RESUMO
BACKGROUND: Miescher's cheilitis, whether occurring alone or as part of Melkersson-Rosenthal syndrome, is a rare type of granulomatous inflammation of unknown cause with an even rarer genital equivalent. Herein, we describe a case of the latter condition developing over a 12-year period. PATIENTS AND METHODS: A 27-year-old woman presented episodes of unilateral vulvar oedema, which initially regressed but resulted over time in permanent vulvar and perineal tumefaction. Histology revealed epithelioid histocytic granulomas so mild that they were not noticed at the start of the disease. The oedema remained the sole sign for 12years and two gastrointestinal screening tests for Crohn's disease proved negative, diagnosis of the latter condition was ruled out. DISCUSSION: Miescher's granulomatous vulvitis requires differential diagnosis, essentially with regard to Crohn's disease. Our case illustrates the difficulty in diagnosing this rare disease as well as the uncertainties surrounding its physiopathology. Diagnosis rests upon repeated sampling and biopsies with repeated levels of sections in order to identify the characteristic perivascular granulomas, which may be very mild.
Assuntos
Granuloma/patologia , Vulvite/patologia , Adulto , Células Epitelioides/patologia , Feminino , Histiócitos/patologia , HumanosRESUMO
Cell-mediated immunity directed against human papillomavirus 16 (HPV-16) antigens was studied in 16 patients affected with classic vulvar intra-epithelial neoplasia (VIN), also known as bowenoid papulosis (BP). Ten patients had blood lymphocyte proliferative T cell responses directed against E6/2 (14-34) and/or E6/4 (45-68) peptides, which were identified in the present study as immunodominant among HPV-16 E6 and E7 large peptides. Ex vivo enzyme-linked immunospot-interferon (IFN)-gamma assay was positive in three patients who had proliferative responses. Twelve months later, proliferative T cell responses remained detectable in only six women and the immunodominant antigens remained the E6/2 (14-34) and E6/4 (45-68) peptides. The latter large fragments of peptides contained many epitopes able to bind to at least seven human leucocyte antigen (HLA) class I molecules and were strong binders to seven HLA-DR class II molecules. In order to build a therapeutic anti-HPV-16 vaccine, E6/2 (14-34) and E6/4 (45-68) fragments thus appear to be good candidates to increase HPV-specific effector T lymphocyte responses and clear classic VIN (BP) disease lesions.
Assuntos
Epitopos de Linfócito T/imunologia , Papillomavirus Humano 16/imunologia , Proteínas Oncogênicas Virais/imunologia , Infecções por Papillomavirus/imunologia , Proteínas Repressoras/imunologia , Linfócitos T/imunologia , Neoplasias Vulvares/imunologia , Neoplasias Vulvares/virologia , Adulto , Idoso , Sequência de Aminoácidos , Proliferação de Células , Epitopos de Linfócito T/metabolismo , Feminino , Antígenos HLA-D/metabolismo , Antígenos de Histocompatibilidade Classe I/metabolismo , Humanos , Interferon gama/metabolismo , Leucócitos Mononucleares/citologia , Leucócitos Mononucleares/imunologia , Leucócitos Mononucleares/metabolismo , Ativação Linfocitária/imunologia , Pessoa de Meia-Idade , Proteínas E7 de Papillomavirus , Infecções por Papillomavirus/virologia , Fragmentos de Peptídeos/imunologia , Fragmentos de Peptídeos/metabolismo , Ligação Proteica/imunologia , Linfócitos T/citologia , Linfócitos T/metabolismo , Fatores de Tempo , Adulto JovemRESUMO
Many patients with dyspareunia suffer from vulvar vestibulitis syndrome. When all conservative treatments have failed, surgery is attempted. We appreciated results and complications of this treatment. During 1993 and 1994, at the gynecological department of the intercommunal hospital of Creteil (France), twelve women suffering from this condition for longer than six months had undergone a posterior crescent-shaped vestibular excision followed by vaginal advancement. Three weeks after surgery, three partial dehiscences have been observed, which secondary healed. The average duration of follow-up was 8 months. The interview of the patients in January 1995 revealed: six patients obtained painless sexual intercourse, four patients improved significantly, one had no improvement and one complained of severe vulvar burning instead of dyspareunia. Our results are weighing against the initial enthusiasm to surgical treatment. Collaboration between gynecologists and psychologists to specify the indications and previous treatment of an associated vaginismus should be helpful for the treatment of these patients.