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

Neuhof, Dirk; Debus, Jürgen

doi:10.1080/02841860600812701

Cited by 41 publications

(44 citation statements)

References 23 publications

(28 reference statements)

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“…Characteristics of unicenter type Castleman's disease are as follows[1]: (1) males and females have the same incidence, with a median age of 37 years; (2) hyaline vascular type is the major pathologic type, accounting for 92%; (3) most lesions are in the mediastinum, neck, axillary, and retroperitoneal lymph nodes, and other rare locations include the intra-orbital region, hepatic portal, subcutaneous region, parotid glands, kidney, and pelvic cavity[2]–[7]; (4) the disease progresses very slowly, intact surgical excision could cure the disease and partial excision could significantly relieve symptoms. However, postoperative adjuvant radiotherapy is recommended due to the possibility of relapse after partial excision[8],[9]. Characteristics of multicenter type Castleman's disease are as follows: (1) males and females have the same incidence, with a median age of 53 years; (2) plasma cell and mixed variant types are the major pathologic types, accounting for 89%; (3) it is mainly treated by chemotherapy with adjuvant local radiotherapy.…”

Section: Discussionmentioning

confidence: 99%

Radiotherapy of unicentric mediastinal Castleman's disease

Li¹,

Liu²,

Zhang³

et al. 2011

Chin J Cancer

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Castleman's disease is a slowly progressive and rare lymphoproliferative disorder. Here, we report a 55-year-old woman with superior mediastinal Castleman's disease being misdiagnosed for a long term. We found a 4.3 cm mass localized in the superior mediastinum accompanied with severe clinical symptoms. The patient underwent an exploratory laparotomy, but the mass failed to be totally excised. Pathologic examination revealed a mediastinal mass of Castleman's disease. After radiotherapy of 30 Gy by 15 fractions, the patient no longer presented previous symptoms. At 3 months after radiotherapy of 60 Gy by 30 fractions, Computed tomography of the chest showed significantly smaller mass, indicating partial remission. Upon a 10-month follow-up, the patient was alive and free of symptoms.

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Section: Discussionmentioning

confidence: 99%

Radiotherapy of unicentric mediastinal Castleman's disease

Li¹,

Liu²,

Zhang³

et al. 2011

Chin J Cancer

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“…Radiotherapy can also achieve clinical response and cure in patients unsuitable for surgery or in patients who have undergone incomplete surgical excision. However, there have been reported possible complications and high recurrence rates [19][20][21][22][23][24].…”

Section: Resultsmentioning

confidence: 99%

“…Authors concluded that in patients with unresectable UCD lesions, radiotherapy will shrink the tumor and eventually lead to regression of the symptoms. Neuhof et al [23] reported the outcome in a small series of five patients with UCD that were managed either with surgery (partial excision) followed by post-operative RT or with RT alone. Patients who had the dual therapy showed no progression of the disease during the follow-up.…”

Section: Resultsmentioning

confidence: 99%

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The Role of Surgical Resection in Unicentric Castleman’s Disease: A Systematic Review

Mitsos

Stamatopoulos

Patrini

et al. 2018

Advances in Respiratory Medicine

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Introduction: Castleman's disease is a rare benign lymphoproliferative disorder of unknown etiology. The disease occurs in two clinical forms with different prognoses, treatments and symptoms: a unicentric form (UCD), which is solitary, localized, and a multicentric form characterized by generalized lymphadenopathy and systemic symptoms. This article aims to review the current literature to consolidate the evidence surrounding the curative potential of surgical treatment to the unicentric type. Material and methods: A systematic review of English-language literature was performed and databases (Medline, Pubmed, the Cochrane Database and grey literature) were searched to identify articles pertaining to the treatment of unicentric form of Castleman's disease. Each article was critiqued by two authors using a structured appraisal tool, and stratified according to the level of evidence. Results: After application of inclusion criteria, 14 studies were included. There were no prospective randomized control studies identified. One meta-analysis including 278 patients with UCD reported that resective surgery is safe and should be considered the gold standard for treatment. Seven retrospective studies enhance this standpoint. Radiotherapy (RT) has been used in six studies with controversial results. Conclusions: We conclude that surgical resection appears to be the most effective treatment for Unicentric Castleman's Disease of the thoracic cavity. Radiotherapy can also achieve clinical response and cure in selected patients.

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“…preoperative embolization to limit intra-operative bleeding and adjuvant radiation therapy in case of incomplete resection (8,9). Radiotherapy has also been offered in a curative intent (10), but the experience remains limited and its long-term efficacy warrants confirmation.…”

Section: CDmentioning

confidence: 99%

Clinical and molecular characteristics of unicentric mediastinal Castleman disease

et al. 2018

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Background: Unicentric mediastinal Castleman disease (CD) is a rare condition, poorly characterized due to the small number of cases and the absence of genomic study. We analyzed clinical, radiological, histological and genomic patterns associated with mediastinal CD in a substantial case series. Methods: We retrospectively reviewed cases of unicentric mediastinal CD managed in 2 French thoracic surgery departments between 1988 and 2012. Clinical, radiological, surgical and pathological data were recorded. On available FFPE blocks we performed mutation screening by next-generation-sequencing, using AmpliSeq™ Cancer Hotspot v2 (Life Technologies) and immunohistochemistry (IHC) (AKT-mTOR pathway). Results: Eleven patients were identified (mean age 41±15 years, sex-ratio 0.8, median follow-up 78 months). Surgical approach was thoracotomy (n=6), sternotomy (n=4), and VATS (n=1). Additional procedures included thymectomy in three cases, mediastinal lymphadenectomy in two cases, and bilobectomy in one case. One patient presented local relapse as a follicular dendritic cell sarcoma, leading to death 48 months after the first resection. Within 9 patients whose FFPE blocks were available, 2 mutations were found: VHL (p.F119L, 35%, n=1) and JAK3 (p.V718L, 53%, n=1). Phospho-AKT and phospho-mTOR stainings were negative in all cases, whereas phospho-S6RP staining was positive in eight cases, mainly in interfollicular cell cytoplasm. Conclusions: From this series of patients with unicentric mediastinal CD, we observed 2 cases of potential driver mutations and 8 cases of phospho-S6RP activation not related to AKT-mTOR. Larger studies are required to decipher more precisely the molecular abnormalities and potential therapeutic targets underlying this uncommon condition.

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

Cited by 41 publications

References 23 publications

Radiotherapy of unicentric mediastinal Castleman's disease

Radiotherapy of unicentric mediastinal Castleman's disease

The Role of Surgical Resection in Unicentric Castleman’s Disease: A Systematic Review

Clinical and molecular characteristics of unicentric mediastinal Castleman disease

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