Castleman's Disease: An Intrapulmonary Form with Intrafissural Development

Abstract

Castleman's disease (CD) is an uncommon, mainly benign, lymphoproliferative disorder of unknown etiology, mostly involving the mediastinum. Parenchymal lung involvement of the disease is exceedingly rare. We describe a case of CD in a 23-year-old woman with a 4-year history of recurring dyspnea and nonproductive cough, whose chest X-ray showed an abnormal shadow of the right hilum. Chest computed tomography confirmed the presence of a tissue-density mass of the right lower lobe, demonstrating poor contrast enhancement, associated with multiple laterotracheal and mediastinal lymphadenopathies. The patient underwent curative surgery, revealing a right hilar compressive mass, with an intrafissural development between the superior and middle lobes. Pneumonectomy was performed due to profuse bleeding. This case of CD is particular because of its unusual intrapulmonary location and its intrafissural development. Poor contrast enhancement is atypical in CD.

Castleman’s disease- a diagnostic dilemma

“…Surgical excision may not be easy in unicentric hyaline vascular type due to high vascularity. It may be associated with massive haemorrhage at excision and pneumonectomy has been reported for massive bleeding [7]. Embolization of the feeding artery before surgery can be considered to prevent intraoperative bleeding.…”

Isolated Intrapulmonary Castleman’s Disease: A Case Report, Review of the Literature

“…CD is clinically classified into unicentric (localized) and multicentric (systemic) and then further subdivided histologically into the hyaline vascular (90% of cases), plazma cell (8%-9% of cases), and mixed types (1%-2% of cases). [1][2][3][4] The localized type is a rare form and usually detected incidentally in asymptomatic cases as in our case.…”

“…2,5) In the diagnosed cases, preoperative arterial embolization may reduce the intraoperative bleeding. 4) Limited resection may be tried in the appropriate cases. In our case, mass was located in the central of the upper lobe, and the branches of upper lobe pulmonary artery were invaded by the mass Thus, we preferred the anatomically resection.…”

“…The value of the PET/CT in the preoperative diagnosis is low because it is inadequate to differentiate the malign diseases and benign diseases. [2][3][4][5] Castleman disease can be a cause of false-positive findings by mimicking metastatic adenopathy. The diagnosis is very difficult in these patients, and a thoracotomy is usually needed for the diagnosis and treatment as our case.…”

Intrapulmonary Castleman’s Disease Pretending to Be a Lung Cancer—Work Up of an Intrapulmonary Tumour

“…Intrapulmonary manifestation of CD is even less common, as only 6 cases have been reported in the literature. [1][2][3][4][5][6] CD histologically presenting as the plasma cell type (95%) is associated with severe systemic symptoms such as weakness (81%), fever (71%), weight loss (58%), anorexia and nausea (42%) and signs as splenomegaly (79%), hepatomegaly (63%), oedema and effusion, hypergammaglobulinemia, anaemia and multicentric lymphadenopathy. 9) The localized variant mostly presents as the hyalinevascular type with typical onion-skin arrangement of the lymphatic tissue.…”