In this very rare case of 43 years-old female, refugee from Kosovo, was admitted as an outpatient to the Department of pulmonary diseases in 2003. because of erythema nodosum and dry cough. She was without fever during admission, smoked up to 30 cigarettes daily and was healthy most of her life. She suffered only of head injury and face trauma acquired during the war (during 1995.) by shelling. Three years before the outbreak of erythema nodosum, two small red subcutaneous tubercles were found on her forehead, in the left eyebrow area. The biopsy test showed chronic granulomatous infection that was described as a reaction to a foreign body.
Laboratory analysis revealed ESR 37 (normal range 0-15 mm/h), WBC 9.030/mm3, RBC 3,7/mm3 and C-reactive protein 9.8 mg/L, AST 60 U/L, ALT 76 U/L, GGT 86 U/L, AP 79 U/L, while urea, creatinine, calcium in serum and 24 hours urine were within normal range. Angiotensin-converting enzyme (ACE) was elevated 108 U/l (normal 0-56). Tuberculosis test (2 IU) was 14 mm of induration in diameter. The result of spirometry measured 85% vital capacity (VC), 94,5% of forced vital capacity (FCV), 77% of forced expiratory volume (FEV),82,4% of forced expiratory volume in first second (FEV1) and FEV1/FCV 87,2% from referral values. Arterial blood gases were pO2 9,8 kPa, pH 7.45 and diffusing capacity of carbon monoxide (DLCO) 72%. Chest radiogram showed fibroindurative changes in the area of both upper lobes with lung traction. In upper lobes subpleurally and more to the left there was a focus with <<round glass>> pattern. Mycronodular interstitial infiltrations were found bilateral on chest radiogram with fibrous changes of hilus (Figure 1). High-resolution computed tomography (HRCT) of thorax showed interlobar and peribronchial thickness in upper lung lobes with subpleural fibroses and deformation of bronchi. There were minor areas of fresh infiltrations in apices on both sides, suggesting a new pathological process. There was a cavity 8 x 12 mm large in apical-posterior segment of the upper left lobe. Hilar lymph nodes were more then 2 cm in diameter, some of them had fibrous changes and some were calcificated. High resolution thorax CT with hilar lymph nodes enlargement and granulomas with reticular interstitial pattern in the lower respiratory part is present on the Figure 2(a, b). Fiberbronchoscopy revealed slightly edematous and hyperemic mucous membrane of bronchi on both sides, while purulent secrete was seen in bronchus left upper lobe and in lingul. The routine bacteriological culture of bronchial aspirate was sterile. Direct smear microscopy examination of the sample for the presence of acid-resistant bacilli by using Ziehl-Neelsen method was negative. Pathohistological findings of lung (Figure 3) and bronchial walls biopsy showed chronic granulomatous infection without caseous necroses. In bronchoalveolar lavage (BAL) CD4+ lymphocytes and alveolar macrophages were predominantly. The ratio of CD4+ and CD8+ lymphocytes was 5.5:1. All relative findings, including pathohistological examination indicate lung sarcoidosis. The patient was stared corticosteroid therapy (out of hospital-out-hospital patient) according to usual scheme for lung sarcoidosis, prednisolone 1 mg/kg body weight. Three weeks after introducing corticosteroid therapy, culturing the sample on Lowenstein-Jensen solid medium by using liquid culture method MGIT (Mycobacterium Growth Indicator Tube), Mycobacterium tuberculosis was isolated. The sensitivity test was performed by using the proportion method on solid Lowenstein-Jansen culture according to CLSI (Clinical and Laboratory Standards Institute) standards. All the four routine tested antituberculotics (isoniazid, rifampicin, ethambutol and streptomycin) responded well to sensitivity test and the susceptibility of pyrasinamide was confirmed in the referral national laboratory. A previous diagnosis was corrected to microbiological confirmed lung tuberculosis. Corticosteroid therapy was excluded after three weeks of treatment and antituberculotic therapy (ATL) was introduced. The patient was treated with rifampicin 600 mg, isoniazid 400 mg, etambuthol 1200 mg and pyrazinamide 1500 mg, on a daily basis during two months, after pyrazinamide was excluded and triple therapy was continued for one month. After three months from the beginning of ATL treatment, ethambuthol was excluded and rifampicin and isoniazid were continued for the next 3,5 months (entirely 6,5 months) with controlling regular hematological and biochemical analysis. The patient was in good clinical condition. However, ACE was continuously raised, 80-100 U/l. After four months of ATL therapy chest radiogram was made showing complete regression of tiny infiltrates in both upper lobes and small regression of nodular infiltrates which was described as unsatisfactory. During ATL therapy new nodes on face and nose, similar to those on the forehead appeared (Figure 4). The biopsy of skin changes was made and the pathohistological confirmation for sarcoidosis is evident on the Figure 5. Antituberculotic therapy was preceded for next two months until negative mycobacterium culture in new collected sputum was obtained. In course of the last month of TB therapy novel bigger, firm nodes appeared on ear lap. Pathohistological tests indicate sarcoidosis of the skin.
Microbiological examinations of the skin substrata in direct microscopy and polymerase chain reaction (PCR) with specific primer had not confirmed presence of M. tuberculosis. After 6 months of the beginning of ATL treatment, the patient received corticosteroid (prednisolone 40 mg daily) and colchicine therapy (200 mg daily) for lung and skin type of sarcoidosis. After four months of the corticosteroid therapy chest radiogram showed almost complete regression of nodular infiltrates in lung parenchyma. The skin changes were considerably smaller, dry on surface, and there were no new lesions on the skin while ACE was in normal rate (30 U/l).
Thorax CT with hilar lymph nodes enlargement.