult
- Child
- Dermatitis
(complications, etiology)
- Humans
- Male
- Rubella
(complications)
- Rubella virus
(genetics)
- Skin Neoplasms
(complications)
- United States
- Virus Diseases
(complications)
- Young Adult
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