Spontaneous Bilateral Torsion of Fallopian Tubes Presenting as Primary Infertility

Abstract

Background: Spontaneous torsion of the fallopian tubes is a rare condition, usually seen in acute settings or as incidental findings during laparoscopy. Presentation of the case: A 34-year-old nulligravid woman with inability to conceive for 7 months presented to our center. Her hysterosalpingography revealed a blind-ending right fallopian tube, and a blocked and dilated left fallopian tube. Laparoscopy showed a long, auto-amputated right fallopian tube, disconnected from the fimbriae and a long, dilated left fallopian tube, with several twists around its axis. Bilateral salpingectomy was performed. The patient subsequently underwent IVF, resulting in an ongoing pregnancy. Conclusion: Spontaneous torsion of the fallopian tubes can manifest as primary infertility without any antecedent symptoms and should be considered in the differential diagnosis of bilateral tubal obstruction.

Case presented at “Stump the Professors” 44th American Association of Gynecologic Laparoscopists Global Congress on Minimally Invasive Gynecology, NV, USA, 15–19 November 2015

Tubal disease is responsible for approximately 25–35% of female infertility [1]. Pelvic inflammatory disease (PID or salpingitis) is the most common cause for tubal disease, representing more than 50% of all cases [1,2]. Other causes of tubal disease include endometriosis, prior pelvic surgery, tubal diverticula or infections such as pelvic tuberculosis [2]. Although bilateral tubal disease has the most impact on fertility, unilateral tubal pathologies, such as the congenital absence of a fallopian tube or torsion with auto-amputation of the fallopian tube, have been reported as causes of infertility [3]. Spontaneous bilateral torsion of the fallopian tubes is an infrequent gynecologic condition that is rarely reported as a cause of infertility. In this context, we report the case of a 34-year-old nulligravid woman with primary infertility due to spontaneous bilateral torsion and occlusion of the fallopian tubes. Following laparoscopic bilateral salpingectomy, the patient underwent ovarian stimulation, IVF and a subsequent frozen-thawed embryo transfer cycle.

Presentation of the case

Patient details

A 34-year-old woman, gravida 0, was referred to our center due to inability to conceive for 7 months. Her menarche was at age 13 years, and she reported regular menstrual cycles, occurring at intervals of 28–30 days. She experienced mild dysmenorrhea, but did not have any dyspareunia. She had no prior history of sexually transmitted infections, and her medical and surgical history was unremarkable. The patient had used oral contraceptive pills until she began attempting to conceive. Her partner was a 44-year-old man with no proven fertility.

Initial assessment

The patient's initial work-up revealed normal hormonal testing – follicle-stimulating hormone: 2.75 mIU/ml, luteinizing hormone: 2.77 mIU/ml, antimüllerian hormone: 3.32 ng/ml, TSH: 1.23 uIU/ml, and prolactin: 4.87 ng/ml. Endocervical cultures were negative for Chlamydia trachomatis and Neisseria gonorrheae. Her partner's semen analysis was normal as follows – volume: 5.1 ml, concentration: 77 × 10⁶/ml, motility: 51%, and morphology: 3% normal forms (strict). A transvaginal pelvic sonogram revealed normal ovaries with an antral follicle count of approximately 30. Adjacent to the left ovary, a 68.3 × 14.3 mm tubular cystic structure was noted (Figure 1). The hysterosalpingogram performed by her gynecologist prior to her consultation with us was reviewed. A normal uterine cavity was visualized; however, the left fallopian tube was markedly dilated without any free intraperitoneal spillage (Figure 2, blue arrow). The right fallopian tube showed minimal fill, with a blind ending through which no intraperitoneal spillage was seen (Figure 2, red arrow). Given these findings of bilateral tubal occlusion, IVF was recommended. Prior to IVF, a diagnostic laparoscopy with excision of the hydrosalpinges was performed to improve chance of treatment success [4].

Figure 1.

Transvaginal ultrasongraphy showing the left adnexal tubular cystic structure.

Figure 2.

Hysterosalpingography findings. The right fallopian tube shows minimal fill, with a blind ending through which no intraperitoneal spillage is seen (red arrow). The left fallopian tube is markedly dilated without any free intraperitoneal spillage (blue arrow).

Treatment

The patient was brought to the operating room where diagnostic laparoscopy was performed. Overall, the pelvis was also noted to be normal, without any evidence of endometriosis or pelvic adhesions. The right fallopian tube was noted to be long, with dilation and blockage at the distal isthmic portion (Figure 3, red arrow). This portion of the fallopian tube was disconnected from the fimbriae (Figure 3, blue arrow). The right fallopian tube was excised with a 5 mm LigaSure^™ device (Covidien Ltd, Dublin, Ireland). Attention was then turned to the left fallopian tube, which was noted to be long with several twists around its axis (Figure 4). The fallopian tube was also dusky with a purple hue and markedly dilated (Figure 5). Although untwisting the fallopian tube was attempted, it was not considered salvageable and therefore, a left salpingectomy was performed with the LigaSure device. Gross pathology was significant for an 18 cm left fallopian tube, which is longer than the average length of a fallopian tube (8–12 cm). Histopathology showed a discontinuous segment of the right fallopian tube and also confirmed bilateral hydrosalpinges. The patient made an uneventful recovery and had no complaints during her postoperative visit 2 weeks later.

Figure 3.

Laparoscopic findings of the right fallopian tube. The right fallopian tube is long, with dilatation and blockage at the distal isthmic portion (red arrow). This portion of the fallopian tube is disconnected from the fimbriae (blue arrow).

Figure 4.

Laparoscopic findings of the left fallopian tube. The left fallopian tube is long with several twists around its axis.

Figure 5.

Laparoscopic findings of the left fallopian tube. The left fallopian tube is dusky with a purple hue, markedly dilated, blocked and necrotic-appearing.

Outcome & implications

The patient underwent controlled ovarian stimulation 6 weeks postoperatively. A total of 28 oocytes were retrieved, of which 22 fertilized with conventional insemination. A single fresh day-5 blastocyst was transferred, but this cycle was unsuccessful. In total, 11 day-5 blastocysts and two day-6 blastocysts were subsequently cryopreserved. The patient returned 2 months later for a frozen–thawed embryo transfer cycle. Frozen–thawed embryo transfer of a single blastocyst was performed 5 days after detection of the luteinizing hormone surge. The patient is currently pregnant with a single intrauterine gestation.

Discussion

Spontaneous torsion of the fallopian tube is a rare gynecologic condition, with an estimated incidence of one in 1.5 million women [5]. Risk factors for spontaneous fallopian tube torsion differ in pediatric and adult patients. In the pediatric population, hydrosalpinges due to endometriosis, bladder exstrophy and Hirschsprung's disease are considered risk factors [6]. However, in adults, intrinsic and extrinsic tubal risk factors have been described. Congenital anomalies of the fallopian tube, tubal ligation, tubal neoplasms, hydrosalpinges, hematosalpinges, excessive length or tortuosity of the tube, abnormal peristalsis and tubal hypermobility or spasm represent the former; paratubal or ovarian masses, pelvic adhesions, congestion of the mesosalpinx, sudden body movements, trauma and pregnancy are included in the latter [6]. It is thought that these risk factors create a pivot about which the fallopian tube can twist one or more times [6].

Typical symptoms associated with fallopian tube torsion, particularly in acute settings, include abdominal pain, nausea and vomiting. Pyrexia, tachycardia or leukocytosis may be also present. Abdominal guarding may develop as blood flow to the fallopian tube is compromised, though relief of pain may gradually ensue due to spontaneous detorsion of the fallopian tube, or nerve and tissue death in the affected area [6]. Furthermore, in cases of intermittent or chronic fallopian tube torsion, patients may remain asymptomatic [6]. Ultrasonographic findings of a dilated fallopian tube with a normal-appearing ipsilateral ovary should raise the suspicion of fallopian tube torsion, especially in the setting of acute pelvic pain [7]. In this context, a dilated fallopian tube appears as a hyperechoic wall with foci of echogenicity protruding into its lumen, and a tapering end leading to the cornua of the uterus [7].

The current patient's case highlights noteworthy aspects about the clinical presentation and pathogenesis of spontaneous fallopian tube torsion. When spontaneous fallopian tube torsion does occurs, it is likely to be unilateral, and more often on the right [6]. Reasons for the laterality include the presence of the sigmoid colon, which may prevent torsion of the left adnexal structures, dextrorotation of the uterus and the differential in the venous return of the fallopian tube. In most case reports, spontaneous fallopian tube torsion is hypothesized to occur in the setting of hydrosalpinges. When the ampullary portion of the fallopian tube is distended with fluid, the tubal fluid generally flows into the peritoneal cavity where it is reabsorbed. If the fimbriated end of the fallopian tube becomes agglutinated, the increasing accumulation of tubal fluid and resulting distention can cause the fallopian tube to twist over itself, especially when the tube is long. Hydrosalpinges are known to occur commonly due to PID, after tubal ligation, and sometimes during ovarian stimulation, which increases tubal secretions. However, hydrosalpinges can occur without any antecedent infection, and have been reported in young girls and virginal females with no specific risk factors [8]. Thus, in our patient, it seems likely that intrinsic factors (long fallopian tubes) and hydrosalpinges contributed to the risk of tubal torsion. Though we remain uncertain about the precise factors causing the hydrosalpinges, we postulate that they could be noninfectious as seen in young girls [8], or due to a remote sub-clinical infection. We also speculate that the torsion occurred asynchronously, in other words, the right before left. It is likely that torsion of the right fallopian tube subsequently led to auto-amputation of the tube, and thereby to discontinuity between the distal isthmic portion and fimbriae. By contrast, the duskiness of the twisted left fallopian tube indicated possible bleeding of tubal blood vessels or impending necrosis, indicating that torsion of this tube occurred at a later point. It is also possible that the patient had symptoms at the time of the torsion, but the symptoms were not severe to seek medical care. Tuboplasty rather than bilateral salpingectomy could have been considered in this patient. However, the disconnected right fallopian tube and the markedly dilated left fallopian tube swayed our intraoperative management towards bilateral salpingectomy over tuboplasty. Furthermore, most professional organizations recommend salpingectomy for hydrosalpinges before pursuing IVF given higher pregnancy rates in patients undergoing salpingectomy [2].

Executive summary

• Background

  • Spontaneous bilateral torsion of the fallopian tubes is an infrequent gynecologic condition that is rarely reported as a cause of infertility.
• Presentation of the case

  • Bilateral tubal occlusion seen on hysterosalpingography should be followed by diagnostic laparoscopy.
  • In the setting of bilateral tubal occlusion, diagnostic laparoscopy with excision of the hydrosalpinges should be performed prior to IVF to improve treatment success.
• Discussion

  • Hydrosalpinges can occur without any antecedent infection.
  • If bilateral fallopian tube torsion does occur, the torsion occurs asynchronously.
• Conclusion

  • Spontaneous bilateral torsion of the fallopian tubes can manifest as primary infertility without any antecedent symptoms, and should therefore be considered in the differential diagnosis of bilateral tubal obstruction or bilateral hydrosalpinges.

Conclusion

Previous reports in the medical literature have described spontaneous torsion of the fallopian tubes in acute settings or as an incidental finding during gynecologic laparoscopy. However, as highlighted by our case, spontaneous bilateral torsion of the fallopian tubes can manifest as primary infertility without any antecedent symptoms, and should therefore be considered in the differential diagnosis of bilateral tubal obstruction or bilateral hydrosalpinges. In such patients, diagnostic laparoscopy with salpingectomy before IVF should be considered standard of care, particularly when concomitant hydrosalpinges are noted.

Informed consent disclosure

The authors state that they have obtained verbal and written informed consent from the patient/patients for the inclusion of their medical and treatment history within this case report.