In February 2011 the 26-year-old male patient ("the case"), a never smoking professional soldier, was referred to the Department of Pneumology and Allergy with disseminated lung parenchymal changes. His chest X-ray was performed as part of a routine medical check-up before admission (Figure 1A). A previous X-ray taken 1 year earlier was normal. After his X-ray had been evaluated, he was admitted to the pulmonary unit of a district hospital, where lung CT scans were performed (Figure 1B). Bronchial aspirate examined for the presence of Mycobacterium tuberculosis taken at this time, both direct smear (Ziehl-Neelsen stain) and in culture (Lowenstein-Jensen medium) were negative. His tuberculin skin test (TST) was also negative (0 mm). Based on the clinical and radiological picture, stage II sarcoidosis was suspected, and he was referred to the Department of Pneumology and Allergy, Medical University of Lodz for further diagnosis. Although BAL fluid cellular pattern and laboratory results were not typical for sarcoidosis (Table 1), transbronchial lung biopsy (TBLB) revealed non-caseating granulomas (Figure 1C), confirming the initial diagnosis. Due to asymptomatic course, normal lung function and lung diffusing capacity for CO (DLCO) and lack of extra-pulmonary locations, the decision of treatment was suspended and the patient was asked to visit the out-patient pulmonary clinic within 3 months. In the meantime, 8 weeks after the collection, BAL fluid culture for Mycobacterium tuberculosis appeared positive. He was referred to a regional tuberculosis clinic and anti-TB treatment composed of isoniazid, rifampicin, pyrazinamide, and ethambutol was started according to directly observed treatment short-course strategy.
The household members (the case's mother and twin brother) were asked to visit a regional TB out-patient clinic for routine examination of household contacts. Chest X-ray revealed disseminated parenchymal changes with enlarged hili in both family members. The previous X-rays of the twin brother and mother had been performed 3 months and a year ago, respectively, and were both normal. The chest X-ray of the case's older brother, who was living separately, was normal. Therefore, the mother and twin brother were referred to the Department of Pneumology and Allergy for further diagnosis. Chest CT scans confirmed the presence of bilateral hilar enlargement and disseminated nodular changes and thickening of bronchovascular bundles with predominance of upper and middle zones (Figures 2, and 3A). BAL cellular pattern was typical for sarcoidosis (Table 1) in both. TBLB of the case's twin brother was non-diagnostic (normal lung structure) and he refused further invasive examinations. The mother's TBLB showed non-ca-seating granulomas (Figure 3B). BACTEC examination of BAL fluid was negative in both the case's mother and twin brother. Due to asymptomatic course, well-preserved lung function, and lack of extrapulmonary symptoms, they were left untreated. Anti-TB treatment was also abandoned.
At a follow-up in November 2011, the chest X-ray of the case was not different from the initial X-ray, but lung HRCT done in March 2012 showed substantial regression of parenchymal changes and sustained slightly enlarged mediastinal lymph nodes (picture not shown). Chest X-ray of the case's twin-brother performed in November showed complete regression of the previously described changes. The mother's chest X-ray performed at this time showed neither progression nor regression. Laboratory markers (SACE, CRP, serum calcium, liver function tests, and peripheral blood count) were within normal limits in all 3 patients. Twenty-four hour urinary calcium loss was increased in the mother but was normal in both twin brothers. Blood for QuantiFERON-TB testing was taken in November 2011 and the results were negative in the whole family. MTB were not found in lung biopsies of any family members (Ziehl-Neelsen stain).
Radiological and histopathological results of thecase's mother;. CT-scan showing enlarged hilar lymph nodes, and ,disseminated micronodular changes.