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Int J Surg Case Rep
Int J Surg Case Rep
International Journal of Surgery Case Reports
2210-2612
Elsevier

S2210-2612(24)01036-8
10.1016/j.ijscr.2024.110255
110255
Case Report
Torsion of distal right hydrosalpinx in a primiparous woman at term: A case report
Toumi Dhekra a
Medemagh Malek a
Ghaddab Imen a
Zoukar Olfa a
Chaouch Mohamed Ali docmedalichaouch@gmail.com
b⁎
Bergaoui Haifa a
a Department of Gynecology, Monastir University Hospital, Monastir, Tunisia
b Department of Visceral and Digestive, Monastir University Hospital, Monastir, Tunisia
⁎ Corresponding author. docmedalichaouch@gmail.com
07 9 2024
10 2024
07 9 2024
123 11025523 7 2024
1 9 2024
5 9 2024
© 2024 Published by Elsevier Ltd on behalf of IJS Publishing Group Limited.
2024

https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Introduction and importance

Adnexal torsion is a significant cause of acute pelvic pain and a common gynaecological emergency. While ovarian cysts are well-recognized predisposing factors, hydrosalpinx is a less common precursor. This case report presents a unique instance of isolated fallopian tube torsion accompanied by hydrosalpinx.

Case presentation

A 27-year-old primiparous woman at 37 weeks gestation presented with severe right lumbar pain. The patient's vital signs were stable, and abdominal and vaginal examinations were conducted. Imaging confirmed a progressing pregnancy and a 7.5 cm hemorrhagic cystic formation on the right side. Suspecting adnexal torsion, she underwent an urgent cesarean section and surgical exploration, which revealed a twisted distal right hydrosalpinx—the treatment involved detorsion, right salpingectomy, and ovarian suspension. Postoperative recovery was uneventful with histopathological confirmation of hydrosalpinx.

Discussion

Isolated fallopian tube torsion is rare, particularly during pregnancy. This condition's etiopathogenesis involves rotation of the tube around its ligamentous supports, possibly exacerbated by factors such as hydrosalpinx, pregnancy, and anatomical variances. Despite the availability of imaging techniques, diagnosis remains challenging, often confirmed only during surgical intervention. The literature highlights the importance of considering this diagnosis in pregnant women with acute pelvic pain and identifying characteristic ultrasound features.

Conclusions

Isolated tubal torsion in pregnancy is an exceptional clinical challenge. Early and accurate diagnosis is critical to prevent irreversible damage to the fallopian tube and preserve fertility. This case underscores the need for awareness among clinicians and provides insights into the effective management of such cases.

Highlights

• The case report details a rare instance of isolated fallopian tube torsion with hydrosalpinx in a 27-year-old woman at 37 weeks of pregnancy, emphasizing the rarity and diagnostic challenges.

• Highlighting the urgency and complexity of surgical management, the patient underwent a cesarean section followed by the successful detorsion and removal of the necrotic fallopian tube, leading to an uncomplicated postoperative recovery and preserving the patient's health.

• Despite advancements in imaging technologies, the diagnosis of isolated tubal torsion remains predominantly intraoperative. This case illustrates the critical need for high clinical suspicion and timely intervention to prevent severe complications such as irreversible ischemia of the fallopian tube.

Keywords

Hydrosalpinx
Case report
Obstetrics and gynecology
Pregnancy
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pmc1 Introduction and importance

Adnexal torsion represents a frequent gynaecological emergency and a notable source of acute pelvic pain in women [1]. Torsion can be favoured by the presence of an adnexal mass (ovarian cyst, hydrosalpinx…). Hydrosalpinx is an uncommon predisposing factor for adnexal or isolated fallopian tube torsion. Infection can lead to damage of the endosalpinx, causing distal occlusion of the tube and accumulation of exudate, which leads to its distention [2]. The diagnosis is confirmed intraoperatively by the presence of a twisted hydrosalpinx with at least one coil. We present an interesting case report, according to SCARE guidelines [3], of a tubal torsion accompanied by hydrosalpinx, diagnosed in a 27-year-old patient who was pregnant.

2 Case presentation

We report the case of a 27-year-old Arabian patient, with no notable medical history, primiparous and pregnant at 37 weeks gestation. The patient was regularly followed up during pregnancy. She presented with intense right lumbar pain for the past 6 h. On examination, the body mass index was 29 kg/m2. The patient was afebrile and hemodynamically stable. She exhibited tenderness on the right lumbar area upon abdominal examination. A vaginal examination revealed a long, closed, posterior cervix. Further investigations included a negative urinary culture. The biological data were normal. The ultrasound revealed a progressing monofetal pregnancy, a fetus with appropriate growth for gestational age, a posterior placenta positioned far from the cervix without signs of detachment, absence of dilation of the renal collecting system, and a 7.5 cm hemorrhagic cystic formation likely of ovarian origin (Fig. 1). Adnexal torsion was suspected. The patient was immediately transferred to the operating room for cesarean section and detorsion. The cesarean section proceeded uneventfully, and intraoperatively, a small amount of hemoperitoneum was observed, along with a twisted (3 turns) distal right hydrosalpinx measuring 5 cm in diameter (Fig. 2). The homolateral utero-ovarian ligament was Sectioned (Fig. 3). The management plan involved detorsion followed by right salpingectomy and ovarian suspension. The postoperative course was uncomplicated with favorable clinical progress. Histopathological analysis confirmed the presence of hydrosalpinx and the destruction of the internal structure of the fallopian tube.Fig. 1 The ultrasound image shows a cystic formation measuring 7.5 cm in diameter (indicated by a red arrow) with likely hemorrhagic content, presumably of ovarian origin.

Fig. 1

Fig. 2 Posterior view of the twisted hydrosalpinx with hemorrhagic content (→) after fetal extraction.

Fig. 2

Fig. 3 Anterior view of the twisted hydrosalpinx with hemorrhagic content and sectioned utero-ovarian ligament.

Fig. 3

3 Discussion

Isolated torsion of the fallopian tube is a rare condition first described in 1890 by Sutton [1]. Its exact incidence remains poorly understood. Several recent literature reviews cite a figure of 1 in 1,500,000, referring to the incidence reported in 1970 by Hansen [2]. The proportion of adnexal torsions occurring during pregnancy varies from 13 % to 28 % [3,4], and its frequency is estimated at 1 in 5000 pregnancies (7 cases per 1000 pregnancies in cases of associated adnexal mass). It can occur during all three trimesters of pregnancy, although the increased size of the uterus in the second and third trimesters reduces the mobility of the adnexa. The etiopathogenesis of tubal torsion is not well understood. It involves isolated fallopian tube torsion around the axis formed by the tubo-ovarian ligament [5]. The ovary is typically not compromised due to vascular collateralization provided by the dual blood supply from the ovarian artery and the uterine artery, which form an arcade around the tube and ovary [6]. Factors favouring this torsion mechanism have been described in the literature and classified into intrinsic factors (such as hydrosalpinx, hematosalpinx, tubal tumour, and tubal malformation) and extrinsic factors (such as ligament mobility, ovarian or para-ovarian mass, uterine volume increase (tumour or pregnancy), post-surgical adhesions, pre-menarche hormonal changes) [7]. It is conceivable that anatomy itself plays a role as a predisposing factor. The fallopian tube is highly mobile, anchored by the uterus, mesosalpinx, and infundibulopelvic ligament (ovarian fimbria). Bernardus described the mechanism of torsion as follows: obstruction of the venous and lymphatic vessels of the adnexa leads to congestion in the pelvis. This results in oedema of the fimbria or ovarian fimbria, promoting its torsion [8]. Right-sided predominance has been reported in several articles. Possible explanations include the lesser mobility of the left tube, which is anchored by the sigmoid colon, and the more frequent evaluation of right-sided pain in search of acute appendicitis [7,10]. In our case, the torsion was localized to the right side. Clinical signs of adnexal torsion include acute and abrupt pain (present in 98 % of cases), nausea and vomiting (in 78 % of cases), secondary to peritoneal irritation, and the presence of an adnexal mass (in 82 % of cases) [3]. During pregnancy, the pain may be confused with uterine contractions. Adnexal torsion can induce uterine contractions, thus posing a risk of miscarriage (early or late) or premature delivery. Similarly, nausea and vomiting are common in the first trimester of pregnancy. Additionally, the adnexal mass may be more challenging to palpate as the pregnancy advances. As the pregnancy progresses, the adnexa ascends with the uterus and is displaced posteriorly. Abdominal ultrasound is the first-line investigation for any woman presenting with acute abdominal pain. The diagnosis of isolated tubal torsion should be considered when a cystic mass is identified between the uterus and the normal ipsilateral ovary. Tubal torsion typically presents as a dilated tubular structure (a normal fallopian tube is usually not visible, and its normal diameter does not exceed 4 mm) [9], folded, developed between the uterus and the ovary. Its thickened, hyperechoic wall is identifiable by longitudinal folds that appear as incomplete septa in the longitudinal section and as small bulges in the transverse section (resembling a “cogwheel sign” or “beads on a string” appearance). Its contents are fluid-filled and may contain debris. Another characteristic sign of torsion is the tapered narrowing or “beak sign” at the junction of the tube and ovary. A third, pathognomonic sign is the “whirlpool sign,” representing the torsion itself [9]. Due to dual blood supply from both the ovarian and uterine arteries, the presence of colour Doppler flow does not definitively exclude torsion. The twisted and dilated tube may also mimic a nonspecific cystic mass. Differential diagnoses may include para-ovarian cysts and peritoneal pseudocysts. In our case, exploration of the cystic mass was limited by the size of the gravid uterus given the advanced stage of pregnancy. Surgical management should be as prompt as possible to prevent irreversible gangrene of the tube and preserve fertility. Treatment of adnexal torsion during pregnancy can be performed laparoscopically, following safety guidelines for the gravid uterus (insufflation in the left upper quadrant or open laparoscopy, moderate CO2 insufflation pressure, appropriate trocar positioning, gentle uterine mobilization, etc.). Treatment should be conservative if the adnexa can be salvaged after detorsion [11]. Salpingectomy could be a common treatment option for torsion accompanied by hydrosalpinx. Torsion accompanied by hydrosalpinx often presents with severe, acute abdominal pain. Salpingectomy can provide definitive relief from pain and remove the source of future potential pain. In addition, hydrosalpinx can cause chronic pelvic pain and discomfort. Removing the affected tube can alleviate these symptoms. We should also mention that this condition is often associated with pelvic inflammatory disease, which can lead to chronic infections and complications. Then, salpingectomy can reduce also the risk of recurrent infections and the potential for abscess formation. Several studies have shown that the presence of hydrosalpinx can negatively impact the success rates of in vitro fertilization. The fluid from a hydrosalpinx can leak back into the uterine cavity, creating a toxic environment for embryo implantation. Salpingectomy improves its outcomes by removing this source of fluid. Occasionally, hydrosalpinx may harbor malignancies or other pathologies that are not detectable through imaging alone. Salpingectomy allows for histopathological evaluation to rule out malignancy, providing a definitive diagnosis. Regarding these findings, salpingectomy is a well-supported treatment for torsion with hydrosalpinx due to its benefits in preventing recurrence, alleviating symptoms, reducing infection risk, and improving in vitro fertilization outcomes. However, treatment decisions should be individualized based on patient circumstances and preferences.

In our case, the pregnancy was at 37 weeks gestation, and laparoscopic surgery was not feasible due to the size of the uterus. The decision was made to perform fetal extraction concurrently. The management plan involved tubal detorsion followed by salpingectomy, as the tube was completely necrotic.

4 Conclusions

Isolated tubal torsion is a rare and poorly understood condition, exceptionally rare in pregnant women. It presents a challenge for clinicians to make an early diagnosis to save the fallopian tube from irreversible ischemia. It should be suspected in pregnant women presenting with acute pelvic pain, with ultrasound revealing a cystic mass adjacent to the uterus developed between the uterus and the normal ipsilateral ovary. Management should be prompt to preserve women's fertility.

Ethical approval

As a case report, it is exempted from ethical approval by the Institutional Board of Review, Monastir University Hospital, Monastir.

Funding

No funding.

Author contribution

All the authors participated in the manuscript and validated the final version of the manuscript.

Guarantor

Mohamed Ali Chaouch.

Research registration number

Not applicable.

Patient consent

Written informed consent was obtained from the patient to publish this case report and accompanying images. On request, a copy of the written consent form is available for review by the editor-in-chief of this journal.

Provenance and peer review

Not commissioned, externally peer-reviewed.

Conflict of interest statement

The authors declare no competing interest.

Acknowledgements

There were no acknowledgements to mention.
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