The Auto-Amputated Adnexa: A Review of Findings in a Pediatric Population

Study ObjectiveTo quantify our experience and that of the literature with diagnosis and management of the auto-amputated adnexa in a pediatric population.DesignCase series and literature review.SettingTertiary care medical center.ParticipantsCase series of pediatric patients (<18 years of age) with surgically documented adnexal auto-amputation collected from our medical center and the literature.InterventionsNone.Main Outcome MeasureAuto-amputated adnexa.ResultsIn addition to the 3 cases discussed from our institution, 91 cases of auto-amputated adnexa were identified in the literature dating back to 1943, for a total of 94 cases. Forty-nine percent (46/94) of the cases involved girls in a pediatric population (<18 years of age). Of these, the majority (n = 26) were identified in a subgroup of girls who were diagnosed with an adnexal cyst by antenatal ultrasound. Most of these neonates were asymptomatic at birth or had a palpable abdominal mass (n = 6) and at the time of surgical exploration were found to have an auto-amputated adnexa. 34 out of 46 cases were analyzed in detail. The right adnexa were involved in 56% of the cases. The most common presenting complaint verbalized by the older girls was pain; however, 8 cases were identified in asymptomatic girls undergoing unrelated diagnostic testing.ConclusionThe auto-amputated adnexa is a rare finding in the pediatric population, but it must be considered as a possible explanation for the incidental finding of absence of the fallopian tube or ovary in the subgroup of patients who undergo surgery for any reason. Patients with an antecedent history of pelvic pain either chronic or intermittent in nature may be diagnosed with torsion or less frequently auto-amputation of the adnexa. A fetal “pelvic mass” or “ovarian cyst” may predispose the adnexa to torsion and subsequent auto-amputation either in-utero or post-delivery. Many of these antenatally diagnosed cysts and even subsequent auto-amputations are completely asymptomatic, however, and do not compromise fertility assuming the contralateral adnexa are normal. Thus expectant management is appropriate for small (less than 4 cm), asymptomatic simple cysts and even suspected auto-amputated adnexa in an asymptomatic patient.     

Ruptured Hemorrhagic Corpus Luteum Cyst in an Undescended Ovary: A Rare Cause of Acute Abdomen

BackgroundUndescended ovaries are typically detected during infertility evaluations and are frequently associated with uterine malformations. Ruptured hemorrhagic corpus luteum cyst of an undescended ovary is an unusual cause of acute abdomen in an adolescent.CaseA 15-year-old girl presented with right lower quadrant pain, nausea, and vomiting, and transabdominal sonography and magnetic resonance imaging of the pelvis showed a 10 cm × 5 cm sized cystic mass at the level of the pelvic brim, anterior to the psoas muscle suggestive of a retroperitoneal hemorrhagic cyst. At surgery, the uterus and left adnexa appeared normal, but the right ovary was not visible within the pelvic cavity, and the right pelvic retroperitoneum was distended. After opening the retroperitoneum and aspirating blood clots, the undescended ovary with a ruptured cyst was visualized within the retroperitoneum. Right ovarian wedge resection was performed and the right ovary was repositioned in the pelvic cavity.Summary and ConclusionRupture of a corpus luteum cyst in an undescended ovary should be included in the differential diagnosis of acute abdomen in adolescents.     

Extragonadal mature cystic teratoma at anterior uterine wall coexisting with a pararectal corpus luteum cyst and agenesis of left fallopian tube and left ovary

ObjectiveMature cystic teratomas are mostly confined to the ovaries, but several authors have reported findings of extragonadal occurrences along the migration pathway of primordial germ cells. Extragonadal mature cystic teratomas are extremely rare; their occurrences and pathogenesis are unknown.Case reportWe report the case of a 26-year-old woman who was admitted for scheduled laparoscopic right ovarian tumor excision. An anterior uterine wall mature cystic teratoma and a pararectal corpus luteum cyst were found intraoperatively with coexistence of left adnexal agenesis.ConclusionThe existence of an extragonadal mature cystic teratoma over the anterior uterine serosal layer may be caused by autoamputation and reimplantation as a result of ovarian torsion or displacement of primordial germ cells along their migration path. The existence of an ovarian mass over the sigmoid colon combined with left adnexal agenesis may be the result of ovarian torsion with remnant tissue attached to the sigmoid colon.     

Oophoropexy to Prevent Adnexal Torsion: How,When, and for Whom?

Study ObjectiveTo assess the efficacy of oophoropexy in obviating recurrent torsion and its possible long-term effects.DesignCase series and review of the literature (Canadian Task Force classification III).SettingUniversity hospital.PatientsWomen who underwent oophoropexy for recurrent torsion of normal adnexa between 2003 and 2008.Measurements and Main ResultsRetrieved information included the indication for oophoropexy, surgical methods, recurrence, and follow-up. Seven women underwent oophoropexy during the study period because of recurrent torsion of normal adnexa. One additional patient had experienced 3 torsion events of cystic adnexa. Surgical methods included suturing of the ovary to the pelvic sidewall or to the round ligament and plication of the utero-ovarian ligaments. Recurrence occurred in 1 of 6 patients for whom follow-up was available. All 6 patients reported spontaneous menstruation, and 2 conceived spontaneously and gave birth. Ultrasound at long-term follow-up (9–58 months) demonstrated normal ovaries.ConclusionOophoropexy seems to be efficacious in preventing recurrent torsion. It is our impression that plication of the utero-ovarian ligaments has advantages over other approaches insofar as surgical feasibility and anatomical conservation.     

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.     

Management of Isolated Tubal Torsion in a Premenarchal Adolescent Female with Prior Oophoropexy: A Case Report and Review of the Literature

BackgroundIsolated tubal torsion in a premenarchal adolescent girl is a rare phenomenon. Preoperative diagnosis remains a challenge.CaseA 14-year-old premenarchal girl, with a history of bilateral ovarian torsion treated by laparoscopic detorsion and oophoropexy two years prior, presented to the emergency room with lower abdominal pain accompanied by nausea and vomiting. Pelvic ultrasound demonstrated an enlarged left adnexa. Diagnostic laparoscopy revealed an isolated left tubal torsion. Surgical evidence of previous bilateral plication of the utero-ovarian ligaments was confirmed. Untwisting of the left fallopian tube immediately restored the vascular supply. Subsequently, her symptoms resolved.Summary and ConclusionClinicians should consider torsion of the fallopian tube in the differential diagnosis of lower abdominal pain in all female patients. Prompt laparoscopic intervention is essential. Oophoropexy, while usually efficacious, may not prevent recurrence.     

Torsion of a functional ovarian cyst in a premenopausal patient receiving tamoxifen.

We report a case of torsion of an ovarian follicular cyst that developed during treatment with tamoxifen for breast cancer. A 40-year-old Japanese woman was admitted complaining of acute lower abdominal pain. Eight months earlier, she had undergone a partial mastectomy and local irradiation for ductal carcinoma of her left breast, estrogen receptor-positive stage I (T(1a) N(1b) M(0)). The administration of tamoxifen, 20 mg/day, and doxifluridine, 600 mg/day, were started immediately postoperatively. Pelvic examination after admission revealed the left ovarian cyst and enlarged uterus. Transvaginal ultrasonography and computed tomography revealed a multilocular cystic mass in the pelvic cavity. The pathological diagnosis of the tumor after total hysterectomy and bilateral salpingo-oophorectomy was a typical follicular cyst with torsion and uterine leiomyoma. This ovarian cyst was believed to have developed during tamoxifen administration.     

Laparoscopic management of a ruptured benign dermoid cyst during advanced pregnancy

Benign cystic teratomas in pregnant women may be responsible for complications such as torsion, rupture and obstruction of labor. A woman in her 31st week of pregnancy with torsion of a large dermoid cyst and lipogranulomatosis peritonitis due to spilled cyst contents was managed laparoscopically with a favorable outcome. Trocar sites were selected according to the uterine size. Open laparoscopy allowed protection of the gravid uterus from penetrative injuries. Laparoscopic management of a voluminous adnexal mass may be safely performed during advanced pregnancy.     

Torsion of Fallopian Tube Remnant Associated with Noncommunicating Rudimentary Horn in Adolescent Girl with Unicornuate Uterus

BackgroundThe prevalence of müllerian anomalies may be as high as 7% in the general population, yet there is scant published literature on adnexal torsion occurring in these patients.CaseA 14-year-old female presented with right lower quadrant pain. Pelvic ultrasonography demonstrated a 2-cm simple right adnexal cyst. Diagnostic laparoscopy revealed a unicornuate uterus with a normal left uterine horn and fallopian tube but atretic and cordlike müllerian structures on the right side. Torsion of the right tubal remnant and two paratubal cysts were noted and the structures were then excised.Summary and ConclusionAgenesis, hypoplasia, or maldevelopment of müllerian structures may predispose patients to an increased risk of adnexal torsion secondary to looser ligamentous attachments and consequent lack of fixation to the pelvic sidewall.     

Coexistence of borderline ovarian epithelial tumor, primary pelvic hydatid cyst, and lymphoepithelioma-like gastric carcinoma

ObjectivesBorderline ovarian tumors (BOTs) represent a heterogeneous group of ovarian epithelial neoplasms. Despite a favorable prognosis, 10–20% of BOTs exhibit progressively worsening clinic. Primary involvement of pelvic organs with echinococcus is very rare. Lymphoepithelioma-like gastric carcinoma is a rare neoplasm of the stomach.Case ReportA 58-year-old woman referred with abdominal swelling and gastric complaints. Imaging studies revealed a huge cystic mass with multiple septations and solid component, another cystic mass with an appearance of cyst hydatid in the pelvis, and thickening of the small curvature of stomach. Gastroscopy revealed an ulcer with a suspicious malignant appearance, and histology of the endoscopic specimen showed severe chronic inflammation and lymphocytic infiltration. No other involvement of hydatid cyst was detected. In the exploration, there was a 25 cm cystic lesion with solid components arising from right ovary, another 6 cm cyst over the former, 7 cm cystic lesion arising from left ovary, and 10 cm mass near the small curvature of the stomach. Excision of the masses; total gastrectomy with esophagojejunal anastomosis; total abdominal hysterectomy; bilateral salpingo-oophorectomy; omentectomy; appendectomy; splenectomy; and pelvic, paraaortic, and coeliac lympadenectomy were performed. Final pathology revealed lymphoepithelioma-like gastric carcinoma, bilateral serous BOT, and hydatid cyst.DiscussionHydatid cyst should always be considered in the differential diagnosis of abdominopelvic masses in endemic regions of the world. Preoperative diagnosis of primary pelvic hydatid disease is difficult and awareness of its possibility is very important especially in patients residing in or coming from endemic areas.     

Laparoscopic Single-site “In-bag” Ovarian Dermoid Cystectomy in a 16-week- pregnant Patient

Study ObjectiveTo demonstrate a novel “in-bag” ovarian cystectomy technique for a large adnexal mass in pregnancy.DesignStepwise demonstration with narrated video.SettingAn academic tertiary care hospital. The patient was a 26-year-old woman, gravida 1, para 0, at gestational age of 7 weeks and 3 days who presented to the emergency department with persistent left pelvic pain and was diagnosed with a 16 cm × 10 cm × 12 cm dermoid cyst. She re-presented at gestational age of 16 weeks and 3 days with worsening pelvic pain, and the decision was made to proceed with surgical intervention.InterventionsLaparoscopic transumbilical single-site surgery for the surgical management of adnexal masses in pregnancy has been demonstrated to be feasible and safe 1, 2, 3. However, single-site laparoscopic ovarian cystectomy can be very challenging in pregnancy, especially when the need for suturing arises. Exteriorizing the ovary and cyst after intraperitoneal drainage may allow for extracorporeal suturing that is faster and easier; however, it may increase the probability of spillage of cystic contents if it is not performed in a bag, which can then cause peritonitis in cases of dermoid cysts. A combination of in-bag and extracorporeal ovarian cystectomy is a novel alternative minimally invasive approach that is cosmetic, safe, and effective.Several helpful techniques in this novel combination technique include the following: • Creating an umbilical incision of at least 2 cm or one that is large enough for better manipulation of both the surgical bag and adnexal mass. • Tightening the bag appropriately around the infundibulopelvic ligament so that it is not too tight leading to compromised blood supply and tissue necrosis, yet not too loose resulting in leakage of cystic contents. • Ensuring that the infundibulopelvic ligament is stabilized within the surgical bag. • Inserting small-sized wound retractor into the bag for better exposure during cystectomy. • Having a double-suction irrigation setup for large adnexal masses, as demonstrated in this patient, to reduce the spillage of cystic contents.The procedure was successfully performed in approximately 110 minutes, and the fetal heart rate postprocedure was 128 bpm through bedside transabdominal ultrasound. Estimated blood loss was 5 mL, and the patient was discharged the same day with an uneventful 4-week postoperative follow-up.ConclusionLaparoscopic single-site “in-bag” ovarian dermoid cystectomy is feasible, effective, and safe in pregnant patients with a large adnexal mass. This technique results in better stabilization of the ovarian cyst and reduction of cystic content spillage.     

Clinical and Computed Tomographic Features of Ovarian Lesions in Infants,Children, and Adolescents: A Series of 222 Cases

Study ObjectiveTo investigate the clinical and computed tomography (CT) characteristics of ovarian lesions in infants, children, and adolescents.Design, Setting, and ParticipantsA retrospective analysis of the clinical and CT data was performed in 222 patients who were 20 years or younger with ovarian lesions. Patients’ age, medical history, symptoms, tumor marker levels, and CT imaging findings were recorded.InterventionsNone.Main Outcome MeasuresIdentification of the clinical and CT features of ovarian lesions in infants, children, and adolescents.ResultsA total of 136 patients had abdominal pain, and 73 patients had palpable abdominal mass. The β-HCG was elevated in 4 and AFP was elevated in 16 of the 222 cases. A total of 235 lesions were found in 222 cases, including 75 non-neoplastic and 160 neoplastic lesions. Ovarian cyst exhibited homogeneous low density. The torsion of a normal-sized ovary demonstrated mild or no enhancement. The torsion associated with an ovarian mass demonstrated a thickened, hyperdense wall. Mature teratoma presented as a cystic mass, with bulk fat and coarse calcification. Immature teratoma appeared as a solid mass with foci of fat and fine calcification. Yolk sac tumor was shown as cystic–solid mass with intense enhancement of solid component. Wall and septation of benign epithelial tumors were relatively uniform in thickness; mural nodule was detected in borderline tumor; and malignant epithelial tumor was predominantly a solid mass with intense enhancement.ConclusionOvarian cyst is the most common non-neoplastic lesion. Torsion of a normal-sized ovary was the second most common non-neoplastic lesion, almost always causing abdominal pain. Germ cell tumor has the highest incidence among neoplastic lesions. Fat and calcification are highly specific for germ cell tumor. The elevation of AFP and HCG levels in serum indicates germ cell tumor. Ovarian epithelial tumor is usually large, benign, and predominantly cystic. The combination of clinical and imaging features is helpful for correct diagnosis.     

Non-Surgical Management of the Auto-Amputated Adnexa in the Neonate: A Report on Two Cases

BackgroundPrenatal ovarian torsion is a rare but significant gynecologic abnormality. Current literature has yet to establish standard management in the case of auto-amputated adnexa secondary to ovarian torsion in the neonate.CasesWe report 2 cases of abdominal masses that were diagnosed in the antenatal period and were clinically consistent with auto-amputated adnexa followed with serial ultrasonography until resolution.Summary and ConclusionTo our knowledge this is the first report in the literature to document resolution of 2 pelvic masses due to auto-amputated adnexa with expectant management. This suggests expectant management is an appropriate alternative to surgical management in carefully selected cases.     

Recurrent giant Gartner's duct cysts. A report of two cases

BACKGROUND: Gartner's duct cysts are cystically dilated wolffian duct remnants found in the upper anterolateral part of the vagina. Many such giant cysts are diagnosed during childhood and result from ectopic communication with the ureter or cervix. There is a paucity of literature on recurrent and giant cysts presenting among older women. CASES: A 43-year-old woman presented in 1981 with a 7 x 14-cm, left, paravaginal, cystic mass. This was initially drained vaginally, then marsupialized vaginally. Following marsupialization, the patient began to note large gushes of fluid from the vagina. Ultrasound demonstrated a 3-cm cyst thought to arise within the broad ligament. The patient required total abdominal hysterectomy/bilateral salpingo-oophorectomy for endometrial hyperplasia. Exploration revealed neither a broad ligament nor vaginal mass. Postoperatively, vaginal drainage continued. Computed tomography demonstrated a multiloculated, cystic mass left of the vaginal cuff. Exploratory laparotomy revealed the mass to be within the paravaginal space. The cyst was marsupialized into the peritoneal cavity. A 32-year-old woman was diagnosed in 1992 with an 8 x 10-cm right pelvic mass found on examination and confirmed by computed tomography. At exploratory laparotomy the mass was found to be within the paravaginal space and was resected vaginally. In 1999 the patient returned, complaining of rectal pain. Examination and ultrasound revealed a right, multiloculated pelvic mass displacing the rectum, uterus and vagina. Magnetic resonance imaging demonstrated that the mass was entirely inferior to the levator plate. The cyst was resected vaginally. CONCLUSION: Giant Gartner's cysts tend to be misdiagnosed as pelvic masses. Magnetic resonance imaging is the best imaging modality for localizing these cysts. Recurrences of giant cysts tend to be multiloculated. Management strategies for multiloculated recurrences include periodic surveillance, schlerotherapy and marsupialization into the peritoneal cavity.     

Asynchronous Bilateral Ovarian Torsion. A Case Report and Mini Review

BackgroundAdnexal torsion is a serious condition and delay in surgical intervention may result in loss of ovary. Children and adolescents who have suffered from uterine adnexal torsion may be at risk for asynchronous torsion of the contralateral adnexa.CaseWe report the case of asynchronous bilateral ovarian torsion in a 9-year-old girl, resulting in right and subsequently left salpingo-oophorectomy.ConclusionThe diagnosis of ovarian torsion often is delayed. When ovarian torsion is suspected, laparoscopy should be performed without delay, and conservative management should be strongly considered to prevent surgical castration. Oophoropexy of the ipsilateral and contralateral ovary should be considered to prevent a recurrent torsion.     

Hydrosalpinx with adnexal torsion in an adolescent virgin patient--A diagnostic dilemma: case report and review of the literature

Hydrosalpinx is one of the predisposing factors of adnexal torsion. However, because the incidence of hydrosalpinx in adolescent virgin patients is very rare, it may cause diagnostic dilemma, leading sometimes to suboptimal treatment. We present the case of an 18-yr-old female, not sexually active, presenting with acute lower right abdominal pain. The working diagnosis was of a simple ovarian cyst, so aspiration was performed. Abdominal symptoms reoccurred and sonography revealed a large hemorrhagic cystic mass adjacent to an edematous right ovary. The patient was referred to immediate laparoscopy due to suspected right adnexal torsion. On laparoscopy, the right adnexa was twisted three times causing an edematous ovary with a hematosalpinx. Detorsion was performed. Five weeks later, transabdominal ultrasound reviled normal bilateral ovaries and the hematosalpinx disappeared. In conclusion, hydrosalpinx, although very rare in adolescence, must be considered in the differential diagnosis. Aspiration in such cases is not the treatment of choice and moreover, it may cause complications.     

Ectopic ovary: a case of autoamputated ovary with mature cystic teratoma into the cul-de-sac

An ovary with a mature cystic teratoma which was autoamputated into the cul-de-sac and confirmed by laparoscopy is described. A 24-year-old woman with a history of chronic pelvic pain for 5 years presented with left abdominal pain. Magnetic resonance imaging revealed a left ovarian mass of 5 cm in diameter. The pain was relieved spontaneously after a few weeks. Laparoscopy was performed 5 months later. The mass was identified in the cul-de-sac partly enveloped in the omentum without any ligamentous or direct connection with the pelvic organs. There was no left ovary in its proper anatomical location. Histopathologic study revealed a mature cystic teratoma with viable ovarian tissue. These findings suggested autoamputation of the ovary either by inflammation or torsion, which is one of the mechanisms for the formation of an ectopic ovary.     

Multiloculated peritoneal inclusion cyst treated successfully with laparoscopic surgery

Background: Multiloculated peritoneal inclusion cysts or benign cystic mesotheliomas are most commonly found in women of reproductive years; the disease involves the abdomen, pelvis and retroperitoneum. Case: A 33-year-old, para 3-0-0-3, Thai woman presented to the hospital upon an incidental finding of left ovarian cyst and a mild pelvic pain for three months. Her pelvic examination revealed left ovarian cyst. Transvaginal ultrasonogram demonstrated a multiloculated cyst size 61×40×55 mm³ adherent to the normal left ovary. Laparoscopic surgery was performed. There was a multiloculated cyst 6 cm in diameter in the left pelvic cavity. The cyst was excised and the histopathology revealed inclusion cysts. She was well at discharge and throughout the four-week and six-month follow-up periods. Conclusion: Although multiloculated peritoneal inclusion cyst is uncommon, it should be included in the differential diagnosis of adnexal masses. Surgical excision remains the current recommended treatment for symptomatic disease. Surgical excision by laparoscopy might be an alternative method for successful management.     

Unilateral Ovarian Dermoid Cyst Accompanied by an Ipsilateral Paratubal Cyst in a Girl with Proteus Syndrome Discovered by Laparoscopic Surgery

BackgroundProteus syndrome is a rare hamartomatous disorder characterized by the overgrowth of multiple tissues in a mosaic pattern. Tumors of genitourinary tract in Proteus syndrome are uncommon.CaseWe here report a 5-year-old girl with Proteus syndrome who developed a cystic mass in the pelvic cavity. The cyst was discovered by ultrasonographic examination and finally proved to be a unilateral ovarian dermoid cyst accompanied by an ipsilateral paratubal cyst by laparoscopic surgery.Summary and ConclusionProteus syndrome accompanied by an ovarian cyst is rare in girls. The possibilities of underlying ovarian cyst required for surgical intervention should be considered in Proteus syndrome.     

Absence of unilateral adnexa associated with asymptomatic adnexal torsion and autoamputation

We present the case of a patient who had a pelvic mobile mass and unilateral absence of unilateral ovary and ipsilateral partial fallopian tube, which may be associated with asymptomatic adnexal torsion. She had had no previous abdominal operations and no history of acute abdominal pain, nausea or vomiting. There are two possible reasons for unilateral absence of adnexa: one is adnexal torsion and the other is congenital malformation.