A 55-year-old woman with cough-variant asthma presented for 1 month of worsening wheezing, cough, and dyspnea refractory to treatment. Initial laboratory findings revealed profound peripheral eosinophilia, and a chest computed tomography showed bi-apical consolidation. Bronchio-alveolar lavage demonstrated alveolar eosinophilia. She was diagnosed with idiopathic chronic eosinophilic pneumonia (ICEP). Her peripheral eosinophilia and respiratory symptoms improved rapidly with high-dose systemic corticosteroid therapy. However, she was intolerant to corticosteroid monotherapy due to non-compliance and psychological adverse effects. Mepolizumab was initiated as a steroid-sparing agent, resulting in successful therapy for 2 years without relapse or adverse effects. Mepolizumab is an interleukin-5 (IL-5) antagonist monoclonal antibody, which is a targeted therapy for diseases mediated by eosinophil activity and eosinophil proliferation. Mepolizumab is typically used in ICEP refractory to steroids, but this case supports its use in cases of glucocorticoid intolerance. Further study of IL-5 antagonist therapies for ICEP may identify an alternative treatment modality for patients in whom the adverse effects of corticosteroids pose a challenge.
Juvenile idiopathic arthritis (JIA) is the most common cause of arthritis in children. It is characterized by inflammatory cell infiltration of synovial membranes leading to synovitis and synovial membrane thickening. Synovial chondromatosis is rare sequela of synovitis in which foci of cartilage develop within the synovial membrane of a joint capsule. We report a case of a 35-month old boy who developed tumefactive synovial hypertrophy and hyperplasia that mimicked synovial chondromatosis on MRI. The reactive synovium was arthroscopically excised and the patient's arthritis improved. Tumefactive synovial hypertrophy and hyperplasia mimicking synovial chondromatosis on MRI in the setting of JIA, has not been reported in this young of a patient in the literature to date. A discussion on imaging in oligoarticular JIA and synovial chondromatosis is presented herein.
A woman in her sixth decade presented with several months of abdominal cramping, decreased appetite, bloating, and increased constipation. Radiologic imaging revealed a 28 cm, multilocular, heterogeneous cystic neoplasm involving the right adnexa. An intraoperative frozen section showed mucinous glandular epithelium, with and without foci of goblet cells, embedded in apparent ovarian stroma. The findings were concerning at least borderline mucinous cystadenoma with possible invasion. Subsequent surgical management and staging were performed. Permanent sections showed a moderately to poorly differentiated Sertoli-Leydig cell tumor (SLCT) with retiform foci and heterologous elements. The discrepancy between frozen and permanent sections was attributable to solely sampling a focus of heterologous elements during intraoperative consultation. The rarity of SLCT and even rarer presence of both heterologous and retiform elements make this concerning frozen section diagnostic pitfall.
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