Claire Scrivani1, Melissa A Merideth, Tajana Klepac Pulanic, Steven Pavletic, Richard W Childs, Matthew M Hsieh, Pamela Stratton. 1. 1Hematology Branch, National Heart Lung and Blood Institute, National Institutes of Health, Bethesda, MD; 2Medical Genetics Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD; 3Intramural Office of Rare Diseases, Office of the Director, National Institutes of Health, Bethesda, MD; 4Community Health Center Zagreb East, Department of Gynecology, Zagreb, Croatia; 5Graft-versus-Host and Autoimmunity Unit, Experimental Transplantation and Immunology Branch, National Cancer Institute, National Institutes of Health, Bethesda, MD; 6Molecular and Clinical Hematology Branch, National Institute of Diabetes and Digestive and Kidney Diseases, National Institutes of Health, Bethesda, MD; and 7Eunice Kennedy Shriver National Institute of Child Health and Human Development, National Institutes of Health, Bethesda, MD.
Abstract
OBJECTIVE: The aim of the study was to describe the presentation and successful treatment of labial fusion in women after allogeneic hematopoietic cell transplantation (HCT). MATERIALS AND METHODS: During routine posttransplant gynecologic evaluation, labial fusion was identified in 5 female patients. Clinical data were collected regarding underlying disease, transplant regimen, genital symptoms, systemic sites of chronic graft-versus-host disease (cGvHD) and treatment, and follow-up. RESULTS: At presentation, women had a median age of 40 years (range = 35-50) and were 23-month to 8-year post-HCT. Four of the 5 patients with labial fusion had evidence of active cGvHD; 3 patients had severe sclerotic cGvHD, and 1 patient had bronchiolitis obliterans. One had rheumatoid arthritis and had recently stopped taking etanercept, but had no sites of cGvHD. One patient presented with only a pinpoint opening for passage of urine. Her complete labial fusion was lysed under general anesthesia. Three of the 4 others presented with dyspareunia. Their labia were fused between the clitoris and urethra narrowing the vaginal opening without obstructing the urethra. These labial adhesions were successfully lysed during an office procedure. Once the labial mucosa healed, the patients applied topical clobetasol and estrogen to prevent reagglutination. On follow-up, 1 month to 1 year later, all women were significantly improved. CONCLUSIONS: These patients highlight the importance of asking women who have undergone allogeneic HCT, especially those with severe cGvHD, about dyspareunia and dysuria. Those with genital symptoms warrant referral to a gynecologist. These cases illustrate that labial fusion, if diagnosed early enough, may be treated successfully with an office procedure and medical therapy.
OBJECTIVE: The aim of the study was to describe the presentation and successful treatment of labial fusion in women after allogeneic hematopoietic cell transplantation (HCT). MATERIALS AND METHODS: During routine posttransplant gynecologic evaluation, labial fusion was identified in 5 female patients. Clinical data were collected regarding underlying disease, transplant regimen, genital symptoms, systemic sites of chronic graft-versus-host disease (cGvHD) and treatment, and follow-up. RESULTS: At presentation, women had a median age of 40 years (range = 35-50) and were 23-month to 8-year post-HCT. Four of the 5 patients with labial fusion had evidence of active cGvHD; 3 patients had severe sclerotic cGvHD, and 1 patient had bronchiolitis obliterans. One had rheumatoid arthritis and had recently stopped taking etanercept, but had no sites of cGvHD. One patient presented with only a pinpoint opening for passage of urine. Her complete labial fusion was lysed under general anesthesia. Three of the 4 others presented with dyspareunia. Their labia were fused between the clitoris and urethra narrowing the vaginal opening without obstructing the urethra. These labial adhesions were successfully lysed during an office procedure. Once the labial mucosa healed, the patients applied topical clobetasol and estrogen to prevent reagglutination. On follow-up, 1 month to 1 year later, all women were significantly improved. CONCLUSIONS: These patients highlight the importance of asking women who have undergone allogeneic HCT, especially those with severe cGvHD, about dyspareunia and dysuria. Those with genital symptoms warrant referral to a gynecologist. These cases illustrate that labial fusion, if diagnosed early enough, may be treated successfully with an office procedure and medical therapy.
Authors: Simonetta Spinelli; Sandra Chiodi; Sergio Costantini; Maria Teresa Van Lint; Anna Maria Raiola; Gian Battista Ravera; Andrea Bacigalupo Journal: Haematologica Date: 2003-10 Impact factor: 9.941
Authors: Pamela Stratton; Maria L Turner; Richard Childs; John Barrett; Michael Bishop; Alan S Wayne; Steven Pavletic Journal: Obstet Gynecol Date: 2007-11 Impact factor: 7.661
Authors: Mary E D Flowers; Pablo M Parker; Laura J Johnston; Alice V B Matos; Barry Storer; William I Bensinger; Rainer Storb; Frederick R Appelbaum; Stephen J Forman; Karl G Blume; Paul J Martin Journal: Blood Date: 2002-07-15 Impact factor: 22.113