Successful Pregnancy Following Surgery in the Obstructed Uterus in a Uterus Didelphys with Unilateral Distal Vaginal Agenesis and Ipsilateral Renal Agenesis: Case Report and Literature Review

SynopsisAfter surgical correction of unilateral distal vaginal agenesis, the obstructed didelphic uterus was able to harbor 2 subsequent pregnancies.BackgroundThere was a congenital anomaly syndrome of uterus didelphys, unilateral distal vaginal aplasia, and ipsilateral renal aplasia. Intuition suggests that later pregnancy would be in the unobstructed uterus.ObjectiveThe purpose is to report pregnancy occurring twice in a previously obstructed didelphic uterus after surgical correction.CaseA girl aged 13 years, 8 months presented with the syndrome of didelphic uterus, upper right hematocolpos, and right renal aplasia. Right vaginal marsupialization was done. Subsequently, she had 2 pregnancies in the right didelphic uterus.ConclusionPregnancy occurred in the obstructed uterus despite a large hematometra, extensive right pelvic peritoneal endometriosis, and hematocolpos. The surgeon should make every effort to preserve the obstructed uterus.     

Unilateral groin and pelvic irradiation for unilaterally node-positive women with vulval carcinoma

It is essential that any patient with resected vulval cancer and significant nodal disease receive optimal adjuvant treatment with radiation. Adequate radiotherapy for such patients with unilateral positive groin nodes has not been defined. Whether both groins and pelvic sidewalls should be irradiated or only the affected (node positive) side remains unclear. From our registry, we identified all patients with primary, previously untreated squamous cell carcinoma of the vulva undergoing bilateral inguinofemoral lymphadenectomy (superficial and deep nodes) and having unilaterally positive groin nodes treated with unilateral groin and pelvic radiotherapy (44 Gy in 22 fractions). Clinical and pathologic records were reviewed to identify the anatomical site and timing of recurrences in these patients and determine whether unilateral groin and pelvic irradiation was sufficient for disease control on the node-negative side. From 1983 to 2002, 20 patients with unilateral positive nodes treated with unilateral groin and pelvic irradiation were identified. Nineteen patients were classed as having FIGO stage III disease and one as FIGO stage IV due to involvement of the rectal mucosa. There were nine patients with disease recurrences in this group (45%). The disease-free interval ranged from 4 to 31 months (median time to recurrence, 9 months). All nine patients had local or regional failures, the most common site being the ipsilateral groin (six of nine patients). One patient was also found to have distant metastases. There were no recurrences noted in the contralateral (nonirradiated) groin or pelvic sidewall. Recurrence was generally fatal. Eight of the nine patients subsequently died of their disease. The ninth patient died of another cause. There was a high incidence of regional failure after unilateral groin and pelvic radiotherapy, but there were no recurrences on the nonirradiated, node-negative side. Although a small series, we speculate that there is no apparent disadvantage to administering unilateral adjuvant radiotherapy for unilaterally positive groin nodes and encourage further studies in order to more confidently determine whether the tendency observed in our center holds true.     

Uterus Didelphys Associated with Unilateral Cervical Atresia and Renal Agenesis

A case is reported of uterus didelphys associated with unilateral cervical atresia and renal agenesis on the same side. Two successful pregnancies occurred in the affected side after surgical correction.     

Unilateral Hydroureteronephrosis After a Mesh Procedure

Synthetic mesh has become a popular treatment of pelvic floor relaxation and pelvic organ prolapse, with low complication rates. We describe the case of a woman with unilateral hydroureteronephrosis after a mesh procedure (Gynecare Prolift; Ethicon Women's Health & Urology, Ethicon, Inc., Somerville, New Jersey) successfully treated by neoureterocystostomy. A 39-year-old woman with pelvic organ prolapse underwent the mesh procedure. Two months later, she had left flank pain, and hydroureteronephrosis was diagnosed on the same side despite cystoscopic confirmation of ureteral passage at the first operation. The arm of the mesh was removed surgically, and neoureterocystostomy was performed successfully. Mesh surgery is not without serious complications, and surgeons should bear in mind the possible complications associated with this surgical procedure.     

Non-vascular transplantation of the rabbit uterus

Transplantation of the uterus may be an optimal solution in young hysterectomized patients, in women with agenesis of the uterus or in severe uterine anomalies. In the human model, attempted tubal and ovarian homografts have usually failed. Eighteen rabbits underwent unilateral non-vascular uterine transplantation. Four rabbits underwent total unilateral uterine autograft. Three uteri showed severe inflammation and necrosis 72 h, 1 week and 1 month postoperatively. Thereafter, the operative technique was modified to avoid contamination from the vagina and four rabbits underwent supravaginal unilateral non-vascular autograft. Three uteri had preserved endometrial and myometrial structure on relaparotomy. The following four rabbits underwent homotransplantation of the uterus and showed acute rejection of the transplant. Under cyclosporine therapy three homotransplanted rabbits developed pelvic abscesses while three other rabbits showed preserved myometrial and endometrial structures.     

Unilateral tuboovarian abscess: A distinct entity

In a 30 month interval at the Los Angeles County-University of Southern California Medical Center, 85 patients had tuboovarian abscesses removed that were unrelated to complications of pregnancy. Thirty-seven patients (44 per cent) of the 85 had unilateral abscesses. Twenty patients (54 per cent) of the 37 patients with a unilateral abscess were using an intrauterine contraceptive device (IUD). In 13 (65 per cent) of the 20 women using an IUD, the foreign body was a Dalkon Shield. Unilateral pelvic abscesses can occur with or without the presence of an IUD.     

Unilateral primary ovarian leiomyoma in adolescent: a case report

Background Primary ovarian leiomyoma is a rare, usually small and incidentally detected neoplasm, clinically indistinguishable from subserous leiomyomas and ovarian fibromas, until histopathological confirmation. Case report We report a rare case of unilateral, ovarian leiomyoma that was not associated with uterine tumor in a 17-year-old woman. Trans abdominal ultrasonography and magnetic resonance imaging (MRI) revealed a pelvic mass (15 cm × 12 cm). Conclusion At laparotomy, unilateral salpingo-oophorectomy was performed and histologic examination revealed a leiomyoma arising primarily in the ovary.     

Uterus didelphys with blind hemivagina and ipsilateral renal agenesis complicated by pyocolpos and presenting as acute abdomen 11 years after menarche: presentation of a rare case with review of the literature

BACKGROUND: Congenital anomaly of the Müllerian duct system can result in various urogenital anomalies including uterus didelphys with blind hemivagina and ipsilateral renal agenesis. The diagnosis of this condition is usually made after menarche, but its rarity and variable clinical features may contribute to a diagnostic delay for years after menarche. CASE: A 24-year-old woman presented to the emergency room of the Department of Obstetrics and Gynecology complaining of severe worsening lower abdominal pain, vomiting and pus-like vaginal discharge. Physical examination revealed acute abdomen with diffuse lower abdominal tenderness, rebound and muscular resistance. Cervical and vaginal observation was impossible because of the patient's discomfort. Bimanual gynecological examination showed high tenderness cervical motion. Transabdominal ultrasound scan was performed and the radiologist interpreted the ultrasonographic findings as existence of a pelvic mass with mixed echogenicity. The preoperative diagnosis was ruptured tubo-ovarian abscess and emergency laparotomy was performed. Free pus in the pelvis was found. Also, a double uterus with an elongated and inflammatory right fallopian tube with pus passing out of its fimbrial end was observed. Vaginal examination under general anesthesia revealed an obstructed right hemivagina with vaginal pus-like discharge from a small fistula hole on the septate vagina. The final diagnosis was uterus didelphys with unilateral imperforate right hemivagina and pyocolpos. Transvaginal resection of the vaginal septum was performed and a large amount of pus and blood was spilled out. Postoperatively, intravenous pyelography demonstrated agenesis of the right kidney. CONCLUSION: We demonstrated the difficulty in making a correct diagnosis of this rare congenital anomaly of the female genital tract, especially after many years since menarche. This condition should be considered to prevent misdiagnosis or suboptimal treatment and decrease morbidity and unnecessary surgical procedures.     

Infected tuboovarian hydatid cyst: a rare cause of tuboovarian abcess

Pelvic hydatid cysts, although rare, must be considered when evaluating a pelvic mass in women living in an endemic area. The pelvis may become secondarily involved as a result of a rupture of the cyst in another location or be the only localization of the disease. If the cyst becomes secondarily infected, it may mimic a tuboovarian abscess. A 49-year-old multipara was admitted to the emergency department with the complaint of fever, generalized abdominal pain and distension. Abdominal ultrasound revealed a 4 cm cystic structure in the liver and the gynecological examination was normal. The patient's abdominal pain receded spontaneously, so she was prescribed albendazole and discharged from the hospital. Ten days later, she complained of pelvic pain, pressure and vaginal discharge. The uterus and adnexa were tender on pelvic examination. Ultrasound revealed an 8 cm uniloculated cyst with free floating internal echogenities located between the bladder and the uterus. At surgery a 10 cm right-sided tuboovarian mass was present. A germinative membrane was present inside the abscess and pericystectomy with unilateral salphingo-oophorectomy was performed.     

Unilateral twin ectopic pregnancy managed with laparoscopic salpingectomy

We describe a rare case of unilateral twin tubal ectopic pregnancy in a multiparous lady in the right fallopian tube that was confirmed on laparoscopy and managed by laparoscopic salpingectomy.     

Ovarian malposition – Mullerian anomalies revisited

A chronic pelvic pain case is presented with right unicornuate uterus, non-communicating left uterus, missing left fallopian tube, left uterosacral ligament lateral insertion to the periosteum of iliac bone, and ectopic left ovary, located under the sigmoid colon. Neither MRI, nor Laparoscopic evaluation may not be a sufficient tool alone to diagnose these rare cases.     

Leiomyosarcoma of the broad ligament: a case report and literature review

Leiomyosarcoma of the broad ligament is a rare tumor, with only 16 cases. In this paper we examine the case of a 52-year-old women consulted for pelvic pain. Clinical examination revealed a solid mass on the right side of the uterus. Abdominal ultrasonography and CT scan confirmed a mass presumably ovarian in origin. Laparotomy found a tumor of the right broad ligament, independent from uterus, tubes and ovaries. A total hysterectomy with bilateral salpingo-oophorectomy was performed and the histologic examination led to the diagnosis of leiomyosarcoma of the broad ligament. No evidence of metastasis is noted after 42 months' follow-up.     

Isolated tubal torsion at menarche- a case report

Background: Adnexal torsion is a well-recognized cause of acute pelvic pain. Isolated tubal torsion with ovarian sparing has certainly been documented, but is uncommon. Although risk factors for the latter include a menstrual period, menarche in particular is not known to predispose a patient to this event. Severe unilateral pelvic pain with first menses is more likely to herald a congenital mullerian anomaly and cryptomenorrhea, particularly when accompanied by a pelvic mass. We present a case of tubal torsion where a coincidental, yet misleading temporal relation to menarche led to a delay in laparoscopy and ultimate diagnosis.Case: KG, an eleven-year-old female, experienced severe right-sided dysmenorrhea with her first and second menses in August and September 1999 respectively. Between episodes, pain, although still present, was more tolerable and the patient never required hospitalization. Ultrasound revealed a lobulated inhomogeneous mass posterior to the uterus and extending from one normal ovary to the other (Figures). MRI further described the mass as pseudoencapsulated with inhomogeneous areas of high attenuation on T1 and T2 images (Figures). Findings were consistent with an endometrioma, but admittedly could have represented a hemorrhagic cystic mass. No definite mullerian anomaly was seen to explain advanced endometriosis, but two focal areas within the endometrial canal raised the possibility of a uterine septum. Examination of the patient (one week after presentation) was not very helpful although she was pubertal, did have a hymenal septum and was mildly tender on bimanual examination in the Pouch of Douglas. The patient had been started on continuous oral contraceptives while undergoing investigations. Pain only recurred during an episode of break-through bleeding. Ultimately she came to laparoscopy and hysteroscopy where chronic right tubal torsion and necrosis was identified with an inflammatory/hemorrhagic reaction in the pelvis (Photos). There were no identifiable fimbria of the right tube which was densely adherent distally to perirectal fat (Photo). No obvious precipitant was found. Laparoscopic lysis of adhesions and right distal salpingectomy was performed (Photo). Her uterine cavity was in fact normal (Photo)Conclusion: Whether or not this patient's right tube was originally normal will never be known. Congenital abnormalities of fallopian tubes do occur and can predispose to torsion. Nonetheless, adnexal torsion must always be kept in mind whenever a woman presents with unilateral pelvic pain. Early diagnosis is paramount in children and women of reproductive age in order to improve the likelihood of adnexal salvage and future fertility. A "gold-standard" radiological investigative tool continues to elude us. Laparoscopy, albeit more invasive, remains an invaluable procedure in this context with relatively low morbidity as compared to the consequences of delayed diagnosis.     

The Unicornuate Uterus with an Occult Adenomyotic Rudimentary Horn

We report 2 case of an atypical variant of unicornuate uterus in 2 adolescent patients with severe dysmenorrhea. Pelvic ultrasonography and magnetic resonance imaging identified a normal uterine contour. On the right side within the uterine fundus, a nodule was detected with a small hypoechogenic content. At laparoscopy the uterus and adnexae appeared to be normal. No endometriotic lesions were identified. Hysteroscopy identified a single regular cervical canal and a uterine cavity resembling that of a left unicornuate uterus, with a single regular left tubal ostium. Complete resection of the right uterine nodule along with an ipsilateral salpingectomy was performed. The nodule contained a small endometrial cavity and hematometra. Histologic study showed a cavitated adenomyotic uterine rudiment. The patients were discharged on the second postoperative day. No intraoperative or postoperative complications or recurrence of pelvic pain occurred.     

Sacrococcygeal neurofibroma: rare cause for chronic pelvic pain

Pelvic pain is a common gynecologic complaint. Retroperitoneal pelvic tumors are rarely a cause of pelvic pain. Neurofibroma is an uncommon pelvic retroperitoneal tumor, and only 17 cases are reported to date. A 38-year-old woman with chronic pelvic pain had a soft fixed mass that was the size of an orange in the right posterolateral fornix, with a normal uterus on pelvic examination, and a mass of 6.3 × 5.2 cm with mixed echotexture on the right side separate from both ovaries on transvaginal ultrasonography. A provisional diagnosis of retroperitoneal mass probably a retroperitoneal teratoma was made. Laparoscopy was performed; an ill-defined retroperitoneal soft tissue mass of about 6 cm was seen on the right pararectal and presacral area, displacing the rectum toward the left side. The mass was soft and jellylike without a cyst wall. Histopathologic study and immunohistochemistry results were consistent with neurofibroma of the sacrococcygeal regions. To our knowledge this is the third case of sacrococcygeal neurofibroma treated by complete laparoscopic excision. Gynecologists should keep sacrococcygeal neurofibroma as a differential diagnosis of pelvic pain with atypical location of a pelvic mass. A high index of suspicion and an appropriate imaging technique are needed for accurate diagnosis. Laparoscopy seems to be a safe and effective method of managing retroperitoneal presacral neurofibromas.     

Acute Abdominal Pain as a Result of a Ruptured Hematosalpinx: A Rare Complication of an Unusual Müllerian Anomaly

Case ReportA 15-year-old virgin Caucasian female presented to the emergency room with a 40-hour history of acute left lower quadrant abdominal pain and nausea. Evaluation suggested a left pelvic kidney with obstructed ureter being the etiology. Her pain continued to escalate so further workup with laparoscopy was performed. This demonstrated a left pelvic sidewall hemi uterus with ruptured hematosalpinx. This is an unusual clinical presentation of a müllerian anomaly not previously documented.DiscussionThe differential diagnosis of acute unilateral abdominal pain in adolescent females should include müllerian anomalies. The incidence of this diagnosis is low but the evaluation and treatment can be performed in an expeditious manner if the diagnosis is considered. The laparoscopic excision of a unilateral noncommunicating uterine horn is a valid and recommended treatment approach of this rare malformation.     

Low-grade endometrial stromal sarcoma with retroperitoneal metastases: an unusual case report

Background Endometrial stromal sarcoma (ESS) is an uncommon malign neoplasm, and its occurrence outside the uterus is extremely rare in the absence of metastasis or extension of a primary uterine neoplasm. When arising in the pelvis or abdominal cavity, ESS is associated with uterine adnexa or serosal surface of various organs. Case We present the case of a 46-year-old woman with lower abdominal pain and regular menstruation who underwent laparotomy after a diagnosis of pelvic mass mimicking a right adnexial tumor. Exploration of the pelvis revealed a retroperitoneal mass of 15 cm in diameter in the right illiac fossa without accompanying pelvic or paraaortic lymphadenopathy. Uterus and ovaries were bilaterally normal in size. The pathology showed low-grade ESS of the uterus with direct spread to retroperitoneum without serosal metastases. Conclusion This case shows that despite its well-known good prognostic nature, low-grade ESS may behave as an aggressive malignancy.     

Intravascular large B-cell lymphoma of the uterus: a diagnostic challenge.

A 63-year-old woman presented with postmenopausal bleeding. The initial curettage material was reported as "undifferentiated malignant tumor." A total abdominal hysterectomy with bilateral salpingo-oophorectomy was performed with pelvic lymph node dissection and omental biopsy, which showed an intravascular large B-cell lymphoma in the lumina of small blood vessels of the uterus, left tube, and right ovary. This is the first reported case of an intravascular lymphoma of the uterus.     

Unilateral absence of distal tube and ovary with migratory calcified intraperitoneal mass

A case of unilateral absence of a distal portion of Fallopian tube and ovary in association with a migratory calcified intraperitoneal mass is described. A review of the literature concerning this rare occurrence is summarized, and a discussion of the etiology of such an entity is presented. The surgical technique for removal of such a pelvic lesion through a posterior colpotomy incision and use of the laparoscope is also described.     

Combined laparoscopic and radical vaginal treatment of primary vaginal leiomyosarcoma in a patient with unicornuate uterus and pelvic kidney

We report a case of primary vaginal leiomyosarcoma occurring in a patient with a unicornuate uterus and pelvic kidney. The patient was treated with radical partial vaginectomy, unilateral groin dissection, total laparoscopic hysterectomy, and left pelvic laparoscopic lymphadenectomy, followed by adjuvant chemotherapy. Twenty-five months after her original procedure, the patient is alive and free of disease. The exact association between müllerian anomalies and primary malignancies of the genitourinary tract is unclear. It is nonetheless important to be aware of the close embryonic association between the genital and urinary tracts when evaluating such patients and choosing the appropriate surgical and therapeutic approach.