Case Reports in Rheumatology最新文献

Genital Tuberculosis and Serous Cystadenoma in a 58-Year-Old Female With Rheumatoid Arthritis and Sjögren's Syndrome: A Case Report. 58岁女性类风湿性关节炎合并Sjögren综合征并发生殖器结核和浆液性囊腺瘤1例。

Case Reports in Rheumatology Pub Date : 2025-05-06 eCollection Date: 2025-01-01 DOI: 10.1155/crrh/9372058

Sheila De la Cruz-Aragón, Itzel Guadalupe Castillo-Duarte, Abril Camacho-Cervantes, Alfredo Saad-Ganem, Francisco Mario García Rodríguez, Alan Antonio Leija-Torres

{"title":"Genital Tuberculosis and Serous Cystadenoma in a 58-Year-Old Female With Rheumatoid Arthritis and Sjögren's Syndrome: A Case Report.","authors":"Sheila De la Cruz-Aragón, Itzel Guadalupe Castillo-Duarte, Abril Camacho-Cervantes, Alfredo Saad-Ganem, Francisco Mario García Rodríguez, Alan Antonio Leija-Torres","doi":"10.1155/crrh/9372058","DOIUrl":"10.1155/crrh/9372058","url":null,"abstract":"Genital tuberculosis (GT) is a rare but significant extrapulmonary tuberculosis form, often mimicking ovarian malignancy. We report a case of a 58-year-old woman with Sjögren's syndrome and rheumatoid arthritis, previously treated with infliximab, who presented with abdominal distension, weight loss, night sweats, and intermittent abdominal pain. Initial imaging and elevated CA-125 levels suggested ovarian cancer. However, intraoperative findings revealed a frozen pelvis with granulomatous inflammation, caseating granulomas, and Langhans' giant cells. Histopathological analysis and RT-PCR confirmed GT coexisting with a serous cystadenoma. GT should be considered in the differential diagnosis of pelvic masses, especially in immunocompromised patients. This case emphasizes the importance of thorough diagnostic evaluation using molecular, serological, and imaging techniques to avoid misdiagnosis and unnecessary surgical interventions. Prompt initiation of antituberculosis treatment led to significant clinical improvement. Early and accurate diagnosis of GT is crucial to prevent morbidity associated with misdiagnosis and to provide effective treatment. This case underscores the need for heightened clinical awareness and multidisciplinary approaches in managing complex cases where GT mimics malignancy, ensuring optimal patient outcomes.","PeriodicalId":9622,"journal":{"name":"Case Reports in Rheumatology","volume":"2025 ","pages":"9372058"},"PeriodicalIF":0.0,"publicationDate":"2025-05-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12074851/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143985278","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Secukinumab Leading to Rapid Improvement in Pyogenic Arthritis, Acne, Pyoderma Gangrenosum, and Hidradenitis Suppurativa (PAPASH) Syndrome: A Case Report and Review of Treatment Modalities for PAPASH Patients. Secukinumab可快速改善化脓性关节炎、痤疮、坏疽性脓皮病和化脓性汗腺炎（PAPASH）综合征：一个病例报告和PAPASH患者治疗方式的回顾。

Case Reports in Rheumatology Pub Date : 2025-04-22 eCollection Date: 2025-01-01 DOI: 10.1155/crrh/7720064

Inga N Shevtsova, James J Abbott, Pavel N Shevtsov, Guiset Carvajal Bedoya, Diana C Norton

{"title":"Secukinumab Leading to Rapid Improvement in Pyogenic Arthritis, Acne, Pyoderma Gangrenosum, and Hidradenitis Suppurativa (PAPASH) Syndrome: A Case Report and Review of Treatment Modalities for PAPASH Patients.","authors":"Inga N Shevtsova, James J Abbott, Pavel N Shevtsov, Guiset Carvajal Bedoya, Diana C Norton","doi":"10.1155/crrh/7720064","DOIUrl":"https://doi.org/10.1155/crrh/7720064","url":null,"abstract":"PAPASH syndrome, a rare autoinflammatory condition characterized by pyogenic arthritis, pyoderma gangrenosum, acne, and hidradenitis suppurativa, presents significant treatment challenges due to its rarity and complex multisystem involvement. Since its initial description in 2013, only 14 cases have been documented, leading to limited treatment experience. Although IL-1 and TNF-alpha blocking agents have shown efficacy, responses vary due to genetic and pathogenetic differences, with some cases being resistant. Therefore, alongside summarizing prior treatment experiences, new treatment modalities need to be explored. This report presents the case of a 46-year-old Native American male with PAPASH syndrome who responded successfully to IL-17 inhibition with secukinumab. The patient experienced marked improvement in both dermatologic and rheumatologic symptoms, highlighting the potential role of IL-17 in the pathogenesis of PAPASH. This case suggests that IL-17 inhibition could be a promising treatment modality for PAPASH syndrome.","PeriodicalId":9622,"journal":{"name":"Case Reports in Rheumatology","volume":"2025 ","pages":"7720064"},"PeriodicalIF":0.0,"publicationDate":"2025-04-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12041644/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143969000","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Anifrolumab in Refractory Dermatomyositis and Antisynthetase Syndrome. 难治性皮肌炎和抗合成酶综合征的无性瘤抗体。

Case Reports in Rheumatology Pub Date : 2025-04-21 eCollection Date: 2025-01-01 DOI: 10.1155/crrh/5560523

Ryan Bonaventure Soares, Jihad Ben Gabr, Mark Ash, Gregory Hosler

{"title":"Anifrolumab in Refractory Dermatomyositis and Antisynthetase Syndrome.","authors":"Ryan Bonaventure Soares, Jihad Ben Gabr, Mark Ash, Gregory Hosler","doi":"10.1155/crrh/5560523","DOIUrl":"https://doi.org/10.1155/crrh/5560523","url":null,"abstract":"Dermatomyositis (DM) and antisynthetase syndrome are rare autoimmune disorders within the spectrum of inflammatory myopathies, typically characterized by cutaneous and muscular inflammation along with systemic manifestations. This case report highlights the evolving treatment strategies for inflammatory myopathies, with a focus on the use of anifrolumab, a type-1 interferon receptor blocker, in a 29 year-old female with refractory DM/antisynthetase syndrome. The patient presented with classic DM features, including heliotrope rash, Gottron's papules, and malar erythema, but lacked significant myopathy. Initial therapies with methotrexate and prednisone were ineffective, and her condition worsened despite adding intravenous immunoglobulin (IVIg) and tofacitinib. Following persistent disease progression, therapy was switched to a combination of IVIg, apremilast, and anifrolumab as an off-label drug for DM. Within 5 months, the patient showed significant improvement in myopathy and skin manifestations. Anifrolumab targets the interferon (IFN) axis, which plays a crucial role in DM pathogenesis. This case underscores the potential of targeted therapies like anifrolumab in managing DM, especially in cases not responsive to conventional standard of care therapies. It also highlights the need for further research into the IFN pathway as a therapeutic target in inflammatory myopathies. Trial Registration: ClinicalTrials.gov identifier: NCT06455449.","PeriodicalId":9622,"journal":{"name":"Case Reports in Rheumatology","volume":"2025 ","pages":"5560523"},"PeriodicalIF":0.0,"publicationDate":"2025-04-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12037237/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143974093","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Concomitant Systemic Lupus Erythematosus and Glanzmann Thrombocytopenia: A Case Report and Literature Review. 伴发系统性红斑狼疮和血小板减少：1例报告及文献复习。

Case Reports in Rheumatology Pub Date : 2025-04-15 eCollection Date: 2025-01-01 DOI: 10.1155/crrh/1543762

Maryam Noory, Haniyeh Hajian, Farzad Kompani, Azadeh Kiumarsi, Mohammad Shahrooei, Vahid Ziaee

{"title":"Concomitant Systemic Lupus Erythematosus and Glanzmann Thrombocytopenia: A Case Report and Literature Review.","authors":"Maryam Noory, Haniyeh Hajian, Farzad Kompani, Azadeh Kiumarsi, Mohammad Shahrooei, Vahid Ziaee","doi":"10.1155/crrh/1543762","DOIUrl":"https://doi.org/10.1155/crrh/1543762","url":null,"abstract":"Background: Glanzmann thrombasthenia (GT) is a rare disease that manifests with bleeding in different parts such as epistaxis and bruising. GT can be congenital or acquired. Systemic lupus erythematosus (SLE) is an autoimmune disorder. It is mentioned that the acquired type can be associated with other disorders like malignancies and autoimmune disorders. There is no report about the co-occurrence of congenital GT with SLE. Case Report: In this report, we present this co-occurrence in a girl. An 11-year-old girl was referred to our clinic with severe and uncontrolled epistaxis. She had a history of recurrent epistaxis, gastrointestinal bleeding, and bruising. She also had a malar rash and generalized body pain. She was admitted, and after clinical and laboratory assessments, a co-occurrence of congenital GT with SLE was diagnosed. Conclusion: The co-occurrence of congenital GT and SLE has not been reported until now. Patients with this presentation should be closely followed up because the risk of bleeding is high for them.","PeriodicalId":9622,"journal":{"name":"Case Reports in Rheumatology","volume":"2025 ","pages":"1543762"},"PeriodicalIF":0.0,"publicationDate":"2025-04-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12014255/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143977629","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

A Case of Hydralazine-Induced ANCA Vasculitis/Lupus Overlap Syndrome Presenting as Persistent Bicytopenia. 肼嗪诱导的ANCA血管炎/狼疮重叠综合征1例，表现为持续性双环减少。

Case Reports in Rheumatology Pub Date : 2025-04-09 eCollection Date: 2025-01-01 DOI: 10.1155/crrh/9276592

Madiha Naqsh Siddiqui, Stephanie Norris

{"title":"A Case of Hydralazine-Induced ANCA Vasculitis/Lupus Overlap Syndrome Presenting as Persistent Bicytopenia.","authors":"Madiha Naqsh Siddiqui, Stephanie Norris","doi":"10.1155/crrh/9276592","DOIUrl":"https://doi.org/10.1155/crrh/9276592","url":null,"abstract":"Background: Hydralazine is a commonly used arteriolar vasodilator that is associated with autoimmune side effects, including drug-induced lupus. A less well-recognized drug-induced vasculitis can be seen, often accompanying drug-induced lupus. This syndrome can cause long-standing vague symptoms, leading to missed diagnoses, and can result in permanent end-organ damage. We describe here such a case of hydralazine-induced vasculitis and lupus overlap syndrome. Case Presentation: An 85-year old male presented with chronic fatigue and weight loss associated with anemia, leukopenia, and acute renal injury in the setting of longstanding hydralazine use. Serologic studies were notable for a positive antinuclear antibody, antihistone antibody, along with anti-myeloperoxidase (MPO) and anti-proteinase 3 (PR3) antibodies. Hydralazine was discontinued, and treatment was initiated with high-dose prednisone. A renal biopsy revealed antineutrophil cytoplasmic antibody (ANCA)-associated focal necrotizing pauci-immune glomerulonephritis. The patient's clinical course was complicated by the development of oral ulcerations and recurrent hydrocele secondary to serositis. Rituximab was then employed without clinical improvement, with eventual progression to end-stage renal disease requiring hemodialysis. Conclusions: This case report helps highlight the vague symptoms that can be associated with hydralazine-induced vasculitis/lupus overlap syndrome. This case will increase clinician awareness for early recognition of such a syndrome, prompting early diagnosis, preventing end-organ damage, reducing hospitalizations and improving quality of life.","PeriodicalId":9622,"journal":{"name":"Case Reports in Rheumatology","volume":"2025 ","pages":"9276592"},"PeriodicalIF":0.0,"publicationDate":"2025-04-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12003037/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143968997","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Ruptured Tubercular Baker's Cyst: A Case Report. 结核性贝克囊肿破裂1例报告。

Case Reports in Rheumatology Pub Date : 2025-03-26 eCollection Date: 2025-01-01 DOI: 10.1155/crrh/8840886

Md Rashid Al Mahmood, A B M Mehedi

引用次数: 0

Small-Vessel Thrombotic Vasculopathy Secondary to Paradoxical Emboli Traversing a Patent Foramen Ovale: A Vasculitis Mimic. 小血管血栓性血管病变继发于异位栓子穿过卵圆孔未闭：血管炎模拟。

Case Reports in Rheumatology Pub Date : 2025-02-26 eCollection Date: 2025-01-01 DOI: 10.1155/crrh/8840496

Trevor Kwan, Steven Tu, Carlos El-Haddad

引用次数: 0

Subcutaneous Emphysema and Severe Interstitial Lung Disease in the Setting of Anti-MDA 5 Positive Dermatomyositis in a Hispanic Patient. 皮下肺气肿和严重间质性肺疾病在抗mda - 5阳性皮肌炎西班牙患者的设置。

Case Reports in Rheumatology Pub Date : 2025-01-23 eCollection Date: 2025-01-01 DOI: 10.1155/crrh/2017703

Sweta Subhadarshani, Brad Woodie, Emerson Bookal, Justin Reed

引用次数: 0

Late Presentation of McArdle's Disease Mimicking Polymyalgia Rheumatica: A Case Report and Review of the Literature. 模仿风湿性多肌痛的McArdle病的晚期表现：1例报告及文献回顾。

Case Reports in Rheumatology Pub Date : 2025-01-03 eCollection Date: 2025-01-01 DOI: 10.1155/crrh/8148736

Maiar Elghobashy, Ute Pohl, James Bateman

引用次数: 0

Unilateral Anterior Scleritis Following the Booster Shot of Inactivated COVID-19 (Sinopharm) Vaccine in a 52-Year-Old Woman: A Case Report. 52岁女性新冠肺炎（国药）灭活疫苗加强注射后单侧前巩膜炎1例

Case Reports in Rheumatology Pub Date : 2024-12-28 eCollection Date: 2024-01-01 DOI: 10.1155/crrh/6614757

Kimia Jazi, Mahnaz Rahimi, Fatemeh Hasani, Maryam Shirmohammadi, Maryam Masoumi

{"title":"Unilateral Anterior Scleritis Following the Booster Shot of Inactivated COVID-19 (Sinopharm) Vaccine in a 52-Year-Old Woman: A Case Report.","authors":"Kimia Jazi, Mahnaz Rahimi, Fatemeh Hasani, Maryam Shirmohammadi, Maryam Masoumi","doi":"10.1155/crrh/6614757","DOIUrl":"https://doi.org/10.1155/crrh/6614757","url":null,"abstract":"The only way to mitigate the spread of coronavirus disease 2019 (COVID-19) pandemic was vaccines. While effective in decreasing the rate and severity of the disease, there also have been considerable adverse events. Since the birth of vaccines, adverse reactions accompanied the immunity, and COVID-19 vaccines are no exceptions. This is a report about a 52-year-old female patient who presented with bilateral redness of the eyes, with normal bilateral visual acuity, postbooster dose of the Sinopharm COVID-19 vaccine. She had no significant past history of any disease or any similar reactions after previous doses. All her physical examinations were normal. Ophthalmic examination disclosed diffuse erythema, and mild scleral edema consistent with bilateral anterior diffused scleritis with negative phenylephrine test. Thereafter, with a course of tapering doses of prednisolone (30 mg at the onset) combined with azathioprine (100 mg/day), over a 2-week period, the condition completely resolved. Very few vaccination-related adverse events may manifest an unrecognized underlying autoimmune vasculopathy which may also require urgent management. As in this case, ocular adverse events, as highlighted, are highly associated with undiagnosed autoimmune diseases and therefore warrant careful assessment by clinicians.","PeriodicalId":9622,"journal":{"name":"Case Reports in Rheumatology","volume":"2024 ","pages":"6614757"},"PeriodicalIF":0.0,"publicationDate":"2024-12-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11699981/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142930662","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0