{"title":"A lymph node lesion of hyper IL-6 syndrome mimicking plasmacytoma, IgG4-related disease, and Castleman disease.","authors":"Kenta Matsui, Hiroshi Imai, Toshiaki Kobayashi, Minoru Mizutani, Midori Filiz Nishimura, Yasuharu Sato, Kennosuke Karube, Manato Okada, Miki Usui, Keiki Kawakami, Tetsuya Murata","doi":"10.3960/jslrt.24039","DOIUrl":"https://doi.org/10.3960/jslrt.24039","url":null,"abstract":"<p><p>A 72-year-old male patient presented fatigue, anemia, elevated total protein, IgG, IgG4, IL-6, and vascular endothelial growth factor (VEGF) levels. Initial diagnostics suspected multiple myeloma. A plane computed tomography (CT) scan showed pneumonia and the enlargement of generalized lymph nodes. A lymph node biopsy revealed a mix of immature and mature plasma cells, significant IgG4-positive cells, and IL-6-secreting plasma cells, mimicking plasmacytoma, IgG4-related disease, and Castleman disease, ultimately leading to a diagnosis of hyper IL-6 syndrome. Treatment with antibiotics improved the patient's condition without the need for additional therapies, including tocilizumab.</p>","PeriodicalId":45936,"journal":{"name":"Journal of Clinical and Experimental Hematopathology","volume":"64 4","pages":"307-312"},"PeriodicalIF":0.9,"publicationDate":"2024-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142899256","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}
{"title":"CD30- and CD56-positive atypical intravascular lymphocytes of the uterine cervix, mimicking intravascular lymphoma: A case report and review of the literature.","authors":"Daisuke Yamashita, Munemichi Otani, Hayato Maruoka, Takuya Aoki, Shigeo Hara","doi":"10.3960/jslrt.24058","DOIUrl":"https://doi.org/10.3960/jslrt.24058","url":null,"abstract":"<p><p>Intravascular accumulation of atypical large lymphoid cells is a rare condition that necessitates a differential diagnosis of intravascular lymphoma (IVL). Recently, a non-neoplastic condition known as benign atypical intravascular CD30+ T-cell proliferation (BAITP) has been identified. This condition is characterized by CD30+ and CD3+ or CD4+ atypical T-cells and is often associated with trauma and chronic inflammation. This case highlights atypical intravascular lymphoid cells of the uterine cervix that are positive for CD30 and CD56 and can mimic IVL.</p>","PeriodicalId":45936,"journal":{"name":"Journal of Clinical and Experimental Hematopathology","volume":"64 4","pages":"328-331"},"PeriodicalIF":0.9,"publicationDate":"2024-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142899257","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}
{"title":"Epstein-Barr virus-positive plasmacytoma in an immunocompetent female: A case report.","authors":"Hiyo Okuno, Motoha Miura, Naoki Oishi, Megumi Koshiishi-Yamada, Fumihiko Tanioka, Keita Kirito, Tetsuo Kondo","doi":"10.3960/jslrt.24038","DOIUrl":"10.3960/jslrt.24038","url":null,"abstract":"<p><p>Plasmacytoma is defined as a plasma cell neoplasm forming a solitary osseous or extramedullary tumor without evidence of myeloma or organ damage related to a plasma cell neoplasm. Epstein-Barr virus (EBV) is associated with various B-cell neoplasms, particularly in patients with immune dysregulation; however, plasmacytoma is typically negative for EBV. Here, a case of EBV-positive sternal plasmacytoma in an immunocompetent female is presented. A 76-year-old female with no immunodeficiency presented with a tumor on the anterior thoracic wall. Imaging analysis revealed a 6.3 cm-sized tumor at the manubrium, and a needle biopsy was performed. The tumor in the bone was composed of a diffuse proliferation of plasmacytes with eccentric nuclei and a perinuclear halo. By immunohistochemistry and in situ hybridization, tumor cells were CD20-, CD3-, CD138+, κ+, λ-, EBER+, and the Ki67-labeling index was approximately 20%. Subsequent studies identified IgG κ monoclonal protein in serum but no evidence of plasma cell neoplasm-related organ damage, such as hypercalcemia, anemia, or renal dysfunction. No plasma cell neoplasm was detected in the bone marrow in the morphological and flowcytometric studies. Accordingly, the diagnosis was EBV-positive plasmacytoma. The patient was treated with local radiation therapy and achieved complete remission. EBV-positive plasmacytoma is rare in immunocompetent patients and should be carefully distinguished from plasmablastic lymphoma, another EBV-positive neoplasm with a plasma cell phenotype and an aggressive clinical course. This case also raises an important question: \"when to perform EBER in situ hybridization in diagnosing plasma cell neoplasm?\", which prompts further large case-series studies.</p>","PeriodicalId":45936,"journal":{"name":"Journal of Clinical and Experimental Hematopathology","volume":"64 3","pages":"268-272"},"PeriodicalIF":0.9,"publicationDate":"2024-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11528257/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142356093","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}
{"title":"Anticancer immune reaction and lymph node sinus macrophages: a review from human and animal studies.","authors":"Yukio Fujiwara, Hiromu Yano, Cheng Pan, Takuya Shiota, Yoshihiro Komohara","doi":"10.3960/jslrt.24017","DOIUrl":"10.3960/jslrt.24017","url":null,"abstract":"<p><p>Lymph nodes are secondary lymphoid organs localized throughout the body that typically appear as bean-like nodules. Numerous antigen-presenting cells, including dendritic cells and macrophages, that mediate host defense responses against pathogens, such as bacteria and viruses, reside within lymph nodes. To react to cancer cell-derived antigens in a variety of cancers, antigen-presenting cells induce cytotoxic T lymphocytes (CTLs). In relation to anticancer immune responses, macrophages in the lymph node sinus have been of particular interest because a number of studies involving both human specimens and animal models have reported that lymph node macrophages expressing CD169 play a key role in activating anticancer CTLs. Recent studies have indicated that dysfunction of lymph node macrophages potentially contributes to immune suppression in elderly patients and immunological \"cold\" tumors. Therefore, in anticancer therapy, the regulation of lymph node macrophages is a potentially promising approach.</p>","PeriodicalId":45936,"journal":{"name":"Journal of Clinical and Experimental Hematopathology","volume":"64 2","pages":"71-78"},"PeriodicalIF":0.9,"publicationDate":"2024-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11303962/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141459901","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}
{"title":"Atypical lymphoplasmacytic and immunoblastic proliferation: A Systematic Review.","authors":"Midori Filiz Nishimura, Toshiaki Takahashi, Kensuke Takaoka, Sharina Macapagal, Chalothorn Wannaphut, Asami Nishikori, Hiroko Toda, Yoshito Nishimura, Yasuharu Sato","doi":"10.3960/jslrt.24007","DOIUrl":"10.3960/jslrt.24007","url":null,"abstract":"<p><p>Atypical lymphoplasmacytic and immunoblastic proliferation (ALPIBP) was first reported in 1984 as characteristic histological findings in lymph nodes associated with autoimmune diseases, but it has not been clearly defined to date. To summarize the histological characteristics and clinical diagnoses associated with ALPIBP, we searched MEDLINE and EMBASE for all peer-reviewed articles using keywords including \"atypical lymphoplasmacytic and immunoblastic lymphadenopathy\" from their inception to December 27, 2023. We also summarized the courses of three cases with a pathological diagnosis of ALPIBP. Nine articles with 52 cases were included. Among the total of 55 cases, including the three from our institution, the median age of the cases was 63.5 years with a female predominance (69.5%). Lymphadenopathy was generalized in 65.6% and regional in 34.4% of cases. RA (24.4%), SLE (24.4%), and autoimmune hemolytic anemia (20.0%), were common clinical diagnoses. A combination of cytotoxic chemotherapy was used in 15.6% of cases due to the suspicion of malignancy. Nodal T-follicular helper cell lymphoma, angioimmunoblastic type, methotrexate-associated lymphoproliferative disorders, and IgG4-related diseases were listed as important diseases that need to be pathologically differentiated from ALPIBP. This review summarizes the current understanding of the characteristics of ALPIBP. Given that underrecognition of ALPIBP could lead to overdiagnosis of hematological malignancy and unnecessary treatment, increased awareness of the condition in pathologists and clinicians is crucial.</p>","PeriodicalId":45936,"journal":{"name":"Journal of Clinical and Experimental Hematopathology","volume":"64 2","pages":"97-106"},"PeriodicalIF":0.9,"publicationDate":"2024-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11303958/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141459902","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}
{"title":"Complex karyotype determined using conventional cytogenetic analysis is a poor prognostic factor in patients with multiple myeloma.","authors":"Hideki Uryu, Yuko Mishima, Yuko Ishihara, Yuko Shirouchi, Nobuhiko Yamauchi, Mitsuhito Hirano, Kei Hirano, Yukako Teramoto, Kikuaki Yoshida, Dai Maruyama","doi":"10.3960/jslrt.23047","DOIUrl":"10.3960/jslrt.23047","url":null,"abstract":"<p><p>High-risk cytogenetic abnormalities (HRCAs) influence the prognosis of multiple myeloma (MM). However, additional cytogenetic aberrations can lead to poor outcomes. This study aimed to clarify whether HRCAs and additional chromosomal abnormalities affect MM prognosis. Patients with newly diagnosed MM who were treated with novel agents were retrospectively evaluated. The primary objective was to assess the difference in progression-free survival (PFS) and overall survival (OS) between patients with/without HRCAs and between patients with/without complex karyotype (CK). The secondary objectives were to identify factors affecting PFS/OS and factors related to CK. HRCAs were defined as del(17p), t(4;14), t(14;16), and gain/amplification(1q) assessed using fluorescence in situ hybridization. CK was defined as ≥3 chromosomal abnormalities on G-banding. Among 110 patients, 40 had HRCAs and 15 had CK. In this study, survival durations between patients with/without HRCAs were similar, while the CK group had significantly poorer PFS/OS than the no-CK group (median PFS: 9 vs. 24 months and median OS: 29 vs. 97 months, respectively), and a poor prognostic impact of CK was maintained in patients with HRCAs. In multivariate analysis, CK was correlated with poor PFS/OS (hazard ratio [HR]: 2.39, 95% confidence interval [95% CI]: 1.22-4.66 and HR: 2.66, 95% CI: 1.10-6.45, respectively). Bone marrow plasma cell (BMPC) ≥60% (odds ratio [OR] = 6.40, 95% CI: 1.50-27.2) and Revised International Staging System III (OR = 7.53, 95% CI: 2.09-27.1) were associated with CK. Our study suggests that CK may contribute to the poor prognosis of MM. Aggressive disease status including high BMPC proliferation could be relevant to CK.</p>","PeriodicalId":45936,"journal":{"name":"Journal of Clinical and Experimental Hematopathology","volume":"64 1","pages":"10-20"},"PeriodicalIF":1.5,"publicationDate":"2024-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11079984/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140307301","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}
{"title":"Histiocytic neoplasms: a brief review and differential diagnosis.","authors":"Rin Yamada, Yoshihiro Komohara","doi":"10.3960/jslrt.24031","DOIUrl":"10.3960/jslrt.24031","url":null,"abstract":"<p><p>Histiocytic neoplasms (HNs) include juvenile xanthogranuloma, Erdheim-Chester disease, Rosai-Dorfman disease, ALK-positive histiocytosis, and histiocytic sarcoma in the 5th edition of the World Health Organization Classification of Haematolymphoid Tumours. These entities are clinicopathologically distinctive, and typical histological findings have been established. However, the common feature of a proliferation of histiocytic cells often leads to morphological overlap among HNs, and also necessitates a differential diagnosis from several non-HNs or non-neoplastic conditions. In this review, we provide a brief summary of the clinical findings, molecular features, histopathologies, and immunophenotypes of HNs, as well as to discuss their differential diagnosis.</p>","PeriodicalId":45936,"journal":{"name":"Journal of Clinical and Experimental Hematopathology","volume":"64 3","pages":"156-165"},"PeriodicalIF":0.9,"publicationDate":"2024-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11528248/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142356094","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}
{"title":"TTMV::RARA-positive acute promyelocytic leukemia with marrow necrosis and central nervous system involvement at disease recurrence.","authors":"Zhao Wang, Jiaqi Chen, Juanxia Meng, Mingfeng Zhao, Hongxing Liu, Xia Xiao","doi":"10.3960/jslrt.24015","DOIUrl":"10.3960/jslrt.24015","url":null,"abstract":"<p><p>Since the identification of the TTMV::RARA fusion in pediatric cases resembling acute promyelocytic leukemia (APL) by Astolfi et al. in 2021, several similar cases have been reported worldwide. In this report, we present a case of relapsed APL in an adolescent patient, who exhibited the TTMV::RARA fusion gene. This patient exhibited extensive central nervous system involvement and experienced bone marrow necrosis during disease recurrence. Despite achieving complete remission after re-induction chemotherapy, the patient experienced a rapid second relapse, highlighting the extremely aggressive nature of this subtype. These clinical manifestations contribute to the growing recognition of this rare disease.</p>","PeriodicalId":45936,"journal":{"name":"Journal of Clinical and Experimental Hematopathology","volume":"64 3","pages":"237-241"},"PeriodicalIF":0.9,"publicationDate":"2024-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11528256/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142356097","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}
{"title":"Salivary gland swelling as a characteristic manifestation of local cytokine release syndrome after anti-CD19 chimeric antigen receptor T cell therapy: A case series.","authors":"Saki Kawase, Masatoshi Sakurai, Kyoko Masuda, Yusuke Kubota, Takahide Shindo, Ami Inokuchi, Hiroyoshi Hayashi, Misa Nakayasu, Yuka Shiozawa, Tomohiro Hirai, Takahiro Inoue, Takayuki Fujii, Haryoon Kim, Yuya Koda, Jun Kato, Keisuke Kataoka","doi":"10.3960/jslrt.24035","DOIUrl":"10.3960/jslrt.24035","url":null,"abstract":"<p><p>Cytokine release syndrome (CRS) is the most common adverse event of chimeric antigen receptor T (CAR-T) cell therapy and is usually characterized by systemic symptoms such as fever, hypotension, and hypoxia. However, there have been several recent reports of local CRS characterized by cervical swelling. This localized syndrome can cause life-threatening laryngeal edema and requires early diagnostic treatment. Here we report 3 cases of local CRS where bilateral salivary gland swelling emerged following anti-CD19 CAR-T cell therapy for relapsed or refractory diffuse large B-cell lymphoma. Following tocilizumab treatment for systemic CRS, all patients exhibited cervical swelling. Physical examinations revealed significant swelling of the bilateral submandibular glands, and computed tomography scans showed substantial enlargement of the bilateral parotid and submandibular glands. Immediate treatment with dexamethasone effectively managed the potentially life-threatening laryngeal or pharyngeal edema, thereby preventing severe airway obstruction. This study has demonstrated, for the first time to our knowledge, that salivary gland enlargement is a common finding in local CRS. This observation suggests that physicians should continue to closely monitor the risk of developing cervical edema leading to life-threatening airway obstruction after systemic CRS, even in patients treated with tocilizumab. If salivary gland swelling is observed, it would be better to consider prompt evaluation and dexamethasone administration.</p>","PeriodicalId":45936,"journal":{"name":"Journal of Clinical and Experimental Hematopathology","volume":"64 3","pages":"261-267"},"PeriodicalIF":0.9,"publicationDate":"2024-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11528259/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142356096","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}
{"title":"Real-world outcomes of venetoclax and rituximab for chronic lymphocytic leukemia/small lymphocytic lymphoma: A retrospective analysis of nine Japanese cases.","authors":"Masuho Saburi, Takumi Nishikawa, Yasuhiko Miyazaki, Kazuhiro Kohno, Masanori Sakata, Kazuki Okuhiro, Toshiyuki Nakayama, Eiichi Ohtsuka, Masao Ogata","doi":"10.3960/jslrt.24014","DOIUrl":"10.3960/jslrt.24014","url":null,"abstract":"","PeriodicalId":45936,"journal":{"name":"Journal of Clinical and Experimental Hematopathology","volume":"64 2","pages":"152-155"},"PeriodicalIF":0.9,"publicationDate":"2024-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11303959/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141459805","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}