Pub Date : 2025-10-18eCollection Date: 2025-01-01DOI: 10.1177/2050313X251381576
Carola Petino, Marta Moreno Solé, Mairi Ziaka
Metamizole (dipyrone) is a non-opioid analgesic and antipyretic agent belonging to the pyrazolone class. While it is widely used in many countries due to its favourable safety profile compared to non-steroidal anti-inflammatory drugs and opioids, its use has been associated with rare but potentially life-threatening haematologic adverse effects, including neutropaenia and agranulocytosis. These complications typically occur within 6-14 days of treatment initiation but may also arise earlier or significantly later, even after discontinuation of the drug. Given that late-onset neutropaenia after metamizole discontinuation may be underdiagnosed, we present the case of a 92-year-old woman who developed transient, likely drug-induced neutropaenia 10 days later, with spontaneous haematologic recovery. The patient remained asymptomatic, with no signs of infection or evidence of inflammatory or neoplastic systemic disease.
{"title":"A probable case of metamizole-induced neutropaenia presenting 10 days after drug discontinuation.","authors":"Carola Petino, Marta Moreno Solé, Mairi Ziaka","doi":"10.1177/2050313X251381576","DOIUrl":"10.1177/2050313X251381576","url":null,"abstract":"<p><p>Metamizole (dipyrone) is a non-opioid analgesic and antipyretic agent belonging to the pyrazolone class. While it is widely used in many countries due to its favourable safety profile compared to non-steroidal anti-inflammatory drugs and opioids, its use has been associated with rare but potentially life-threatening haematologic adverse effects, including neutropaenia and agranulocytosis. These complications typically occur within 6-14 days of treatment initiation but may also arise earlier or significantly later, even after discontinuation of the drug. Given that late-onset neutropaenia after metamizole discontinuation may be underdiagnosed, we present the case of a 92-year-old woman who developed transient, likely drug-induced neutropaenia 10 days later, with spontaneous haematologic recovery. The patient remained asymptomatic, with no signs of infection or evidence of inflammatory or neoplastic systemic disease.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251381576"},"PeriodicalIF":0.6,"publicationDate":"2025-10-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12547106/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145378566","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}
Pub Date : 2025-10-16eCollection Date: 2025-01-01DOI: 10.1177/2050313X251388724
Grant M Tinsley, Spencer Nadolsky
GLP-1 receptor agonists (e.g., semaglutide) and dual GLP-1/GIP receptor agonists (e.g., tirzepatide) are effective for reducing body weight and fat mass, though lean soft tissue loss comprised 26%-40% of weight loss in recent trials. This case series describes three patients (two female, one male; body mass index: 32.9-51.9 kg m-2) who prioritized lean soft tissue preservation strategies during treatment with semaglutide or tirzepatide. Patients engaged in intentional exercise or structured physical activity 4-7 days·week-1, including resistance training 3-5 days·week-1. Typical protein intakes were 0.7-1.7 g·kg-1·day-1 relative to body mass and 1.6-2.3 g·kg-1·day-1 relative to fat-free mass. Changes in weight, fat mass, and lean soft tissue were: -33.0%, -53.4%, and -6.9% (case 1); -26.8%, -61.6%, and +2.5% (case 2); and -13.2%, -46.9%, and +5.8% (case 3). Accordingly, one patient lost 8.7% of weight as lean soft tissue, while two increased lean soft tissue. These findings highlight the potential for some individuals to preserve or even increase lean soft tissue during treatment with semaglutide or tirzepatide alongside supportive lifestyle strategies.
GLP-1受体激动剂(如西马鲁肽)和双GLP-1/GIP受体激动剂(如替西帕肽)对减轻体重和脂肪量有效,尽管在最近的试验中,瘦软组织损失占体重减轻的26%-40%。本病例系列描述了3名患者(2名女性,1名男性;体重指数:32.9-51.9 kg m-2),他们在使用西马鲁肽或替西肽治疗期间优先考虑瘦软组织保存策略。患者进行有意锻炼或有组织的体育活动4-7天·周-1,其中阻力训练3-5天·周-1。典型蛋白质摄入量相对于体重为0.7 ~ 1.7 g·kg-1·day-1,相对于无脂质量为1.6 ~ 2.3 g·kg-1·day-1。体重、脂肪量和瘦软组织的变化分别为-33.0%、-53.4%和-6.9%(病例1);-26.8%, -61.6%和+2.5%(病例2);以及-13.2%,-46.9%和+5.8%(案例3)。相应的,1例患者瘦软组织减轻8.7%,2例患者瘦软组织增加。这些发现强调了一些个体在使用西马鲁肽或替西帕肽治疗期间保留甚至增加瘦软组织的潜力,以及支持的生活方式策略。
{"title":"Preservation of lean soft tissue during weight loss induced by GLP-1 and GLP-1/GIP receptor agonists: A case series.","authors":"Grant M Tinsley, Spencer Nadolsky","doi":"10.1177/2050313X251388724","DOIUrl":"10.1177/2050313X251388724","url":null,"abstract":"<p><p>GLP-1 receptor agonists (e.g., semaglutide) and dual GLP-1/GIP receptor agonists (e.g., tirzepatide) are effective for reducing body weight and fat mass, though lean soft tissue loss comprised 26%-40% of weight loss in recent trials. This case series describes three patients (two female, one male; body mass index: 32.9-51.9 kg m<sup>-2</sup>) who prioritized lean soft tissue preservation strategies during treatment with semaglutide or tirzepatide. Patients engaged in intentional exercise or structured physical activity 4-7 days·week<sup>-1</sup>, including resistance training 3-5 days·week<sup>-1</sup>. Typical protein intakes were 0.7-1.7 g·kg<sup>-1</sup>·day<sup>-1</sup> relative to body mass and 1.6-2.3 g·kg<sup>-1</sup>·day<sup>-1</sup> relative to fat-free mass. Changes in weight, fat mass, and lean soft tissue were: -33.0%, -53.4%, and -6.9% (case 1); -26.8%, -61.6%, and +2.5% (case 2); and -13.2%, -46.9%, and +5.8% (case 3). Accordingly, one patient lost 8.7% of weight as lean soft tissue, while two increased lean soft tissue. These findings highlight the potential for some individuals to preserve or even increase lean soft tissue during treatment with semaglutide or tirzepatide alongside supportive lifestyle strategies.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251388724"},"PeriodicalIF":0.6,"publicationDate":"2025-10-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12536186/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145346714","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}
Pub Date : 2025-10-15eCollection Date: 2025-01-01DOI: 10.1177/2050313X251377218
Alexandra Metz, Ash Cooper, Meredith Garst, Genevra Addis, Susan Flesher
We present a case of a 2-month-old female with an acute onset and rapidly progressing diffuse erythroderma involving the scalp, face, neck folds, torso, and groin. She had a history of seborrheic and diaper dermatitis and developed a worsening rash that initially appeared as a "blister-like" lesion on the cheek. Examination revealed greasy yellow scaling and intertriginous erythema. Workup ruled out herpes simplex virus/varicella-zoster virus, and blood cultures grew presumed contaminant Staphylococcus epidermidis. She was treated empirically with antibiotics, antivirals, anti-fungals, and topical steroids. Dermatology suspected inverse psoriasis. The patient improved within 48 h of treatment. Diagnoses included inverse psoriasis, fungal diaper dermatitis with secondary bacterial infection, and possible early staphylococcal scalded skin syndrome. This case emphasizes the diagnostic complexity of infantile erythroderma and the importance of a broad differential and multidisciplinary approach. Recognition of inverse psoriasis in this age group is crucial, especially in the setting of superimposed fungal and bacterial infections.
{"title":"Two-month-old with diffuse erythema: A case report.","authors":"Alexandra Metz, Ash Cooper, Meredith Garst, Genevra Addis, Susan Flesher","doi":"10.1177/2050313X251377218","DOIUrl":"https://doi.org/10.1177/2050313X251377218","url":null,"abstract":"<p><p>We present a case of a 2-month-old female with an acute onset and rapidly progressing diffuse erythroderma involving the scalp, face, neck folds, torso, and groin. She had a history of seborrheic and diaper dermatitis and developed a worsening rash that initially appeared as a \"blister-like\" lesion on the cheek. Examination revealed greasy yellow scaling and intertriginous erythema. Workup ruled out herpes simplex virus/varicella-zoster virus, and blood cultures grew presumed contaminant <i>Staphylococcus epidermidis</i>. She was treated empirically with antibiotics, antivirals, anti-fungals, and topical steroids. Dermatology suspected inverse psoriasis. The patient improved within 48 h of treatment. Diagnoses included inverse psoriasis, fungal diaper dermatitis with secondary bacterial infection, and possible early staphylococcal scalded skin syndrome. This case emphasizes the diagnostic complexity of infantile erythroderma and the importance of a broad differential and multidisciplinary approach. Recognition of inverse psoriasis in this age group is crucial, especially in the setting of superimposed fungal and bacterial infections.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251377218"},"PeriodicalIF":0.6,"publicationDate":"2025-10-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12536091/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145346738","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}
Pacemaker lead infections with large vegetation and extensive adhesions pose significant procedural risks during surgical extraction. A 71-year-old woman with a 21-year-old pacemaker developed recurrent Staphylococcus aureus bacteremia. She underwent successful surgical lead extraction performed under cardiopulmonary bypass, with concomitant tricuspid valve repair, due to the presence of a large vegetation and strong adhesions to the right ventricular myocardium and tricuspid valve. Surgical lead extraction under cardiopulmonary bypass should be considered a viable option in patients with long-standing pacemaker lead implantation or large vegetation to reduce the risk of life-threatening complications.
{"title":"Surgical lead extraction for pacemaker infection with tricuspid valve repair: A case report.","authors":"Yoichi Hisata, Shunsuke Taguchi, Shoichiro Furukawa, Yasuaki Koga, Mitsutaka Yamamoto, Takafumi Yamada","doi":"10.1177/2050313X251368057","DOIUrl":"10.1177/2050313X251368057","url":null,"abstract":"<p><p>Pacemaker lead infections with large vegetation and extensive adhesions pose significant procedural risks during surgical extraction. A 71-year-old woman with a 21-year-old pacemaker developed recurrent <i>Staphylococcus aureus</i> bacteremia. She underwent successful surgical lead extraction performed under cardiopulmonary bypass, with concomitant tricuspid valve repair, due to the presence of a large vegetation and strong adhesions to the right ventricular myocardium and tricuspid valve. Surgical lead extraction under cardiopulmonary bypass should be considered a viable option in patients with long-standing pacemaker lead implantation or large vegetation to reduce the risk of life-threatening complications.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251368057"},"PeriodicalIF":0.6,"publicationDate":"2025-10-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12536103/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145346660","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}
Pub Date : 2025-10-01eCollection Date: 2025-01-01DOI: 10.1177/2050313X251381565
Jiale Chen, Jieyi Zhou, Chengyang Xu, Bo Zheng, Jie He, Rong Li
To report the management and outcome of an elderly, high-risk lymphoplasmacytic lymphoma/Waldenström macroglobulinemia patient presenting with severe symptomatic anemia, marked hyperleukocytosis, a complex karyotype, dual MYD88/CXCR4 mutations, and significant comorbidities, highlighting the therapeutic challenges and the efficacy of targeted therapy after chemoimmunotherapy intolerance. This case report details a 78-year-old male diagnosed with high-risk lymphoplasmacytic lymphoma/Waldenström macroglobulinemia (MYD88 L265P+, pathogenic CXCR4+, complex karyotype: 46,X,-Y,add(6)(q224),+18/47,XY,+18/47,X,Y,+4,+18/48,XY,+4,+18/48,XY,+3,+18/46,XY) and significant comorbidities including hypertension and suspected cardiac amyloidosis. Initial treatment with bendamustine-rituximab was administered but led to significant toxicity. Subsequently, therapy was switched to the second-generation Bruton's tyrosine kinase inhibitor, zanubrutinib. Clinical progress, hematologic response, and adverse events were monitored. Initial treatment with bendamustine-rituximab was discontinued due to severe toxicity, including a grade 3 infusion reaction and subsequent neutropenic fever. Following the switch to zanubrutinib, he achieved a sustained partial hematologic response and clinical improvement. Disease-related complications (hyperviscosity retinopathy) and treatment-related adverse events (neutropenia) occurred but were managed appropriately while continuing zanubrutinib. Zanubrutinib demonstrated efficacy and manageable toxicity in an elderly, high-risk lymphoplasmacytic lymphoma/Waldenström macroglobulinemia patient with a complex karyotype, dual MYD88/CXCR4 mutations, hyperleukocytosis, and significant comorbidities, following intolerance to standard chemoimmunotherapy. This case supports the use of targeted Bruton's tyrosine kinase inhibition in achieving favorable outcomes in complex lymphoplasmacytic lymphoma/Waldenström macroglobulinemia presentations and contributes to understanding the significance of complex karyotypes in the era of novel therapies.
{"title":"Zanubrutinib for high-risk Waldenström macroglobulinemia with complex karyotype and hyperleukocytosis: A case report and literature review.","authors":"Jiale Chen, Jieyi Zhou, Chengyang Xu, Bo Zheng, Jie He, Rong Li","doi":"10.1177/2050313X251381565","DOIUrl":"10.1177/2050313X251381565","url":null,"abstract":"<p><p>To report the management and outcome of an elderly, high-risk lymphoplasmacytic lymphoma/Waldenström macroglobulinemia patient presenting with severe symptomatic anemia, marked hyperleukocytosis, a complex karyotype, dual <i>MYD88/CXCR4</i> mutations, and significant comorbidities, highlighting the therapeutic challenges and the efficacy of targeted therapy after chemoimmunotherapy intolerance. This case report details a 78-year-old male diagnosed with high-risk lymphoplasmacytic lymphoma/Waldenström macroglobulinemia (<i>MYD88</i> L265P<sup>+</sup>, pathogenic <i>CXCR4</i> <sup>+</sup>, complex karyotype: 46,X,-Y,add(6)(q224),+18/47,XY,+18/47,X,Y,+4,+18/48,XY,+4,+18/48,XY,+3,+18/46,XY) and significant comorbidities including hypertension and suspected cardiac amyloidosis. Initial treatment with bendamustine-rituximab was administered but led to significant toxicity. Subsequently, therapy was switched to the second-generation Bruton's tyrosine kinase inhibitor, zanubrutinib. Clinical progress, hematologic response, and adverse events were monitored. Initial treatment with bendamustine-rituximab was discontinued due to severe toxicity, including a grade 3 infusion reaction and subsequent neutropenic fever. Following the switch to zanubrutinib, he achieved a sustained partial hematologic response and clinical improvement. Disease-related complications (hyperviscosity retinopathy) and treatment-related adverse events (neutropenia) occurred but were managed appropriately while continuing zanubrutinib. Zanubrutinib demonstrated efficacy and manageable toxicity in an elderly, high-risk lymphoplasmacytic lymphoma/Waldenström macroglobulinemia patient with a complex karyotype, dual <i>MYD88/CXCR4</i> mutations, hyperleukocytosis, and significant comorbidities, following intolerance to standard chemoimmunotherapy. This case supports the use of targeted Bruton's tyrosine kinase inhibition in achieving favorable outcomes in complex lymphoplasmacytic lymphoma/Waldenström macroglobulinemia presentations and contributes to understanding the significance of complex karyotypes in the era of novel therapies.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251381565"},"PeriodicalIF":0.6,"publicationDate":"2025-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12489237/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145233504","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}
Pub Date : 2025-09-28eCollection Date: 2025-01-01DOI: 10.1177/2050313X251381645
Jiayin Zhang, Zhe Wang, Long Yan
Aberrant right subclavian artery is one of the most common congenital anatomical variations of the aortic arch, but its occlusion is extremely rare. Although femoral artery access for endovascular treatment has become the mainstream approach for subclavian artery stenosis or occlusion, when the femoral artery approach fails to identify the arterial stump, the radial artery approach becomes an alternative. This report presents a case in which, after failure to treat via the femoral artery, a self-expanding stent was successfully deployed through the radial artery approach to treat a long segment occlusion of the aberrant right subclavian artery. The case involved a 71-year-old female patient who presented with a 3-month history of recurrent dizziness, right upper limb weakness, and episodic coldness in the right hand. Clinical and Duplex ultrasound findings confirmed subclavian steal syndrome secondary to aberrant right subclavian artery occlusion. The patient underwent successful stent implantation via the radial artery approach, and postoperatively, the symptoms resolved, and antegrade flow in the vertebral artery was restored. This case highlights the rare long-segment occlusion of the aberrant right subclavian artery and suggests that the radial artery approach may serve as a potential method for occlusion recanalization.
{"title":"Transradial angioplasty and stent placement for total occlusion of aberrant right subclavian artery: A case report.","authors":"Jiayin Zhang, Zhe Wang, Long Yan","doi":"10.1177/2050313X251381645","DOIUrl":"10.1177/2050313X251381645","url":null,"abstract":"<p><p>Aberrant right subclavian artery is one of the most common congenital anatomical variations of the aortic arch, but its occlusion is extremely rare. Although femoral artery access for endovascular treatment has become the mainstream approach for subclavian artery stenosis or occlusion, when the femoral artery approach fails to identify the arterial stump, the radial artery approach becomes an alternative. This report presents a case in which, after failure to treat via the femoral artery, a self-expanding stent was successfully deployed through the radial artery approach to treat a long segment occlusion of the aberrant right subclavian artery. The case involved a 71-year-old female patient who presented with a 3-month history of recurrent dizziness, right upper limb weakness, and episodic coldness in the right hand. Clinical and Duplex ultrasound findings confirmed subclavian steal syndrome secondary to aberrant right subclavian artery occlusion. The patient underwent successful stent implantation via the radial artery approach, and postoperatively, the symptoms resolved, and antegrade flow in the vertebral artery was restored. This case highlights the rare long-segment occlusion of the aberrant right subclavian artery and suggests that the radial artery approach may serve as a potential method for occlusion recanalization.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251381645"},"PeriodicalIF":0.6,"publicationDate":"2025-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12477387/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145200583","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}
This case report explores the ethical complexities faced by physicians when providing medical advice and care to close family members. It focuses on a 29-year-old Indian woman residing in Western Europe who, while visiting family in Kerala, India, was diagnosed with IgA nephropathy during a routine health check. Several family members involved in her care are also practicing physicians, raising significant ethical questions about confidentiality, professional objectivity, and the boundaries of familial care. The case highlights the tension between patient autonomy and informal medical involvement by relatives, especially in a country such as India, where cultural expectations encourage familial responsibility. Although clinical care was efficiently delivered, the situation raised concerns about the formal decision-making process, the lack of clinical documentation, and the absence of established ethical frameworks within the Indian healthcare context. This report emphasizes the need for clearer guidelines and practical tools to help physicians navigate dual-role relationships while upholding confidentiality and clinical integrity. The patient's perspective and the follow-up are discussed, with implications for both policy and practice.
{"title":"Ethical challenges in treating family members: A case study on confidentiality and clinical decision-making in the Indian context.","authors":"Mohamed Ali Kalathingal, Nazia Edathola Kottasseri, Jasim Uluvan, Shajitha Thekke Veettil","doi":"10.1177/2050313X251380521","DOIUrl":"10.1177/2050313X251380521","url":null,"abstract":"<p><p>This case report explores the ethical complexities faced by physicians when providing medical advice and care to close family members. It focuses on a 29-year-old Indian woman residing in Western Europe who, while visiting family in Kerala, India, was diagnosed with IgA nephropathy during a routine health check. Several family members involved in her care are also practicing physicians, raising significant ethical questions about confidentiality, professional objectivity, and the boundaries of familial care. The case highlights the tension between patient autonomy and informal medical involvement by relatives, especially in a country such as India, where cultural expectations encourage familial responsibility. Although clinical care was efficiently delivered, the situation raised concerns about the formal decision-making process, the lack of clinical documentation, and the absence of established ethical frameworks within the Indian healthcare context. This report emphasizes the need for clearer guidelines and practical tools to help physicians navigate dual-role relationships while upholding confidentiality and clinical integrity. The patient's perspective and the follow-up are discussed, with implications for both policy and practice.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251380521"},"PeriodicalIF":0.6,"publicationDate":"2025-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12477383/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145200643","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}
C3 glomerulopathies, including dense deposit disease, are rare kidney disorders caused by dysregulation of the alternative complement pathway. Recurrence after kidney transplantation is common and can threaten graft survival. Iptacopan, an oral factor B inhibitor, was recently Food and Drug Administration-approved as the first targeted therapy for C3 glomerulopathies. We describe a 19-year-old woman with childhood-onset dense deposit disease who progressed to kidney failure and underwent deceased donor transplantation. Post-transplant, she experienced delayed graft function and persistently low serum C3. A biopsy on day 7 revealed C3-only deposits, indicating early recurrent disease. Iptacopan was initiated at 200 mg twice daily. Her serum C3 normalized within 1 week and remained stable over 6 months. Proteinuria decreased significantly, and renal function improved and stabilized. This case highlights the potential of iptacopan as a disease-specific therapy for post-transplant C3 glomerulopathies recurrence, reinforcing its clinical utility and mirroring outcomes from the APPEAR-C3G trial.
{"title":"Iptacopan treatment for recurrent dense deposit disease after kidney transplant: A case report.","authors":"Lean AlKhatib, Rahaf Omaish, Neriman Gokden, Sushma Bhusal","doi":"10.1177/2050313X251380533","DOIUrl":"10.1177/2050313X251380533","url":null,"abstract":"<p><p>C3 glomerulopathies, including dense deposit disease, are rare kidney disorders caused by dysregulation of the alternative complement pathway. Recurrence after kidney transplantation is common and can threaten graft survival. Iptacopan, an oral factor B inhibitor, was recently Food and Drug Administration-approved as the first targeted therapy for C3 glomerulopathies. We describe a 19-year-old woman with childhood-onset dense deposit disease who progressed to kidney failure and underwent deceased donor transplantation. Post-transplant, she experienced delayed graft function and persistently low serum C3. A biopsy on day 7 revealed C3-only deposits, indicating early recurrent disease. Iptacopan was initiated at 200 mg twice daily. Her serum C3 normalized within 1 week and remained stable over 6 months. Proteinuria decreased significantly, and renal function improved and stabilized. This case highlights the potential of iptacopan as a disease-specific therapy for post-transplant C3 glomerulopathies recurrence, reinforcing its clinical utility and mirroring outcomes from the APPEAR-C3G trial.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251380533"},"PeriodicalIF":0.6,"publicationDate":"2025-09-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12476504/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145192633","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}
Pub Date : 2025-09-25eCollection Date: 2025-01-01DOI: 10.1177/2050313X251377265
Ilana Walters, Caroline Malcolmson, Renee Potashner, Cassandra Nelson, Romy Cho
Vitamin B12 deficiency is typically characterized by megaloblastic anemia, but can also rarely present with pseudomicroangiopathic hemolytic anemia or multilineage cytopenias secondary to dyserythropoiesis and apoptotic cell death. We describe a 12-year-old male with autism spectrum disorder who presented with fever, night sweats, and weight loss. Laboratory testing identified severe neutropenia and hemolytic anemia, and an undetectably low vitamin B12 level. Dietary history confirmed significant food restriction over the preceding 6 months. He received 1000 µg of intramuscular vitamin B12 daily for 7 days and subsequently transitioned to oral therapy. His hemoglobin, hemolytic markers, and neutrophil count normalized within 13 days of vitamin B12 initiation. Our report describes a rare and severe manifestation of vitamin B12 deficiency in a patient with autism spectrum disorder and emphasizes the importance of screening for micronutrient deficiencies among vulnerable pediatric populations to avoid unnecessary and invasive testing.
{"title":"Pseudothrombotic microangiopathy resulting from severe vitamin B12 deficiency in a pediatric patient with autism: A case report.","authors":"Ilana Walters, Caroline Malcolmson, Renee Potashner, Cassandra Nelson, Romy Cho","doi":"10.1177/2050313X251377265","DOIUrl":"10.1177/2050313X251377265","url":null,"abstract":"<p><p>Vitamin B12 deficiency is typically characterized by megaloblastic anemia, but can also rarely present with pseudomicroangiopathic hemolytic anemia or multilineage cytopenias secondary to dyserythropoiesis and apoptotic cell death. We describe a 12-year-old male with autism spectrum disorder who presented with fever, night sweats, and weight loss. Laboratory testing identified severe neutropenia and hemolytic anemia, and an undetectably low vitamin B12 level. Dietary history confirmed significant food restriction over the preceding 6 months. He received 1000 µg of intramuscular vitamin B12 daily for 7 days and subsequently transitioned to oral therapy. His hemoglobin, hemolytic markers, and neutrophil count normalized within 13 days of vitamin B12 initiation. Our report describes a rare and severe manifestation of vitamin B12 deficiency in a patient with autism spectrum disorder and emphasizes the importance of screening for micronutrient deficiencies among vulnerable pediatric populations to avoid unnecessary and invasive testing.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251377265"},"PeriodicalIF":0.6,"publicationDate":"2025-09-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12464435/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145186640","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}
Stroke is the leading cause of death among Chinese residents, a common form of which is acute large-vessel occlusion ischemic stroke, characterized by high incidence, disability, and mortality rates. To improve stroke prognosis, mechanical thrombectomy has been promoted as an effective treatment, but it involves risks of such complications as hemorrhagic transformation (symptomatic or asymptomatic bleeding), new distal embolism, reocclusion, restenosis, intraoperative vascular dissection, perforation, spasm, and hyperperfusion syndrome. As a case study, this article reports a 66-year-old male patient who suddenly developed acute cerebral infarction with acute occlusion of the left middle cerebral artery M1 segment. After mechanical thrombectomy, the patient experienced a rare complication of Purtscher-like retinopathy, causing significant vision loss in the left eye. Fundus fluorescein angiography indicated occlusion of the branch retinal artery, considered to be caused by microemboli fragmentation, escape, and blockage of the branch retinal artery during mechanical thrombectomy. Follow-up at 1, 6 months, and 1 year after discharge showed no significant improvement in the patient's vision. An in-depth analysis of its mechanism and imaging features suggests that optimization of mechanical thrombectomy devices can significantly improve procedural outcomes. For instance, the use of intermediate catheters-such as the Penumbra ACE series or Navien catheters-in combination with stent retrievers can enhance thrombus engagement and retrieval efficiency, thereby reducing the risk of distal embolization and lowering the incidence of procedure-related complications.
{"title":"Purtscher-like retinopathy following mechanical thrombectomy: A case report.","authors":"Shuangyun Lv, Ronghui Liu, Jiapeng Sun, Hongmei Liu, Kun Li, Chong Li, Jinhui Bian, Changxia Ding, Xin Guo","doi":"10.1177/2050313X251381564","DOIUrl":"10.1177/2050313X251381564","url":null,"abstract":"<p><p>Stroke is the leading cause of death among Chinese residents, a common form of which is acute large-vessel occlusion ischemic stroke, characterized by high incidence, disability, and mortality rates. To improve stroke prognosis, mechanical thrombectomy has been promoted as an effective treatment, but it involves risks of such complications as hemorrhagic transformation (symptomatic or asymptomatic bleeding), new distal embolism, reocclusion, restenosis, intraoperative vascular dissection, perforation, spasm, and hyperperfusion syndrome. As a case study, this article reports a 66-year-old male patient who suddenly developed acute cerebral infarction with acute occlusion of the left middle cerebral artery M1 segment. After mechanical thrombectomy, the patient experienced a rare complication of Purtscher-like retinopathy, causing significant vision loss in the left eye. Fundus fluorescein angiography indicated occlusion of the branch retinal artery, considered to be caused by microemboli fragmentation, escape, and blockage of the branch retinal artery during mechanical thrombectomy. Follow-up at 1, 6 months, and 1 year after discharge showed no significant improvement in the patient's vision. An in-depth analysis of its mechanism and imaging features suggests that optimization of mechanical thrombectomy devices can significantly improve procedural outcomes. For instance, the use of intermediate catheters-such as the Penumbra ACE series or Navien catheters-in combination with stent retrievers can enhance thrombus engagement and retrieval efficiency, thereby reducing the risk of distal embolization and lowering the incidence of procedure-related complications.</p>","PeriodicalId":21418,"journal":{"name":"SAGE Open Medical Case Reports","volume":"13 ","pages":"2050313X251381564"},"PeriodicalIF":0.6,"publicationDate":"2025-09-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12464436/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145186657","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}