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SARCOIDOSIS VASCULITIS AND DIFFUSE LUNG DISEASES 2012; 29; 151-154 Sarcoidosis: vaginal wall and vulvar involvement F. Xu1*, Y. Cheng1*, R. Diao1, X. Zhou1, X. Wang1, Y. Ma2, W. Lv2, H. Shen11 Department of Respiratory and Critical Care Medicine, and 2 Department of Gynecology, Second Affiliated Hospital, Zhejiang Universi-ty School of Medicine, Hangzhou, China Abstract. Sarcoidosis is a non-caseous granulomatous disease which could involve numerous organs includ-
ing lungs, eyes, skin, nervous system, heart, liver. However, the genitourinary tract involvement was rarely re-
ported in sarcoidosis. We report the case of a 45-year-old married woman who presented with 2 months histo-
ry of a vulval mass as large as a soybean, and did not reveal any remarkable pulmonary signs. Biopsy results
showed non-caseous granulomatous inflammation consistent with sarcoidosis in the vulvar lesion. To our
knowledge, this is the first reported case of this entity in the world. Based on the related literature, we highlight
the possibility of gynecologic involvement in sarcoidosis. (Sarcoidosis Vasc Diffuse Lung Dis 2012; 29: 151-154)
Key words: sarcoidosis, gynecologic involvement
Case report
of right Bartholin’s cyst 4 months ago. Thehistopathological results of these two operations A 45-year-old married woman presented with 2 months history of a vulval mass as large as a soybean.
On physical examination, we found a firm and There was no swelling, pain, vulval itching or causal- unpainful mass (1.5 cm × 1 cm) on the edge of an gia, and other complaints. Other symptoms such as episiotomy scar in the outside of the left vulva, and a urinary frequency, cough, chest tightness, shortness callous nodule (0.5 cm × 0.5 cm) in the left vaginal of breath, blurred vision and night sweats were not wall. Other abnormal physical signs were not found.
revealed. The patient had regular menstrual cycles Baseline blood tests and angiotensin-converting and no history of dysmenorrhea. Her menstrual flow enzyme (ACE) level were normal. The level of was moderate, dark red and without clots. She had serum cancer antigen (CA)-125 was 43.1 U/mL one daughter, and no history of cigarette smoking (normal range <35U/mL). The PPD skin test was and alcohol abuse. The patient underwent a right negative. Her spirometry showed an FVC of 3.46 L ovary oophorocystectomy 10 years ago and a section (110% predicted), an FEV1 of 2.94 L (108% pre-dicted), an FEV1 /FVC of 0.85, an DLCO SB of6.63 mmol/min /KPa (79% predicted). The ultra- Received: 1 December 2011Accepted after Revision: 11 January 2012 sound images of the vulva showed a low level echo of a nodule under the skin in the left vulva. There was Department of Respiratory and Critical Care Medicine, no apparent abnormality seen in the Chest X-ray.
Zhejiang University School of Medicine,88 Jiefang Road, Hangzhou, 31009, China The chest enhancement CT revealed reticulonodular infiltrates in bilateral lungs and mildly enlarged me-diastinal lymph nodes (Fig. 1). Resections of the vul- * Dr. Feng Xu and Dr. Yusheng Cheng equally contribute to thismanuscript var mass and vaginal wall nodule were performed.
Fig. 2. H&E staining showed non-caseous granulomatous in-
flammation and necrosis in both the genital masses (magnifica-
tion 40X)
Pathological examinations showed non-caseousgranulomatous inflammation consistent with sar-coidosis in both the genital mass and the vaginal wallnodule (Fig. 2). PAS staining and acid-fast stainingwere negative for fungi and acid fast bacilli. Consis-tently, cultures for fungi and mycobacteria were alsonegative. A diagnosis of vaginal wall and vulval sar-coidosis is made after excluding other potential caus-es of granulomatous inflammation and cancer. Basedon the staging result of the chest radiography (stage0), glucocorticoid was not used to treat the patient.
Presently, the patient is closely followed every 1 Discussion
Sarcoidosis is a multisystem granulomatous dis- ease affecting many organs, mostly the lungs. Thepathological features are noncaseous necrotic granu-loma, which was first described in 1899 by Hutchin-son. (1) The cause of sarcoidosis remains unclear. Itmay be related to the environment, genetic predispo-sition and disturbance of immune function for CD4+T cells.
Sarcoidosis is clinically manifested with pul- monary involvement. Other commonly affected or- Fig. 1. The chest X-ray (A) showed no apparent abnormality
gans include eyes, skin, nervous system, heart, and whereas the chest CT scan (B-C) revealed reticulonodular infil- liver. The male patient with sarcoidosis may present trates of bilateral lungs and mildly enlarged mediastinal lymphnodes (see arrows) with a painless testicular swelling when genitouri- Sarcoidosis: vaginal wall and vulvar involvement Table 1. Published sarcoidosis cases involving the vulva or vagina
nary tract is involved whereas in female patients, sar- (8) The laboratory examination showed elevated coidosis has also been reported with the involvement CA-125, a glycoprotein expressed by a variety of tis- of fallopian tubes, ovaries, and uterus. (2,3) Howev- sues of mesothelial origin. It was reported that pa- er, sarcoidosis of the vulva is a rare condition. Until tients with sarcoidosis in the peritoneum had higher now, only 6 cases of sarcoidosis with vaginal wall or vulval lesions have been reported in the world, and 5 Sarcoidosis generally has a good prognosis. Six- cases published in English (Table 1) (3-7). Among ty percent of patients do not require treatment and them, actually only one case of sarcoidosis with the the disease may spontaneously regress in some pa- vaginal wall involvement has been reported previ- tients. Glucocorticoid and other immunosuppres- ously. (3) Here we report a rare presentation of biop- sants have been used to treat sarcoidosis for many sy-confirmed sarcoidosis involving the vaginal wall years. However, their influence on the natural histo- and vulva. To our knowledge, this is the first report- ry of sarcoidosis is unclear. Our case had no bother- ed case of this entity in the world.
some signs and her vital organs were not at risk, The mass of sarcoidosis was found in the epi- therefore aggressive treatment was not adopted for siotomy scar of the left vulva in this case, in line with her. After 1 year’s follow-up, this patient is still in another study showing a skin scar is often involved.
5. Klein PA, Appel J, Callen JP. Sarcoidosis of the vulva: a rare cutaneous manifestation. J Am Acad Dermatol 1998; 39: 281-3.
6. Ezughah FI, Ghaly AF, Evans A, Green CM. Vulval sarcoid: a sys- 1. Dempsey OJ, Paterson EW, Kerr KM, Denison AR. Sarcoidosis. BMJ temic presentation of sarcoidosis. J Obstet Gynaecol 2005; 25: 730-2.
7. Decavalas G, Adonakis G, Androutsopoulos G, Gkermpesi M, 2. Vasu TS, Lai RS, Amzuta IG, Nasr MR, Lenox RJ. Sarcoidosis pre- Kourounis G. Sarcoidosis of the vulva: a case report. Arch Gynecol senting as intrascrotal mass: case report and review. South Med J 2006; 8. Veien NK, Stahl D, Brodthagen H. Cutaneous sarcoidosis in Cau- 3. Allen SL, Judson MA. Vaginal involvement in a patient with sar- casians. J Am Acad Dermatol 1987; 16: 534-40.
9. Kalluri M, Judson MA. Sarcoidosis associated with an elevated serum 4. Tatnall FM, Barnes HM, Sarkany I. Sarcoidosis of the vulva. Clin Exp CA 125 level: description of a case and a review of the literature. Am


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