Here we present a rare case of an adolescent with hypertension, concurrently diagnosed with fibromuscular dysplasia of the renal artery and Graves’ disease. Although fibromuscular dysplasia and Graves’ disease have distinct pathogenic mechanisms, it is possible to infer the potential correlation between the two from the perspective of vascular involvement. It is believed that transforming growth factor-β, as the shared element of both diseases, may contribute to their development and progression. The overactivation of the sympathetic nervous system in Graves’ disease may induce hyperplasia of vascular smooth muscle cells, similar to that observed in fibromuscular dysplasia. In Graves’ disease, the excessive synthesis and secretion of angiotensin II due to the overactivation of the renin-angiotensin system, along with the up-regulation of angiotensin II receptors, may also induce pathological changes in the vasculature throughout the body. In this regard, exploring the correlation between fibromuscular dysplasia and Graves’ disease is of significant clinical importance.
我们在这里介绍一例罕见的青少年高血压患者，他同时被诊断出患有肾动脉纤维肌性发育不良和巴塞杜氏病。虽然纤维肌性发育不良和巴塞杜氏病的致病机制不同，但从血管受累的角度可以推断出两者之间潜在的相关性。据认为，转化生长因子-β作为这两种疾病的共同因素，可能有助于它们的发展和恶化。巴塞杜氏病中交感神经系统的过度活跃可能会诱发血管平滑肌细胞的增生，这与纤维肌肉发育不良症中观察到的情况类似。在巴塞杜氏病中，由于肾素-血管紧张素系统的过度激活，血管紧张素 II 合成和分泌过多，再加上血管紧张素 II 受体的上调，也可能诱发全身血管的病理变化。因此，探讨纤维肌发育不良与巴塞杜氏病之间的相关性具有重要的临床意义。
{"title":"The Coexistence of Fibromuscular Dysplasia of the Renal Artery and Graves’ Disease in an Adolescent: A Case Report","authors":"Jinsol Jeong, Haesook Kim, Dongwon Lee","doi":"10.15746/sms.23.013","DOIUrl":"https://doi.org/10.15746/sms.23.013","url":null,"abstract":"Here we present a rare case of an adolescent with hypertension, concurrently diagnosed with fibromuscular dysplasia of the renal artery and Graves’ disease. Although fibromuscular dysplasia and Graves’ disease have distinct pathogenic mechanisms, it is possible to infer the potential correlation between the two from the perspective of vascular involvement. It is believed that transforming growth factor-β, as the shared element of both diseases, may contribute to their development and progression. The overactivation of the sympathetic nervous system in Graves’ disease may induce hyperplasia of vascular smooth muscle cells, similar to that observed in fibromuscular dysplasia. In Graves’ disease, the excessive synthesis and secretion of angiotensin II due to the overactivation of the renin-angiotensin system, along with the up-regulation of angiotensin II receptors, may also induce pathological changes in the vasculature throughout the body. In this regard, exploring the correlation between fibromuscular dysplasia and Graves’ disease is of significant clinical importance.","PeriodicalId":22016,"journal":{"name":"Soonchunhyang Medical Science","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-12-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139138578","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}
Kawasaki disease (KD) is an acute systemic vasculitis that involves coronary arteries. Systematized epidermal nevus is a skin disease characterized by extensive verrucous and hyperchromic plaques. A male patient, previously diagnosed with systematized epidermal nevus, was referred for high fever. Eye injection, red lip, strawberry tongue, and enlarged neck node were accompanied. Intravenous immunoglobulin (IVGG) and high-dose aspirin were given for the treatment of KD. An echocardiogram revealed a small aneurysm in the right coronary artery (RCA). Second IVGG was administered because of refractoriness. The size of the aneurysm was increased to medium in RCA and another medium-sized aneurysm was found in the left anterior descending artery (LAD). Clopidogrel was added. After 3 days of defervescence, he was discharged. Warfarin was added for a giant aneurysm in LAD, aggravated 8 weeks later after the presentation. 11 months later, giant aneurysm was changed to medium sized aneurysm. Patient is at following with dual antiplatelet therapy without warfarin.
{"title":"Kawasaki Disease Complicated with Giant Coronary Aneurysm in Case of Systematized Epidermal Nevus","authors":"G. Yoo","doi":"10.15746/sms.23.020","DOIUrl":"https://doi.org/10.15746/sms.23.020","url":null,"abstract":"Kawasaki disease (KD) is an acute systemic vasculitis that involves coronary arteries. Systematized epidermal nevus is a skin disease characterized by extensive verrucous and hyperchromic plaques. A male patient, previously diagnosed with systematized epidermal nevus, was referred for high fever. Eye injection, red lip, strawberry tongue, and enlarged neck node were accompanied. Intravenous immunoglobulin (IVGG) and high-dose aspirin were given for the treatment of KD. An echocardiogram revealed a small aneurysm in the right coronary artery (RCA). Second IVGG was administered because of refractoriness. The size of the aneurysm was increased to medium in RCA and another medium-sized aneurysm was found in the left anterior descending artery (LAD). Clopidogrel was added. After 3 days of defervescence, he was discharged. Warfarin was added for a giant aneurysm in LAD, aggravated 8 weeks later after the presentation. 11 months later, giant aneurysm was changed to medium sized aneurysm. Patient is at following with dual antiplatelet therapy without warfarin.","PeriodicalId":22016,"journal":{"name":"Soonchunhyang Medical Science","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-12-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139138839","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}
D. Moon, Byung-Chan Kim, Young-Ju Kim, Kwang-Soo Lee
Spontaneous intracranial hypotension (SIH) is a condition caused by spontaneous cerebrospinal fluid (CSF) leakage, resulting in orthostatic headache as the main symptom, but other symptoms such as memory loss, nausea, and tinnitus may also be present. Various imaging techniques are used to diagnose SIH, with magnetic resonance imaging myelography being an important tool for detecting the leakage site of CSF. Conservative treatments including hydration, bed rest, and intravenous caffeine have been tried with limited success. Epidural blood patch (EBP) is the most commonly performed treatment for SIH after the failure of conservative management. There are different opinions about the EBP procedure (e.g., blind EBP vs. targeted EBP). This report describes the treatment of SIH with targeted EBP according to imaging diagnosis after the failure of initial blind lumbar EBP.
{"title":"Spontaneous Intracranial Hypotension Treated with Epidural Blood Patch: A Case Report","authors":"D. Moon, Byung-Chan Kim, Young-Ju Kim, Kwang-Soo Lee","doi":"10.15746/sms.23.005","DOIUrl":"https://doi.org/10.15746/sms.23.005","url":null,"abstract":"Spontaneous intracranial hypotension (SIH) is a condition caused by spontaneous cerebrospinal fluid (CSF) leakage, resulting in orthostatic headache as the main symptom, but other symptoms such as memory loss, nausea, and tinnitus may also be present. Various imaging techniques are used to diagnose SIH, with magnetic resonance imaging myelography being an important tool for detecting the leakage site of CSF. Conservative treatments including hydration, bed rest, and intravenous caffeine have been tried with limited success. Epidural blood patch (EBP) is the most commonly performed treatment for SIH after the failure of conservative management. There are different opinions about the EBP procedure (e.g., blind EBP vs. targeted EBP). This report describes the treatment of SIH with targeted EBP according to imaging diagnosis after the failure of initial blind lumbar EBP.","PeriodicalId":22016,"journal":{"name":"Soonchunhyang Medical Science","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-06-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"74435327","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}
To overcome the global pandemic of coronavirus disease 2019 (COVID-19), COVID-19 vaccination has been developed and distributed. Many people have received the vaccination worldwide. However, there are some vaccinated individuals who complain of side effects due to COVID-19 vaccination. We report the case of a patient who developed adult-onset Still’s disease (AOSD) after receiving the messenger RNA COVID-19 vaccine. A 21-year-old male patient without a previous medical history developed a fever on the day of the first dose of the vaccine. He had persistent fever, arthralgia of the knee and wrist, hyperferritinemia, transient skin rash, and negative test results for rheumatoid factor or antinuclear antibody. Positron emission tomography-computed tomography scan showed lymphadenopathies with reactive patterns and no malignancy. His symptoms and laboratory abnormalities gradually improved with glucocorticoid, cyclosporine, methotrexate, and tocilizumab treatment. Although its causality is still not confirmed, AOSD should be considered in a case that meets the diagnostic criteria after COVID-19 vaccination.
{"title":"Adult-Onset Still’s Disease after COVID-19 Vaccination: A Case Report and Review","authors":"J. Park, S. Lee","doi":"10.15746/sms.23.002","DOIUrl":"https://doi.org/10.15746/sms.23.002","url":null,"abstract":"To overcome the global pandemic of coronavirus disease 2019 (COVID-19), COVID-19 vaccination has been developed and distributed. Many people have received the vaccination worldwide. However, there are some vaccinated individuals who complain of side effects due to COVID-19 vaccination. We report the case of a patient who developed adult-onset Still’s disease (AOSD) after receiving the messenger RNA COVID-19 vaccine. A 21-year-old male patient without a previous medical history developed a fever on the day of the first dose of the vaccine. He had persistent fever, arthralgia of the knee and wrist, hyperferritinemia, transient skin rash, and negative test results for rheumatoid factor or antinuclear antibody. Positron emission tomography-computed tomography scan showed lymphadenopathies with reactive patterns and no malignancy. His symptoms and laboratory abnormalities gradually improved with glucocorticoid, cyclosporine, methotrexate, and tocilizumab treatment. Although its causality is still not confirmed, AOSD should be considered in a case that meets the diagnostic criteria after COVID-19 vaccination.","PeriodicalId":22016,"journal":{"name":"Soonchunhyang Medical Science","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-06-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"86181282","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}
Lymphangioleiomyomatosis is a rare disease characterized by the proliferation of atypical smooth muscle cells. Recurrent pneumothorax is a frequent complication of lymphangioleiomyomatosis. We present the case of a 41-year-old woman who presented with recurrent pneumothorax. Video-assisted thoracic surgery was performed. We did not suspect the possibility of lymphangioleiomyomatosis because of the presence of very few cysts on chest computed tomography.
{"title":"Recurrent Pneumothorax Caused by an Unexpected Lymphangioleiomyomatosis: A Case Report","authors":"Y. Seok","doi":"10.15746/sms.23.001","DOIUrl":"https://doi.org/10.15746/sms.23.001","url":null,"abstract":"Lymphangioleiomyomatosis is a rare disease characterized by the proliferation of atypical smooth muscle cells. Recurrent pneumothorax is a frequent complication of lymphangioleiomyomatosis. We present the case of a 41-year-old woman who presented with recurrent pneumothorax. Video-assisted thoracic surgery was performed. We did not suspect the possibility of lymphangioleiomyomatosis because of the presence of very few cysts on chest computed tomography.","PeriodicalId":22016,"journal":{"name":"Soonchunhyang Medical Science","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-06-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"83472332","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}
Kyeoung-Soo Park, Kye-Weol Kim, Y. Kim, Yoon Mee Jeen, Dongho Kim, S. Jin, I. Choi
Nodular histiocytic/mesothelial hyperplasia (NHMH) is a very rare condition. It is generally thought to be associated with repeated mechanical or chemical stimulation. This study reported NHMH with papillary growth pattern in the ovary following high-intensity focused ultrasound (HIFU) treatment for uterine leiomyoma and adenomyosis. A 48-year-old female, who had a history of undergoing HIFU treatment 7 times for adenomyosis and leiomyoma of the uterus 6 months ago, was referred to the hospital. After the hysterectomy and right salpingo-oophorectomy, the patient was confirmedly diagnosed with nodular histiocytic and mesothelial hyperplasia with the papillary growth pattern of the right ovary. This benign reactive inflammatory lesion of the mesothelium mimicking malignancy must be kept in mind to avoid unnecessary treatment.
{"title":"Papillary Histiocytic and Mesothelial Hyperplasia of the Ovary Related to a Huge Uterus: A Case Report","authors":"Kyeoung-Soo Park, Kye-Weol Kim, Y. Kim, Yoon Mee Jeen, Dongho Kim, S. Jin, I. Choi","doi":"10.15746/sms.23.003","DOIUrl":"https://doi.org/10.15746/sms.23.003","url":null,"abstract":"Nodular histiocytic/mesothelial hyperplasia (NHMH) is a very rare condition. It is generally thought to be associated with repeated mechanical or chemical stimulation. This study reported NHMH with papillary growth pattern in the ovary following high-intensity focused ultrasound (HIFU) treatment for uterine leiomyoma and adenomyosis. A 48-year-old female, who had a history of undergoing HIFU treatment 7 times for adenomyosis and leiomyoma of the uterus 6 months ago, was referred to the hospital. After the hysterectomy and right salpingo-oophorectomy, the patient was confirmedly diagnosed with nodular histiocytic and mesothelial hyperplasia with the papillary growth pattern of the right ovary. This benign reactive inflammatory lesion of the mesothelium mimicking malignancy must be kept in mind to avoid unnecessary treatment.","PeriodicalId":22016,"journal":{"name":"Soonchunhyang Medical Science","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-06-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"74785018","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}
Patients with type II Alexander disease have white matter dysplasia, which may cause various symptoms due to nerve conduction impairment. When providing anesthesia to a patient with Alexander disease, careful and patient-specific individualized risk evaluation must be preceded by planning the method of anesthesia and anesthetic drugs. This is the first case report of a patient with type II Alexander disease who underwent general anesthesia. We performed general anesthesia on a 45-year-old male with type II Alexander disease for laparoscopic cholecystectomy, using remimazolam, remifentanil, and rocuronium. Despite the use of reversal agents including flumazenil, naloxone, and sugammadex, the patient manifested a delay in emergence but successfully recovered from anesthesia without postoperative complications. In consideration of the possibility that leukodystrophy may have altered responses to anesthetics, the action of an anesthetic agent may be prolonged or delayed in patients with type II Alexander disease. We recommend using drugs with a short duration of action and which can be reversed immediately by a reversal agent.
{"title":"Anesthetic Management of a Patient with Type II Alexander Disease: A Case Report","authors":"Y. Yoon, J. Yeom","doi":"10.15746/sms.23.004","DOIUrl":"https://doi.org/10.15746/sms.23.004","url":null,"abstract":"Patients with type II Alexander disease have white matter dysplasia, which may cause various symptoms due to nerve conduction impairment. When providing anesthesia to a patient with Alexander disease, careful and patient-specific individualized risk evaluation must be preceded by planning the method of anesthesia and anesthetic drugs. This is the first case report of a patient with type II Alexander disease who underwent general anesthesia. We performed general anesthesia on a 45-year-old male with type II Alexander disease for laparoscopic cholecystectomy, using remimazolam, remifentanil, and rocuronium. Despite the use of reversal agents including flumazenil, naloxone, and sugammadex, the patient manifested a delay in emergence but successfully recovered from anesthesia without postoperative complications. In consideration of the possibility that leukodystrophy may have altered responses to anesthetics, the action of an anesthetic agent may be prolonged or delayed in patients with type II Alexander disease. We recommend using drugs with a short duration of action and which can be reversed immediately by a reversal agent.","PeriodicalId":22016,"journal":{"name":"Soonchunhyang Medical Science","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-06-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"72563311","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}
Tatton-Brown-Rahman syndrome (TBRS) is a relatively new congenital anomaly syndrome manifesting overgrowth and a broad spectrum of intellectual disability. It is caused by pathogenic variants in the DNA methyltransferase 3 alpha (DNMT3A) gene, mainly de novo inheritance. Overgrowth, mild-to-severe intellectual disability, and other clinical features of TBRS may affect the quality of life of patients and their family members. Thus, early diagnosis by genetic testing and management of these symptoms is critical. We report a case of a 17-year-old male patient with hemihypertrophy who suffered back pain since school age, diagnosed with TBRS-identified DNMT3A gene mutation.
{"title":"Tatton-Brown-Rahman Syndrome: A Report of a Case with a Novel DNMT3A Mutation Presented Hemihypertrophy","authors":"Mi Le Kim, Jeongho Lee","doi":"10.15746/sms.23.007","DOIUrl":"https://doi.org/10.15746/sms.23.007","url":null,"abstract":"Tatton-Brown-Rahman syndrome (TBRS) is a relatively new congenital anomaly syndrome manifesting overgrowth and a broad spectrum of intellectual disability. It is caused by pathogenic variants in the DNA methyltransferase 3 alpha (DNMT3A) gene, mainly de novo inheritance. Overgrowth, mild-to-severe intellectual disability, and other clinical features of TBRS may affect the quality of life of patients and their family members. Thus, early diagnosis by genetic testing and management of these symptoms is critical. We report a case of a 17-year-old male patient with hemihypertrophy who suffered back pain since school age, diagnosed with TBRS-identified DNMT3A gene mutation.","PeriodicalId":22016,"journal":{"name":"Soonchunhyang Medical Science","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-06-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"86527989","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}
Imatinib mesylate is an effective, small-molecule, selective tyrosine kinase inhibitor, which inhibits BCR-ABL in patients with chronic myeloid leukemia, c-kit in patients with gastrointestinal tumor (GIST), and platelet-derived growth factors in hypereosinophilic syndrome. Above all, bone marrow necrosis is a rare complication of imatinib usage. If newly developed increased opacity bone lesions are seen in patients with metastatic GIST, it is usual to consider those lesions as bone metastasis. Also, it is true that making a differential diagnosis between bone marrow change (bone marrow necrosis) and disease progression (bone metastasis) is both clinically and radiologically difficult. Because it may alter treatment options according to early distinguish bone metastasis and imatinib related-bone necrosis, a bone biopsy is recommended. Here, we report a case that a biopsy confirmed bone marrow necrosis due to imatinib, even though investigations such as magnetic resonance imaging, and whole-body bone scan indicated more favor to the metastatic lesion.
{"title":"Bone Biopsy Confirmed Imatinib-Induced Bone Marrow Necrosis in Patient with Gastrointestinal Tumor: A Case Report","authors":"H. Kim","doi":"10.15746/sms.23.006","DOIUrl":"https://doi.org/10.15746/sms.23.006","url":null,"abstract":"Imatinib mesylate is an effective, small-molecule, selective tyrosine kinase inhibitor, which inhibits BCR-ABL in patients with chronic myeloid leukemia, c-kit in patients with gastrointestinal tumor (GIST), and platelet-derived growth factors in hypereosinophilic syndrome. Above all, bone marrow necrosis is a rare complication of imatinib usage. If newly developed increased opacity bone lesions are seen in patients with metastatic GIST, it is usual to consider those lesions as bone metastasis. Also, it is true that making a differential diagnosis between bone marrow change (bone marrow necrosis) and disease progression (bone metastasis) is both clinically and radiologically difficult. Because it may alter treatment options according to early distinguish bone metastasis and imatinib related-bone necrosis, a bone biopsy is recommended. Here, we report a case that a biopsy confirmed bone marrow necrosis due to imatinib, even though investigations such as magnetic resonance imaging, and whole-body bone scan indicated more favor to the metastatic lesion.","PeriodicalId":22016,"journal":{"name":"Soonchunhyang Medical Science","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-06-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"74857764","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}
Jun Young Kim, S. Yoon, M. Lee, N. Lee, Jongchan Won, I. Choi, K. Kim, Kyoungha Kim
Castleman’s disease is a rare non-neoplastic lymphoproliferative disorder of unknown origin. It is classified into unicentric or multicentric based on its anatomical distribution. Multicentric Castleman’s disease can be subdivided according to the presence of human herpesvirus-8 (HHV-8) infection. Castleman’s disease has a rare incidence, and HHV-8-positive multicentric Castleman’s disease is even rarer. There are several types of natural course for this disease, and the rapidly progressing type can lead to death within a few weeks, emphasizing the need for prompt diagnosis and treatment. We report a recent case from Korea, presenting with multiple lymphadenopathies, confirmed as HHV-8-positive multicentric Castleman’s disease through biopsy, and achieving complete response with rituximab monotherapy.
{"title":"Human Herpesvirus-8 Positive Multicentric Castleman’s Disease with Complete Response after Rituximab Monotherapy: A Case Report","authors":"Jun Young Kim, S. Yoon, M. Lee, N. Lee, Jongchan Won, I. Choi, K. Kim, Kyoungha Kim","doi":"10.15746/sms.23.008","DOIUrl":"https://doi.org/10.15746/sms.23.008","url":null,"abstract":"Castleman’s disease is a rare non-neoplastic lymphoproliferative disorder of unknown origin. It is classified into unicentric or multicentric based on its anatomical distribution. Multicentric Castleman’s disease can be subdivided according to the presence of human herpesvirus-8 (HHV-8) infection. Castleman’s disease has a rare incidence, and HHV-8-positive multicentric Castleman’s disease is even rarer. There are several types of natural course for this disease, and the rapidly progressing type can lead to death within a few weeks, emphasizing the need for prompt diagnosis and treatment. We report a recent case from Korea, presenting with multiple lymphadenopathies, confirmed as HHV-8-positive multicentric Castleman’s disease through biopsy, and achieving complete response with rituximab monotherapy.","PeriodicalId":22016,"journal":{"name":"Soonchunhyang Medical Science","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2023-06-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"84642837","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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