[Endothoracic localization of the Von Recklinghausen disease. A case report]. / Localizzazione endotoracica della malattia di Von Recklinghausen. Caso clinico.

OBJECTIVE: A case of a 45 years old man affected by the Von Recklinghausen disease with a rare neurofibroma of the thoracic wall is reported. MATERIALS: Starting from 2000 the Authors are using the V.A.T.S. (video assisted thoracic surgery) in their Department of surgery, as minimally invasive approach to diagnosis and treatment of some thoracic diseases: initially the patient was studied by a standard chest x-ray followed by tomography and magnetic resonance to determine the localization of the neoplasm of the chest wall. RESULTS: The neurofibroma was removed by V.A.T.S., through a minimal thoracic access. After treatment we observed the complete remission of symptoms. DISCUSSION: Single localization of the neurofibroma in the Von Recklinghausen disease can transform into a malignant neoplasm. The indication to surgery, in the case presented, was determined by the intense thoracic pain and by the risk of malignant degeneration. The minimally invasive approach showed to be the best option according to the dimensions of the neurofibroma. In the actual literature this approach represents the gold standard in the treatment of small intrathoracic neoplasm. CONCLUSION: In the chest wall localization of the Von Recklinghausen disease the Authors recommend the minimal surgical treatment by using V.A.T.S.

Higher frequency of T-cell response to M. tuberculosis latency antigen Rv2628 at the site of active tuberculosis disease than in peripheral blood.

RATIONALE: Due to the invasive nature of the procedures involved, most studies of Mycobacterium tuberculosis (Mtb)-specific immunity in humans have focused on the periphery rather than the site of active infection, the lung. Recently, antigens associated with Mtb-latency and -dormancy have been described using peripheral blood (PB) cells; however their response in the lung is unknown. The objective of this report was to evaluate, in patients prospectively enrolled with suspected active tuberculosis (TB), whether the latency antigen Rv2628 induces local-specific immune response in bronchoalveolar lavage (BAL) cells compared to PB cells. MATERIAL/METHODS: Among the 41 subjects enrolled, 20 resulted with active TB. Among the 21 without active disease, 9 were defined as subjects with latent TB-infection (LTBI) [Quantiferon TB Gold In-tube positive]. Cytokine responses to Rv2628 were evaluated by enzyme linked immunospot (ELISPOT) assay and flow cytometric (FACS) analysis. RD1-secreted antigen stimulation was used as control. RESULTS: There was a significantly higher frequency of Rv2628- and RD1-specific CD4+ T-cells in the BAL of active TB patients than in PB. However the trend of the response to Rv2628 in subjects with LTBI was higher than in active TB in both PB and BAL, although this difference was not significant. In active TB, Rv2628 and RD1 induced a cytokine-response profile mainly consisting of interferon (IFN)-γ-single-positive over double-IFN-γ/interleukin (IL)-2 T-cells in both PB and BAL. Finally, BAL-specific CD4+ T-cells were mostly effector memory (EM), while peripheral T-cell phenotypes were distributed among naïve, central memory and terminally differentiated effector memory T-cells. CONCLUSIONS: In this observational study, we show that there is a high frequency of specific T-cells for Mtb-latency and RD1-secreted antigens (mostly IFN-γ-single-positive specific T-cells with an EM phenotype) in the BAL of active TB patients. These data may be important for better understanding the pathogenesis of TB in the lung.