Streptomyces infection in Cushing syndrome: A case report and literature review
Patient was a 24-year-old man, admitted to hospital due to sudden dyspnea for 3 days with cough and yellowish sputum from almost 7 days before admission. He had fever for 5 days before admission. He was healthy until 50 days before admission and was sent to the tropical city for military service where he had an insect bite. After the bite, swelling and edema of upper limb occurred followed by edema on the face and body, acne and weakness of proximal muscles 2 days later. During 1 week, body edema decreased while muscle weakness, acne and facial edema remained. Acnes spread to the anterior chest and face.\nOn physical examination: He was ill and had a moon face appearance; a lot of papulopustular rashes were seen in the face and anterior of the chest. He was febrile with 39.3°C oral temperatures, respiratory rate: 30/min and pulse rate 115/min, blood pressure 145/95 mmHg. O2 saturation was 86%, White blood cells: 20500/cm, polymorphonuclear leukocyte: 90%, erythrocyte sedimentation rate: 72 mm at 1 h, k: 2.8 mEq/l, pH: 7.52 and he had a decreased sound in left lung with hyper resonance in upper and dullness in the lower portion of the chest, the force of proximal muscle was 3/5. Chest X-ray showed hydropneumothorax [].\nChest computed tomography scans showed pneumothorax, plural effusion in the left side accompanied by lung collapse. Also, there were several lung consolidations with pleural thickening [].\nThoracentesis was done and analysis for protein, lactate dehydrogenase, glucose, PH, cytology, culture and smear was sent. Blood culture was asked because of systemic inflammatory response syndrome.\nPleural fluid was indicative of empyema with glucose 30 mg/dl and pH: 7, so chest tube was inserted and a broad spectrum was administered until smear and culture of pleural fluid being ready. After chest tube insertion, chest X-ray was taken which showed decreased pleural effusion, but lung expansion did not occur. Bronchoscopy was normal. Thoracoscopy was done for decortication of pleural thickening [Figures and ].\nPleural biopsy showed fibrin deposition and chronic inflammatory cell infiltration, with chronic inflammation [].\nA smear of pleural fluid showed gram positive beaded basil and aerobic culture of pleural fluid confirmed Streptomyces with filamentous appearance. Pleural biopsy culture justified Streptomyces too. Culture was negative for acid fast bacilli. Bacteriogram reveal sensitivity to co-trimoxazole, meropenem, ciprofloxacin, cefotaxim, tetracycline and claritromycin and resistance to ampicillin, ceftazidim, cefotaxim and piperacillin-tazobactam. Blood culture was negative for specific organisms.\nDuring hospitalization polyuria occurred. In work-up, hypokalemia and hyperglycemia happened. Because of symptom, physical examination and laboratory test, a work-up for Cushing was done.\nUrinary free cortisol was 600 μg/24 h, over-night dexametason suppression test was positive justified Cushing syndrome. Adrenocorticotropic hormone (ACTH) was 180 μg/dl that showed ACTH dependent Cushing syndrome. High-dose dexametason suppression test was done and the suppression of cortisol did not occur, brain magnetic resonance imaging (MRI) did not show adenoma. Rapid progression of symptom and normal brain MRI confirm ectopic ACTH, but no origin was found in the work-up.\nBilateral adrenalectomy was done and histopathology confirmed adrenal hyperplasia without malignancy [].\nEmpirical treatment was converted to meropenem for 6 weeks. He was recommended to use claritromycin for 6 months at the time of discharge.