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At least 19 recordsLinked to original sources
PubMed2006

Outcome and late complications of radiotherapy in patients with unicentric Castleman disease.

Castleman disease is a rare lymphoproliferative disorder. Surgery is considered standard therapy for the unicentric type. However, case reports have documented favorable responses to radiotherapy. The aim of this study was to analyse the clinical outcomes of five patients with unicentric Castleman disease treated with radiotherapy between 1991 and 2005. Mediastinal lymph nodes were the most common site of disease (four patients). Three patients were treated with radiotherapy alone, two patients with surgery and radiotherapy. Patients were treated with radiotherapy doses ranging from 40 Gy to 50 Gy. The median follow-up was 12 months (range, 3-175 months). During follow-up only one patient had progressive disease and died of Castleman disease. At the time of last follow-up two patients were in complete remission, one patient in partial remission, and one patient had stable disease. One patient showed serious acute and late toxicities. At the end of radiotherapy a paraneoplastic pemphigus vulgaris occurred, and eight to 11 months after radiotherapy a stenosis of the esophagus, of the left bronchus, and of the trachea due to scars. The study shows that unicentric Castleman disease is successfully treated with radiotherapy. However, for detection of possible complications as pemphigus vulgaris or stenosis of the esophagus or trachea an accurate follow-up is necessary.

Adult↗
PubMed2003

Preoperative embolization as an adjunct to the operative management of mediastinal Castleman disease.

Castleman disease usually presents in children as a localized mass with prominent feeding vessels. The mainstay of treatment of Castleman disease is surgical resection; historically, resection is associated with excessive blood loss. These tumors are well known to have large feeding vessels and, thus, are amenable to preoperative arteriography with embolization. The authors present a case of Castleman disease treated with preoperative embolization as an adjunct to operative management.

Castleman Disease↗
PubMed2005

[Retroperitoneal Castleman disease].

Castleman disease or angiofollicular lymph node hyperplasia is a rare disease of unknown cause with polymorphic clinical features that raises many diagnostic and therapeutic problems. The unifocal, localized, pseudoneoplastic form, first described in 1956, has a good prognosis, but the multifocal form has a more aggressive course. Three histological types have been identified: hyaline vascular, plasma cell and mixed. The authors present a case of hyaline vascular unifocal Castleman disease. A retroperitoneal mass was discovered incidentally during follow-up ultrasound examination in a 38-year-old woman treated for recurrent renal stones. On CT examination, a solid mass measuring 9 x 8 x 6 cm raised a problem of differential diagnosis with other retroperitoneal masses. CT-guided biopsy was inconclusive and the diagnosis was finally established by tumour excision and histological examination. The immediate and subsequent postoperative course was uneventful.

Adult↗
PubMed2005

[Castleman disease].

Castleman disease or angiofollicular hyperplasia is a rare disorder included in the group of lymphoproliferative disorders. This entity was originally described by Castleman in 1956. The etiology remains unknown but it is postulated to be a reactive lymphoid hyperplasia due to chronic antigenic stimulation caused by a viral infection. The disease presents in young adults and is more frequent in women; it is exceptionally rare in the pediatric age group. It is classified into two clinical groups (localized disease and disseminated disease) and there are two histologic variants (hyaline-vascular and plasma cell Castleman disease). Localized disease is usually asymptomatic, has a good prognosis, and is the most common presentation in pediatric patients, usually corresponding to highly vascularized mediastinal masses. Resection of the mass, which is curative, is associated with a high risk of blood loss. Recently, preoperative arteriography with embolization has been used satisfactorily in the preoperative management of these tumors. We present a case of localized Castleman disease in a 12-year-old girl satisfactorily treated with embolization before curative resection.

Castleman Disease↗
PubMed2005

Laparoscopic treatment of an uncommon abdominal localization of Castleman disease.

Castleman disease is a rare lymphoproliferative disorder overall localized in the mediastinum and rarely in the abdomen. It appears as a tumor-like mass characterized by a massive growth of lymphoid tissue. Benign forms are usually associated to a good prognosis even if multifocal variants present more aggressive behavior. Two different histologic types have been described: the hyaline vascular and the plasma cell form. The diagnosis is often achieved only at the histologic evaluation of the surgical specimen. Presented here is the rare occurrence of this disease in the abdominal cavity treated by the laparoscopic approach. No postoperative complications were observed. No recurrence has been detected at 12 months CT scan follow-up. Until now, no reports of this kind of treatment have been available in literature.

Abdomen↗
PubMed2003

Acute myelogenous leukemia M5b developed during clinical remission of Castleman disease.

Castleman disease (CD) is a rare heterogeneous lymphoproliferative disease characterized by clinical symptoms due to an excess of interleukin-6 (IL-6) or IL-6-like activity. We describe the first case of CD associated with acute myelogenous leukemia (AML). A 55-year-old man presented with skin rash on his face and multiple cervical lymphadenopathy. The results of examination of his lymph node biopsy specimen led to a diagnosis of CD. The symptoms resolved after the administration of prednisolone. Three years after the onset of CD, the patient's white blood cell count had increased to 63.4 x 10(9)/L. His bone marrow aspirate showed that approximately 80% of cells were leukemic, including well-differentiated monocytic cells A diagnosis of AML M5b was made. The patient died of invasive pulmonary aspergillosis after chemotherapy.

Antineoplastic Agents, Hormonal↗
PubMed1999

[Localized Castleman disease. Diagnostic difficulties in a surgically treatable disease].

Castleman's disease is a rare cause for a retroperitoneal mass. The disorder can be classified into two histopathological groups: the hyalin-vascular and plasma-cell types. The former type, particularly in its localized form, is often asymptomatic and benign, the surgical therapy may cure the patient. The latter type is frequently associated with systemic manifestations and an uncertain prognosis. We present a case in which the diagnosis of localized Castleman's disease proved to be extremely difficult. In these cases, the correct diagnosis, however, is indispensable, as these patients can be cured by radical surgical tumor excision.

Adult↗
PubMed1998

The clinical behavior of localized and multicentric Castleman disease.

BACKGROUND: Castleman disease, an unusual condition of unknown cause consisting of a massive proliferation of lymphoid tissue, remains a clinicopathologic diagnosis. Three histologic variants (hyaline vascular, plasma-cell, and mixed) and two clinical types (localized and multicentric) of Castleman disease have been described. OBJECTIVE: To analyze the clinical features, management, and outcome of patients with Castleman disease. DESIGN: Case series. SETTING: University referral hospitals. PATIENTS: All patients with Castleman disease who were seen at Texas Medical Center, Houston, Texas, between 1977 and 1995. INTERVENTIONS: Surgical excision for localized disease; surgery, combination chemotherapy, or prednisone for multicentric disease. MEASUREMENTS: Patients were identified according to initial presentation as having localized or multicentric Castleman disease. Patients within each group were further subdivided according to whether they had hyaline vascular, plasma-cell, or mixed disease. RESULTS: Data from 15 patients were analyzed. All 7 patients with localized disease underwent surgical excision and remain free of disease. The 8 patients with multicentric disease were further subdivided according to initial treatment: Three patients who received combination chemotherapy are currently alive and free of disease; 2 patients treated with prednisone are alive but have needed intermittent maintenance therapy for disease reactivations; and 2 patients treated with surgery only have died, 1 of infectious complications and 1 of non-Hodgkin lymphoma. CONCLUSIONS: Localized and multicentric Castleman disease are different clinical disorders with overlapping histologic features. Localized disease can be cured with surgery, but complete remissions in patients with multicentric disease have been achieved only with chemotherapy or prednisone given at the time of diagnosis.

Adolescent↗
PubMed2004

Peripancreatic Castleman disease.

CONTEXT: Castleman disease or giant lymph node hyperplasia is a rare disorder of the lymphoid tissue, usually occurring in the mediastinum. We report a case of localized peripancreatic plasma cell type Castleman disease, which is an unusual site. CASE REPORT: A 45-year-old woman with a history of mild epigastric pain radiating to the back for the previous year was admitted and her physical examination was normal. A raised erythrocyte sedimentation rate, C-reactive protein and hypergammaglobulinemia were found. Abdominal ultrasonography and magnetic resonance imaging demonstrated a homogeneously hypoechoic solid mass having a smooth-surface, 6 cm in diameter, located between the head of the pancreas, the lower margin of the antrum and the left lobe of the liver. The mass was totally excised surgically with repair of the pancreas capsule. Histopathological examination of the tumor showed plasma-cell type Castleman disease. CONCLUSION: Clinicians should be aware that Castleman disease may involve peripancreatic tissue which leads to difficulties in arriving at a differential diagnosis. Surgical excision is both a diagnostic and a curative method for management of the disease.

Blood Sedimentation↗
PubMed2002

High incidence of Kaposi sarcoma-associated herpesvirus-related non-Hodgkin lymphoma in patients with HIV infection and multicentric Castleman disease.

Multicentric Castleman disease (MCD) is a distinct type of lymphoproliferative disorder associated with inflammatory symptoms and interleukin 6 (IL-6) dysregulation. In the context of human immunodeficiency virus (HIV) infection, MCD is associated with Kaposi sarcoma-associated herpesvirus, also called human herpesvirus type 8 (KSHV/HHV8). Within a prospective cohort study on 60 HIV-infected patients with MCD, and a median follow-up period of 20 months, 14 patients developed KSHV/HHV8-associated non-Hodgkin lymphoma (NHL): 3 "classic" KSHV/HHV8(+) Epstein-Barr virus-positive (EBV(+)) primary effusion lymphoma (PEL), 5 KSHV/HHV8(+) EBV(-) visceral large cell NHL with a PEL-like phenotype, and 6 plasmablastic lymphoma/leukemia (3/3 KSHV/HHV8(+) EBV(-)). The NHL incidence observed in this cohort study (101/1000 patient-years) is about 15-fold what is expected in the general HIV(+) population. MCD-associated KSHV/HHV8(+) NHL fell into 2 groups, suggesting different pathogenesis. The plasmablastic NHL likely represents the expansion of plasmablastic microlymphoma from the MCD lesion and progression toward aggressive NHL. In contrast, the PEL and PEL-like NHL may implicate a different original infected cell whose growth is promoted by the cytokine-rich environment of the MCD lesions.

Antiviral Agents↗
PubMed2005

Humanized anti-interleukin-6 receptor antibody treatment of multicentric Castleman disease.

Multicentric Castleman disease (MCD) is an atypical lymphoproliferative disorder characterized by systemic lymphadenopathy and constitutional inflammatory symptoms. Dysregulated overproduction of interleukin-6 is responsible for the clinical abnormalities. This multicenter prospective study was undertaken to evaluate the safety and efficacy of a humanized anti-human interleukin-6 (IL-6) receptor monoclonal antibody (MRA) in patients with MCD. We report here results of the first 60 weeks of the study enrolling 28 patients. The initial dosing period consisted of 8 infusions of 8 mg/kg MRA administered biweekly. Adjustments in the dose and treatment interval were allowed for each patient in an extension phase after 16 weeks. Within 16 weeks, treatment with MRA consistently alleviated lymphadenopathy and all the inflammatory parameters. Hemoglobin, albumin, and total cholesterol levels, high-density lipoprotein cholesterol values, and body mass index all increased significantly. In addition, fatigue diminished. Chronic inflammatory symptoms were successfully managed over 60 weeks. In 8 (28.6%) patients, the MRA dose was decreased or the treatment interval was extended without exacerbation. Eleven (73.3%) of 15 patients who had received oral corticosteroids before study entry were able to do well on a reduced corticosteroid dose. Most adverse events were mild to moderate in severity. MRA was tolerated well and significantly alleviated chronic inflammatory symptoms and wasting in patients with MCD.

Adult↗
PubMed1997

Castleman disease in pregnancy.

BACKGROUND: Castleman disease, a rare disorder characterized by benign proliferation of lymphoid tissues, generally presents as a solitary mediastinal mass. We report the first case of this disease during pregnancy. CASE: A 32-year-old woman presented with a large abdominal mass and vaginal bleeding during the second trimester of pregnancy. Abdominal ultrasound demonstrated a large, retroperitoneal solid mass of mixed echogenicity and increased vascularity. The patient underwent explorative laparotomy that revealed a mesenteric mass, histologically consistent with Castleman disease of the hyaline-vascular type. The mass was excised completely, and the immediate postoperative course was uneventful, although the patient went into spontaneous preterm labor during the 29th week of pregnancy. CONCLUSION: Castleman disease should be considered one of the benign etiologies for an abdominal or retroperitoneal mass during pregnancy.

Adult↗
PubMedDate not supplied

[Castleman disease of the neck].

UNLABELLED: Castleman Disease is a rare disorder characterized by benign lymph node hyperplasia involving lymphatic tissue in the neck, mediastinum, abdomen, axilla. There are three types of Castleman Disease: hyalin-vascular type localized nodal involvement, asymptomatic form; plasma cell type and generalized and multicentric type. MATERIAL AND METHOD: Between 2000-2004, 5 cases with Castleman Disease hyalin-vascular type with cervical lymph node involvement were diagnosed and treated in E.N.T. Department of "St. Spiridon" Hospital Iaşi. 2 of these cases are presented: a 24 year old male and a 13 year old female were admitted with a asymptomatic neck mass with slow progressive growth. Paraclinical exams (radiology, cervical and abdominal echography, CT scanner) confirmed the lymphadenopathy and excluded another localization. Differential diagnosis included tuberculosis, metastatic lymphadenopathy, lymphoma, branchial cyst etc. RESULTS: The treatment of choice was surgical excision by cervicotomy in all cases, followed by vascular proliferation and hyalinization in affected lymph nodes. Postoperative controls no revealed recurrences. CONCLUSIONS: The prognosis of localized form of Castleman Disease hyalin-vascular tape is better than multicentric form surgical excision is both diagnostic and therapeutic.

Adolescent↗
PubMed1994

Case report: Castleman disease in association with POEMS.

Castleman disease, or angiofollicular lymph node hyperplasia, and POEMS (Polyneuropathy, Organomegaly, Endocrinopathy, Monoclonal gammopathy, and Skin changes), are associated and can lead to a clinical conundrum. The physician caring for a patient with Castleman disease should be alert to the development of multiple endocrine deficiencies, including primary hypogonadism, diabetes mellitus, hypothyroidism, and adrenal insufficiency. Avoidance of treating hypothyroidism alone when there is concomitant subclinical adrenal insufficiency is important, to avoid precipitating an adrenal crisis. A better outcome may result from earlier recognition of the endocrinopathies of this syndrome. This article describes a patient with Castleman disease in whom the features of POEMS unfolded over the ensuing years.

Adrenal Cortex Hormones↗
PubMed2005

[Castleman disease in the pelvic cavity].

Although Castleman disease may occur in any lymph node of the body, it is the most commonly found in the thoracic cavity. Castleman disease rarely occurs in the pelvic cavity and only 8 such cases have been reported previously in Japan. In this report, we describe a case of Castleman disease in the pelvic cavity. A 36-year-old man complained of fever. Both serum c-reactive protein (CRP) and serum interleukin-6 (IL-6) were elevated. Computed tomography and magnetic resonance imaging showed a solid mass with calcification in the pelvic cavity. The pelvic mass was surgically excised. The removed mass was solid and 75 x 45 x 30 mm in size. The histological diagnosis was the plasma cell type of Castleman disease. Fever subsided and both serum CRP and serum IL-6 were normalized after the operation. No evidence of disease was detected one year post operation.

Adult↗
PubMed2005

Human herpesvirus 8 and Epstein-Barr virus coinfection in localized Castleman disease during pregnancy.

Castleman's disease is a rare disorder characterized by two distinct entities with similar histology but different time course and therapeutic response. Multicentric plasma cell variant is highly associated with infection by human herpesvirus 8 (HHV-8), but the pathogenesis of the hyaline vascular variant is currently unknown. We report a pregnant patient who develops a localized axillary hyaline-type Castleman's disease in which HHV-8 DNA sequences were detected in the lymph node lesions by nested PCR. In addition, the PCR multiplex also showed positivity for EBV. Immunohistochemical studies confirmed the presence of both viruses. Our results provide the first evidence of the presence of HHV-8 and EBV sequences in localized Castleman's disease, suggesting a possible role of the association of these herpes virus in the pathogenesis of this type of disorder. This case highlights that searching for HHV-8 and EBV sequences in cases of localized Castleman's disease is strongly advised.

Adult↗
PubMed2002

Pelvic Castleman disease presenting as vaginal occlusion.

BACKGROUND: Castleman disease is a lymphoreticular disorder, which may be clinically silent. However, systemic manifestations can occur, which include generalized adenopathy and life-threatening bronchiolitis obliterans. CASE: A woman presented with vaginal mucosal lesions recalcitrant to initial treatment efforts. Progression of the disease required hospitalization for pulmonary involvement. Castleman disease was diagnosed after extensive multidisciplinary evaluation and excision of a pelvic mass. The patient's symptoms dramatically improved postoperatively. CONCLUSION: Castleman disease should be included in the differential diagnosis in patients with paraneoplastic symptoms and a pelvic mass.

Castleman Disease↗