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Behcet syndrome is a systemic disease with venous tropism, generally expressed by thrombosis and phlebitis. Arterial involvement is more exceptional but can lead to aneurysm of the pulmonary artery, generally not more than three. We report a patient with Behcet syndrome who developed multiple aneurysms of the pulmonary artery and discuss the clinical, radiological aspects as well as the disease course and therapeutic management.
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A case of Behcet syndrome with immunologic evaluation, including screening of a vulvar ulcer for IgG, IgM, IgA, and fibrinogen by direct fluorescent microscopy is presented. Attempts were made to demonstrate cellular and humoral immune responses to mucosal antigens by lymphoblast transformation in the presence of cadaver esophageal mucosal extracts and indirect immunofluorescence using autologous serum and mucosal tissue. Serial measurements of percentages of total T, active T, and B lymphocyte populations, and lymphocyte response to phytohemagglutinin (PHA) stimulation during the course of Behcet syndrome are also presented. Clinical evaluation, histology of a Behcet vulvar ulcer, and a 2-year followup with good response to chlorambucil are reviewed.
Sera of patients diagnosed as having the active Vogt-Koyanagi-Harada (VKH) syndrome, Behcet's syndrome or sympathetic ophthalmia as well as normal controls were evaluated by ELISA and by staining of normal human retinal tissue using the avidin-biotin-peroxidase complex (ABC) technique for anti-retinal antibodies. No anti-retinal S-antigen antibodies were detected by ELISA. However, autoimmune antibodies were found against the outer segments of photoreceptors and Müller cells in patients with the VKH syndrome, with lower titers in some patients with Behcet's syndrome, and in a few patients with sympathetic ophthalmia. These results suggested anti-retinal antibodies were present and that retinal autoimmunity may play a role in pathogenesis in varieties of posterior uveitis. In addition, the indirect immunoperoxidase staining technique may facilitate the diagnosis of VKH in uncertain cases.
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The various forms of aphthous stomatitis and Behcet syndrome share common signs that were discussed during a conference workshop. Four markers should be considered in determining the presence of these diseases: hematologic abnormalities, HLA, antibody responses, and immune complexes.
The case of a 14-year-old girl with Behcet syndrome is described. Besides painful and recurrent oral ulcerations, the patient had a cough and intermittent hemoptysis. The initial chest roentgenogram revealed bilateral parahilar opacities. CT and MRI scans of the thorax showed bilateral thrombosing aneurysms of the pulmonary arteries. Pulmonary blood flow imaging was performed after technegas ventilation lung scanning and Tc-99m MAA injection using a first-pass radionuclide angiography procedure. Altered blood flow in the left pulmonary artery was shown. Bilateral and well-defined ventilation/perfusion mismatched areas suggested a high probability of pulmonary embolism. Little additional information was obtained on subsequent contrast pulmonary angiography. The high incidence of pulmonary artery hypertension and associated vascular injury risk makes pulmonary angiography an unsafe procedure in patients with pulmonary Behcet syndrome. The need for pulmonary angiography could be obviated in such cases with the use of high-precision MRI and ventilation/perfusion lung scanning, including radionuclide pulmonary angiography.
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Behcet's syndrome is reported in two families (one English and the other Egyptian). A mother and two of her sons of the English family and two brothers of the Egyptian family are described. The contradictory opinions concerning the incidence of the familial occurrence of this syndrome are partly due to uncertainty about the nosologic relationship of recurrent aphthal and Behcet's syndrome. The question of whether this syndrome should be included in a large group of aphthoses or be regarded as a true clinical entity is discussed.
Behcet's syndrome is a multisystem disease complex, the major manifestions of which are oral and genital ulcers, arthritis, uveitis, and skin eruptions. Less frequently, CNS disturbances, colitis, thrombophlebitis, large-vessel vaculitis, and myocarditis occur. If renal involvement does occur, it is usually manifested by asymptomatic microhematuria and/or proteinuria. Recently, renal amyloidosis and focal necrotizing glomerulonephritis with immune complex deposition have been described. We describe a patient with Behcet's syndrome who experienced diffuse proliferative glomerulonephritis with epithelial cell crescent formation in 75% of glomeruli examined. Immune complexes were not identified. Renal function, impaired on admission, improved with no therapy.
Behcet's syndrome is a systemic illness of unknown etiology characterized by necrotising vasculitis originally described in Turkey. Typical manifestations include urogenital ulcerations, eye inflammation and migratory thrombophlebitis. An unusual course of this disease is described. A 56 year-old man was admitted to the hospital with fever, milk-glass opacities on chest x-ray, mucosal defects on the tongue and penis, hematuria and proteinuria with functional disorder of kidney. Chlamydia pneumoniae, CIK positivity and C 3 complement decrease were found. Antibiotics and antimycotic drugs have shown only slight improvement of pulmonary lesions. An excellent effect in all pathological findings was achieved after prednison administered in dosis of 1 mg/kg and later cyclophosphamide administration (Fig. 7, Ref. 12).
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