{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,22]],"date-time":"2026-08-22T12:50:27Z","timestamp":1787403027606,"version":"build-2736575974"},"reference-count":28,"publisher":"Springer Science and Business Media LLC","issue":"3","license":[{"start":{"date-parts":[[2024,12,12]],"date-time":"2024-12-12T00:00:00Z","timestamp":1733961600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2024,12,12]],"date-time":"2024-12-12T00:00:00Z","timestamp":1733961600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["CEN Case Rep"],"abstract":"<jats:title>Abstract<\/jats:title>\n          <jats:p>Visceral disseminated varicella zoster virus (VZV) infection is a severe complication, characterized by a notably high mortality rate. Herein, we present a case of a 36-year-old-man involving visceral disseminated VZV infection that emerged during remission induction therapy involving high-dose prednisolone (PSL), mycophenolate mofetil (MMF), and hydroxychloroquine for lupus nephritis. Two months after starting the immunosuppressive therapy, he experienced a rapid onset of severe upper abdominal pain. The following day, clinical manifestations and laboratory abnormalities rapidly deteriorated. Hyperferritinemia and hypertriglyceridemia, indicative of hemophagocytic lymphohistiocytosis (HLH), emerged, along with escalating liver and renal impairment and newly appeared disseminated intravascular coagulation, and multiple organ failure is suggested. The patient developed widespread blistering predominantly on the trunk and face, patient\u2019s condition failed to ameliorate, ultimately culminating in his demise a few hours later. At autopsy, the cutaneous lesions with blisters revealed positive immunostaining with anti-VZV antibody, and similar findings were detected in multiple organs. HLH was confirmed in lymph nodes. It is crucial to emphasize the awareness of visceral disseminated VZV, particularly in cases patients are undergoing concurrent PSL therapy alongside MMF for SLE. The progression of this fatal condition usually begins with abdominal pain, followed by a skin rash a few days later. The present case is the first to show evidence of HLH occurring as a result of visceral disseminated VZV infection. This disease is extremely rare but extremely serious, therefore, VZV-DNA should be measured in cases where you suspect this disease for early diagnosis and treatment.<\/jats:p>","DOI":"10.1007\/s13730-024-00950-7","type":"journal-article","created":{"date-parts":[[2024,12,12]],"date-time":"2024-12-12T15:59:49Z","timestamp":1734019189000},"page":"477-485","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Fatal visceral disseminated varicella zoster virus infection during initial remission induction therapy in a patient with lupus nephritis: a case report and review of the literature"],"prefix":"10.1007","volume":"14","author":[{"given":"Runa","family":"Takehara","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0002-1089-6755","authenticated-orcid":false,"given":"Itaru","family":"Ebihara","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yoshifumi","family":"Honda","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Norimasa","family":"Ooba","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiromi","family":"Kurosawa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chihiro","family":"Sato","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Haruo","family":"Ohtani","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yutaka","family":"Tsutsumi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masato","family":"Nose","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masaki","family":"Kobayashi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2024,12,12]]},"reference":[{"key":"950_CR1","doi-asserted-by":"publisher","first-page":"e13062","DOI":"10.1111\/tid.13062","volume":"12","author":"MJ Loftus","year":"2019","unstructured":"Loftus MJ, Yong MK, Wilson S, Peleg AY. Fatal disseminated visceral varicella zoster virus infection in a renal transplant recipient. Transpl Infect Dis. 2019;12:e13062.","journal-title":"Transpl Infect Dis"},{"key":"950_CR2","doi-asserted-by":"publisher","first-page":"361","DOI":"10.1016\/j.ejim.2004.11.018","volume":"16","author":"H Hyvernat","year":"2005","unstructured":"Hyvernat H, Roger PM, Pereira C, Saint-Paul MC, Vandenbos F, Bernardin G. Fatal Varicella hepatitis in an asthmatic adult after short-term corticosteroid treatment. Eur J Intern Med. 2005;16:361.","journal-title":"Eur J Intern Med"},{"key":"950_CR3","doi-asserted-by":"publisher","first-page":"2814","DOI":"10.1016\/j.transproceed.2012.09.090","volume":"44","author":"M Rommelaere","year":"2012","unstructured":"Rommelaere M, Marechal C, Yombi JC, Goffin E, Kanaan N. Disseminated varicella zoster virus infection in adult renal transplant recipients:outcome and risk factors. Transpl Proc. 2012;44:2814\u20137.","journal-title":"Transpl Proc"},{"key":"950_CR4","doi-asserted-by":"publisher","first-page":"810","DOI":"10.1111\/j.1572-0241.1998.230_a.x","volume":"93","author":"DS David","year":"1998","unstructured":"David DS, Tegtmeier BR, O\u2019Donnell MR, Paz IB, McCarty TM. Visceral varicella-zoster after bone marrow transplantation: report of a case series and review of the literature. Am J Gastroenterol. 1998;93:810\u20133.","journal-title":"Am J Gastroenterol"},{"key":"950_CR5","doi-asserted-by":"publisher","first-page":"314","DOI":"10.1111\/tid.12073","volume":"15","author":"N Doki","year":"2013","unstructured":"Doki N, Miyawaki S, Tanaka M, Kudo D, Wake A, Oshima K, et al. Visceral varicella zoster virus infection after allogeneic stem cell transplantation. Transpl Infect Dis. 2013;15:314\u20138.","journal-title":"Transpl Infect Dis"},{"key":"950_CR6","doi-asserted-by":"publisher","first-page":"1758","DOI":"10.1016\/S0041-1345(03)00684-5","volume":"35","author":"R Lauzurica","year":"2003","unstructured":"Lauzurica R, Bayes B, Frias C, Fontsere N, Hernandez A, Matas L, et al. Disseminated varicella infection in adult renal allograft recipients: role of mycophenolate mofetil. Transpl Proc. 2003;35:1758\u20139.","journal-title":"Transpl Proc"},{"key":"950_CR7","doi-asserted-by":"publisher","first-page":"608","DOI":"10.1097\/00007890-200202270-00023","volume":"73","author":"T Fehr","year":"2002","unstructured":"Fehr T, Bossart W, Wahl C, Binswanger U. Disseminated varicella infection in adult renal allograft recipients: four cases and a review of the literature. Transplantation. 2002;73:608\u201311.","journal-title":"Transplantation"},{"key":"950_CR8","doi-asserted-by":"publisher","first-page":"155","DOI":"10.3109\/14397595.2014.948981","volume":"27","author":"H Tsuji","year":"2017","unstructured":"Tsuji H, Yoshifuji H, Fujii T, Matsuo T, Nakashima R, Imura Y, et al. Visceral disseminated varicella zoster virus infection after rituximab treatment for granulomatosis with polyangiitis. Mod Rheumatol. 2017;27:155\u201361.","journal-title":"Mod Rheumatol"},{"issue":"1","key":"950_CR9","doi-asserted-by":"publisher","first-page":"538","DOI":"10.1186\/s12879-020-05254-6","volume":"20","author":"V Vassia","year":"2020","unstructured":"Vassia V, Croce A, Ravanini P, et al. Unusual presentation of fatal disseminated varicella zoster virus infection in a patient with lupus nephritis: a case report. BMC Infect Dis. 2020;20(1):538.","journal-title":"BMC Infect Dis"},{"issue":"1","key":"950_CR10","doi-asserted-by":"publisher","first-page":"164","DOI":"10.1186\/s12882-023-03223-0","volume":"24","author":"I Yuri","year":"2023","unstructured":"Yuri I, Hirotaka F, Shuhei K, et al. A survival case of visceral disseminated varicella zoster virus infection in a patient with systemic lupus erythematosus. BMC Nephrol. 2023;24(1):164.","journal-title":"BMC Nephrol"},{"issue":"1","key":"950_CR11","doi-asserted-by":"publisher","first-page":"165","DOI":"10.1186\/s13104-018-3271-3","volume":"11","author":"H Masato","year":"2018","unstructured":"Masato H, Yoko W, Yoichi K, et al. Fatal visceral disseminated varicella zoster infection during initial remission induction therapy in a patient with lupus nephritis and rheumatoid arthritis\u2014possible association with mycophenolate mofetil and high-dose glucocorticoid therapy: a case report. BMC Res Notes. 2018;11(1):165.","journal-title":"BMC Res Notes"},{"issue":"14","key":"950_CR12","doi-asserted-by":"publisher","first-page":"e33459","DOI":"10.1097\/MD.0000000000033459","volume":"102","author":"T Tian","year":"2023","unstructured":"Tian T, Jun C, Kunlan L, et al. Severe varicella-zoster virus meningoencephalomyelitis coexisting with visceral disseminated varicella-zoster virus infection in a patient with lupus nephritis A case report. Medicine (Baltimore). 2023;102(14):e33459.","journal-title":"Medicine (Baltimore)"},{"key":"950_CR13","first-page":"18","volume":"61","author":"R Saito","year":"2011","unstructured":"Saito R, Yano K, Katsushima F, et al. A case of varicella with severe back pain in a patient with systemic lupus erythematosus. Fukushima Med Mag. 2011;61:18\u201323.","journal-title":"Fukushima Med Mag"},{"issue":"1","key":"950_CR14","doi-asserted-by":"publisher","first-page":"57","DOI":"10.1093\/mrcr\/rxac054","volume":"7","author":"K Hattori","year":"2023","unstructured":"Hattori K, Tanaka S, Ishikawa J, et al. Visceral disseminated varicella zoster virus infection during non-intensive maintenance therapy in a patient with systemic lupus ervthematosus. Mod Rheumatol Case Rep. 2023;7(1):57\u20139. https:\/\/doi.org\/10.1093\/mrcr\/rxac054.","journal-title":"Mod Rheumatol Case Rep"},{"key":"950_CR15","doi-asserted-by":"publisher","first-page":"1725","DOI":"10.1002\/art.1780400928","volume":"40","author":"MC Hochberg","year":"1997","unstructured":"Hochberg MC. Updating the American College of Rheumatology revised criteria for the classification of systemic lupus erythematosus. Arthritis Rheum. 1997;40:1725.","journal-title":"Arthritis Rheum"},{"key":"950_CR16","doi-asserted-by":"publisher","first-page":"2677","DOI":"10.1002\/art.34473","volume":"64","author":"M Petri","year":"2012","unstructured":"Petri M, Orbai AM, Alarcon GS, Gordon C, et al. Derivation and validation of the systemic lupus international collaborating clinics classification criteria for systemic lupus erythematosus. Arthritis Rheum. 2012;64:2677\u201386.","journal-title":"Arthritis Rheum"},{"issue":"4","key":"950_CR17","first-page":"789","volume":"93","author":"MB Ingeborg","year":"2018","unstructured":"Ingeborg MB, Suzanne W, Charles EA, et al. Revision of the International Society of Nephrology\/Renal Pathology Society classification for lupus nephritis: clarification of definitions, and modified National Institutes of Health activity and chronicity indices. Pract Guidel Kidney Intactions. 2018;93(4):789\u201396.","journal-title":"Pract Guidel Kidney Intactions."},{"issue":"2","key":"950_CR18","doi-asserted-by":"publisher","first-page":"124","DOI":"10.1002\/pbc.21039","volume":"48","author":"JI Henter","year":"2007","unstructured":"Henter JI, Horne A, Aric\u00f3 M, Egeler RM, Filipovich AH, Imashuku S, et al. HLH-2004: diagnostic and therapeutic guidelines for hemophagocytic lymphohistiocytosis. Pediatr Blood Cancer. 2007;48(2):124\u201331. https:\/\/doi.org\/10.1002\/pbc.21039.","journal-title":"Pediatr Blood Cancer"},{"key":"950_CR19","doi-asserted-by":"publisher","first-page":"2266","DOI":"10.1056\/NEJMp058091","volume":"352","author":"A Arvin","year":"2005","unstructured":"Arvin A. Aging, immunity, and the varicella-zoster virus. N Engl J Med. 2005;352:2266\u20137.","journal-title":"N Engl J Med"},{"issue":"5","key":"950_CR20","doi-asserted-by":"publisher","first-page":"451","DOI":"10.1093\/qjmed\/hct046","volume":"106","author":"J Merayo-Chalico","year":"2013","unstructured":"Merayo-Chalico J, G\u00f3mez-Mart\u00edn D, Pi\u00f1eir\u00faa-Men\u00e9ndez A, et al. Lymphopenia as risk factor for development of severe infections in patients with systemic lupus erythematosus: a case\u2013control study. QJM. 2013;106(5):451\u20137.","journal-title":"QJM"},{"key":"950_CR21","doi-asserted-by":"publisher","first-page":"2328","DOI":"10.1002\/art.39670","volume":"68","author":"H Yun","year":"2016","unstructured":"Yun H, Yang S, Chen L, et al. Risk of herpes zoster in autoimmune and inflammatory diseases: implications for vaccination. Arthritis Rheumatol. 2016;68:2328\u201337.","journal-title":"Arthritis Rheumatol"},{"key":"950_CR22","doi-asserted-by":"publisher","first-page":"238","DOI":"10.1177\/0961203312470186","volume":"22","author":"EF Chakravarty","year":"2013","unstructured":"Chakravarty EF, Michaud K, Katz R, et al. Increased incidence of herpes zoster among patients with systemic lupus erythematosus. Lupus. 2013;22:238\u201344.","journal-title":"Lupus"},{"issue":"Suppl 1","key":"950_CR23","doi-asserted-by":"publisher","first-page":"S91","DOI":"10.1086\/342963","volume":"186","author":"JW Gnann Jr","year":"2002","unstructured":"Gnann JW Jr. Varicella-zoster virus: atypical presentations and unusual complications. J Infect Dis. 2002;186(Suppl 1):S91\u20138.","journal-title":"J Infect Dis"},{"key":"950_CR24","doi-asserted-by":"publisher","first-page":"119","DOI":"10.1097\/RHU.0b013e3181d52ed7","volume":"16","author":"EF Borba","year":"2010","unstructured":"Borba EF, Ribeiro AC, Martin P, Costa LP, Guedes LK, Bonf\u00e1 E. Incidence, risk factors, and outcome of herpes zoster in systemic lupus erythematosus. J Clin Rheumatol. 2010;16:119\u201322.","journal-title":"J Clin Rheumatol"},{"key":"950_CR25","doi-asserted-by":"crossref","unstructured":"Sayeeda A, Al Arfaj H, Khalil N, Al Arfaj AS. Herpes zoster infections in SLE in a University Hospital in Saudi Arabia: risk factors and outcomes. Autoimmune Dis. 2010.","DOI":"10.4061\/2010\/174891"},{"key":"950_CR26","doi-asserted-by":"publisher","first-page":"746","DOI":"10.1093\/mr\/roab076","volume":"32","author":"T Takeuchi","year":"2022","unstructured":"Takeuchi T, Hashimotob H, Matsumotob M, et al. Long-term safety and effectiveness of mycophenolate mofetil in adults with lupus nephritis: a real-world study in Japan. Mod Rheumatol. 2022;32:746\u201354.","journal-title":"Mod Rheumatol"},{"key":"950_CR27","doi-asserted-by":"publisher","first-page":"158","DOI":"10.1097\/00007890-199907150-00030","volume":"68","author":"WS Rothwell","year":"1999","unstructured":"Rothwell WS, Gloor JM, Morgenstern BZ, Milliner DS. Disseminated varicella infection in pediatric renal transplant recipients treated with mycophenolate mofetil. Transplantation. 1999;68:158\u201361.","journal-title":"Transplantation"},{"issue":"1","key":"950_CR28","doi-asserted-by":"publisher","first-page":"625","DOI":"10.1186\/s12879-019-4193-y","volume":"19","author":"Y Furuto","year":"2019","unstructured":"Furuto Y, Kawamura M, Namikawa A, et al. Successful management of visceral disseminated varicella zoster virus infection during treatment of membranous nephropathy: a case report. BMC Infect Dis. 2019;19(1):625.","journal-title":"BMC Infect Dis"}],"container-title":["CEN Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s13730-024-00950-7.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s13730-024-00950-7\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s13730-024-00950-7.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,6,1]],"date-time":"2025-06-01T15:01:34Z","timestamp":1748790094000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s13730-024-00950-7"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,12,12]]},"references-count":28,"journal-issue":{"issue":"3","published-online":{"date-parts":[[2025,6]]}},"alternative-id":["950"],"URL":"https:\/\/doi.org\/10.1007\/s13730-024-00950-7","relation":{},"ISSN":["2192-4449"],"issn-type":[{"value":"2192-4449","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,12,12]]},"assertion":[{"value":"17 September 2024","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"8 November 2024","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"12 December 2024","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The authors have declared that no Conflict of interest exists.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}},{"value":"This article does not contain any studies with human participants or animals performed by any of the authors.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethical standards"}},{"value":"Written informed consent to publish the case was obtained from the next of kin of the patient (concerning his personal or clinical details along with any identifying images to be published in this study).","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}}]}}