scholarly journals A case of solitary plasmacytoma of bone showing co-expression of both immunoglobulin light chains

2021 ◽  
Vol 26 (1) ◽  
Author(s):  
Ryota Matsuoka ◽  
Noriaki Sakamoto ◽  
Takayasu Kato ◽  
Shigeru Chiba ◽  
Masayuki Noguchi

Abstract Background Solitary plasmacytoma of bone (SPB) is a rare plasma cell neoplasm. It arises in bone as a single locus in the absence of any plasma cell myeloma lesions. Plasma cell neoplasms intrinsically express only one immunoglobulin light chain (IgL)—kappa or lambda—and using this fact, kappa/lambda deviation is the decisive factor for diagnosis. Co-expression of both IgLs in a single tumor cell is extremely rare. Case presentation We report a case of SPB that arose in the vertebra of a 52-year-old Japanese woman. Histologically, the resected mass showed diffuse plasma cell proliferation. Dual IgL expression was detected by flow cytometry, immunohistochemistry, and in situ hybridization (ISH) targeting IgL mRNA. Conclusion We have presented an extremely rare case of SPB showing dual expression of kappa and lambda IgLs. This unusual case of plasma cell neoplasia might represent a possible exceptional example of failure of “IgL isotypic exclusion.”

2021 ◽  
Author(s):  
Ryota Matsuoka ◽  
Noriaki Sakamoto ◽  
Takayasu Kato ◽  
Shigeru Chiba ◽  
Masayuki Noguchi

Abstract Background: Solitary plasmacytoma of bone (SPB) is a rare plasma cell neoplasm. It arises in bone as a single locus in the absence of any plasma cell myeloma lesions. Plasma cell neoplasms intrinsically express only one immunoglobulin light chain (IgL) – kappa or lambda – and using this fact, kappa/lambda deviation is the decisive factor for diagnosis. Coexpression of both IgLs in a single tumor cell is extremely rare.Case presentation: We report a case of SPB that arose in the vertebra of a 52-year-old Japanese woman. Histologically, the resected mass showed diffuse plasma cell proliferation. Dual IgL expression was detected by flow cytometry, immunohistochemistry and in situ hybridization (ISH) targeting IgL mRNA.Conclusion: We have presented an extremely rare case of SPB showing dual expression of kappa and lambda IgLs. This unusual case of plasma cell neoplasia might represent a possible exceptional example of failure of “IgL isotypic exclusion”.


2020 ◽  
Vol 154 (Supplement_1) ◽  
pp. S98-S98
Author(s):  
C N Giraldo ◽  
D Myers ◽  
A Holmes ◽  
J Dodd ◽  
W Wendi

Abstract Introduction/Objective Cutaneous plasmacytosis (CP) is an uncommon condition typically affecting Asian males in the 3rd to 5th decades. It is thought to be a reactive process that classically presents with asymptomatic, red-brown, plaques and nodules on the face and neck. It has been associated with polyclonal hypergammaglobulinemia and systemic involvement. Histologically it is characterized by dense superficial and deep dermal infiltrates of mature plasma cells with polyclonal differentiation on in-situ hybridization (ISH). The differential diagnosis includes neoplastic plasma cell processes, characteristically with monoclonal plasma cell infiltrate, and mature B cell neoplasms with polyclonal plasma cell differentiation. Methods We report a case of a 69 year old Caucasian male who presented with asymptomatic, enlarging red-brown nodules on bilateral nasal ala. Histologic examination revealed dense, superficial plasma cell infiltrate, concerning for a plasma cell neoplasm. CD138 and Kappa/Lambda ISH demonstrated plasma cell polyclonality. Further workup ruled out infectious or systemic involvement and a plasma cell dyscrasia was ruled out by Hematology/Oncology. Results These findings supported the diagnosis of CP. Treatment with intralesional steroids showed initial improvement with regrowth of the nodules. To date, treatment with topical steroids and CO2 laser ablation are being considered. Conclusion CP is reported as type of pseudolymphoma, which is described as a reactive lymphoproliferation that histopathologically and/or clinically imitates cutaneous lymphoma. The pathogenesis is unknown, however, there are studies suggesting an association with increased interleukin-6, which is involved in the differentiation of B cells to mature plasma cells. The majority of patients with CP have a favorable prognosis. There has been variable success with both topical and intralesional treatment to include, cyclophosphamide, topical tacrolimus, prednisone, intralesional steroid therapy, topical psoralens combined with ultraviolet A exposure, and other chemotherapies. Familiarity with this rare entity is imperative to prevent misdiagnosis and overtreatment.


2021 ◽  
Vol 7 (1) ◽  
Author(s):  
Chiharu Kinoshita ◽  
Koichi Matsuda ◽  
Yumiko Kawai ◽  
Takayuki Hagiwara ◽  
Akane Okada

Abstract Background Peritoneal dialysis (PD)-associated peritonitis caused by Sphingomonas paucimobilis (S. paucimobilis) is very rare, and most of the characteristics of such cases are still unknown. Case presentation An 80-year-old Japanese woman on PD was diagnosed with PD-associated peritonitis and received ceftazidime and cefazolin. The number of cells in the peritoneal dialysate decreased quickly. However, because S. paucimobilis was detected, the antibiotic was changed to meropenem according to the susceptibility test results. She was treated with meropenem for two weeks and discharged. After 21 days, she was hospitalized for relapsing peritonitis. S. paucimobilis was detected again, and improvement after the administration of meropenem was poor, eventually resulting in catheter removal. Conclusions S. paucimobilis may be resistant to empirical antibiotics; furthermore, catheter removal may still be required, even with sensitive-antibiotic treatment.


2017 ◽  
Vol 2017 ◽  
pp. 1-4 ◽  
Author(s):  
Georgia Mitropoulou ◽  
Adamantia Zizi-Sermpetzoglou ◽  
Hippokrates Moschouris ◽  
Athanasios Kountourogiannis ◽  
Despoina Myoteri ◽  
...  

Introduction. Plasmacytoma is an uncommon plasma cell neoplasm and its localized form is solitary plasmacytoma of the bone and solitary extramedullary plasmacytoma. Solitary plasmacytoma of the mesentery is extremely rare, reported only in a handful of cases. Case Presentation. A 47-year-old man with nonspecific abdominal complains was found to have an ill-defined mass on his mesenteric root. Laparoscopic biopsy and stepwise histopathological examination revealed a mesenteric plasmacytoma, and extensive imaging and laboratory investigations led to the diagnosis of the solitary mesenteric plasmacytoma. The patient underwent definitive radiotherapy and remains under remission one year later. Discussion. Plasma cell dyscrasias include a variant of proliferative disease, characterized by clonal expansion of bone marrow plasma cells, producing a massive quantity of monoclonal immunoglobulin called paraprotein or M-protein. Solitary extramedullary plasmacytoma accounts for only 3–5% of all plasma cell neoplasms. Meticulous adherence to the established diagnostic criteria helps the clinician to set the correct, yet very unusual and unexpected diagnosis.


Author(s):  
Hari Ram ◽  
◽  
Sneha Gupta ◽  
Praveen Kumar Singh ◽  
Shivani Sharma ◽  
...  

Multiple myeloma (MM) is a malignant proliferation of plasma cells with multiple foci. Plasmacytoma is a solitary plasma cell neoplasm involving a single bone. The most commonly involved bone is vertebra. Jaw bones are rarely involved as a first bone as they have lesser hematopoietic marrow. A solitary plasmacytoma may progress to multiple myeloma within few months to year. We present a case of a swelling of mandible that on further investigations confirmed the diagnosis of multiple myeloma. We have discussed the course of treatment given and its prognosis. Keywords: multiple myeloma; plasmacytoma of jaw; bence jones Protein; abnormal plasma cells; CD138.


2013 ◽  
Vol 137 (5) ◽  
pp. 625-631 ◽  
Author(s):  
Gary Lu ◽  
Ramya Muddasani ◽  
Robert Z. Orlowski ◽  
Lynne V. Abruzzo ◽  
Muzaffar H. Qazilbash ◽  
...  

Context.—Methods for plasma cell enrichment of bone marrow (BM) specimens can increase the sensitivity of fluorescence in situ hybridization (FISH) for detecting cytogenomic abnormalities. There are no published reports using these methods to evaluate high-risk cytogenomic abnormalities in patients with plasma cell neoplasms (PCNs) after therapy. Objective.—To evaluate the utility of plasma cell enrichment combined with FISH for detection of high-risk cytogenomic abnormalities in patients with PCNs after therapy. Design.—Twenty-eight patients with PCNs, of whom 22 received treatment, were included in this study. Plasma cells were enriched in BM aspirates by using a magnetic cell-sorting procedure to select CD138+ cells. Probes were chosen to assess for del(17p13/TP53), del(13q14/RB1), 1q21/CKS1B gain, IgH/FGFR3, and IgH/MAF. Clinicopathologic data were collected during clinical follow-up after plasma cell enrichment. Results.—Plasma cells in nonenriched BM specimens ranged from 1% to 28% (median, 8%) compared with 28% to 96% (median, 73%) in enriched BM specimens (P < .001). In a subset of treated patients in clinical remission, FISH detected high-risk cytogenomic abnormalities only in plasma cell–enriched samples. This approach also detected abnormalities in cases of solitary plasmacytoma and monoclonal gammopathy of undetermined significance. Conclusions.—Plasma cell enrichment of BM specimens increases FISH sensitivity for detecting high-risk cytogenomic abnormalities, particularly in treated patients, and these results, in combination with clinical follow-up data, can be of value to improve risk stratification and patient management.


1984 ◽  
Vol 98 (9) ◽  
pp. 929-938 ◽  
Author(s):  
Thomas A. Mustone ◽  
Marvin P. Fried ◽  
Max L. Goodman ◽  
James H. Kelly ◽  
Marshall Strome

AbstractPlasma cell neoplasms have been classified as multiple myeloma, solitary plasmacytoma and extramedullary plasmacytoma. They are usually considered as osteolytic lesions of bone except for the rare occurrence of osteosclerotic lesions. This paper describes the first reported osteosclerotic plasmacytoma of the maxillary bone and orbital floor. The difficulties in establishing a diagnosis and the relationship to other plasma cell neoplasms are discussed.Osteosclerotic plasmacytomas are a rare variant of plasma cell tumors which usually produce osteolytic lesions rather than bony sclerosis. Sixty-eight patients with the osteosclerotic variant have appeared in the world literature, with an overall incidence of about 1 per cent in a large series of plasma cell neoplasms (Dreidger and Pruzanski, 1979). There have been only six previous cases of solitary osteosclerotic plasmacytomas reported (Morley and Schweiger, 1964; Roberts et al., 1974; Rodriguez et al. 1976; Rushton, 1965; Schneinker, 1938; Brigham Medical Review, 1961) involving spine, sternum, or rib. None have previously been reported in the head and neck area.Plasma cell tumors have been classified into multiple myeloma, solitary plasmacytomas of bone, and extramedullary plasmacytomas. Multiple myeloma is a disseminated plasma cell malignancy characterized by the production of homogeneous immunoglobulins (whole or fragments) which appear in the serum and urine. Plasma cell tumors can also occur as solitary plasmacytomas, usually in bone, but also in soft tissue. With time, most solitary plasmacytomas develop disseminated disease with all the characteristics of multiple myeloma (Wiltshaw, 1976). Extramedullary plasmacytomas arise in soft tissue rather than bone, and primarily occur in the head and neck region. Clinically, they remain localized and less frequently develop into disseminated myeloma.


2020 ◽  
Vol 14 (1) ◽  
Author(s):  
Daisuke Katsura ◽  
Suzuko Moritani ◽  
Shunichiro Tsuji ◽  
Kounosuke Suzuki ◽  
Kazutaka Yamada ◽  
...  

Abstract Background Uterine inversion may cause massive hemorrhage, resulting in maternal deterioration and death. Replacement of the inverted uterus must be performed as soon as possible. As time passes, the inverted uterus becomes atonic and necrotic, and a surgical approach may be required. Case presentation A 27-year-old Japanese woman was admitted to our hospital 4 hours postpartum with increased hemorrhage after the replacement of an inverted uterus. Recurrent inversion was diagnosed, and though the atonic uterus was replaced again by the Johnson maneuver, hemorrhage persisted. Balloon tamponade was not successful in stopping the hemorrhage, and uterine artery embolization was performed. Bleeding resumed the next day on removal of the balloon, and hysterectomy was performed. Massive hemorrhage, coagulopathy, and uterine necrosis caused uterine atony, and the reperfused blood flow on replacement of the ischemic uterus increased hemorrhage. Conclusions Cases of uterine inversion with coagulopathy lasting for more than 4 hours may require a surgical intervention, and uterine replacement may have to be delayed until the maternal hemodynamic condition is stabilized. Uterine replacement under laparotomy may be also be considered due to the risk of increased hemorrhage.


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