Clinical spectrum of intrathoracic Castleman disease: a retrospective analysis of 48 cases in a single Chinese hospital

Luo, Jin Mei; Li, Shan; Huang, Hui; Cao, Jian; Xu, Kai; Bi, Ya Lan; Feng, Rui; Huang, Cheng; Qin, Ying; Xu, Zhigang; Xiao, Yi

doi:10.1186/s12890-015-0019-x

Cited by 39 publications

(44 citation statements)

References 43 publications

(90 reference statements)

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“…But this ratio is much lower than the ratio reported in HIV-positive patients (Table 2). On the contrary, a study from China found female preponderance in CD [8]. The reasons for lower male to female ratio in our study and female predominance in a study from China remain unclear.…”

Section: Discussioncontrasting

confidence: 99%

“…Therefore, many of the HIV positive patients die of other complications before developing uncommon neoplasm-like CD, and this phenomenon can create an impression that CD is uncommon in Asian patients suffering from HIV. Our series and the data from China [8] also reflect the same as none of the patients with CD reported in these studies was positive for HIV. A pertinent question can arise by this observation.…”

Section: Discussionsupporting

confidence: 83%

“…When we compared baseline characteristics between the two subtypes of CD, cases with MCD had a median age of presentation about a decade higher than in the UCD group. A similar difference was also observed by Table 2 Comparative summarisation of prominent large case series published in the recent past Mylona et al [1] Gerard et al [6] Bower et al [10] Luo et al [8] Luo et al [8], who noted median age for UCD at 30 years and for MCD at 41 years. A strong predilection of CD for male patients was reported in earlier studies with M:F ratio as high as 9:1 [1,6,17].…”

Section: Discussionsupporting

confidence: 70%

“…Significantly higher number of patients with MCD had plasmas cell variant (p = 0.03). Luo et al also showed similar trends in differences in between UCD and MCD [8].…”

Section: Discussionmentioning

confidence: 61%

“…1). For the plasma cell Castleman's disease (PCCD) variant, retained lymph node architecture, variable germinal center hyperplasia, with expanded mantle zones with marked plasmacytosis was recorded [8,9]. On the basis of the extent of organ involvement, two subtypes of CD were classified.…”

Section: Case Definitionsmentioning

confidence: 99%

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Presentation and Outcome of Castleman’s Disease in Immunocompetent Hosts

Prakash¹,

Bal

Malhotra³

et al. 2015

Indian J Hematol Blood Transfus

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Castleman's disease (CD) is a heterogeneous lymphoproliferative disorder of unknown aetiology. Mostly, this disorder is seen in immunocompromised hosts. It is known to be associated with systemic disorders like HIV, HHV-8, lymphoma, and Kaposi sarcoma. As of today, the clinical behaviour and outcome of CD in immunocompetent host remains suboptimally studied. We analyzed consecutively treated cases of CD presented to our centre in last 12 years. Case record files were studied for patient's characteristics, clinical presentation, baseline laboratory and pathologic parameters, therapy and outcome. This study describes presentation and treatment outcome of CD in immunocompetent patients. Total 16 patients of CD were treated during the study period. The median age of patients at the time of presentation was 40.5 years (range 13-72 years). An equal number of patients (8 each) had unicentric and multicentric CD. Sixty-three percent patients had hyaline vascular subtype while 37 % patients had plasma cell or mixed variant.Majority of the patients had good performance status (ECOG PS 0, 1 in 10 (62.5 %) patients; PS2-4 in 6 (37.5 %) patients). The median duration of symptoms was 6 months (range 2-36 months). None of the patients in our study had associated HIV infection. Six patients presented with fever, out of which four had plasma cell variant of CD and three of them had multicentric involvement. In comparison to unicentric CD, patients with multicentric CD had lower albumin levels (4.15 vs. 3.38 g/dl, p = 0.006), haemoglobin levels (11.3 vs. 9.8, p = 0.06), and lower complete remission rates (62.5 % vs. none). Patients were treated according to the stage and clinical status with surgery, chemotherapy or combination of both modalities. Surgery was the predominant treatment for unicentric CD while multicentric CD was treated with various chemotherapy regimens. Eight patients were treated with chemotherapy (CHOP-based regimen-5, melphalan-prednisolone thalidomide-1, chlorambucil-prednisolone-1, and only corticosteroids-1). In the entire study group, ORR was 72 % (CR 36 %, PR 36 %), one patient died of progressive disease during chemotherapy. Two patients lost to followup before assessment of treatment response. Five-year overall survival was 100 and 87 % for unicentric and multicentric CD respectively. Castleman's disease is a rare lymphoproliferative disorder amongst Indian patients. It more commonly presents as hyaline vascular variant and affects middle-aged individuals. The most remarkable fact in our analysis was a lack of HIV positivity in any of the patient that is in contrast to the majority of the published literature. With multimodality therapy, high response rates and long-term survival were noted in the entire study group.

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Section: Discussioncontrasting

confidence: 99%

Section: Discussionsupporting

confidence: 83%

Section: Discussionsupporting

confidence: 70%

“…Significantly higher number of patients with MCD had plasmas cell variant (p = 0.03). Luo et al also showed similar trends in differences in between UCD and MCD [8].…”

Section: Discussionmentioning

confidence: 61%

Section: Case Definitionsmentioning

confidence: 99%

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Presentation and Outcome of Castleman’s Disease in Immunocompetent Hosts

Prakash¹,

Bal

Malhotra³

et al. 2015

Indian J Hematol Blood Transfus

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Differential Diagnosis (2): Castleman Disease

Zoshima

Kawano

Hara

et al. 2016

IgG4-Related Kidney Disease

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Intrathoracic tumor of the chest wall: A case of Castleman’s disease mimicking myositis of the lower extremities

Tampakis

Tampaki

Daikeler

et al. 2017

Gen Thorac Cardiovasc Surg

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Castleman's disease refers to a group of uncommon lymphoproliferative disorders which exhibit common lymph-node histological features. A 72-year-old male patient presented with signs of lower limb myositis. Detailed work-up focused initially on evaluating hematological malignancies, the presence of a solid tumor, autoimmune diseases and degenerative disorders of the peripheral nerves. Finally, a PET-CT scan was performed to exclude paraneoplastic manifestations of a primary tumor, revealing however a tumor of the thoracic wall. The definite histological diagnosis confirmed the presence of unicentric Castleman's disease of the chest wall. The manifestations of the present case suggest that a systemic inflammation might occur in the unicentric form of the disease possibly due to cytokine hypersecretion. The unicentric manifestation of the disease should be well distinguished from the multicentric appearance. Unicentric disease is a surgical condition and warrants a follow-up based on the systemic inflammation that might occur.

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Clinical spectrum of intrathoracic Castleman disease: a retrospective analysis of 48 cases in a single Chinese hospital

Cited by 39 publications

References 43 publications

Presentation and Outcome of Castleman’s Disease in Immunocompetent Hosts

Presentation and Outcome of Castleman’s Disease in Immunocompetent Hosts

Differential Diagnosis (2): Castleman Disease

Intrathoracic tumor of the chest wall: A case of Castleman’s disease mimicking myositis of the lower extremities

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