CEN Case Reports最新文献_第7页

A case of lupus nephritis achieving early remission using a treatment protocol similar to the AURORA trial with voclosporin and successfully discontinuing corticosteroids early. 狼疮性肾炎的病例实现早期缓解使用治疗方案类似AURORA试验与氯菌素和成功停用皮质类固醇早期。

IF 1

CEN Case Reports Pub Date : 2026-05-22 DOI: 10.1007/s13730-026-01133-2

Akira Mima, Masasuke Kumagai, Shizuka Fukumoto, Yuta Funabiki, Sakura Kure, Takashi Hirata, Yuta Saito, Keishi Matsumoto, Shinji Lee

{"title":"A case of lupus nephritis achieving early remission using a treatment protocol similar to the AURORA trial with voclosporin and successfully discontinuing corticosteroids early.","authors":"Akira Mima, Masasuke Kumagai, Shizuka Fukumoto, Yuta Funabiki, Sakura Kure, Takashi Hirata, Yuta Saito, Keishi Matsumoto, Shinji Lee","doi":"10.1007/s13730-026-01133-2","DOIUrl":"10.1007/s13730-026-01133-2","url":null,"abstract":"Lupus nephritis (LN) is characterized by immune system attacks on self-tissues, triggered by the production of pathological autoantibodies, leading to damage in multiple organs and tissues throughout the body. The second-generation calcineurin inhibitor, voclosporin, has recently been recommended in guidelines as an adjunct to basic immunosuppressive interventions for managing active LN in adult patients. However, there are still no real-world data showing that remission was achieved by administering voclosporin in LN using the same protocol as the AURORA trial. We report the case of a 19-year-old woman with proteinuria and microscopic hematuria. The patient developed nephrotic syndrome three years prior to presentation to our department and achieved complete remission with corticosteroids. In the year of presentation, hypocomplementemia and anti-ds-DNA antibodies were detected. Renal biopsy revealed a thickened glomerular basement membrane with spike formation, granular C1q deposition in the mesangial regions, and high electron-density deposits on the epithelial side of the basement membrane. The patient was diagnosed with LN (ISN V) and started treatment with voclosporin combined with corticosteroids, hydroxychloroquine, and mycophenolate mofetil to decrease corticosteroids, which was similar to the regimen as in the AURORA study. Complete remission was achieved 30 days after treatment initiation, and the corticosteroids were discontinued 25 weeks after treatment initiation. This study represents the first clinical case report demonstrating complete remission of LN achieved by implementing the AURORA regimen.","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":"15 3","pages":""},"PeriodicalIF":1.0,"publicationDate":"2026-05-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13197560/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147986773","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

Ten-year clinical course of familial lecithin-cholesterol acyltransferase deficiency caused by Cys74Tyr mutation leading to dialysis and multiple systemic complications: a case report. Cys74Tyr突变致家族性卵磷脂-胆固醇酰基转移酶缺乏导致透析及多重全身并发症的10年临床病程1例

IF 1

CEN Case Reports Pub Date : 2026-05-20 DOI: 10.1007/s13730-026-01128-z

Takuya Yamazaki, Shokichi Naito, Shun Sakurabayashi, Keiko Sano, Tetsuya Abe, Mariko Kamata, Togo Aoyama, Yasuo Takeuchi

{"title":"Ten-year clinical course of familial lecithin-cholesterol acyltransferase deficiency caused by Cys74Tyr mutation leading to dialysis and multiple systemic complications: a case report.","authors":"Takuya Yamazaki, Shokichi Naito, Shun Sakurabayashi, Keiko Sano, Tetsuya Abe, Mariko Kamata, Togo Aoyama, Yasuo Takeuchi","doi":"10.1007/s13730-026-01128-z","DOIUrl":"10.1007/s13730-026-01128-z","url":null,"abstract":"Familial lecithin-cholesterol acyltransferase deficiency (FLD) is a rare hereditary disorder. In FLD, a clinical triad of corneal opacity, anemia, and proteinuria typically precedes progressive renal dysfunction. Although early renal involvement has been widely described, there are limited reports on the long-term course leading to dialysis initiation, including systemic complications. We present a genetically confirmed case of FLD caused by the Cys74Tyr mutation, which had a clinical course exceeding 10 years with the emergence of multisystem complications. The patient initially showed a transient stabilization of renal function following a fat-restricted and protein-restricted diet and renin-angiotensin system blockade; however, subsequent decline occurred despite continued management. During this deterioration phase, systemic complications emerged, including progressive aortic valve stenosis, complete atrioventricular block, heart failure, and cerebellar infarction, in close temporal association with worsening renal function. Hemodialysis initiation led to rapid resolution of erythropoietin-resistant anemia, which may be attributable to the improvement in the uremic milieu following dialysis initiation. This case offers valuable insights into the late-stage progression of FLD. The observations may help determine the point at which renoprotective and metabolic interventions become less effective. Furthermore, the temporal association between renal deterioration and the development of cardiovascular and neurovascular complications suggests the need for careful clinical surveillance during advanced stages of renal dysfunction.","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":"15 3","pages":""},"PeriodicalIF":1.0,"publicationDate":"2026-05-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13190904/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147970777","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

Severe mesangiolysis with prominent microaneurysm formation following total body irradiation conditioning for bone marrow transplantation. 骨髓移植术后全身照射后严重的系膜松解伴明显的微动脉瘤形成。

IF 1

CEN Case Reports Pub Date : 2026-05-20 DOI: 10.1007/s13730-026-01130-5

Yosuke Nakagawa, Masahiro Koizumi, Norisuke Shimamura, Naoto Hamano, Go Ogura, Takehiko Wada, Makoto Onizuka, Hirotaka Komaba

{"title":"Severe mesangiolysis with prominent microaneurysm formation following total body irradiation conditioning for bone marrow transplantation.","authors":"Yosuke Nakagawa, Masahiro Koizumi, Norisuke Shimamura, Naoto Hamano, Go Ogura, Takehiko Wada, Makoto Onizuka, Hirotaka Komaba","doi":"10.1007/s13730-026-01130-5","DOIUrl":"10.1007/s13730-026-01130-5","url":null,"abstract":"Patients who undergo hematopoietic stem cell transplantation (HSCT) face a high risk of acute kidney injury (AKI), which significantly increases the likelihood of developing chronic kidney disease (CKD). Proteinuria is also frequently observed after HSCT and is associated with an increased risk of CKD progression. Thrombotic microangiopathy (TMA), stemming from endothelial injury caused by factors such as total body irradiation (TBI), calcineurin inhibitors, tyrosine kinase inhibitors, and graft-versus-host disease, is a major contributor to both AKI and CKD. We report a case of a woman in her early thirties who developed progressive kidney dysfunction and subnephrotic-range proteinuria three months after HSCT. Kidney biopsy findings included marked mesangiolysis, endothelial cell detachment, subendothelial expansion, and diffuse segmental capillary microaneurysms, which are highly suggestive of TMA due to TBI administered as part of the myeloablative conditioning regimen prior to HSCT. She underwent conservative management for chronic kidney disease, but her kidney function progressively declined. In patients presenting with a progressive decline in kidney function following HSCT, TMA should be included in the differential diagnosis. A kidney biopsy is valuable for determining the underlying cause of HSCT-associated TMA, as treatment strategies vary depending on the etiology. Prominent mesangiolysis with capillary microaneurysms, as observed in this case and the previously reported irradiated patients, may serve as a distinctive histological marker of radiation nephropathy.","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":"15 3","pages":""},"PeriodicalIF":1.0,"publicationDate":"2026-05-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13190939/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147970703","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

Adenovirus infection associated with focal segmental glomerulosclerosis following autologous hematopoietic stem cell transplantation. 腺病毒感染与自体造血干细胞移植后局灶节段性肾小球硬化相关。

IF 1

CEN Case Reports Pub Date : 2026-05-20 DOI: 10.1007/s13730-026-01110-9

Yasser Isaac Arana-Escandón, Paula Eliana Ramírez-Arboleda, Lina María Gaviria-Jaramillo, Mauricio Andrés Álzate-Arias, Oliver Gerardo Perilla-Suarez, María Ximena Lagos-Buitrago, Joaquín Roberto Rodelo-Ceballos, Mayra Alejandra Estacio, Alejandra Taborda-Murillo

{"title":"Adenovirus infection associated with focal segmental glomerulosclerosis following autologous hematopoietic stem cell transplantation.","authors":"Yasser Isaac Arana-Escandón, Paula Eliana Ramírez-Arboleda, Lina María Gaviria-Jaramillo, Mauricio Andrés Álzate-Arias, Oliver Gerardo Perilla-Suarez, María Ximena Lagos-Buitrago, Joaquín Roberto Rodelo-Ceballos, Mayra Alejandra Estacio, Alejandra Taborda-Murillo","doi":"10.1007/s13730-026-01110-9","DOIUrl":"10.1007/s13730-026-01110-9","url":null,"abstract":"Disseminated adenovirus infection is a rare but often fatal complication of hematopoietic stem cell transplantation (HSCT), particularly uncommon in the autologous setting. We report the case of a 59-year-old man with stage IVB diffuse large B-cell lymphoma who underwent autologous HSCT after multiple lines of therapy. Following conditioning regimen, he developed persistent fever, mucositis, dysuria, and gross hematuria, with imaging showing bilateral pyelonephritis, ureteritis, and cystitis. Despite broad-spectrum antibiotics, symptoms persisted. Quantitative PCR revealed rapidly rising adenovirus viremia (> 1,000,000 copies/mL), and renal biopsy demonstrated viral cytopathic changes on light microscopy together with focal segmental glomerulosclerosis (FSGS). The patient progressed to fulminant oliguric renal failure and died before dialysis could be initiated. This case highlights a rare presentation of adenovirus infection with renal involvement consistent with adenovirus-associated nephropathy and concurrent FSGS after autologous HSCT, highlighting the potential convergence of viral, neoplastic, and transplant-related mechanisms in catastrophic renal injury.","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":"15 3","pages":""},"PeriodicalIF":1.0,"publicationDate":"2026-05-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13190899/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147970737","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

Evaluation of continuous renal replacement therapy efficacy in severe caffeine intoxication: concentration-dependent variation in caffeine clearance. 重度咖啡因中毒持续肾脏替代治疗的疗效评价：咖啡因清除率的浓度依赖性变化。

IF 1

CEN Case Reports Pub Date : 2026-05-12 DOI: 10.1007/s13730-026-01127-0

Saki Takeda, Haruki Chiba, Takeru Yamazaki, Yasuhiko Tsukada, Makoto Onodera, Ken Iseki, Kazuki Harada

{"title":"Evaluation of continuous renal replacement therapy efficacy in severe caffeine intoxication: concentration-dependent variation in caffeine clearance.","authors":"Saki Takeda, Haruki Chiba, Takeru Yamazaki, Yasuhiko Tsukada, Makoto Onodera, Ken Iseki, Kazuki Harada","doi":"10.1007/s13730-026-01127-0","DOIUrl":"10.1007/s13730-026-01127-0","url":null,"abstract":"Hemodialysis is an effective treatment option for acute caffeine intoxication. However, the efficacy of continuous renal replacement therapy remains a subject of debate. We encountered a case of acute caffeine intoxication that was managed using high-flow continuous hemodiafiltration. To evaluate the effectiveness of high-flow continuous hemodiafiltration, we compared it with other continuous renal replacement therapy approaches in managing cases of caffeine intoxication. In this study, we aimed to elucidate the caffeine-removal ability of continuous renal replacement therapy and assess its effectiveness. Additionally, we aimed to investigate the underlying reasons for the conflicting reports regarding the effectiveness of continuous renal replacement therapy. The time course of the blood caffeine concentration and elimination half-life were used for the evaluation. A woman in her 40s attempted suicide by ingesting an overdose of caffeine hydrate tablets. She presented with sweating, vomiting, tachycardia, sinus tachycardia, and tremors. The serum caffeine concentration in this case decreased from 103 to 50 μg/mL after high-flow continuous hemodiafiltration. The initial symptoms at the time of admission also disappeared post-treatment. Caffeine clearance by continuous renal replacement therapy may vary depending on the blood caffeine concentration, and this concentration-dependent variation may lead to discrepancies in the evaluation of continuous renal replacement therapy. The discrepancies in the ongoing debate regarding the efficacy of continuous renal replacement therapy were likely attributable to the selection of inappropriate comparators. We inferred that continuous renal replacement therapy provides a therapeutic benefit because continuous renal replacement therapy may facilitate caffeine clearance.","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":"15 3","pages":""},"PeriodicalIF":1.0,"publicationDate":"2026-05-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13168395/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147873530","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

Refractory minimal change disease associated with Mycobacterium marinum infection. 与海洋分枝杆菌感染相关的难治性微小改变疾病。

IF 1

CEN Case Reports Pub Date : 2026-05-12 DOI: 10.1007/s13730-026-01131-4

Yuto Matsui, Kan Katayama, Yuka Mitamura, Keiichi Nagata, Koyo Ohara, Toru Hibi, Yuhei Ito, Naohiro Kawamura, Mayuko Yamakawa, Yuri Oue, Mutsuki Mori, Masahiro Yamawaki, Ryosuke Saiki, Tomohiro Murata, Kaoru Dohi

{"title":"Refractory minimal change disease associated with Mycobacterium marinum infection.","authors":"Yuto Matsui, Kan Katayama, Yuka Mitamura, Keiichi Nagata, Koyo Ohara, Toru Hibi, Yuhei Ito, Naohiro Kawamura, Mayuko Yamakawa, Yuri Oue, Mutsuki Mori, Masahiro Yamawaki, Ryosuke Saiki, Tomohiro Murata, Kaoru Dohi","doi":"10.1007/s13730-026-01131-4","DOIUrl":"10.1007/s13730-026-01131-4","url":null,"abstract":"This case report describes a rare instance of minimal change disease (MCD) associated with Mycobacterium marinum infection. MCD is typically idiopathic but can occasionally occur secondary to infections. The patient was a 61-year-old fisherman with a history of relapsing MCD who developed a subcutaneous abscess in the lower leg while undergoing rituximab and glucocorticoid therapy. Ziehl-Neelsen staining revealed acid-fast bacilli, and culture at 30 °C isolated M. marinum, a slow-growing nontuberculous mycobacterium (NTM) commonly found in aquatic environments. The infection was attributed to the combination of occupational exposure and immunosuppression. Targeted antimicrobial therapy with clarithromycin, minocycline, and rifampicin led to resolution of the abscess and complete remission of MCD, suggesting a causal link between the infection and disease relapse. This case highlights several key clinical considerations. First, secondary infectious causes should be evaluated in patients with treatment-resistant nephrotic syndrome. Second, appropriate laboratory techniques, including optimized culture conditions, are critical for identifying rare NTM. Third, a detailed assessment of lifestyle and occupational exposures can provide essential diagnostic clues. The report underscores the importance of vigilance in atypical or refractory MCD cases and supports the role of targeted antimicrobial therapy in resolving both infection and associated nephrotic syndrome.","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":"15 3","pages":""},"PeriodicalIF":1.0,"publicationDate":"2026-05-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13168420/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147873509","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

An unusual presentation of dialysis-related amyloidosis: a large tumor extending from the gingiva to the palate. 透析相关淀粉样变的不寻常表现：一个从牙龈延伸到上颚的大肿瘤。

IF 1

CEN Case Reports Pub Date : 2026-05-12 DOI: 10.1007/s13730-026-01129-y

Sachio Iwanari, Mari Tanaka, Toshihisa Akimune, Yuri Suzuki, Akira Morita, Yoshinao Matsuyama, Mako Takayoshi, Hiroto Yamamoto, Yutaro Nishiyama, Shuhei Iwamoto, Akane Murata, Yuki Ishida, Mariko Teragaki, Masaki Ikeda, Hiroya Takeoka

{"title":"An unusual presentation of dialysis-related amyloidosis: a large tumor extending from the gingiva to the palate.","authors":"Sachio Iwanari, Mari Tanaka, Toshihisa Akimune, Yuri Suzuki, Akira Morita, Yoshinao Matsuyama, Mako Takayoshi, Hiroto Yamamoto, Yutaro Nishiyama, Shuhei Iwamoto, Akane Murata, Yuki Ishida, Mariko Teragaki, Masaki Ikeda, Hiroya Takeoka","doi":"10.1007/s13730-026-01129-y","DOIUrl":"10.1007/s13730-026-01129-y","url":null,"abstract":"Dialysis-related amyloidosis (DRA) is a clinically significant complication of long-term hemodialysis, primarily affecting the bones and joints. Involvement of oral cavity is uncommon, and almost all reported cases are localized to the tongue. Here, we report a rare case of DRA presenting as a large tumor originating from the maxillary gingiva in a hemodialysis patient. We also review previously reported cases of oral DRA. A 77-year-old Japanese woman with diabetic kidney disease had been receiving maintenance hemodialysis for 11 years. She was referred to our hospital for evaluation of a large tumor extending from the maxillary gingiva to the palate. Although she had been completely edentulous in the maxilla for years, she did not wear dentures. A biopsy specimen from the tumor revealed diffuse subepithelial deposition of eosinophilic, amorphous material, which was positive for direct fast scarlet stain and exhibited apple-green birefringence under polarized light. Immunohistochemistry confirmed the presence of β2-microglobulin, leading to the diagnosis of DRA. The tumor was surgically resected, and no recurrence was noted during follow-up. Our case and literature review suggest that chronic mechanical stimulation in the oral cavity may contribute to the development of oral DRA. These findings highlight the importance of regular oral examinations and the elimination of mechanical irritation in patients receiving maintenance hemodialysis.","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":"15 3","pages":""},"PeriodicalIF":1.0,"publicationDate":"2026-05-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13168372/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147873524","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

Kidney-limited thrombotic microangiopathy caused by relative hypertension in a hemodynamically vulnerable kidney allograft, mimicking malignant hypertension. 在血流动力学易损的同种异体肾移植中，由相对高血压引起的肾限制性血栓性微血管病，模仿恶性高血压。

IF 1

CEN Case Reports Pub Date : 2026-05-11 DOI: 10.1007/s13730-026-01120-7

Shinnosuke Sugihara, Tatsuhiko Azegami, Shinya Morita, Akinori Hashiguchi, Kaori Hayashi

{"title":"Kidney-limited thrombotic microangiopathy caused by relative hypertension in a hemodynamically vulnerable kidney allograft, mimicking malignant hypertension.","authors":"Shinnosuke Sugihara, Tatsuhiko Azegami, Shinya Morita, Akinori Hashiguchi, Kaori Hayashi","doi":"10.1007/s13730-026-01120-7","DOIUrl":"10.1007/s13730-026-01120-7","url":null,"abstract":"Malignant hypertension is traditionally defined as severe systemic blood pressure elevation accompanied by end-organ damage. In kidney transplant recipients, however, kidney allografts may be particularly vulnerable to hypertensive injury even at lower blood pressure levels, owing to impaired autoregulation associated with denervation and calcineurin inhibitor exposure. We report a 52-year-old kidney transplant recipient presenting with severe kidney injury (serum creatinine, 4.90 mg/dL) 16 months after transplantation. Although systemic blood pressure remained within the range of 140-170/90-105 mmHg-below the classical threshold of malignant hypertension-an allograft biopsy revealed the lesions suggestive of thrombotic microangiopathy (TMA), including mesangiolysis and edematous intimal thickening of the arteries. After exclusion of antibody-mediated rejection, calcineurin inhibitor toxicity, and primary TMAs, the patient was diagnosed with kidney-limited TMA attributable to relative hypertension producing a malignant hypertension-like phenotype in the setting of graft-specific vulnerability. Given the presumed contribution of the renin-angiotensin system (RAS)-mediated vascular injury, an angiotensin II receptor blocker was initiated despite severe kidney dysfunction, resulting in marked improvement in kidney function and proteinuria. This case illustrates that kidney allografts may develop a malignant hypertension-like pathophysiology with kidney-limited TMA at blood pressure levels below the classical malignant range. Awareness of this discrepancy may help clinicians recognize atypical hypertensive injury in transplant recipients, and early RAS blockade may be considered as a potential strategy to preserve allograft function, even in the setting of severe kidney injury.","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":"15 3","pages":""},"PeriodicalIF":1.0,"publicationDate":"2026-05-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13161382/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147866884","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

Acute tubulointerstitial nephritis following a coronavirus disease 2019 vaccine booster dose: a pediatric case report. 2019冠状病毒病疫苗加强剂后急性肾小管间质性肾炎：一份儿科病例报告

IF 1

CEN Case Reports Pub Date : 2026-05-08 DOI: 10.1007/s13730-026-01126-1

Haruhide Sakaguchi, Daishi Hirano, Yoichi Takemasa, Yuna Hiwatari, Shiho Yoshizawa, Yosuke Shoji, Aya Saito, Chisato Umeda, Saori Miwa, Akira Ito, Kimihiko Oishi

{"title":"Acute tubulointerstitial nephritis following a coronavirus disease 2019 vaccine booster dose: a pediatric case report.","authors":"Haruhide Sakaguchi, Daishi Hirano, Yoichi Takemasa, Yuna Hiwatari, Shiho Yoshizawa, Yosuke Shoji, Aya Saito, Chisato Umeda, Saori Miwa, Akira Ito, Kimihiko Oishi","doi":"10.1007/s13730-026-01126-1","DOIUrl":"10.1007/s13730-026-01126-1","url":null,"abstract":"Messenger ribonucleic acid vaccines against coronavirus disease 2019 (COVID-19) have been temporally associated with various renal adverse events, including acute tubulointerstitial nephritis (ATIN). Herein, we present the case of a 14-year-old male who developed biopsy-confirmed ATIN following a booster dose of the BNT162b2 (Pfizer-BioNTech) severe acute respiratory syndrome coronavirus 2 vaccine. Ten days after vaccination, the patient presented with fever, fatigue, and nausea and was referred to our hospital because of impaired kidney function. Kidney biopsy demonstrated diffuse tubulointerstitial inflammation consistent with ATIN, and gallium-67 scintigraphy showed diffuse bilateral kidney uptake. The lymphocyte transformation test (LTT) showed a stronger positive response to the BNT162b2 vaccine than to concomitant medications, suggesting a possible association with vaccination. Corticosteroid therapy led to rapid improvement in kidney function and normalization of tubular injury markers. This case demonstrated that ATIN may rarely occur after booster vaccination, and highlights the potential role of the LTT as a useful tool when multiple possible triggers are present.","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":"15 3","pages":""},"PeriodicalIF":1.0,"publicationDate":"2026-05-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13156369/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147834170","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

Familial hypomagnesemia with hypercalciuria and nephrocalcinosis treated with living-donor kidney transplantation: case report. 活体肾移植治疗家族性低镁血症伴高钙尿和肾钙质沉着症1例。

IF 1

CEN Case Reports Pub Date : 2026-05-07 DOI: 10.1007/s13730-026-01103-8

Akari Kaba, Keizo Kaku, Kodai Shingaki, Shinsuke Kubo, Yu Hisadome, Hiroshi Noguchi, Kei Nishiyama, Yasuhiro Okabe, Masafumi Nakamura

{"title":"Familial hypomagnesemia with hypercalciuria and nephrocalcinosis treated with living-donor kidney transplantation: case report.","authors":"Akari Kaba, Keizo Kaku, Kodai Shingaki, Shinsuke Kubo, Yu Hisadome, Hiroshi Noguchi, Kei Nishiyama, Yasuhiro Okabe, Masafumi Nakamura","doi":"10.1007/s13730-026-01103-8","DOIUrl":"10.1007/s13730-026-01103-8","url":null,"abstract":"Familial hypomagnesemia with hypercalciuria and nephrocalcinosis (FHHNC) is a rare genetic disorder characterized by renal magnesium wasting, hypercalciuria, and progressive renal function decline, often culminating in end-stage renal disease (ESRD). This report presents the case of a successful living-donor kidney transplantation from a mother to her 22-year-old son, who was diagnosed with FHHNC. The patient initially exhibited hematuria at age two years, progressing to recurrent nephrolithiasis and hypercalciuria. Genetic analysis at age nine years confirmed FHHNC because of a CLDN-16 gene mutation. The CLDN-16 gene, which codes for claudin-16, is a crucial protein involved in renal magnesium and calcium reabsorption. While both parents and one sibling were identified as heterozygous carriers of the mutation, only the patient experienced severe symptoms. As his renal function progressively declined, ultimately reaching ESRD, a living-donor transplant was deemed necessary. His mother, with normal renal function and no history of nephrolithiasis, was evaluated as a suitable donor. Post-transplantation, the patient demonstrated rapid improvement in renal function, with magnesium levels stabilizing to baseline without supplementation, indicating effective graft function. This case underscores the complexities of managing FHHNC, particularly regarding kidney transplantation options and outcomes. Given the rarity of FHHNC, this report highlights the critical role of genetic diagnosis, family screening, and timely intervention. Additionally, it contributes to the limited literature on post-transplant outcomes in FHHNC, emphasizing the potential for living-donor transplantation to restore renal function and enhance quality of life in affected patients.","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":"15 3","pages":""},"PeriodicalIF":1.0,"publicationDate":"2026-05-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13153341/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147834244","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