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Rheumatologic manifestations and vasculitis are considered rare manifestations in MSMD patients.<\/jats:p><\/jats:sec><jats:sec><jats:title>Case presentation<\/jats:title><jats:p>In this study, we reported a 20-year-old female who was presented with recurrent lymphadenitis following bacillus Calmette-Gu\u00e9rin (BCG) vaccination and a history of recurrent disseminated rash diagnosed as leukocytoclastic vasculitis (LCV). A slight reduction in lymphocyte subsets including CD4+, CD19+, and CD 16\u2009+\u200956\u2009T-cell count, as well as an elevation in immunoglobulins level (IgG, IgA, IgM, IgE), were observed in the patient. Whole exome sequencing revealed a homozygous Indel-frameshift mutation, c.527_528delCT (p. S176Cfs*12), at the exon 5 of the<jats:italic>IL12B<\/jats:italic>gene. She experienced symptom resolution after treatment with anti-mycobacterial agents and subcutaneous IFN-\u03b3. We conducted a manual literature search for MSMD patients reported with vasculitis in PubMed, Web of Science, and Scopus databases. A total of 18 MSMD patients were found to be affected by a variety of vasculitis phenotypes mainly including LCV and Henoch-Sch\u00f6nlein purpura (HSP) with often skin involvement. Patients were all involved with vasculitis at the median age of 6.8 (2.6\u20137.7) years, nearly 6.1\u2009years after the initial presentations. Sixteen patients (88.9%) had<jats:italic>IL12RB1<\/jats:italic>defects and concurrent<jats:italic>Salmonella<\/jats:italic>infection was reported in 15 (88.2%) patients.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>The lack of IL-12 and IL-23 signaling\/activity\/function and salmonella infection may be triggering factors for the development of leukocytoclastic vasculitis. IL12B or IL12RB1 deficiency and salmonellosis should be considered in MSMD patients with vasculitis.<\/jats:p><\/jats:sec>","DOI":"10.1186\/s12969-021-00623-0","type":"journal-article","created":{"date-parts":[[2021,8,13]],"date-time":"2021-08-13T07:03:24Z","timestamp":1628838204000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["Leukocytoclastic vasculitis in patients with IL12B or IL12RB1 deficiency: case report and review of the literature"],"prefix":"10.1186","volume":"19","author":[{"given":"Niusha","family":"Sharifinejad","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6224-4797","authenticated-orcid":false,"given":"Seyed Alireza","family":"Mahdaviani","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mahnaz","family":"Jamee","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zahra","family":"Daneshmandi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Afshin","family":"Moniri","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Majid","family":"Marjani","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Payam","family":"Tabarsi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Parisa","family":"Farnia","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mahsa","family":"Rekabi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mazdak","family":"Fallahi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Seyedeh Atefeh","family":"Hashemimoghaddam","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masoumeh","family":"Mohkam","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jacinta","family":"Bustamante","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jean-Laurent","family":"Casanova","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Davood","family":"Mansouri","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ali Akbar","family":"Velayati","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,8,13]]},"reference":[{"issue":"5368","key":"623_CR1","doi-asserted-by":"publisher","first-page":"1432","DOI":"10.1126\/science.280.5368.1432","volume":"280","author":"F Altare","year":"1998","unstructured":"Altare F, Durandy A, Lammas D, Emile JF, Lamhamedi S, Le Deist F, et al. 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