Case Report: Kaposi Sarcoma in Anti-Glomerular Basement Membrane Disease

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Its management requires aggressive immunosuppressive therapy, including corticosteroids, cyclophosphamide, and plasmapheresis. Kaposi sarcoma (KS) is a vascular neoplasm linked to human herpes virus 8 (HHV-8), most frequently seen in HIV-infected or iatrogenically immunosuppressed patients. We report the case of a 39-year-old man with anti-GBM vasculitis who developed cutaneous Kaposi sarcoma two months after initiation of immunosuppressive treatment. HHV-8–positive spindle cell proliferation was confirmed on skin biopsy. HIV testing was negative, and the lesions regressed following the tapering of corticosteroids and withdrawal of cyclophosphamide. This case highlights the importance of considering KS as a differential diagnosis in patients developing skin lesions under immunosuppressive therapy for autoimmune vasculitis." } { "@context": "http://schema.org", "@type": "BreadcrumbList", "itemListElement": [ { "@type": "ListItem", "position": "1", "item": { "@id": "https://f1000research.com/", "name": "Home" } }, { "@type": "ListItem", "position": "2", "item": { "@id": "https://f1000research.com/browse/articles", "name": "Browse" } }, { "@type": "ListItem", "position": "3", "item": { "@id": "https://f1000research.com/articles/14-878/v1", "name": "Case Report: Kaposi Sarcoma in Anti-Glomerular Basement Membrane Disease" } } ] } Home Browse Case Report: Kaposi Sarcoma in Anti-Glomerular Basement Membrane Disease ALL Metrics - Views Downloads Get PDF Get XML Cite How to cite this article Guesmi R, Boukadida R, Mrabet S et al. Case Report: Kaposi Sarcoma in Anti-Glomerular Basement Membrane Disease [version 1; peer review: 1 approved with reservations] . F1000Research 2025, 14 :878 ( https://doi.org/10.12688/f1000research.165476.1 ) NOTE: If applicable, it is important to ensure the information in square brackets after the title is included in all citations of this article. Close Copy Citation Details Export Export Citation Sciwheel EndNote Ref. Manager Bibtex ProCite Sente EXPORT Select a format first Track Share ▬ ✚ Case Report Case Report: Kaposi Sarcoma in Anti-Glomerular Basement Membrane Disease [version 1; peer review: 1 approved with reservations] Rahma Guesmi https://orcid.org/0000-0002-1706-9181 1,2 , Raja Boukadida 2,3 , Sanda Mrabet 2,3 , Dorsaf Zellama 2,3 , Zohra Sassi Jalleli 1 Rahma Guesmi https://orcid.org/0000-0002-1706-9181 1,2 , Raja Boukadida 2,3 , [...] Sanda Mrabet 2,3 , Dorsaf Zellama 2,3 , Zohra Sassi Jalleli 1 PUBLISHED 08 Sep 2025 Author details Author details 1 Nephrology Department, Sidi Bouzid Regional Hospital, Sidi Bouzid, Sidi Bouzid, Tunisia 2 Faculty of Medicine of Sousse, University of Sousse, Sousse, Sousse, Tunisia 3 Nephrology Department, Sahloul Hospital, Sousse, Sousse, Tunisia Rahma Guesmi Roles: Conceptualization, Investigation, Methodology, Resources, Visualization, Writing – Original Draft Preparation Raja Boukadida Roles: Conceptualization, Visualization Sanda Mrabet Roles: Investigation, Supervision Dorsaf Zellama Roles: Supervision Zohra Sassi Jalleli Roles: Investigation, Supervision OPEN PEER REVIEW DETAILS REVIEWER STATUS This article is included in the Rare diseases collection. Abstract Anti-glomerular basement membrane (anti-GBM) disease is a rare and life-threatening autoimmune vasculitis characterized by rapidly progressive glomerulonephritis, with or without pulmonary hemorrhage. Its management requires aggressive immunosuppressive therapy, including corticosteroids, cyclophosphamide, and plasmapheresis. Kaposi sarcoma (KS) is a vascular neoplasm linked to human herpes virus 8 (HHV-8), most frequently seen in HIV-infected or iatrogenically immunosuppressed patients. We report the case of a 39-year-old man with anti-GBM vasculitis who developed cutaneous Kaposi sarcoma two months after initiation of immunosuppressive treatment. HHV-8–positive spindle cell proliferation was confirmed on skin biopsy. HIV testing was negative, and the lesions regressed following the tapering of corticosteroids and withdrawal of cyclophosphamide. This case highlights the importance of considering KS as a differential diagnosis in patients developing skin lesions under immunosuppressive therapy for autoimmune vasculitis. READ ALL READ LESS Keywords Anti-glomerular basement membrane disease, Kaposi sarcoma, vasculitis, immunosuppressive treatment Corresponding Author(s) Rahma Guesmi ( [email protected] ) Close Corresponding author: Rahma Guesmi Competing interests: No competing interests were disclosed. Grant information: The author(s) declared that no grants were involved in supporting this work. Copyright: © 2025 Guesmi R et al . This is an open access article distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. How to cite: Guesmi R, Boukadida R, Mrabet S et al. Case Report: Kaposi Sarcoma in Anti-Glomerular Basement Membrane Disease [version 1; peer review: 1 approved with reservations] . F1000Research 2025, 14 :878 ( https://doi.org/10.12688/f1000research.165476.1 ) First published: 08 Sep 2025, 14 :878 ( https://doi.org/10.12688/f1000research.165476.1 ) Latest published: 08 Sep 2025, 14 :878 ( https://doi.org/10.12688/f1000research.165476.1 ) Introduction Anti-glomerular basement membrane (anti-GBM) disease, also known as Goodpasture’s disease, is a rare but severe autoimmune vasculitis characterized by autoantibodies targeting the non-collagenous domain of the alpha-3 chain of type IV collagen (α3[IV]NC1) in glomerular and alveolar basement membranes. 1 It typically presents as rapidly progressive glomerulonephritis (RPGN), often accompanied by pulmonary hemorrhage, constituting the classic pulmonary–renal syndrome. 2 The condition is life-threatening without prompt diagnosis and aggressive treatment. Standard therapy combines high-dose corticosteroids, cytotoxic agents such as cyclophosphamide, and plasmapheresis to remove circulating antibodies. 3 Kaposi sarcoma (KS) is an angioproliferative neoplasm associated with latent infection by human herpesvirus 8 (HHV-8). It most commonly affects individuals with acquired immunodeficiency, such as people living with HIV/AIDS or organ transplant recipients under long-term immunosuppression. 4 Iatrogenic KS can also emerge in patients receiving immunosuppressive therapy for autoimmune diseases, although this presentation remains uncommon. The cutaneous form of KS typically manifests as violaceous macules, papules, or plaques, predominantly on the lower limbs. Histologically, KS is characterized by spindle cell proliferation with neoangiogenesis and erythrocyte extravasation, and HHV-8 immunohistochemistry is diagnostic. 5 We report a rare case of iatrogenic Kaposi sarcoma in a patient treated for anti-GBM vasculitis. This case illustrates the potential for opportunistic neoplasms to develop in non-transplant autoimmune settings, underscoring the need for dermatologic vigilance in patients undergoing intensive immunosuppressive therapy. Case presentation A 39-year-old man with no significant past medical history presented with gross hematuria that appeared four days after an episode of acute tonsillitis. Physical examination was unremarkable. Laboratory tests revealed acute kidney injury (serum creatinine 196 μmol/L), mild normocytic anemia (hemoglobin 11.9 g/dL), leukocytosis (11,800/mm 3 ), and elevated C-reactive protein (CRP) at 110 mg/L. Urinalysis showed a urinary albumin/creatinine ratio of 0.5 g/g and macroscopic hematuria (600,000 red blood cells/mL), with a sterile urine culture. Abdominal ultrasound was normal. Given the presentation of glomerulonephritis without extrarenal manifestations, an immunologic workup was performed. Antinuclear antibodies and antineutrophil cytoplasmic antibodies were negative, while anti-glomerular basement membrane (anti-GBM) antibodies were strongly positive (+++). Kidney biopsy revealed extracapillary proliferation (crescent formation) in over 60% of glomeruli (9 out of 14) and acute tubular necrosis with early signs of regeneration. A chest computed tomography scan ruled out alveolar hemorrhage. The diagnosis of anti-GBM vasculitis was established, and treatment was initiated with intravenous steroid pulses followed by high-dose oral prednisone, daily oral cyclophosphamide, and a course of plasmapheresis. Despite treatment, the patient’s renal function deteriorated, progressing to anuria and requiring initiation of hemodialysis. Anti-GBM antibodies remained positive, requiring a total of 29 plasmapheresis sessions. During hospitalization, the patient developed multiple infections, including pyelonephritis, infectious diarrhea, and esophageal candidiasis, all of which were treated with broad-spectrum antimicrobials. Approximately two months after starting immunosuppressive therapy, the patient developed violaceous skin lesions on the lower extremities ( Figure 1 ). At that time, he was on oral cyclophosphamide and prednisone 40 mg/day. He remained otherwise asymptomatic. A skin biopsy from the left foot revealed HHV-8–positive spindle cell vascular proliferation without vasculitis, consistent with Kaposi sarcoma ( Figures 2 , 3 , 4 ). HIV testing was negative. Figure 1. Violaceous maculopapular skin lesions on the legs, suggestive of Kaposi sarcoma. Figure 2. Hematoxylin and Eosin staining: a dermal spindle cell proliferation with hemorrhage and pigment deposition. Figure 3. HHV-8 immunohistochemistry showing strong nuclear staining in atypical endothelial cells (x400). Figure 4. Dense capillary proliferation in the dermis with red blood cell extravasation and spindle cells (H&E). Shortly afterward, the patient was admitted for upper gastrointestinal bleeding. Gastroscopy revealed actively bleeding lesions, but biopsy excluded gastrointestinal Kaposi sarcoma. The bleeding was attributed to direct oral anticoagulant, which was prescribed after arteriovenous fistula placement, and resolved with octreotide and proton pump inhibitors. In light of the Kaposi sarcoma diagnosis and absence of renal recovery, cyclophosphamide was discontinued and prednisone was rapidly tapered to 10 mg/day. The patient was referred to dermatology for follow-up but he declined further evaluation. His skin lesions regressed spontaneously over the following weeks. Discussion This case illustrates a rare but significant complication of immunosuppressive therapy in a patient with anti-glomerular basement membrane (anti-GBM) vasculitis. Anti-GBM disease is classically associated with rapidly progressive glomerulonephritis and pulmonary hemorrhage. Therefore, its management requires aggressive immunosuppression that may predispose patients to opportunistic infections and, more rarely, virus-associated malignancies. 1 , 2 Kaposi sarcoma (KS) is a vascular tumor caused by human herpesvirus 8 (HHV-8), which can persist in a latent form and reactivates under immunosuppressive conditions. 4 Iatrogenic KS is typically reported in organ transplant recipients, but it may also occur in patients treated for autoimmune diseases such as systemic lupus erythematosus or vasculitis. 6 In the context of anti-GBM vasculitis, such presentations remain exceptional. Our patient developed KS two months after initiation of immunosuppressive therapy combining corticosteroids and cyclophosphamide. The diagnosis was confirmed histologically by the presence of HHV-8–positive spindle cell vascular proliferation. The regression of skin lesions following reduction of immunosuppression supports the iatrogenic origin of the disease. He had no other recognized risk factors such as HIV infection. While the majority of iatrogenic KS cases occur in transplant medicine, the literature describes rare occurrences in autoimmune vasculitis. Tiong et al. reviewed several such cases, mostly associated with ANCA-associated vasculitis, treated with cyclophosphamide and corticosteroids. 7 The proposed mechanism involves a combination of impaired T-cell–mediated immunity and HHV-8 reactivation. In the absence of systemic involvement, reducing or discontinuing immunosuppression is often sufficient for KS regression. 4 , 6 More advanced or visceral forms may require chemotherapy or antiviral therapy. In our case, the spontaneous regression after drug tapering and the patient’s refusal of further dermatologic management guided a conservative approach. This case underlines the need to include KS among the differential diagnosis of skin lesions in immunosuppressed patients, even outside the context of HIV or organ transplantation. Early recognition allows timely therapeutic adjustment and may avoid unnecessary interventions. Conclusion This case highlights a rare but clinically important complication of immunosuppressive therapy in anti-GBM vasculitis. Although Kaposi sarcoma is typically associated with HIV infection or solid organ transplantation, it may also occur in patients treated for autoimmune diseases. In this context, the development of cutaneous Kaposi sarcoma should prompt consideration of iatrogenic immunosuppression as a precipitating factor. Early dermatologic evaluation and timely tapering of immunosuppressive therapy can lead to spontaneous regression of lesions, as illustrated in this case. Vigilance for unusual cutaneous manifestations is therefore essential during follow-up of patients receiving potent immunosuppressive regimens. Consent to publish A written consent to publish has been obtained from the patient. Data availability statement No data are associated with this article. References 1. McAdoo SP, Pusey CD: Anti-Glomerular Basement Membrane Disease. Clin. J. Am. Soc. Nephrol. 2017; 12 (7): 1162–1172. PubMed Abstract | Publisher Full Text | Free Full Text 2. Hellmark T, Segelmark M: Diagnosis and classification of Goodpasture’s disease (anti-GBM). J. Autoimmun. 2014; 48-49 : 108–112. PubMed Abstract | Publisher Full Text 3. Geetha D, Jefferson JA: ANCA-Associated Vasculitis: Core Curriculum 2020. Am. J. Kidney Dis. 2020; 75 (1): 124–137. PubMed Abstract | Publisher Full Text 4. Lebbé C, et al. : Kaposi sarcoma. Nat. Rev. Dis. Primers. 2019; 5 (1): 9. 5. Pantanowitz L, Dezube BJ: Kaposi sarcoma in unusual locations. BMC Cancer. 2008; 8 : 190. PubMed Abstract | Publisher Full Text | Free Full Text 6. Cattelan AM, et al. : Iatrogenic Kaposi’s sarcoma in a patient with systemic lupus erythematosus. Clin. Exp. Rheumatol. 2001; 19 (5): 557–560. 7. Tiong BK, Singh AS, Sarantopoulos GP, et al. : Kaposi sarcoma in anti-neutrophil cytoplasmic antibody-associated vasculitis: a case-based review. Rheumatol. Int. 2021; 41 (7): 1357–1367. PubMed Abstract | Publisher Full Text | Free Full Text Comments on this article Comments (0) Version 1 VERSION 1 PUBLISHED 08 Sep 2025 ADD YOUR COMMENT Comment Author details Author details 1 Nephrology Department, Sidi Bouzid Regional Hospital, Sidi Bouzid, Sidi Bouzid, Tunisia 2 Faculty of Medicine of Sousse, University of Sousse, Sousse, Sousse, Tunisia 3 Nephrology Department, Sahloul Hospital, Sousse, Sousse, Tunisia Rahma Guesmi Roles: Conceptualization, Investigation, Methodology, Resources, Visualization, Writing – Original Draft Preparation Raja Boukadida Roles: Conceptualization, Visualization Sanda Mrabet Roles: Investigation, Supervision Dorsaf Zellama Roles: Supervision Zohra Sassi Jalleli Roles: Investigation, Supervision Competing interests No competing interests were disclosed. Grant information The author(s) declared that no grants were involved in supporting this work. Article Versions (1) version 1 Published: 08 Sep 2025, 14:878 https://doi.org/10.12688/f1000research.165476.1 Copyright © 2025 Guesmi R et al . This is an open access article distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Download Export To Sciwheel Bibtex EndNote ProCite Ref. Manager (RIS) Sente metrics Views Downloads F1000Research - - PubMed Central info_outline Data from PMC are received and updated monthly. - - Citations open_in_new 0 open_in_new 0 open_in_new SEE MORE DETAILS CITE how to cite this article Guesmi R, Boukadida R, Mrabet S et al. Case Report: Kaposi Sarcoma in Anti-Glomerular Basement Membrane Disease [version 1; peer review: 1 approved with reservations] . F1000Research 2025, 14 :878 ( https://doi.org/10.12688/f1000research.165476.1 ) NOTE: If applicable, it is important to ensure the information in square brackets after the title is included in all citations of this article. COPY CITATION DETAILS track receive updates on this article Track an article to receive email alerts on any updates to this article. TRACK THIS ARTICLE Share Open Peer Review Current Reviewer Status: ? Key to Reviewer Statuses VIEW HIDE Approved The paper is scientifically sound in its current form and only minor, if any, improvements are suggested Approved with reservations A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit. Not approved Fundamental flaws in the paper seriously undermine the findings and conclusions Version 1 VERSION 1 PUBLISHED 08 Sep 2025 Views 0 Cite How to cite this report: Jia X. Reviewer Report For: Case Report: Kaposi Sarcoma in Anti-Glomerular Basement Membrane Disease [version 1; peer review: 1 approved with reservations] . F1000Research 2025, 14 :878 ( https://doi.org/10.5256/f1000research.182115.r412840 ) The direct URL for this report is: https://f1000research.com/articles/14-878/v1#referee-response-412840 NOTE: it is important to ensure the information in square brackets after the title is included in this citation. Close Copy Citation Details Reviewer Report 16 Sep 2025 Xiaoyu Jia , Peking University First Hospital, Beijing, China Approved with Reservations VIEWS 0 https://doi.org/10.5256/f1000research.182115.r412840 The authors presented an interesting case of anti-GBM disease combined skin lesions, which was subsequently diagnosed as KS. The case is overall well written. I have the following minor comments: 1. Please provide more detailed information about ... Continue reading READ ALL The authors presented an interesting case of anti-GBM disease combined skin lesions, which was subsequently diagnosed as KS. The case is overall well written. I have the following minor comments: 1. Please provide more detailed information about the immunosuppressive treatment, including the dose of intravenous pulse corticosteroids, the dose of oral prednisone and how it was tampered during the disease course, and the cumulative dose of cyclophosphamide. 2. It is interesting that this patient was not HIV positive, since there are a few case reports of HIV associated kidney diseases, including anti-GBM disease. Particularly, a proportion of the HIV patients can have positive anti-GBM antibodies, but don't develop kidney injuries. Expand a bit further the discussion on impaired T cell mediated immunity associated with KS and anti-GBM disease. Is the background of the case’s history and progression described in sufficient detail? Yes Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes? Partly Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment? Yes Is the case presented with sufficient detail to be useful for other practitioners? Yes Competing Interests: No competing interests were disclosed. Reviewer Expertise: autoimmune kidney diseases I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however I have significant reservations, as outlined above. Close READ LESS CITE CITE HOW TO CITE THIS REPORT Jia X. Reviewer Report For: Case Report: Kaposi Sarcoma in Anti-Glomerular Basement Membrane Disease [version 1; peer review: 1 approved with reservations] . F1000Research 2025, 14 :878 ( https://doi.org/10.5256/f1000research.182115.r412840 ) The direct URL for this report is: https://f1000research.com/articles/14-878/v1#referee-response-412840 NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article. COPY CITATION DETAILS Report a concern Respond or Comment COMMENT ON THIS REPORT Comments on this article Comments (0) Version 1 VERSION 1 PUBLISHED 08 Sep 2025 ADD YOUR COMMENT Comment keyboard_arrow_left keyboard_arrow_right Open Peer Review Reviewer Status info_outline Alongside their report, reviewers assign a status to the article: Approved The paper is scientifically sound in its current form and only minor, if any, improvements are suggested Approved with reservations A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit. Not approved Fundamental flaws in the paper seriously undermine the findings and conclusions Reviewer Reports Invited Reviewers 1 Version 1 08 Sep 25 read Xiaoyu Jia , Peking University First Hospital, Beijing, China Comments on this article All Comments (0) Add a comment Sign up for content alerts Sign Up You are now signed up to receive this alert Browse by related subjects keyboard_arrow_left Back to all reports Reviewer Report 0 Views copyright © 2025 Jia X. This is an open access peer review report distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. 16 Sep 2025 | for Version 1 Xiaoyu Jia , Peking University First Hospital, Beijing, China 0 Views copyright © 2025 Jia X. This is an open access peer review report distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. format_quote Cite this report speaker_notes Responses (0) Approved With Reservations info_outline Alongside their report, reviewers assign a status to the article: Approved The paper is scientifically sound in its current form and only minor, if any, improvements are suggested Approved with reservations A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit. Not approved Fundamental flaws in the paper seriously undermine the findings and conclusions The authors presented an interesting case of anti-GBM disease combined skin lesions, which was subsequently diagnosed as KS. The case is overall well written. I have the following minor comments: 1. Please provide more detailed information about the immunosuppressive treatment, including the dose of intravenous pulse corticosteroids, the dose of oral prednisone and how it was tampered during the disease course, and the cumulative dose of cyclophosphamide. 2. It is interesting that this patient was not HIV positive, since there are a few case reports of HIV associated kidney diseases, including anti-GBM disease. Particularly, a proportion of the HIV patients can have positive anti-GBM antibodies, but don't develop kidney injuries. Expand a bit further the discussion on impaired T cell mediated immunity associated with KS and anti-GBM disease. Is the background of the case’s history and progression described in sufficient detail? Yes Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes? Partly Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment? Yes Is the case presented with sufficient detail to be useful for other practitioners? Yes Competing Interests No competing interests were disclosed. Reviewer Expertise autoimmune kidney diseases I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however I have significant reservations, as outlined above. reply Respond to this report Responses (0) Jia X. Peer Review Report For: Case Report: Kaposi Sarcoma in Anti-Glomerular Basement Membrane Disease [version 1; peer review: 1 approved with reservations] . F1000Research 2025, 14 :878 ( https://doi.org/10.5256/f1000research.182115.r412840) NOTE: it is important to ensure the information in square brackets after the title is included in this citation. 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