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Successful treatment with bortezomib for refractory cryoglobulinemic vasculitis triggered by ischemic non-obstructive coronary artery disease. 硼替佐米成功治疗缺血性非阻塞性冠状动脉疾病引发的难治性冷球蛋白性血管炎。
IF 1
CEN Case Reports Pub Date : 2025-01-06 DOI: 10.1007/s13730-024-00963-2
Yui Ohta, Takaaki Tsuchiya, Masatoshi Oka, Moriaki Tachibana, Yoshitaka Kondo, Kaoruko Fukushima, Shiho Matsuno, Noriko Yamanaka, Noriyuki Suzuki, Akiko Komatsu, Hirofumi Rokutan, Wako Yumura, Tomio Arai, Akihito Ishigami, Mitsuyo Itabashi, Takashi Takei
{"title":"Successful treatment with bortezomib for refractory cryoglobulinemic vasculitis triggered by ischemic non-obstructive coronary artery disease.","authors":"Yui Ohta, Takaaki Tsuchiya, Masatoshi Oka, Moriaki Tachibana, Yoshitaka Kondo, Kaoruko Fukushima, Shiho Matsuno, Noriko Yamanaka, Noriyuki Suzuki, Akiko Komatsu, Hirofumi Rokutan, Wako Yumura, Tomio Arai, Akihito Ishigami, Mitsuyo Itabashi, Takashi Takei","doi":"10.1007/s13730-024-00963-2","DOIUrl":"https://doi.org/10.1007/s13730-024-00963-2","url":null,"abstract":"<p><p>Type I and mixed cryoglobulinemic vasculitis differ in pathophysiology, clinical presentation, and therapeutic response. We report a case of refractory cryoglobulinemic vasculitis diagnosed following ischemic non-obstructive coronary artery disease (INOCA). The patient presented with dyspnea, as well as abdominal pain due to ischemic enteritis, purpura, and renal failure requiring dialysis. Despite the patient's IgG λ-type monoclonal gammopathy of undetermined significance (MGUS) and negative hepatitis C virus, the presence of rheumatoid factor (RF) activity and the possibility of IgM involvement were suggested by cryoglobulin analysis and strong glomerular IgM deposition. The condition was diagnosed as mixed cryoglobulinemia, and various immunomodulatory treatments, including methylprednisolone, rituximab and plasmapheresis, were administered without achieving cryoglobulin negativity. However, treatment with bortezomib and dexamethasone ultimately led to cryoglobulin negativity and clinical improvement although the patient was not weaned off dialysis, resulting in remission of the cryoglobulinemic vasculitis. This case suggests that bortezomib, a proteasome inhibitor, may be a promising treatment for refractory cryoglobulinemic vasculitis.</p>","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":" ","pages":""},"PeriodicalIF":1.0,"publicationDate":"2025-01-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142930776","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
The application of omalizumab in children with atopic diseases complicated by nephrotic syndrome: a case report. 奥玛珠单抗在儿童特应性疾病并发肾病综合征中的应用:1例报告
IF 1
CEN Case Reports Pub Date : 2024-12-31 DOI: 10.1007/s13730-024-00962-3
Yi Mao, Jialu Lu, Lei Yin, Chong Liu, Jinhong Wu, Youying Mao
{"title":"The application of omalizumab in children with atopic diseases complicated by nephrotic syndrome: a case report.","authors":"Yi Mao, Jialu Lu, Lei Yin, Chong Liu, Jinhong Wu, Youying Mao","doi":"10.1007/s13730-024-00962-3","DOIUrl":"https://doi.org/10.1007/s13730-024-00962-3","url":null,"abstract":"<p><p>Nephrotic syndrome (NS) and atopic diseases are linked through shared immunological pathways, with allergic triggers often contributing to NS relapses, particularly in immunoglobulin E (IgE)-mediated pathways. Omalizumab, a humanized monoclonal antibody targeting free and cell-bound IgE, is commonly used in treating atopic diseases. We presented a pediatric case with a history of eczema, asthma, and recurrent atopic rhinitis, who first developed NS at age three, responding well to steroid therapy. The patient experienced four NS relapses, three of which were associated with significant atopic symptoms, elevated eosinophil counts, and increased IgE levels, suggesting allergic triggers. After the latest relapse achieved remission, steroid therapy was discontinued, and Omalizumab was initiated two months later. The patient received monthly subcutaneous Omalizumab (300 mg) with regular urine protein monitoring. To date, the patient has shown marked improvement in atopic symptoms, sustained NS control, and reduced steroid dependence. This case highlights the potential of Omalizumab in treating NS triggered by allergic reactions, further prospective pilot studies and randomized controlled trials are warranted to rigorously evaluate its efficacy and safety.</p>","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":" ","pages":""},"PeriodicalIF":1.0,"publicationDate":"2024-12-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142909491","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
A case of neuron-derived neurotrophic factor-positive, syphilis-related membranous nephropathy that achieved spontaneous remission. 一例神经源性神经营养因子阳性,梅毒相关的膜性肾病实现自发缓解。
IF 1
CEN Case Reports Pub Date : 2024-12-28 DOI: 10.1007/s13730-024-00960-5
Yuko Yoshida, Kenji Ueki, Yuta Matsukuma, Akihiro Tsuchimoto, Eri Ataka, Hirofumi Okamoto, Kumiko Torisu, Yuichirou Hara, Hirofumi Ikeda, Takanari Kitazono, Toshiaki Nakano
{"title":"A case of neuron-derived neurotrophic factor-positive, syphilis-related membranous nephropathy that achieved spontaneous remission.","authors":"Yuko Yoshida, Kenji Ueki, Yuta Matsukuma, Akihiro Tsuchimoto, Eri Ataka, Hirofumi Okamoto, Kumiko Torisu, Yuichirou Hara, Hirofumi Ikeda, Takanari Kitazono, Toshiaki Nakano","doi":"10.1007/s13730-024-00960-5","DOIUrl":"https://doi.org/10.1007/s13730-024-00960-5","url":null,"abstract":"<p><p>Neuron-derived neurotrophic factor (NDNF) was discovered as a target antigen in membranous nephropathy (MN) caused by syphilis. However, there have been few reports of NDNF-positive MN in Japan. A 19-year-old female patient was admitted to our hospital with nephrotic syndrome and acute kidney injury. After admission, she presented with a skin rash and was serologically positive for syphilis. Kidney biopsy showed MN positive not only for immunoglobulin (Ig) G, but also for IgA and complement C1q. IgG subclass analysis revealed positivity for IgG1 and IgG3. Immunohistochemistry for glomerular NDNF was positive, and a diagnosis of syphilis-related MN was confirmed. Prior to treatment of the infection, her urinary protein decreased to achieve complete remission and her kidney function improved. Benzylpenicillin was administered, and no recurrence of nephrotic syndrome or kidney dysfunction was observed thereafter. This case illustrates the importance of considering syphilis infection when encountering a case of nephrotic syndrome with skin rash. In addition, urinalysis of syphilitic patients should be performed to avoid missing MN that may be in spontaneous remission. Further elucidation of the pathogenesis of NDNF-positive, syphilis-related MN in Japan is needed.</p>","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":" ","pages":""},"PeriodicalIF":1.0,"publicationDate":"2024-12-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142892388","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Effective combination of corticosteroid and cyclosporine A for immunoglobulin A nephropathy with membranoproliferative glomerulonephritis features: a case report. 皮质类固醇和环孢素A有效联合治疗免疫球蛋白A肾病伴膜增生性肾小球肾炎1例
IF 1
CEN Case Reports Pub Date : 2024-12-28 DOI: 10.1007/s13730-024-00961-4
Saeko Hatanaka, Go Kanzaki, Kentaro Koike, Hiroyuki Ueda, Nanae Matsuo, Yukio Maruyama, Nobuo Tsuboi, Takashi Yokoo
{"title":"Effective combination of corticosteroid and cyclosporine A for immunoglobulin A nephropathy with membranoproliferative glomerulonephritis features: a case report.","authors":"Saeko Hatanaka, Go Kanzaki, Kentaro Koike, Hiroyuki Ueda, Nanae Matsuo, Yukio Maruyama, Nobuo Tsuboi, Takashi Yokoo","doi":"10.1007/s13730-024-00961-4","DOIUrl":"https://doi.org/10.1007/s13730-024-00961-4","url":null,"abstract":"<p><p>Immunoglobulin A nephropathy (IgAN) is the most common primary glomerulonephritis worldwide with heterogeneous histopathological phenotypes. Although IgAN with membranoproliferative glomerulonephritis (MPGN)-like features has been reported in children and adults, treatment strategies for this rare IgAN subtype have not been established. Here, we present the case of a 56-year-old man with no history of kidney disease who initially presented with nephrotic syndrome. Renal biopsy revealed MPGN-like features with a negative serological workup for secondary causes. Immunofluorescent staining was predominantly positive for IgA in the glomerular mesangial and capillary walls. Galactose-deficient IgA1 staining showed a distribution pattern similar to IgA staining. Electron microscopy revealed disorganized structural deposits in the mesangial and subendothelial regions. Based on clinical and histopathological findings, the patient was diagnosed with primary IgAN. The nephrotic syndrome resolved completely after six months of combined corticosteroids and cyclosporine A (CsA) therapy. Although corticosteroids and CsA were tapered off, hematuria and proteinuria remained in complete remission for years of follow-up. This case demonstrates the importance of recognizing IgAN with MPGN-like features as a histopathological subtype that may benefit from intensive immunosuppressive therapy.</p>","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":" ","pages":""},"PeriodicalIF":1.0,"publicationDate":"2024-12-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142892389","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Granulomatosis with polyangiitis with rapidly progressive glomerulonephritis treated with a multipronged approach-a case based review. 肉芽肿病合并多血管炎合并快速进展的肾小球肾炎的多管齐下治疗-基于病例的回顾。
IF 1
CEN Case Reports Pub Date : 2024-12-20 DOI: 10.1007/s13730-024-00959-y
Ujjwal Madan, Vishesh Goel, Jignesh Shah, Hameed Ahmad, Clarissa Cassol, Amr Edrees
{"title":"Granulomatosis with polyangiitis with rapidly progressive glomerulonephritis treated with a multipronged approach-a case based review.","authors":"Ujjwal Madan, Vishesh Goel, Jignesh Shah, Hameed Ahmad, Clarissa Cassol, Amr Edrees","doi":"10.1007/s13730-024-00959-y","DOIUrl":"https://doi.org/10.1007/s13730-024-00959-y","url":null,"abstract":"<p><p>Granulomatosis with polyangiitis is an ANCA-associated vasculitis that involves small to medium-sized vessels. The extent of renal involvement varies, which is also associated with disease prognosis, with aggressive renal involvement having worse outcomes. Rapidly progressive glomerulonephritis with severe inflammatory features and extensive crescent formation can be challenging to treat. Usually, induction regimes utilize a combination of pulse dose methylprednisolone followed by rituximab or cyclophosphamide. Resistant diseases pose additional treatment challenges, and individualized treatment regimens have been described without accumulated outcome data. Cyclophosphamide, rituximab, azathioprine, methotrexate, and mycophenolate with or without plasmapheresis have been variably used, but there is a lack of consensus on a standardized regime in literature. Our case adds to the existing literature on the treatment-refractory granulomatosis with polyangiitis, which was treated with high-dose corticosteroid in combination with rituximab, low-dose cyclophosphamide, plasmapheresis, and brief use of hemodialysis. It also reiterates that the use of a variety of low-dose cyclophosphamide with rituximab could be beneficial for treatment-refractory cases or patients with severe renal involvement, in addition to better tolerance with low dose cyclophosphamide in comparison with full-dose cyclophosphamide.</p>","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":" ","pages":""},"PeriodicalIF":1.0,"publicationDate":"2024-12-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142863524","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Parvovirus B19-related membranoproliferative glomerulonephritis presenting with positive glomerular staining for nephritis-associated plasmin receptor: a case report and review of the literature. 细小病毒b19相关膜增生性肾小球肾炎表现为肾炎相关纤溶酶受体肾小球染色阳性:1例报告和文献复习
IF 1
CEN Case Reports Pub Date : 2024-12-15 DOI: 10.1007/s13730-024-00956-1
Haruka Takahashi, Yukihiro Wada, Takuya Yamazaki, Kazuhiro Takeuchi, Tetsuya Abe, Shokichi Naito, Togo Aoyama, Takashi Sano, Rika Moriya, Takashi Oda, Yasuo Takeuchi
{"title":"Parvovirus B19-related membranoproliferative glomerulonephritis presenting with positive glomerular staining for nephritis-associated plasmin receptor: a case report and review of the literature.","authors":"Haruka Takahashi, Yukihiro Wada, Takuya Yamazaki, Kazuhiro Takeuchi, Tetsuya Abe, Shokichi Naito, Togo Aoyama, Takashi Sano, Rika Moriya, Takashi Oda, Yasuo Takeuchi","doi":"10.1007/s13730-024-00956-1","DOIUrl":"https://doi.org/10.1007/s13730-024-00956-1","url":null,"abstract":"<p><p>Several cases of glomerulonephritis occurring after infection with human parvovirus B19 (PVB19) have been reported. However, the pathogenesis and clinicopathological features of PVB19-related glomerulonephritis remain elusive. We describe the case of a 34 year-old woman who showed nephrotic syndrome and microscopic hematuria 10 days after PVB19 infection. Blood pressure and renal function were within normal ranges. Laboratory tests showed positive results for anti-PVB19 immunoglobulin (Ig)M antibody and complement 3 (C3) hypocomplementemia. Antibody to streptolysin O (ASO) was slightly elevated, but bacterial cultures yielded no colonies. Light microscopy of renal biopsy was compatible with membranoproliferative glomerulonephritis (MPGN). Immunofluorescence microscopy showed intense staining for C3 and faint staining for IgG on the glomerular capillary wall and paramesangial area. Electron micrography showed subendothelial electron-dense deposits (EDDs), but hump-shaped subepithelial EDDs were not evident. PBV19-DNA was absent from renal tissue. Moreover, glomeruli showed positive staining for nephritis-associated plasmin receptor (NAPlr) and plasmin activity with similar distribution. Around 6 months after PVB19 infection, levels of anti-PVB19 IgM antibody spontaneously tuned negative with an apparent reduction of proteinuria and improvement of hypocomplementemia, although ASO level remained unchanged. This appears to represent the first description of positive glomerular staining for NAPlr in MPGN after PVB19 infection. Based on a review of 27 cases, including our own case, the MPGN lesions could be attributable to PVB19 infection. Clinicopathological features of this case were incompatible with post-streptococcal acute glomerulonephritis. We presume that a PBV19-derived glomerular pathogen that cross-reacts with anti-NAPlr antibody might be involved in the development of PVB19-related MPGN.</p>","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":" ","pages":""},"PeriodicalIF":1.0,"publicationDate":"2024-12-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142827555","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Tsukamurella conjunctivitidis peritonitis diagnosed by 16S rRNA gene sequencing in a patient undergoing peritoneal dialysis. 通过16S rRNA基因测序诊断腹膜透析患者的tsukamurela结膜炎腹膜炎。
IF 1
CEN Case Reports Pub Date : 2024-12-15 DOI: 10.1007/s13730-024-00958-z
Masato Habuka, Kosuke Matsunaga, Asa Ogawa, Takashi Yaguchi, Suguru Yamamoto, Yoshinari Tanabe
{"title":"Tsukamurella conjunctivitidis peritonitis diagnosed by 16S rRNA gene sequencing in a patient undergoing peritoneal dialysis.","authors":"Masato Habuka, Kosuke Matsunaga, Asa Ogawa, Takashi Yaguchi, Suguru Yamamoto, Yoshinari Tanabe","doi":"10.1007/s13730-024-00958-z","DOIUrl":"https://doi.org/10.1007/s13730-024-00958-z","url":null,"abstract":"<p><p>Peritoneal dialysis (PD)-associated peritonitis remains a serious and life-threatening complication in patients undergoing PD. Majority of peritonitis cases are caused by bacteria, with coagulase-negative Staphylococcus being the most common cause. Tsukamurella species are obligate aerobic gram-positive bacilli found in various environments; however, peritonitis caused by Tsukamurella species in association with PD is rare, with few reports of infections caused by T. conjunctivitidis, including PD-associated peritonitis. We describe a rare case of peritonitis caused by T. conjunctivitidis in a patient undergoing PD. A 62-year-old man undergoing PD was referred to our hospital for fever and abdominal pain lasting for 4 days and was diagnosed with PD-associated peritonitis based on abdominal pain, cloudy effluent, elevated white blood cell count in the peritoneal fluid, and presence of gram-positive rods in the effluent culture. Yellow-grayish, dry, membrane-like colonies were observed on a blood agar plate. The isolated strain was identified as T. conjunctivitidis through 16S rRNA gene sequencing. Because the PD-associated peritonitis was refractory to antibiotics, the PD catheter was removed, and the patient was switched to hemodialysis on day 21 of admission. Clinicians should consider peritonitis caused by T. conjunctivitidis as a differential diagnosis in cases where diagnosis using routine examination is challenging. Notably, 16S rRNA gene sequencing is a useful diagnostic tool for identifying T. conjunctivitidis. Further analyses of similar cases are required to understand the characteristics of such infections and establish adequate diagnostic methods and treatment regimens.</p>","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":" ","pages":""},"PeriodicalIF":1.0,"publicationDate":"2024-12-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142823984","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Diffuse large B-cell lymphoma with rapid kidney enlargement after induction of hemodialysis in a patient with IgG4-related disease. 一名患有 IgG4 相关疾病的患者在接受血液透析诱导后,出现伴有肾脏快速肿大的弥漫大 B 细胞淋巴瘤。
IF 1
CEN Case Reports Pub Date : 2024-12-14 DOI: 10.1007/s13730-024-00957-0
Shintaro Hara, Daisuke Morita, Ryoko Shibata, Yuki Yasui, Yoshiki Naito, Noriyoshi Fukushima, Seiya Kato, Noriko Uesugi, Yasuhiro Abe, Kosuke Masutani
{"title":"Diffuse large B-cell lymphoma with rapid kidney enlargement after induction of hemodialysis in a patient with IgG4-related disease.","authors":"Shintaro Hara, Daisuke Morita, Ryoko Shibata, Yuki Yasui, Yoshiki Naito, Noriyoshi Fukushima, Seiya Kato, Noriko Uesugi, Yasuhiro Abe, Kosuke Masutani","doi":"10.1007/s13730-024-00957-0","DOIUrl":"https://doi.org/10.1007/s13730-024-00957-0","url":null,"abstract":"<p><p>A 76-year-old Japanese man was incidentally diagnosed with a pancreatic head tumor on computed tomography after surgery for colon cancer. He underwent pancreatoduodenectomy and was diagnosed with IgG4-related autoimmune pancreatitis. Concurrent chronic kidney disease gradually progressed and chronic hemodialysis was introduced 2 years later. Six months after the introduction of hemodialysis, follow-up abdominal computed tomography revealed marked enlargement of bilateral kidneys compared with previous images. Blood tests revealed persistent high IgG and IgG4 levels, and IgG4-related kidney disease was suspected. Thus, percutaneous kidney biopsy was performed. No evidence of IgG4-related kidney disease was detected, and a diagnosis of diffuse large B-cell lymphoma was made. Six courses of combination chemotherapy consisting of rituximab, cyclophosphamide, doxorubicin, vincristine, and prednisone was effective, and the patient achieved and maintained complete remission for five years. This case highlights the need to consider the possible development of malignant lymphoma within several years after IgG4-related disease, especially in cases of autoimmune pancreatitis.</p>","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":" ","pages":""},"PeriodicalIF":1.0,"publicationDate":"2024-12-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142823980","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Fatal visceral disseminated varicella zoster virus infection during initial remission induction therapy in a patient with lupus nephritis: a case report and review of the literature. 狼疮肾炎患者初始缓解诱导治疗期间致死性内脏弥散性水痘带状疱疹病毒感染:1例报告及文献回顾。
IF 1
CEN Case Reports Pub Date : 2024-12-12 DOI: 10.1007/s13730-024-00950-7
Runa Takehara, Itaru Ebihara, Yoshifumi Honda, Norimasa Ooba, Hiromi Kurosawa, Chihiro Sato, Haruo Ohtani, Yutaka Tsutsumi, Masato Nose, Masaki Kobayashi
{"title":"Fatal visceral disseminated varicella zoster virus infection during initial remission induction therapy in a patient with lupus nephritis: a case report and review of the literature.","authors":"Runa Takehara, Itaru Ebihara, Yoshifumi Honda, Norimasa Ooba, Hiromi Kurosawa, Chihiro Sato, Haruo Ohtani, Yutaka Tsutsumi, Masato Nose, Masaki Kobayashi","doi":"10.1007/s13730-024-00950-7","DOIUrl":"https://doi.org/10.1007/s13730-024-00950-7","url":null,"abstract":"<p><p>Visceral disseminated varicella zoster virus (VZV) infection is a severe complication, characterized by a notably high mortality rate. Herein, we present a case of a 36-year-old-man involving visceral disseminated VZV infection that emerged during remission induction therapy involving high-dose prednisolone (PSL), mycophenolate mofetil (MMF), and hydroxychloroquine for lupus nephritis. Two months after starting the immunosuppressive therapy, he experienced a rapid onset of severe upper abdominal pain. The following day, clinical manifestations and laboratory abnormalities rapidly deteriorated. Hyperferritinemia and hypertriglyceridemia, indicative of hemophagocytic lymphohistiocytosis (HLH), emerged, along with escalating liver and renal impairment and newly appeared disseminated intravascular coagulation, and multiple organ failure is suggested. The patient developed widespread blistering predominantly on the trunk and face, patient's condition failed to ameliorate, ultimately culminating in his demise a few hours later. At autopsy, the cutaneous lesions with blisters revealed positive immunostaining with anti-VZV antibody, and similar findings were detected in multiple organs. HLH was confirmed in lymph nodes. It is crucial to emphasize the awareness of visceral disseminated VZV, particularly in cases patients are undergoing concurrent PSL therapy alongside MMF for SLE. The progression of this fatal condition usually begins with abdominal pain, followed by a skin rash a few days later. The present case is the first to show evidence of HLH occurring as a result of visceral disseminated VZV infection. This disease is extremely rare but extremely serious, therefore, VZV-DNA should be measured in cases where you suspect this disease for early diagnosis and treatment.</p>","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":" ","pages":""},"PeriodicalIF":1.0,"publicationDate":"2024-12-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142812006","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Fanconi syndrome following administration of oral supplements containing red yeast rice: several months follow-up of three cases. 服用含红曲米的口服补品后的范可尼综合征:三个病例的数月随访。
IF 1
CEN Case Reports Pub Date : 2024-12-11 DOI: 10.1007/s13730-024-00955-2
Reina Matsui-Hosoya, Koji Sato, Motohiro Yagasaki, Hitomi Hirose, Yusuke Fukao, Toshiki Kano, Hiroaki Io, Yusuke Suzuki
{"title":"Fanconi syndrome following administration of oral supplements containing red yeast rice: several months follow-up of three cases.","authors":"Reina Matsui-Hosoya, Koji Sato, Motohiro Yagasaki, Hitomi Hirose, Yusuke Fukao, Toshiki Kano, Hiroaki Io, Yusuke Suzuki","doi":"10.1007/s13730-024-00955-2","DOIUrl":"https://doi.org/10.1007/s13730-024-00955-2","url":null,"abstract":"<p><p>To date, the treatment strategy and long-term prognosis of acute kidney injury (AKI) after taking Benikoji CholesteHelp<sup>®</sup>, a red yeast rice supplement, remains unclear. We present three cases wherein renal dysfunction improved within a few months of supplement discontinuation, without immunosuppressive therapy. Case 1: A 59-year-old woman with a history of hypertension, treated with telmisartan (serum creatinine [sCr]: 0.65 mg/dL; estimated glomerular filtration rate [eGFR]: 71.3 mL/min/1.73 m<sup>2</sup>) and Benikoji CholesteHelp<sup>®</sup> for 7 months, developed Fanconi syndrome (FS) and severe renal impairment (sCr: 2.32 mg/dL; eGFR: 17.7 mL/min/1.73 m<sup>2</sup>). Renal biopsy and gallium-67 scintigraphy revealed no active drug-induced interstitial nephritis. Her condition improved significantly after supplement discontinuation. Her renal function gradually improved, with 3-month follow-up sCr and eGFR values of 0.96 mg/dL and 46.5 mL/min/1.73 m<sup>2</sup>, respectively; however, these were still worse than the pre-onset values. Case 2: A 48-year-old man had mild renal dysfunction (sCr: 1.12 mg/dL; eGFR: 56.76 mL/min/1.73 m<sup>2</sup>) after taking Benikoji CholesteHelp<sup>®</sup> for approximately 2 years; this was reversed upon supplement discontinuation. Case 3: A 47-year-old man with FS and mild renal dysfunction (sCr: 1.09 mg/dL; eGFR: 58.5 mL/min/1.73 m<sup>2</sup>) after taking Benikoji CholesteHelp<sup>®</sup> for approximately 4 months, showed notable improvement in FS after supplement discontinuation; however, the mild renal dysfunction persisted. The primary intervention in all cases was immediate supplement discontinuation, leading to rapid improvement in renal function, without need for immunosuppressive therapy. These findings increase our understanding of renal impairment caused by red yeast rice, with improvement after withdrawal, sometimes after several months.</p>","PeriodicalId":9697,"journal":{"name":"CEN Case Reports","volume":" ","pages":""},"PeriodicalIF":1.0,"publicationDate":"2024-12-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142812033","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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