scholarly journals Multicentric Hyaline-Vascular Castleman Disease: A Case Report

2015 ◽  
Vol 16 (1) ◽  
pp. 48-50
Author(s):  
Aparna Das ◽  
Sanjana Tarannum ◽  
Tahera Kona ◽  
Santanu Kumar Saha ◽  
MA Kahhar

Castleman disease is a rare lymphoproliferative disorder. Contrary to its closest differential of lymphoma, the disease tends to run a benign course. However the multicentric variety, which is usually of the plasma cell type, has a more sinister prognosis. It is commonly associated with HIV infection. We report a case of a 65 year old man presenting with recurrent episodes of swelling of the whole body and diarrhoea for 3 years. Physical examination revealed generalized lymphadenopathy with hepato-splenomegaly and ascites. Lymph node biopsy revealed histopathological changes consistent with hyaline-vascular type of Castleman disease (multicentric). He was HIV negative. Patient was treated with high dose corticosteroids and discharged with follow up advice. DOI: http://dx.doi.org/10.3329/jom.v16i1.22402 J MEDICINE 2015; 16 : 48-50

2018 ◽  
Vol 4 (4) ◽  
Author(s):  
Nazia Rashid ◽  
Aamna Hassan ◽  
Noreen Akhter ◽  
Abdul Hameed

A 27-year-old male underwent total thyroidectomy for thyroid swelling. Histopathology showed papillary thyroid carcinoma [T3 - 6.0 cm] with extra-thyroidal extension. The patient was treated with 150 mCi radioactive iodine(RAI) as adjuvant ablative therapy. Radioiodine refractory disease was identified 1-year post-RAI therapy with elevated thyroglobulin levels and negative I-131 whole body scan. F-18 FDG positron emission tomography/computedtomography scan showed activity in the right thyroid bed and multilevel right cervical nodes. Right-sided modified neck dissection was done, which showed Castleman disease (hyaline vascular type) in right cervical nodes. The most probable cause of elevated tumour markers was found out to be 0.6 cm right thyroid bed nodule on follow-up ultrasonography. Our patient also had coexistent conditions as; osteopoikilosis and Hepatitis C along with thyroid carcinoma.Key words: Castleman disease, lymph node, radioiodine, thyroid cancer


Blood ◽  
2008 ◽  
Vol 112 (11) ◽  
pp. 4998-4998
Author(s):  
Makoto Ide ◽  
Yasunori Kawachi ◽  
Yoichiro Izumi

Abstract Background: Multicentric Castleman’s disease (MCD) is an indolent lymphoproliferative disorder of unknown etiology, and is classified into three types: hyaline-vascular type, plasma-cell type, and mixed type. Recently, we reported the use of rituximab therapy for HIV-negative patients with MCD, after obtaining the informed consent of the patient and the approval of the Institutional Review Board (Br J Haematology 121, 818–819, 2003, Eur J Haematology 76. 119–123. 2006). Therefore, we tried to review the long-term follow-up (nine to 75 months) of all patients at a single institute. Patients and Methods: Over seven years, 5 HIV-negative patients with MCD (3 men and 2 women; median age: 39, range: 26–61 years) were referred to Takamatsu Red Cross hospital. After clinical evaluation (chest and abdominal computed tomography scans, magnetic resonance imaging and Ga67 citrate scintigrams), lymphnode biopsy was performed for histological diagnosis. Rituximab was administered intravenously at a standard dose of 375 mg/m2 weekly, for 4 or 8 times without chemotherapy, and we analyzed the clinical course of MCD after rituximab therapy. During the follow-up, clinical symptoms were recorded. At a median follow-up of 49 months, biologic parameters (complete blood count, C-reactive protein, immunoglobulin G), and cytokines (soluble interleukin-2 receptor, Interleukin-6) were determined at different time points by standard procedures. Results: Three of the five patients had histological evidence of hyaline-vascular type MCD, and two patients had plasma-cell type MCD. Two (both with hyaline vascular type) of the five MCD patients (40%) with MCD achieved almost complete remission with rituximab therapy when followed up for 50 to 75 months. Three (one with hyaline vascular type and two with plasma-cell type) of the five patients did not show clinical remission after riruximab administration. Conclusion;Rituximab treatment provides a durable response in a some MCD (especially those with hyaline-vascular type) patients, but is not always successful.


2017 ◽  
Vol 74 (4) ◽  
pp. 367-370
Author(s):  
Bosko Milev ◽  
Borka Milev ◽  
Zoran Kostic ◽  
Darko Mirkovic ◽  
Nenad Perisic ◽  
...  

Introduction. Castleman disease is a rare disease of the unknown etiology, occuring in two clinical forms: unicentric or multicentric. It is characterized by the hyperplasia of lymph glands. In literature the four pathohistological forms were described: hyaline vascular type, plasma cell type, mixed type and a recently recognized plasmablastic type. The most frequent changes are localized in the mediastinum, while the abdominal localization is with significatly rare occurrence, and that was the motive for presentation of this case. Case report. In a 41-year old male magnetic resonance (MR) enterography showed a change in the ileocecal area without the presence of subjective symptoms of digestive tract and without loss of body mass. Due to the suspicion of stromal tumor, surgical intervention was indicated. Pathohistological findings showed Castleman lymphadenopathia reactiva mesenterii (plasma cell type) which was in the unicentric form. There were present only anaemia and the increased value of sedimentation from the laboratory analyses. Conclusion. Abdominal localization of unicentric plasma cell form occurs rarely and the surgical method of treatment presents the golden standard as it was shown in the presented case.


2021 ◽  
Vol 2021 ◽  
pp. 1-4
Author(s):  
Adelaide Moutinho ◽  
Rita Gamboa Cunha ◽  
Sheila Koch Jamal ◽  
Marta Meleiro Lisboa ◽  
Sandra Tavares

Castleman disease is a rare lymphoproliferative disorder presenting with localized or disseminated lymphadenopathy and systemic symptoms. It can be categorized clinically as unicentric or multicentric, histopathologically as hyaline vascular, plasma cell, or mixed variant, and etiologically, considering the subtypes based on causative viral agents and associated syndromes. The multicentric type can mimic other haematological malignancies, ranging from asymptomatic to multiple organ involvement. Although its pathophysiology is not well known, the current approved treatments are directed towards interleukin-6, CD-20, and viral agents. The authors present an 82-year-old leucodermic man presented with a 2-week history of constitutional symptoms. Examination revealed pallor, hepatosplenomegaly, and palpable left axillary lymphadenopathy. Investigation showed anaemia, thrombocytopenia, polyclonal hypergammaglobulinemia, hypoalbuminemia, and high acute phase reactants, with image study revealing multiple axillary, mediastinal, inguinal, and pelvic lymphadenopathies. The lymph node biopsy was consistent with hyaline vascular-type Castleman disease without human herpersvirus-8 markers. He started prednisolone with initial improvement evolved poorly on a short term. Castleman disease has a broad spectrum of clinical manifestations, associations, and complications that bring a diagnostic challenge, requiring a multidisciplinary approach. Clinicians should be familiar with its features because proper diagnosis and aggressive targeted treatment are the pillars of proper management of these patients.


2008 ◽  
Vol 136 (1-2) ◽  
pp. 46-49
Author(s):  
Jelena Stojsic ◽  
Svetlana Krstic ◽  
Dragan Subotic ◽  
Tatjana Eminovic ◽  
Jelena Radojicic

INTRODUCTION Castleman disease was for the first time described in 1956 as a mediastinal tumour mass. Etiology of this disease is still unknown. The disease can be solitary and multicentric or rarely of a mixed type. The former is often of hyaline vascular type, while the latter is of plasma cell type. CASE REPORT Castleman disease was diagnosed in a 26-year old male patient when a well defined shadow was incidentally detected in the middle lobe of the right lung. A year before, he was diagnosed with allergic rhinitis to Ambrosia. Two years after surgery the patient was feeling well, and was without any recurrence, however, allergic rhinitis still persisted. CONCLUSION Castleman disease can occur in any organ containing lymph tissues. Most frequently the disease is described as mediastinal, rarely as an intrapulmonary tumorous mass, and it is most frequently seen in younger persons. The solitary type of Castleman disease is surgical treatable with a prospect of good prognosis, while the multicentric and mixed types recur despite treatment with cortisone, irradiation and cytostatics. As the association between Castleman disease and allergic diseases has not been confirmed up-tonow, it could be concluded that this patient suffered from two separated diseases.


2019 ◽  
Vol 4 (4) ◽  

Castleman Disease (CD) is a lymphoproliferative disorder characterized by enlarged hyperplastic lymph nodes with regressed follicles surrounded by expanded mantle zones of small lymphocytes, and interfollicular vascular proliferation in the hyaline-vascular type. There are two types: unicentric and multicentric. Unicentric, hyalinevascular type of Castleman’s disease can be treated successfully with complete surgical resection with monitoring for reoccurrence. Here we report a case of a patient originally diagnosed with sarcoidosis who was found to have Unicentric Castleman’s disease.


2020 ◽  
Vol 45 (4) ◽  
pp. 623-630 ◽  
Author(s):  
Peng Xia ◽  
Lu Zhang ◽  
Menglian Zou ◽  
Tengyue Zhang ◽  
Ran Li ◽  
...  

Introduction: Thrombocytopenia, ascites, myelofibrosis, renal dysfunction, and organomegaly (TAFRO) syndrome is a newly recognized and rare clinical subtype of Castleman disease. Renal involvement in TAFRO syndrome usually presents with mild proteinuria, microscopic hematuria, and acute renal injury requiring temporary renal replacement. There is no standard therapy available and treatment failures are common, leading to a poor prognosis. We report a case of acute renal failure caused by TAFRO syndrome, successfully managed by long-term corticosteroids combined with bortezomib and cyclophosphamide. Case Presentation: The patient was a 52-year-old female who presented with fever, anasarca, oliguria, and abdominal distension at first. She progressed rapidly to anuric renal failure requiring hemodialysis. She also demonstrated thrombocytopenia, anemia, coagulopathy, and a hyperinflammatory status. Her CT scan showed severe polyserositis, splenomegaly, and lymphadenopathy. Her serum vascular epithelial growth factor level was significantly elevated. Axillary lymph node biopsy showed hyaline-vascular type Castleman disease, supporting the diagnosis of TAFRO syndrome. Her renal function recovered after high-dose steroids and supportive treatment. A weekly dosing regimen of bortezomib, cyclophosphamide, and dexamethasone combined with medium dose prednisone in between were deployed. Her blood cell count and renal function remained stable after 6 months. The inflammation was suppressed and the polyserositis resolved completely. Conclusion: TAFRO syndrome is rare and has a poor prognosis due to the lack of standard treatment. Our patient might be the first TAFRO case successfully treated by bortezomib, cyclophosphamide, and corticosteroids.


1996 ◽  
Vol 35 (06) ◽  
pp. 230-265 ◽  
Author(s):  
E. Kresnik ◽  
P. Mikosch ◽  
W. Pipam ◽  
Iris Gomez ◽  
P. Lind ◽  
...  

Summary Aim: The usefulness of the myocardial perfusion agent, Tc-99m-tetrofosmin (Myoview®) in the follow-up of differentiated thyroid carcinoma was evaluated in a clinical study of 15 patients, primary treated with thyroidectomy and high-dose I-131-therapy (2960-3700 MBq), 12 with suspected recurrence and metastases and three patients without any suspicion and compared with other non-specific tracers like TI-201 and Tc-99m-sestamibi. Method: Twelve patients with elevated thyroglobulin (Tg) levels of more than 10 ng/ml (group A), four of these had negative I-131 scans, and three patients with Tg levels less than 10 ng/ml (group B) were examined under TSH suppressive L-Thyroxine treatment. Whole body scans were taken with TI-201 (74 MBq; 20 min post injection), Tc-99m-sestamibi (370 MBq; 20-60 min post injection) and Tc-99m-tetrofosmin (370 MBq; 20-60 min post injection). Tumor/background ratios and optional time/activity analyses (up to 150 min post injection) were evaluated using the region of interest approach. Results: Compared with TI-201 (T/BG: 1.59, ± 0.396), Tc-99m-tetrofosmin showed slightly but not significant better T/BG ratios and detection rates (T/BG: 1.76, ± 0.345).Tc-99m-sestamibi (1.51, ± 0.31 p = 0.05) showea significantly lower values than Tc-99m-tetrofosmin. Conclusion: In the light of these results, scintigraphy with Tc-99m-tetrofosmin seems to be a possibly sensitive imaging modality in the follow-up of DTC with possible advantages concerning T/Bg ratio, background clearance, detection rate and dosimetry compared with TI-201 and Tc-99m-sestamibi, especially in patients with elevated Tg level and no iodine uptake , but further investigations are needed to confirm our preliminary results.


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