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Blackwell Science, LtdOxford, UKJOGThe Journal of Obstetrics and Gynaecology Research1341-80762003 Asia and Oceania Federation of Obstetrics and GynaecologyDecember 2003296395398Original ArticleVaginal myoma: mimicking gluteal abscessR. Gowri et al. J. Obstet. Gynaecol. Res. Vol. 29, No. 6: 395–398, December 2003 Leiomyoma of the vagina: An unusual presentation Raju Gowri1, Subramanium Soundararaghavan1, Asha Oumachigui1, Sarath Chandra Sistla2 and Krishnan R. Iyengar3 1 Department of Obstetrics and Gynecology, 2Department of Surgery, and 3Department of Pathology, Jawaharlal Nehru Institute of Postgraduate Medical Education and Research (JIPMER), Pondicherry, India Abstract Vaginal leiomyomas are rare. They usually arise from the anterior vaginal wall. We report a case of a vaginal leiomyoma arising from the right lateral wall that presented as a gluteal swelling with pus discharging per vagina, creating a clinical dilemma in diagnosis. Preoperative imaging and biopsy may help to rule out malignancy. The size of the tumor necessitated abdominoperineal approach and hysterectomy for better surgical access. To the best of our knowledge such a clinical presentation of a vaginal leiomyoma has not been reported in the literature. Key words: ischiorectal fossa tumor, vaginal leiomyoma. Introduction The vagina is a rare site for leiomyoma, and they are usually located in the anterior wall and rarely in the lateral wall.1 They usually present as a mass per vaginum or dyspareunia or pressure symptoms on the urinary tract.2,3 We report a case of myoma from the lateral vaginal wall that presented like an inflammatory mass in the gluteal region. Surgical excision through the vaginal route has been the traditional approach. Abdominoperineal route may be necessary for huge tumors as in this case. Case Report A 35-year-old woman presented to our outpatient department on March 31 2000 with a gluteal swelling on the right side of 3 months duration. She also had purulent vaginal discharge for 7 days. There was no fever. She had pain over the region of the swelling. There was no history of sexually transmitted disease in her or her husband. She had a normal menstrual his- tory, the last menstrual period was on March 21, 2000. She had three children. All were spontaneous vaginal deliveries. Examination revealed her to be undernourished and anemic. She was not febrile. Pulse was 100/min and blood pressure normal. Breasts were normal and there was no significant lymphadenopathy. There was a swelling in the right gluteal region. The skin over the swelling was shiny, warm, indurated and tender. Speculum examination revealed a bulge through the right vaginal wall. There was an opening in the vaginal wall through which the swelling had ruptured and there was offensive purulent discharge. The vagina appeared compressed and shifted to the left side. The cervix could not be visualized or felt. Pelvic examination was difficult because of the large swelling in the right side that seemed to extend upwards towards the pelvic cavity. The uterus was not clearly made out. A clinical diagnosis of ischiorectal abscess was made and in consultation with surgeons the decision was taken to drain the abscess. Patient was admitted. A cruciate incision was made on the right gluteal region on Received: 7 January 2003. Accepted: 25 July 2003. Reprint request to: Dr Raju Gowri, D-II/13 Dhanvantari Nagar, JIPMER, Pondicherry – 605006, India. Email: gowridorai@hotmail.com 395 R. Gowri et al. Figure 1 Clinical photograph showing the fleshy tumor protruding from the gluteal incision site. the most prominent part of the swelling. When the skin was cut, it retracted and a smooth fleshy mass protruded through the incision (Fig. 1). A soft tissue malignant tumor was suspected and a biopsy from the growth was taken. Histopathology revealed it to be a leiomyoma. Investigations revealed moderate anemia. Other hematological and biochemical indicators were normal. Ultrasound examination revealed a normal uterus. The bladder was displaced anteriorly and to the left. There was a large hypo-echoic mass in the pelvic floor region extending into the right gluteal region close to the cervix. Intravenous pyelogram revealed a mild pressure effect on the right side. Computed tomography revealed a large solid tumor with variable density from the level of the cervix lifting up the bladder, extending posterior to the bladder up to the right ischiorectal fossa and the right gluteal region. There were no calcifications. A course of antibiotics was given according to the culture report of the pus discharging from vagina. Preoperative blood transfusions were given. In consultation with the surgeons the patient was scheduled for an abdominoperineal resection of the tumor. Laparotomy was performed on May 19 2000 and the uterus, both fallopian tubes and ovaries were normal. There was a large extra-peritoneal mass filling the entire pelvis and extending downwards in the right side to the gluteal region. Surgical access to the tumor necessitated hysterectomy. When the vagina was opened it was found that the tumor was arising from the right vaginal wall. It was extending almost up to the left pelvic wall. On the right side, it was passing from the hiatus urogenitalis, through the pelvic diaphragm into the 396 Figure 2 Line diagram showing the extent of the tumor. OI, obturator internus muscle; PD, pelvic diaphragm; U, uterus; V, vagina; S, sinus discharging pus; G, tumor protruding at the gluteal region. Figure 3 Arrow indicates the plane of dissection. PD, pelvic diaphragm; OI, obturator internus; VA, vaginal artery; U, ureter; V, vagina; T, tumor. right ischiorectal fossa and down to the gluteal region (Fig. 2). The dissection was begun from the vault, below the reflection of the ureter, to identify a plane between the tumor and the surrounding structures all around (Fig. 3). The right vaginal artery was clamped cut and ligated. Lateral fibers of levator ani were identified. Thus the tumor was mobilized to the pelvic diaphragm. An incision was made on the right gluteal region to mobilize the tumor medially from the rectum (Figs 4,5). The internal pudendal vessels in the ischiorectal fossa were clamped, cut and ligated. The tumor was mobilized up to the pelvic diaphragm. A tumor measuring 25 ¥ 20 cm and weighing 1500 g was removed from below. The pelvic floor was repaired on the right side and the right wall and vault of vagina Vaginal myoma: mimicking gluteal abscess Figure 4 Incision made around the tumor protruding at the gluteal region. Figure 5 Dissection in the ischiorectal fossa to free the tumor. were reconstructed. Complete hemostasis was obtained. A drain was left through the gluteal incision in the dead space. Abdomen was closed in layers. Blood loss was approximately 2 L and 6 packs of compatible blood were transfused. The surgery took 4 h. Postoperatively the patient recovered uneventfully except for superficial infection of the gluteal wound. Histology confirmed the tumor to be leiomyoma with infarction (Fig. 6). Discussion Over 300 cases of vaginal leiomyomas have been reported in the literature. They have varied clinical presentation, the most common being vaginal mass.4,5 Patients may primarily have symptoms of urinary bladder outlet obstruction.6 Shaw reported a case of a sub urethral leiomyoma who presented with pelvic Figure 6 Photomicrograph showing leiomyoma with infarction (HE ¥ 120). pain and had cystitis cystica.7 Tumors with slow growth and small size may not produce any symptom and regress after menopause.8 These tumors are usually localized and vary from solid to cystic in consistency. They are usually firm and can undergo degenerative changes and feel soft. Ulceration of the overlying mucosa with subsequent necrosis can produce purulent discharge as happened in our case. A huge leiomyoma occupying both the ischiorectal fossae has been reported in a pregnant patient.9 She had presented with vaginal mass and the tumor was found to be adherent to the rectum at the time of operation. Our case had an unusual presentation. Such a presentation has not previously been reported. Vaginal leiomyomas may be confused with a variety of benign vaginal tumors. A preoperative diagnosis is seldom made. To discriminate between benign and malignant tumor and to assess surrounding structures ultrasound, biopsy and CT scan have been recommended.10 Leiomyoma with variable cell size and shape, hyperchromatic and multilobulated nuclei but without marginal infiltration or mitotic activity has been called bizarre leiomyoma. Only one such case has been reported so far.11 The benign behavior of these bizarre leiomyoma has been firmly established.12 Vaginal leiomyoma especially the borderline variety may undergo sarcomatous change.13,14 Liu reported an incidence of 9.1% for malignant change in his series.8 Hence these tumors should be operated as soon as they are recognized. Surgery through the vaginal route has been reported as the treatment of choice for these tumors. However, huge tumors as in our case may require combined 397 R. Gowri et al. abdominal and perineal approach and may necessitate hysterectomy. Though morcellation of the tumor might render the surgery through the vaginal route easier, recurrence of the tumor is of concern. Other authors have specifically recommended removal of the tumor en bloc to avoid any possible recurrence.15 References 1. Kaufman RH, Gardner HL. Benign mesodermal tumors. Clin Obstet Gynecol 1965; 8: 978. 2. Sangwan K, Khosla AH, Hazra PC. Leiomyoma of the vagina. Aust N Z J Obstet Gynaecol 1996; 36: 494–495. 3. Njeh M, Barkia A, Jemni M, Zermani R, Ben Milad K, Ayed M. Vaginal leiomyoma. The female prostate. Acta Urol Belg 1993; 61 (3): 31–32. 4. Ruggieri AM, Brody JM, Curhan RP. Vaginal leiomyoma. A case report with imaging findings. J Reprod Med 1996; 41 (11): 875–877. 5. Nel CP, Tiltman AJ. Leiomyoma of the vagina. S Afr Med J 1978; 54: 816–817. 6. Freed Z, Haleem SA, Wiener I, Feldman J. Bladder outlet obstruction caused by vaginal fibromyoma: the female prostate. J Urol 1975; 113 (10): 30–31. 398 7. Shaw CT. Vaginal leiomyoma as a cause of pelvic pain and cystitis cystica. J Am Osteopatho Assoc 1989; 89: 1330–1331. 8. Liu MM. Fibromyoma of the vagina. Eur J Obstet Gynecol Reprod Biol 1988; 29: 321. 9. Sadan O, Kruger S, van Iddekinge B. Vaginal tumors in pregnancy. Case report and review of literature. Acta Obstet Gynecol Scand 1987; 66: 559–562. 10. Bolt JM, Schutter EM. A tumor in the paracolpium. A case report. Eur J Obstet Gynecol Reprod Biol 1998; 76 (2): 233–236. 11. Biankin SA, O’Toole VE, Fung C, Rassell P. Bizarre leiomyoma of the vagina: report of a case. Int J Gynecol Pathol 2000; 19: 186–187. 12. Downes KA, Hart NR. Bizarre leiomyoma of the uterus. a comprehensive pathological study of 24 cases with long term follow up. Am J Surg Pathol 1997; 21: 1261–1270. 13. Cobanoglu O, Gurkan Zorlu C, Ergun Y, Kutluay L. Leiomyosarcoma of the vagina. Eur J Obstet Gynecol Reprod Biol 1996; 70 (2): 205–207. 14. Miyakawa I, Yasuda H, Taniyama K, Mori N, Uehara Y, Sumiyeshi A. Leiomyosarcoma of the vagina. Int J Gynaecol Obstet 1985; 23 (3): 213–216. 15. Dhaliwal LK, Das I, Gopalan S. Recurrent leiomyoma of the vagina. Int J Gynaecol Obstet 1992; 37: 281–283.