<?xml version="1.0" encoding="UTF-8"?> <!DOCTYPE article PUBLIC "-//NLM//DTD JATS (Z39.96) Journal Publishing DTD v1.2d1 20170631//EN" "JATS-journalpublishing1.dtd"> <ArticleSet> <Article> <Journal> <PublisherName>jmedicalcasereports</PublisherName> <JournalTitle>Frontiers in Medical Case Reports</JournalTitle> <PISSN>I</PISSN> <EISSN>S</EISSN> <Volume-Issue>Volume 4; Issue 3</Volume-Issue> <PartNumber/> <IssueTopic>Multidisciplinary</IssueTopic> <IssueLanguage>English</IssueLanguage> <Season>(May-Jun, 2023)</Season> <SpecialIssue>N</SpecialIssue> <SupplementaryIssue>N</SupplementaryIssue> <IssueOA>Y</IssueOA> <PubDate> <Year>-0001</Year> <Month>11</Month> <Day>30</Day> </PubDate> <ArticleType>Medical Case Reports</ArticleType> <ArticleTitle>Digital Gangrene: A Rare Cutaneous Manifestation of Systemic Lupus Erythematosus in The Absence of Anti-Phospholipid Antibodies</ArticleTitle> <SubTitle/> <ArticleLanguage>English</ArticleLanguage> <ArticleOA>Y</ArticleOA> <FirstPage>1</FirstPage> <LastPage>9</LastPage> <AuthorList> <Author> <FirstName>Lamia</FirstName> <LastName>Bengherbia</LastName> <AuthorLanguage>English</AuthorLanguage> <Affiliation/> <CorrespondingAuthor>N</CorrespondingAuthor> <ORCID/> <FirstName>Said</FirstName> <LastName>Taharboucht</LastName> <AuthorLanguage>English</AuthorLanguage> <Affiliation/> <CorrespondingAuthor>Y</CorrespondingAuthor> <ORCID/> <FirstName>Oussama</FirstName> <LastName>Souas</LastName> <AuthorLanguage>English</AuthorLanguage> <Affiliation/> <CorrespondingAuthor>Y</CorrespondingAuthor> <ORCID/> <FirstName>Nadia</FirstName> <LastName>Touati</LastName> <AuthorLanguage>English</AuthorLanguage> <Affiliation/> <CorrespondingAuthor>Y</CorrespondingAuthor> <ORCID/> <FirstName>Hassina</FirstName> <LastName>Chicha</LastName> <AuthorLanguage>English</AuthorLanguage> <Affiliation/> <CorrespondingAuthor>Y</CorrespondingAuthor> <ORCID/> <FirstName>Ahcene</FirstName> <LastName>Chibane</LastName> <AuthorLanguage>English</AuthorLanguage> <Affiliation/> <CorrespondingAuthor>Y</CorrespondingAuthor> <ORCID/> </Author> </AuthorList> <DOI/> <Abstract>Distal gangrene is a rare and serious manifestation of systemic lupus (LS). Secondary to vasculitis, this complication is most frequently associated with the presence of antipospholipid antibodies. Gangrene affecting all four extremities at the same time is exceptional in addition in the absence of antiphospholide antibodies. We report the observation of a 52-year-old patient with an LS for 16 years, who presented for necrosis of the four extremities of simultaneous installation evolving for 01 month. In addition to the noted dry gangrene, she had peripheral sensorimotor neuropathy in the lower extremities. Despite general corticosteroid therapy, its management required amputation of the fingers and forefeet. We also propose a review of the literature with an analysis of the cases reported to date, i.e. 85 cases including our observation.</Abstract> <AbstractLanguage>English</AbstractLanguage> <Keywords>Lupus,Digital Necrosis,Gangrene,Vasculitis,Peripheral Neuropathy</Keywords> <URLs> <Abstract>https://www.jmedicalcasereports.org/ubijournal-v1copy/journals/abstract.php?article_id=14613&title=Digital Gangrene: A Rare Cutaneous Manifestation of Systemic Lupus Erythematosus in The Absence of Anti-Phospholipid Antibodies</Abstract> </URLs> <References> <ReferencesarticleTitle>References</ReferencesarticleTitle> <ReferencesfirstPage>16</ReferencesfirstPage> <ReferenceslastPage>19</ReferenceslastPage> <References>Adelowo O, Olaosebikan H, Ajani W, Omosebi DT. Digital gangrene as the initial presentation of systemic lupus erythematosus. BMJ Case Rep 2012; 2012: bcr2012006259. Alalawi ZM, Alkenany S, Almahroos F, Albloushi B. Peripheral Gangrene as the Initial Presentation of Systemic Lupus Erythematosus in Emergency Department. Cureus 2020; 12: e6667. Alghamdi M. Digital gangrene as the initial presentation of systemic lupus erythematosus without secondary antiphospholipid syndrome: case report. Zagazig University Medical Journal 2015; 21: 1-4. Alzughayyar TZ, Zalloum JS, Elqadi MN, Abukhalaf SA, Abunejma FM, Alkhanafsa MMY, Haif MHM, Alqarajeh F, Mesk MR, Misk RA. Systemic Lupus Erythematosus with Multiple Autoimmune Disease Presented with Extensive Peripheral Gangrene. Case Rep Rheumatol 2020; 2020: 8278275. Ando T, Yamasaki Y, Takakuwa Y, Iida H, Asari Y, Suzuki K, Uchida M, Kotoku N, Tanabe Y, Chosokabe M, Takahashi M, Suzuki K, Akashi YJ, Yamada H, Kawahata K. Concurrent onset of acute lupus myocarditis, pulmonary arterial hypertension and digital gangrene in a lupus patient: a possible role of vasculitis to the rare disorders. Mod Rheumatol Case Rep 2020; 4: 21-27. Asherson RA, Cervera R, Shoenfeld Y. Peripheral vascular occlusions leading to gangrene and amputations in antiphospholipid antibody positive patients. Ann N Y Acad Sci 2007; 1108: 515-529. Cervera R, Khamashta MA, Font J, Sebastiani GD, Gil A, Lavilla P, Mejand;iacute;a JC, Aydintug AO, Chwalinska-Sadowska H, de Ramand;oacute;n E, Fernand;aacute;ndez-Nebro A, Galeazzi M, Valen M, Mathieu A, Houssiau F, Caro N, Alba P, Ramos-Casals M, Ingelmo M, Hughes GR; European Working Party on Systemic Lupus Erythematosus. Morbidity and mortality in systemic lupus erythematosus during a 10-year period: a comparison of early and late manifestations in a cohort of 1,000 patients. Medicine (Baltimore) 2003; 82: 299-308. Cheah JS. Systemic lupus erythematosus in a Chinese woman presenting with gangrene of the fingers. Aust N Z J Med 1973; 3: 197-199. Dawson GW. Extensive Lupus Erythematosus associated with Symmetrical Gangrene in a Man, aged 49. Proc R Soc Med 1911; 4: 101. Dubois EL, Arterberry JD. Gangrene as a manifestation of systemic lupus erythematosus. JAMA 1962; 181: 366-374. Ha-Ou-Nou FZ, Zahlane M, Benjilali L, Essaadouni L. Extensive digital gangrene revealing late-onset systemic lupus erythematosus. J Med Vasc 2017; 42: 50-53. Hochberg MC. Updating the American College of Rheumatology revised criteria for the classification of systemic lupus erythematosus. Arthritis Rheum 1997; 40: 1725. Igaki N, Moriguchi R, Hirota Y, Sakai M, Akiyama H, Tamada F, Oimomi M, Goto T. Calciphylaxis in a patient with end-stage renal disease secondary to systemic lupus erythematosus associated with acral gangrene and mesenteric ischemia. Intern Med 2001; 40: 1232-1237. Jeffery RC, Narshi CB, Isenberg DA. Prevalence, serological features, response to treatment and outcome of critical peripheral ischaemia in a cohort of lupus patients. Rheumatology (Oxford) 2008; 47: 1379-1383. Jindal BK, Martin MF, Gayner A. Gangrene developing after minor surgery in a patient with undiagnosed systemic lupus erythematosus and lupus anticoagulant. Ann Rheum Dis 1983; 42: 347-349. Keat EC, Shore JH. Gangrene of the legs in disseminated lupus erythematosus. Br Med J 1958; 1: 25-27. Kurnia E, Hardi F, Dewiasty E. Digital gangrene in systemic lupus erythematosus. Acta Med Indones 2012; 44: 335-336. Lee HY, Jang JS, Kim HW, Kim YS, Kim YO, Yoon SA. A case of digital gangrene complicated by atypical hemolytic uremic syndrome in a patient with systemic lupus erythematosus. Korean J Nephrol 2010: 392-397. Liu A, Zhang W, Tian X, Zhang X, Zhang F, Zeng X. Prevalence, risk factors and outcome of digital gangrene in 2684 lupus patients. Lupus 2009; 18: 1112-1118. Nagai Y, Shimizu A, Suto M, Tanaka S, Yasuda M, Tago O, Hasegawa M, Ishikawa O. Digital gangrene in systemic lupus erythematosus. Acta Derm Venereol 2009; 89: 398-401. Nkeck JR, Endomba FTA, Ndoadoumgue AL, Kalbassou FB. Gangrene of All Digits and Toes Attributable to Systemic Lupus Erythematosus with Negative Antiphospholipid Antibodies. J Clin Rheumatol 2019; 25: e79-e80. Omair MA, Bookman A, Mittoo S. Digital gangrene in a patient with systemic lupus erythematosus and systemic sclerosis. J Rheumatol 2012; 39: 1895-1897. Rahman A, Isenberg DA. Systemic lupus erythematosus. N Engl J Med 2008; 358: 929-939. Rajasekhar L, Jayachandran NV, Prabu VN, Narsimulu G. Comment on: Prevalence, serological features, response to treatment and outcome of critical peripheral ischaemia in a cohort of lupus patients. Rheumatology (Oxford) 2009; 48: 451-452. Ribero S, Sciascia S, Borradori L, Lipsker D. The Cutaneous Spectrum of Lupus Erythematosus. Clin Rev Allergy Immunol 2017; 53: 291-305. Shetty VB, Rao S, Krishnamurthy PN, Shenoy VU. Peripheral gangrene during infancy: a rare presentation of systemic lupus erythematosus. Arch Dis Child 2001; 85: 335-336. Sonkar SK, Kumar S, Atam V, Chaudhary SC. Digital dry gangrene as a primary manifestation of systemic lupus erythematosus. BMJ Case Rep 2019; 12: e230869. Tuffanelli DL, Dubois EL. Cutaneous Manifestations of Systemic Lupus Erythematosus. Arch Dermatol 1964; 90: 377-386. Vijay S, Imthiaz VK, Hitesh S. Peripheral Gangrene Complicating Systemic Lupus Erythematosus in a Patient with Spina Bifida: A Case Report. Malays Orthop J 2017; 11: 68-70. Vocks E, Welcker M, Ring J. Digital gangrene: a rare skin symptom in systemic lupus erythematosus. J Eur Acad Dermatol Venereol 2000; 14: 419-421. Whiting DA. Systemic lupus erythematosus with cryoglobulinaemia, peripheral gangrene, and transplacental passage of the LE factor. A case report. S Afr Med J 1967; 41: 479-482. Yang SG, Kim KH, Park KC, Youn SW, Joh GY. A case of systemic lupus erythematosus showing acute gangrenous change of fingertips. Br J Dermatol 1996; 134: 185-187. Yasmin F, Jawaid H, Batra S, Vighio AA, Shaikh MA. A rare case of gangrenous foot in juvenile systemic lupus erythematosus. J Pak Med Assoc 2020; 70: 1869-1873.</References> </References> </Journal> </Article> </ArticleSet>