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1.  Castleman's Disease: A Case Report of the Unicentric Type 
Case Reports in Surgery  2012;2012:175272.
Castleman's disease, or angiofollicular lymph node hyperplasia, is a relatively rare disorder characterized by the benign proliferation of lymphoid tissue related to the chronic human herpes virus 8 (HHV-8) infection and the human immunodeficiency virus (HIV). Two clinical entities have been described: a unicentric presentation with the disease confined to a single anatomic lymph node and a multicentric presentation characterized by generalized lymphadenopathy and a more aggressive clinical course. Also, three histopathological subtypes have been described: hyaline-vascular, plasma cell, and a mixed variant. Preoperative diagnosis of hyaline-vascular Castleman's disease is difficult, and the definitive result is based on postoperative pathological findings. The gold standard therapy is the complete surgical excision.
doi:10.1155/2012/175272
PMCID: PMC3443986  PMID: 22991681
2.  Localized retroperitoneal Castleman’s disease: a case report and review of the literature 
Introduction
Castleman’s disease, also known as angiofollicular lymph node hyperplasia, is a rare disease with two known expansion types, unicentric and multicentric, which play a major role in determining therapy. We focus here on the unicentric type, which can be treated and cured by surgery. To date, approximately 1000 cases of Castleman’s disease have been reported in the literature.
Case presentation
A 50-year-old Caucasian woman presented to our Department of Hematology and Internal Oncology with increasing fatigue as her sole symptom. Diagnostic investigations including laboratory studies, ultrasound, computed tomography and magnetic resonance imaging were performed. These revealed an interaortocaval, retroperitoneal tumor mass in her upper abdomen as the only manifestation of the disease. No enlarged lymph nodes were detected. We conducted a laparotomy with radical extirpation of the tumor mass (10×9×5.7cm). Complete tumor resection with clear margins was achieved. A pathological analysis of the resected sample showed atypical lymphoid tissue of small to medium cells with some clearly visible nucleoli, enlarged sinusoidal vessels, pleomorphic calcifications and focally preserved germinal-center-like structures. Histological and immunohistochemical analysis confirmed the diagnosis of Castleman’s disease: staining for CD3, CD5, CD10, CD20, CD23, CD79 and Ki-67 was strongly positive in the germinal-center-like structures. Histological findings clearly showed the disease to be the hyaline vascular subtype. Staining for cyclin D1 and CD30 was negative. Expression of CD15 was positive in the enlarged sinusoidal vessels. A supplementary clonality analysis was without pathological findings. Tests for human immunodeficiency virus and human herpes virus 8 were negative and results from a bone marrow biopsy were normal. Our patient recovered well from surgery and was discharged from our hospital. To date, no recurrence of the disease has been detected.
Conclusion
Castleman’s disease is a rare disorder that remains a diagnostic challenge. Radical surgical resection is considered to be the gold standard for treating the unicentric variant of this disease.
doi:10.1186/1752-1947-8-93
PMCID: PMC3977947  PMID: 24612848
Castleman’s disease; Hyaline vascular variant; Surgery; Unicentric
3.  An unusual unifocal presentation of Castleman’s disease in a young woman with a detailed description of sonographic findings to reduce diagnostic uncertainty: a case report 
BMC Research Notes  2013;6:97.
Background
Castleman’s disease is a rare lymphoproliferative disorder. It typically presents as mediastinal masses and causes a wide range of clinical symptoms. Histologically, Castleman’s disease is classified as either a hyalinic vascular or plasma cell variant. The prognosis mainly depends on the histological type and broadly varies. We herein report our sonographic findings in a patient with Castleman’s disease, including gray-scale ultrasonography, color Doppler ultrasonography, and sonoelastography ultrasonography, which have not been previously reported in the literature. These findings allowed for a preoperative diagnosis and avoidance of overly aggressive therapy.
Case presentation
A 28-year-old European female patient with unicentric Castleman’s disease of hyalinic vascular type (HV) restricted to the axilla was referred to us because of a 4-month history of a painless, solitary mass located in the left axilla. The patient’s medical history was unremarkable.
Conclusion
Castleman’s disease is a pathologic entity of unknown etiology and pathogenesis. In this case report of unicentric HV-type CD, we demonstrate that typical sonographic findings can lead to a preoperative diagnosis of Castleman’s disease. Core needle biopsy usually allows for a final diagnosis and helps to avoid unnecessary operations and overtreatment.
doi:10.1186/1756-0500-6-97
PMCID: PMC3601982  PMID: 23497665
Castleman’s disease; Giant lymph node hyperplasia; Ultrasonography; Core needle biopsy
4.  Hyaline vascular- type Castleman's disease in the hilum of liver: a case report 
Cases Journal  2010;3:74.
Background
Castleman's disease or angiofollicular lymphoid hyperplasia is a rare benign lymph node hyperplasia usually presenting as an asymptomatic mediastinal mass in children. The disease can present at any extra thoracic site with lymphoid tissue such as retroperitoneal, mesentery, axilla, and pelvis. Hepatic localization castleman disease is very rare in children. Herein, we reported a case of Castleman's disease arising from the lymph node in hilum of liver.
Case presentation
A 5 -year-old girl with chief complaint of abdominal pain for two months which exaggerated in last three days was referred to the hospital. On routine physical examination, only a generalized abdominal pain was noticed. Routine laboratory investigations and Chest X-Ray were normal. Abdominal Sonography revealed a 3.7 × 3.1 cm solid mass in the hilum of the liver. On the MRI images, a lobulated mass in the portal hepatic associated with mass effect on the portal vein was visible. Histological examination revealed expansion of mantle zone in lymphatic nodules accompanied by burnt out germinal centers. This pattern was matched with the diagnosis of the hyaline-vascular type of Castleman disease. The patient underwent a laparotomy. The patient had an uneventful postoperative course.
Conclusion
This pattern was matched with the diagnosis of the hyaline-vascular type of Castleman disease.
doi:10.1186/1757-1626-3-74
PMCID: PMC2845114  PMID: 20193070
5.  A multicentric, hyaline vascular variant of Castleman's disease associated with B cell lymphoma: a case report 
Cases Journal  2009;2:8183.
Introduction
Three histological variants (hyaline vascular, plasma cell, and mixed) and two clinical types (localized and multicentric) of Castleman's disease have been described. The risk of progression to lymphoma is higher in multicentric Castleman's disease and is associated with poorer outcomes and higher mortality rate. Multicentric Castleman's disease often requires systemic therapy. Complete resection of the involved node in localized Castleman's disease is curative, with no reported recurrences.
Case presentation
We report a case of a 66-year-old female with systemic symptoms and bilateral cervical lymph nodes which were initially diagnosed as the hyaline vascular variant of Castleman's disease and two years later after the initial diagnosis she was confirmed to B cell lymphoma. Following the treatment with radiation therapy to the cervical area and combination chemotherapy complete response was achieved.
Conclusion
Although it has rarely been reported, the malignant potential of the Castleman's disease must be kept in mind.
doi:10.4076/1757-1626-2-8183
PMCID: PMC2740273  PMID: 19830060
6.  A Rare Coexistence of Retroperitoneal Pararenal Castleman's Disease with Focal Nodular Hyperplasia 
Case Reports in Surgery  2013;2013:537593.
Castleman's disease is a distinct form of lymph node hyperplasia divided into a solitary and a multicentric type. The solitary type occurs most commonly in the mediastinum and is usually asymptomatic. We present a patient with Castleman's disease of the hyaline-vascular solitary type located in the retroperitoneum. The patient was a 38-year-old male, who presented to our hospital with fever. The imaging workup revealed a retroperitoneal mass, measuring 4 × 6 cm, located lateral to the aorta, inferior to the left renal artery and vein, and posterior to the left testicular vein. At workup, a solid hepatic lesion, 3 cm in diameter, located in the left lobe of the liver, segment IV, was also identified. Both lesions were surgically excised. The retroperitoneal tumor had the features of angiofollicular hyperplasia (Castleman's disease), hyaline-vascular type, whereas a diagnosis of focal nodular hyperplasia was made for the hepatic lesion. The patient is well at fourty months followup postoperatively. Surgical excision is the treatment of choice for unifocal Castleman's disease.
doi:10.1155/2013/537593
PMCID: PMC3603638  PMID: 23533915
7.  Extrathoracic castleman’S disease — presenting as a neck mass 
Castleman’s disease, also known as angiofollicular hyperplasia, is a disease of unsettled complexity and morbidity, usually occurs in the mediastinum and less frequently in the neck as a solitary mass and is often associated with other illnesses. Two variants have been recognized, the hyaline vascular type which is mare common and the less common plasma cell type with a third hybrid or intermediate type that shares features with both. It has been described with AIDS and Kaposi’s sarcoma as well as follicular dendritic cell tumours.
We report a case of Castleman’s disease of the hyaline vascular type arising in the left supra-clavicular region in 41 year old male without any associated systemic illness. The neck is an uncommon location for this rare disease.
doi:10.1007/BF03006199
PMCID: PMC3451106  PMID: 23119691
Giant lymphnode hyperplasia; Hyalin; Plasma cell
8.  Unicentric mixed variant Castleman disease associated with intrabronchial plasmacytoma 
Diagnostic Pathology  2014;9:64.
Virtual slides
The virtual slide(s) for this article can be found here: http://www.diagnosticpathology.diagnomx.eu/vs/2872096831190851.
Castleman disease (CD), described as a heterogeneous lymphoproliferative disorder, can be divided into different subtypes according to clinical appearance (unicentric and multicentric form) and histopathological features (hyaline vascular, plasma cell, mixed type, human herpesvirus 8–associated and multicentric not otherwise specified). Unicentric CD is known to be usually of the hyaline vascular variant, plasma cell and mixed type of this form are quite uncommon. Malignancies are mainly associated with the multicentric form. We report a rare case of unicentric mixed variant CD evolving into intrabronchial, extramedullary plasmacytoma.
Intrabronchial mass with consequential obstruction of the left main bronchus, left lung atelectasis and mediastinal lymphadenomegaly was detected by chest CT in our patient suffering from cough and hemoptysis. Pulmonectomy was performed, histopathological and immunhistochemical analysis of lymph nodes revealed mixed type of CD with interfollicular monotypic plasma cell proliferation. The intrabronchial mass consisted of monotypic plasma cells confirming plasmacytoma. Systemic involvement was not confirmed by further tests.
Although malignancies more often present in multicentric CD that usually belongs to the plasma cell subtype, this case confirms the neoplastic potential of the rarest, unicentric mixed variant of CD.
doi:10.1186/1746-1596-9-64
PMCID: PMC3994877  PMID: 24649966
Castleman disease; Unicentric; Mixed variant; Extramedullary plasmacytoma; Intrabronchial plasmacytoma
9.  Cytologic picture of Castleman’s disease: A report of two cases 
Castleman’s disease (CD), also called giant lymph nodal hyperplasia, is a lymphoproliferative disorder secondary to lymphoid follicle hyperplasia and marked capillary proliferation with endothelial hyperplasia. It presents as enlarged lymph nodes in the mediastinum, neck, groin, axilla and other sites. On clinical examination as well as gross examination, this disease mimics lymphomas and tuberculosis. Since cytological appearances vary depending on the type and extent of hyperplasia, fine needle aspiration cytology (FNAC) findings may not always be conclusive in all cases. We studied aspirates in two cases of CD, one of which presented with multiple enlarged axillary lymph nodes and the other with enlarged cervical lymph node. Cytology revealed reactive lymphadenitis with hyalinized capillaries and other features. Lymph node excision in both cases confirmed the diagnosis.
doi:10.4103/0970-9371.73306
PMCID: PMC3001206  PMID: 21157570
Cytology; Castleman’s disease; lymph node; fine needle aspiration cytology
10.  Neoadjuvant radiotherapy of primary irresectable unicentric Castleman's disease: a case report and review of the literature 
Background
Castleman disease (CD) is a rare benign disorder characterised by hyperplasia of lymphoid tissue that may develop at a single site or throughout the body. The etiology of this disorder is unclear, although the histopathological presentation can be differentiated into a hyaline vascular variant, a plasma cell variant and a mixed variant. Clinically, it has been recorded that 3 manifestations of CD are characterized: a localized unicentric type, a generalized multicentric type and a mixed form. Surgery remains the main treatment for resectable unicentric CD, since removal of the large node is possible without further complications. No consensus has been reached concerning the most adequate treatment for irresectable unicentric CD.
Methods
Case report of a 67 year old woman.
Results
This report, describes the case of a 67-year-old woman with unicentric Castleman disease located in the right lower abdomen. The patient had symptoms of fatigue, dyspnoea and pain in the right lower abdomen. Computed tomography (CT)- examination revealed a tumour, which had grown to form a close relationship with the common iliac vessels and the sacral bone. A Laparotomy procedure revealed that the tumour was an irresectable mass. Neo-adjuvant radiotherapy (40 Gy) was administered in order to downsize the tumour. Six weeks later a new CT-scan revealed a major reduction of the tumour, which enabled a successful radical resection of the tumour to be performed. Histopathological analysis of the tumour showed the hyaline vascular type of CD.
Conclusions
Neo-adjuvant radiotherapy should be considered in case of an irresectable unicentric CD.
doi:10.1186/1748-717X-5-7
PMCID: PMC2827478  PMID: 20122250
11.  Castleman’s disease in a patient with melanoma: the role of VEGF 
BMJ Case Reports  2012;2012:bcr0720114519.
Castleman’s disease is a rare lymphoproliferative disorder characterised by lymph node follicular hyperplasia with abnormal interfollicular vascular growth. The authors report a female patient presenting with erythematous and pruritic skin lesion. A shave biopsy of the lesion revealed a Clark level III melanoma and sentinel lymph node biopsy revealed a microscopic focus. Staging positron emission tomography scan revealed an abnormal soft tissue mass with increased 18F-fluorodeoxyglucose accumulation in the anterior mediastinum, raising suspicions of malignancy. The patient underwent an elective resection of the mass and histological sections revealed features of Castleman’s disease of the hyaline vascular type. Vascular endothelial growth factor (VEGF) has been associated with the increased vessel density in this condition. VEGF secretion by tumour cells in solid tumours such as melanomas could be a predisposing factor for Castleman’s disease. Future research is needed to find an association between VEGF-secreting solid tumours and the development of lymphoproliferative tumours such as Castleman’s disease.
doi:10.1136/bcr.07.2011.4519
PMCID: PMC3351633  PMID: 22605002
12.  Castleman's Disease: Systematic Analysis of 416 Patients from the Literature 
The Oncologist  2011;16(9):1316-1324.
Clinical, pathological, and viral markers were used to classify patients with Castleman's disease into groups with markedly different outcomes and disease associations. This system could aid in providing physicians and patients with a realistic prognostic outlook.
Learning Objectives
After completing this course, the reader will be able to: Describe the centricity and histopathology profiles of each of the three classes of HIV-negative patients identified in this analysis.Correlate nosological classification and outcomes in patients with Castleman's disease.
This article is available for continuing medical education credit at CME.TheOncologist.com
Background.
Castleman's disease is a rare primary disease of the lymph nodes with limited available clinical information.
Methods.
A systematic literature search identified 416 cases amenable to detailed analysis.
Results.
In HIV− patients, centricity, pathology type, the presence of symptoms, gender, and age all predict outcome in univariate analyses. The 3-year disease-free survival (DFS) rate for patients with unicentric hyaline vascular disease (49.5% of cases, class I) was 92.5%, versus 45.7% for those with multicentric plasma cell disease (20.2% of cases, class III) and 78.0% for those with any other combination (22.6% of cases, class II) (p < .0001). HIV+ patients (class IV) exclusively presented with multicentric plasma cell disease and had a 3-year DFS rate of only 27.8%. Kaposi's sarcoma and lymphoma were observed in 59.3% and 9.4% of HIV+ patients and in 2.6% and 3.6% of HIV− patients (p < .0001). Paraneoplastic pemphigus and the syndrome of polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin changes were observed exclusively in HIV− patients at a rate of 1.3% and 1.8%, respectively.
Conclusion.
Clinical, pathological, and viral markers allow for the classification of Castleman's disease into groups with markedly different outcomes and disease associations.
doi:10.1634/theoncologist.2011-0075
PMCID: PMC3228165  PMID: 21765191
Castleman's disease; Lymph node; HIV; Viral infection
13.  Intestinal lymphangiectasia associated with angiofollicular lymph node hyperplasia (Castleman's disease). 
Gut  1992;33(1):135-137.
A patient presenting with predominantly gastrointestinal symptoms and a history of myocardial infarction was found to have ascites, hepatosplenomegaly, para-aortic lymphadenopathy, thrombocytosis, and a paraproteinaemia. A jejunal biopsy specimen showed lymphangiectasia and histology of the spleen and lymph nodes showed angiofollicular hyperplasia or Castleman's disease of the hyaline vascular type. This association has not previously been described and, moreover, systemic symptoms are unusual in this variant of Castleman's disease.
Images
PMCID: PMC1373880  PMID: 1740268
14.  Hyaline Vascular Castleman Disease Involving Renal Parenchyma and a Lymph Node: A Case Report 
Korean Journal of Pathology  2012;46(1):79-82.
Castleman disease is a rare lymphoproliferative lesion that is predominantly found in the mediastinum. Retroperitoneal and pararenal localizations are very rare. We describe a 36-year-old man with a hyaline vascular type of Castleman disease involving renal parenchyma and a paraaortic lymph node. Most reported renal Castleman disease was plasma cell type with systemic symptoms. Herein, we report the first Korean case of the hyaline vascular type of Castleman disease involving the renal parenchyma and the paraaortic lymph node simultaneously.
doi:10.4132/KoreanJPathol.2012.46.1.79
PMCID: PMC3479711  PMID: 23109983
Kidney; Hyaline vascular type, Multicentric; Giant lymph node hyperplasia
15.  An exrathoracic presentation of castleman's disease 
Castleman's disease, also known as angiofollicular hyperplasia, usually occurs in the mediastinum and rarely in cervical region as solitary mass. Histopathologically four variants have been recognized (Castleman et al. Cancer 1954;9:822–30) hyaline vascular type—most common type (Keller et al. Cancer 1972;29:670–83) plasma cell type (Salisbury, Pediatric Pathol 1990;10:609–15) transitional type (Shahidi, Mayo Clinic Proc 1995;70:969–77) stromal rich type. We report a case of Castleman's disease of typical histopathological picture of angio follicular hyperplasia arising from the right carotid triangle of neck in 12-year-old child without any associated systemic illness.
doi:10.1007/BF02907683
PMCID: PMC3450978  PMID: 23120165
Castlman's disease; angio follicular hyperplasia; neck
16.  Three patients with both Hodgkin's lymphoma and Castleman's disease: Clinicopathologic correlations and lack of association with HHV-8 
Background:
The relationship between Hodgkin's lymphoma (HL) and plasma cell-type Castleman's disease (PCD) has been well documented. There have been over 20 cases reported in the literature and nearly all of them were either diagnosed concurrently, or were initially diagnosed as PCD and upon review were found to have interfollicular HL. Human herpes virus type 8 (HHV-8) is present in about 40% of cases with PCD. It predisposes patients to a much higher risk of other malignancies, including Kaposi's sarcoma and non-Hodgkin's lymphoma. Cases linked to HHV-8 are associated with a different morphology than cases that are not linked to HHV-8. It has been proposed that patients with both HL and CD will have lymph nodes with HHV-8-negative morphology.
Materials and Methods:
We present a series of three cases in a retrospective study where patients had both HL and PCD. Surgical pathology reports, clinical histories, and H and E and various immunohistochemical stains on initial work-up were examined and subsequent immunohistochemical stains for HHV-8 were obtained from the Methodist Hospital.
Results:
Patient 1 was diagnosed with PCD and interfollicular HL in the same lymph node. Patient 2 was first diagnosed with classic HL and 2 years later returned with enlarged lymph nodes clinically suspected to be recurrent HL. Histology showed angiofollicular hyperplasia and interfollicular plasmacytosis without Reed-Sternberg cells and a diagnosis of PCD was rendered. Patient 3, a male in his third decade, was diagnosed with nodular sclerosing HL in the thymus, and concurrently PCD in the mediastinal lymph nodes. All three cases had architectural features consistent with an HHV-8-negative morphology. Immunohistochemical stains for HHV-8 were done retrospectively and were negative.
Conclusion:
All three of our patients with both HL and CD had HHV-8-negative lymph node morphology and absence of HHV-8 by immunohistochemistry. These patients, therefore, are not at an increased risk for the development of subsequent malignancies, when compared to HHV-8-positive patients. Included in our series is one unique case where the diagnosis of HL preceded CD by 2 years.
doi:10.4103/0971-5851.60052
PMCID: PMC2885879  PMID: 20596307
Castleman's disease; Hodgkin's lymphoma; HHV-8; plasma cell variant of Castleman's disease
17.  Retroperitoneal Castleman’s disease: advocating a multidisciplinary approach for a rare clinical entity 
Background
Castleman’s disease is a rare and poorly understood disease entity that may resemble more common conditions and represents a clinical challenge to the treating surgeon.
Case presentation
In this report, we describe a case of a 61-year-old Caucasian woman with a symptomatic retroperitoneal mass. The specimen obtained from her resection contained a protuberant encapsulated mass, exhibiting microscopic features consistent with localized, unicentric Castleman’s disease. These characteristics included architectural features and immunohistochemical findings consistent with the hyaline vascular variant of Castleman’s disease.
Conclusion
We report a very rare case of a retroperitoneal hyaline vascular type of Castleman’s disease. We discuss the diagnostic dilemma Castleman’s disease may present to the surgeon, with an emphasis on multidisciplinary management of these patients. We also review current data on pathogenesis, treatment and outcomes.
doi:10.1186/1477-7819-12-30
PMCID: PMC3975995  PMID: 24495409
Angiofollicular lymph node hyperplasia; Castleman’s disease; Management; Multidisciplinary care
18.  Castleman's Disease of the Renal Sinus Presenting as a Urothelial Malignancy: A Brief Case Report 
Korean Journal of Pathology  2012;46(5):503-506.
Castleman's disease is a rare benign lymphoproliferative disorder that frequently affects lymph nodes of the mediastinal thorax and the neck. It very rarely affects the renal sinus. We report a case of Castleman's disease arising in the renal sinus in a 64-year-old man. The patient visited the hospital with the chief complaint of hematuria. Abdominal computed tomography revealed a homogeneous mass in the sinus of the left kidney, radiologically interpreted as a malignant urothelial tumor. Subsequently, nephroureterectomy was performed, after which microscopic examination of the specimen revealed a diffuse lymphoproliferative lesion with reactive lymphoid follicles of various sizes and prominent plasma cell infiltration of interfollicular spaces, highlighted by immunohistochemical staining for CD138. The lesion was diagnosed as Castleman's disease of the plasma cell type. Although preoperative diagnosis of Castleman's disease is difficult and the incidence is exceedingly rare, it should be considered in the differential diagnosis of renal sinus tumors.
doi:10.4132/KoreanJPathol.2012.46.5.503
PMCID: PMC3490114  PMID: 23136580
Giant lymph node hyperplasia; Plasma cell type; Renal sinus; Urothelial malignancy; Kidney
19.  Castleman’s Disease of the Neck: Report of Four Cases with Unusual Presentations 
Purpose
Most of the previously reported cases of Castleman’s disease (CD) in the neck were of the hyaline vascular type and the most common sign was an asymptomatic neck mass. To achieve a more general recognition of CD in the neck, we present four cases of CD in the neck with unusual presentations.
Patients and Methods
We describe four cases of CD in the neck with unusual clinical manifestations, different histopathologic subtypes and discuss the problems arising in their clinical findings.
Results
Two cases were plasma cell type with multiple bilateral enlarged lymph nodes and systemic manifestations. Two cases presented as a solitary mass were hyaline vascular type. One case underwent twice local recurrence and evolved from the hyaline vascular CD to follicular dendritic cell sarcoma.
Conclusions
Cervical plasma cell CD may have a tendency to present as multiple masses on the both sides with systemic manifestations. It is necessary to pay more attention on the overgrowth of follicular dendritic cells in the hyaline vascular CD, which might represent a precursor of follicular dendritic cell sarcoma.
doi:10.1016/j.joms.2010.03.007
PMCID: PMC3010435  PMID: 20708326
Castleman’s disease; Neck; POEMS syndrome; Follicular dendritic cell sarcoma; Clinical manifestation
20.  Induction of B Cell Hyperplasia in Simian Immunodeficiency Virus–Infected Rhesus Macaques with the Simian Homologue of Kaposi's Sarcoma–Associated Herpesvirus 
A simian homologue of Kaposi's sarcoma–associated herpesvirus (KSHV), the eighth human herpesvirus (HHV8), was isolated from a simian immunodeficiency virus (SIV)-infected rhesus macaque (Macaca mulatta) that developed a multicentric lymphoproliferative disorder (LPD). This simian rhadinovirus is genetically similar to a recently described rhesus rhadinovirus (RRV) (Desrosiers, R.C., V.G. Sasseville, S.C. Czajak, X. Zhang, K.G. Mansfield, A. Kaur, R.P. Johnson, A.A. Lackner, and J.U. Jung. 1997. J. Virol. 71:9764–9769) and is designated RRV 17577. RRV 17577 was experimentally inoculated into rhesus macaques with and without SIVmac239 infection to determine if RRV played a role in development of the LPD observed in the index case. In contrast to control animals inoculated with SIVmac239 or RRV alone, two animals coinfected with SIVmac239 and RRV 17577 developed hyperplastic LPD resembling the multicentric plasma cell variant of Castleman's disease, characterized by persistent angiofollicular lymphadenopathy, hepatomegaly, splenomegaly, and hypergammaglobulinemia. Hypergammaglobulinemia was associated with severe immune-mediated hemolytic anemia in one RRV/SIV-infected macaque. Both RRV/SIV-infected macaques exhibited persistent RRV viremia with little or no RRV-specific antibody response. The macaques inoculated with RRV alone displayed transient viremia followed by a vigorous anti-RRV antibody response and lacked evidence of LPD in peripheral blood and lymph nodes. Infectious RRV and RRV DNA were present in hyperplastic lymphoid tissues of the RRV/SIV-infected macaques, suggesting that lymphoid hyperplasia is associated with the high levels of replication. Thus, experimental RRV 17577 infection of SIV-infected rhesus macaques induces some of the hyperplastic B cell LPDs manifested in AIDS patients coinfected with KSHV.
PMCID: PMC2195633  PMID: 10499921
B cell hyperplasia; simian immunodeficiency virus; rhesus rhadinovirus; lymphoproliferative disorders; nonhuman primate
21.  The management of Castleman's disease of the mediastinum: a case report 
Cases Journal  2008;1:330.
Castleman's disease or angiofollicular lymph node hyperplasia is a rare clinical entity that may present in many sites and with a variety of symptoms. We report here a case of unicentric Castleman's disease (hyaline vascular pattern) located in the mediastinum as a solitary mass. The patient was a Caucasian female 58 years old presented after incidental discovery of the mass in a x-ray. In Castleman's disease surgery is generally recommended for localised lesions to remove the mass as completely as possible reserving other treatment modalities for unresectable cases or multicentric disease.
doi:10.1186/1757-1626-1-330
PMCID: PMC2596111  PMID: 19019227
22.  Irreversible Kidney Damage due to Multicentric Castleman's Disease 
The Libyan Journal of Medicine  2008;3(2):101-103.
Castleman's Disease (CD) is a rare lymphoproliferative disorder accompanied by marked systemic inflammatory response. Morphological diagnosis of CD requires biopsy of the whole of the involved lymph node tissue. Three histologic variants have already been described in CD morphology (hyaline vascular, plasma-cell, and mixed). In this study, we report a case of a multicentric Castleman's disease of the plasma cell variant type with negative Herpes Virus 8. The clinical presentation of this patient was of systemic amyloidosis as a result of both a delayed diagnosis and medical management. Previously described cases of CD with secondary amyloidosis have been of the localized type. Regardless, long-standing clinical remission of CD by cytotoxic drugs and anti-CD20 antibody therapy was achieved, but the nephrotic syndrome remained irreversible.
doi:10.4176/080108
PMCID: PMC3074288  PMID: 21499466
23.  Castleman’s Disease of the Porta Hepatis 
Castleman’s disease is an uncommon cause of lymph node enlargement that mostly involves the mediastinum and lung hilum. It is divided into 2 types: localized, which is usually asymptomatic and presents with a mass lesion; and multicentric, which is characterized by chills, anemia, generalized lymphadenopathy and hepatosplenomegaly. An extrathoracic site of involvement is very uncommon, and may be located in the mesentery of the intestines, axilla, and pelvis. Hepatic localization of this disease is a rare event. Herein, we report our experience with a symptomatic case of Castleman’s disease in the porta hepatis who has been treated successfully by excision of the hilar lymph nodes, but recurred after 2 years with the same clinical picture plus abnormal liver function tests. However, at the time of recurrence ,no lymphadenopathy was detected and liver biopsy showed giant lymphoid follicles with germinal centers. She was treated with steroids and showed a dramatic response.
PMCID: PMC3990119  PMID: 24829655
Castleman’s disease; Liver hilum; Recurrence; Lymphoid nodular hyperplasia
24.  A case report of syndrome of inappropriate antidiuretic hormone secretion with Castleman’s disease and lymphoma 
Background
Syndrome of inappropriate antidiuretic hormone secretion (SIADH) is a common cause of hyponatremia in hospitalized patients and is often described in patients with small-cell carcinoma of the lung. In this report, we described both Castleman’s disease and lymphoma coexisting in one patient with SIADH.
Case presentation
A 70-year-old Chinese woman with a history of diabetes mellitus and insulin therapy had severe hyponatremia and gastrointestinal symptoms. Through a series of examinations, common causes such as pulmonary carcinoma were excluded. An abdominal mass was detected by computed tomography. Although the peripheral lymph node biopsy showed the pathological result as Castleman’s disease, the pathology of the abdominal lymph node revealed diffuse large B-cell lymphoma. After chemotherapy, the hyponatremia was treated during a period of follow-up.
Conclusion
This patient presented with the rare clinical condition of inappropriate antidiuretic hormone secretion alongside Castleman’s disease and lymphoma. Asymptomatic hyponatremia may persist for some time suggesting that clinical physicians should pay attention to the mild cases of hyponatremia. We also hypothesized that Castleman’s disease is a condition of pre-lymphoma with both having the ability to cause SIADH. The possibility of lymphoma as well as Castleman’s disease triggering the development of SIADH should also be taken into consideration for conducting recurrent biopsies.
doi:10.1186/1472-6823-13-19
PMCID: PMC3679987  PMID: 23734852
Castleman’s disease; Hyponatremia; Lymphoma; Syndrome of inappropriate antidiuretic hormone secretion (SIADH)
25.  Retroperitoneal Castleman’s disease mimicking soft tissue tumour 
The Indian Journal of Surgery  2008;69(4):153-154.
Castleman’s disease is a type of non-neoplastic lymphoproliferative disease having lymph nodal hyperplasia. It has two distinct microscopic types: hyaline-vascular type and plasma cell type. Clinically, it may present either as a solitary mass, most commonly in the mediastinum, or as a multicentric form whose features are generalized lymph-adenopathy, splenomegaly and involvement of other organs like the lungs and kidneys. Here we report a case of isolated retroperitoneal Castleman’s disease, which presented as a lump in the iliac fossa in a young female. A clinico-radio-logical diagnosis of retroperitoneal soft tissue tumour was made and the patient underwent complete surgical excision. The exact diagnosis was only obtained at histopathology and there is no evidence of recurrence at six months follow-up.
doi:10.1007/s12262-007-0009-1
PMCID: PMC3452467  PMID: 23132970
Retroperitoneal; Castleman’s disease; Isolated; Hyaline vascular

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