Amyloidosis secondary to intrapulmonary Castleman disease mimicking pulmonary hyalinizing granuloma-like clinical features

Abstract

Rationale: Amyloidosis secondary to intrapulmonary Castleman disease (CD) is a rare benign disease diagnosed by histopathology. It seems to be associated with chronic inflammation, and large amounts of IL-6 produced in the germinal center of CD may enhance the production of precursor of amyloid. Patient concerns: We reported a case of an 18-year-old woman presenting with dry cough and dyspnea on exertion for 6 months and detailed exams revealed multiple pulmonary nodules, positive antinuclear antibodies, hypocomplementemia, and thrombocytopenia. Diagnoses: A computed tomography-guided percutaneous lung biopsy revealed the histopathological features of pulmonary hyalinizing granuloma (PHG), but video-assisted pulmonary wedge resection for biopsy with immunohistochemical stains finally demonstrated a corrected diagnosis of intrapulmonary CD with secondary amyloidosis. Interventions: The patient had received prednisone and Tacrolimus for 6 months. Outcomes: There was no significant improvement in pulmonary lesions or platelet level. Chemotherapy to CD was needed. Lessons: Intrapulmonary CD should be considered in patients with multiple pulmonary nodules irresponsive to corticosteroid and diagnosis of PHG should be carefully considered based on small lung biopsy sample. The treatment of amyloidosis secondary to CD remains to be uncertain.

WITHDRAWN: Lung Involvement Multicentric Castleman of Hyaline-vascular Variant: 3 cases

“…Lung involvement are more common than HV-UCD cases, with cough, sputum, shortness of breath and other respiratory symptoms. Hypoxemia is common in blood gas analysis, and pulmonary function is mixed ventilation disorder [12][13][14][15], suggesting interstitial lung damage and small airway lesions, and the pathological features of BO and lymphocytic interstitial pneumonia have also been con rmed in literature [16]. CT ndings include lymphadenopathy, often with diffuse hilar and mediastinal lymphadenopathy, and pulmonary parenchyma changes, including centrilobular nodules, bronchovesicular bundle and septal thickening, cysts, and pleural effusion.…”

Multicentric Castleman disease of hyaline‑vascular variant with paraneoplastic pemphigus results in abnormal lung function: Report of 3 cases

“…Lung involvement is more common than in HV-UCD, with respiratory symptoms including cough, sputum and shortness of breath. Hypoxemia is commonly found in the blood gas analysis and pulmonary function analysis indicates mixed ventilation disorder ( 15-18 ), suggesting interstitial lung damage and small airway lesions, and the pathological features of BO and lymphocytic interstitial pneumonia have also been confirmed in the literature ( 19 ). CT findings include lymphadenopathy, often with diffuse hilar and mediastinal lymphadenopathy, and pulmonary parenchyma changes, including centrilobular nodules, bronchovesicular bundle and septal thickening, cysts and pleural effusion.…”

Pulmonary Hyalinizing Granuloma: A Rare Cause of a Benign Lung Mass

“…It is also considered to be related to other fibro-sclerosing conditions such as retroperitoneal fibrosis, sclerosing mediastinitis, and sclerosing cholangitis [ 10 ]. Some associations of PHG with lymphoproliferative diseases like lymphoma and Castleman’s disease has also been demonstrated [ 13 ]. More importantly, it can mimic a primary lung cancer or metastatic disease warranting due investigation and management.…”