Quadruple torsion of the fallopian tube in an 18-year-old virgin: a rare twist.

in fertility problems when left untreated. The majority of these cases occur in combination with ovarian torsion due to ovarian pathology. However, isolated torsion of the fallopian tube is a more uncommon condition with an incidence estimated at 1 in 1.5 milli on women. 1 Possible risk factors include intrinsic factors and extrinsic factors. Intrinsic factors include abnormalities of the fallo- pian tube, for example, hydrosalpinx, paratubal cyst, neoplasm, tubal ligation device, ectopic preg- nancy and congenital anomaly. Extrinsic factors are defined as changes or abnormalities in organs close by the adnexa, mechanical factors and pelvic congestion such as ovarian mass, infection, adhe- sions and endometriosis. 2 3 Ca Se pre Sen TaT ion An 18-year-old Caucasian, nulliparous woman with no medical history, presented herself at the emer - gency department with abdominal pain in the right lower quadrant, along with nausea and vomiting. Her symptoms had started 3 days prior to her presentation and intensified over the last 2 hours. The pain was described as continuous pain with intermittent bouts of increased pain every 15 min. She was not sexually active yet and her menarche was at age 13. Her menstrual cycle was regular (28 days) with the first day of the last menstruation 9 days before presentation. She reported no dysmen- orr hea, any vaginal discharge or intermenstrual bleeding. Neither did she have any symptoms of the urinary or gastrointestinal tract. inve STiga T ion S On physical examination, the blood pressure was 126/58 mm Hg, with a heart rate of 53/min and a temperature of 37.2°C. There was normal tympanic sound and norm al bowel sounds on auscultation. The lower right quadrant of the abdominal region was painful during examination, without signs of peritoneal irritation. Given the patient’s virgo status, transvaginal ultrasound was not the first choice of imaging. Therefore, transabdominal ultrasound was performed 30 min after admission to the emergency department. The result was inconclusive, showing two echogenic unilocular cysts possibly related to the right ovary. The total size of the ovary was measured 74×56 mm. A fter thorough consideration and counselling of the patient, a shared decision was made to perform a transvaginal ultrasound to strive for better diagnostic accuracy. This revealed an echogenic unilocular cyst measuring 31×44 mm with possible relation to the right ovary. Doppler ultrasonography revealed blood flow on the edge of the right ovary. There was some free fluid in the pouch of Douglas, the deepest pocket measuring 11 mm. The lab oratory results showed a normal haemoglobin level (15.1 g/dL) and normal inflam- matory parameters (leucocytes 9.9×10 9/L, C reac- tive protein (CRP) 1.0 mg/L). Urinalysis revealed no signs of urinary tract infection. The urine preg- nancy test was negative. differen Tial diagno SiS Based on the patient’s history, the physical exam- ination and the ultrasound findings, ovarian torsion seemed to be the most likely diagnosis. However, presentation was atypical since the symptoms where subacute, there was no leucocytosis, and ultrasonic examination was inconclusive. Our differential diagnosis included painful ovulation, ovulation bleeding, ruptured ovarian cyst, haemor- rhagic follicle or issues related to another tract, for example, gastrointestinal. Trea T men T The patient was admitted to our gynaecology ward for observation. She received painkillers (paracetamol, diclofenac and morphine). At first, the symptoms seemed to be relieved, but due to increasing pain, a laparoscopy was performed Protected by copyright. on 24 October 2025 at Universiteit van Amsterdam.http://casereports.bmj.com/ BMJ Case Reports: first published as 10.1136/bcr-2018-224671 on 20 August 2018. Downloaded from 2 Zaat  tr , et al. BMJ Case Rep 2018. doi:10.1136/bcr-2018-224671 r are disease the same day, 11 hours after admission to the emergency department. Surgical findings revealed that the right fallopian tube was dark red in colour and enlarged, with an estimated size of 80×40 mm. There was a quadruple torsion of the right fallopian tube, with the fallopian fimbriae showing signs of necrosis (figure 1). The contralateral fallopian tube was normal. The right ovary was slightly enlarged with an otherwise normal appearance and without any signs of ovarian torsion. The uterus, left ovary, appendix, liver and diaphragm were all normal. Detorsion was easy to perform. However, after detorsion, the fallopian tube immediately twisted again due to substantial swelling. We decided to relieve the pressure in the hydrosalpinx by making a diathermic incision in the distal part of the right fallopian tube (figure 2). After the incision, serosal fluid leaked out and a successful detorsion could be performed. The distal part of the tube remained slightly dark but possibly viable, the proximal part appeared viable (figure 3). The total blood loss was minimal, and the postoperative period was uneventful. o uTCome and follow-up After the surgery, the symptoms subsided rapidly, and the patient was discharged on the same day. Ultrasonic evaluation 38 days later showed normal ovaries and an echogenic area on the right side measuring 2×3 cm, suspicious for persistent hydrosalpinx ( figure 4). Because of the asymptomatic postoperative period, the patient was treated conservatively. The patient did not report any issues during the 90 days following surgery. She was informed about an increased risk of an ectopic preg- nancy in the future and therefore the necessity of an early ultra- sonic diagnosis to localise the pregnancy.

Isolated torsion of the fallopian tube due to hydrosalpinx is a very rare condition. Most of the published case reports and case series concern women in the reproductive period. The diag- nosis of an isolated torsion of the fallopian tube is challenging. The clinical presentation is usually not specific and resembles other, more common causes of lower abdominal pain, such as appendicitis, ovarian torsion or rupture of an ovarian cyst or haemorrhagic follicle. Sixty per cent of patients have symptoms of acute onset of abdominal pain in the lower quadrant. Signs of peritoneal irritation are present in 30% of the patients. Other symptoms such as anorexia, nausea, vomiting, fever or vaginal bleeding are inconsistent. 4 Laboratory test may show a mild leucocytosis and an increased CRP level. Diagnosis is most commonly confirmed by diagnostic laparoscopy. Ideally, diagnosis is made preoperatively. However, f igure 1 P erioperative photograph of the right enlarged fallopian tube (estimated measuring 80×40 mm). There was a quadruple torsion of the right fallopian tube and the proximal part with the fallopian fimbriae showing signs of necrosis. f igure 2 P erioperative photograph immediately after the diathermic incision in the distal portion of the right fallopian tube. f igure 3 P erioperative photograph after incision and detorsion. The distal part of the tube remained somewhat dark but possibly viable, the proximal part appeared viable. f igure 4 Ultrasonic examination 38 days after the surgery. Echogenic area on the right side measuring 2×3 cm, suspicious for persistent hydrosalpinx. Protected by copyright. on 24 October 2025 at Universiteit van Amsterdam.http://casereports.bmj.com/ BMJ Case Reports: first published as 10.1136/bcr-2018-224671 on 20 August 2018. Downloaded from 3 Zaat TR, et al. BMJ Case Rep 2018. doi:10.1136/bcr-2018-224671 r are disease diagnosis of isolated fallopian tube torsion with imaging modal- ities, such as ultrasound, CT and MRI, remains challenging. Sun et al analysed the ultrasonic features of isolated fallopian tube torsion in 11 patients, only 4 cases were diagnosed correctly before surgery (36.4%). The most common findings where cystic masses (36.4%) and tubular structures (27.3%). 5 Also, in a recently published retrospective cohort study, Raban et al eval- uated the diagnostic value of ultrasound imaging in 27 patients with a surgical diagnosis of isolated fallopian tube torsion. In 29.6%, isolated fallopian tube torsion was correctly diagnosed by ultrasound before surgery. Fallopian tube oedema and the presence of a paraovarian cyst were the most common findings in patients with fallopian tube torsion. 6 Even though colour Doppler ultrasound can be useful, the presence of normal flow does not rule out torsion, due to the double vascularisation of the fallopian tube. 7 A sonographic whirlpool sign, which is used to describe the finding of a small heterogeneous mass with a concentric ring that is provoked by rocking movement of the probe over the mass, might be the most specific sign for fallo- pian tube torsion. 5 8 9 In the study of Raban et al, the whirlpool sign was reported in six patients, of whom two had an accu - rate diagnosis of isolated fallopian tube torsion. 6 Unfortunately, in our presented case, evaluation of the whirlpool sign was not performed. Alternatively, CT may provide additional informa- tion compared with ultrasound and is often performed to exclude other causes of acute lower abdominal pain such as appendi- citis. Suggestive CT features for isolated fallopian tube torsion include a dilated fluid-filled structure adjacent to the ovary and a thickened enhancing wall of the fallopian tube. 10 11 However, CT is rarely performed because of limited contrast, and findings are non-specific. 12 Finally, Sakuragi et al retrospectively evalu- ated the specific MRI findings in 12 cases of surgically proven isolated fallopian tube torsion. The key specific findings in this study where the whirlpool sign and plicae tubariae which is the presence of mucosal folds on a twisted fallopian tube. 12 MRI is a time-consuming and costly imaging modality, and its use for preoperative diagnosis of fallopian tube torsion has rarely been reported. In conclusion, preoperative diagnosis of isolated fallopian tube torsion is difficult. The key to improving diagnosis based on imaging modalities is increasing awareness of this rare disorder among clinicians. Several risk factors of fallopian tube torsion have been identi- fied which are divided into intrinsic or extrinsic factors. 3 Intrinsic factors include abnormalities of the fallopian tube, and extrinsic factors are defined as changes or abnormalities in organs close by the adnexa. As described in a case series, torsion of the right- sided fallopian tube is more commonly diagnosed compared with the left side. There are two possible explanations for this finding. First, the left fallopian tube has a preventive anatomic position in between the sigmoid colon and uterus. In addition, there is a relatively increased venous return to the left compared with the right fallopian tube, reducing possible congestion and subsequent risk of enlargement. Second, right-sided abdominal pain is more frequently evaluated because of the differential diagnosis which includes appendicitis, and therefore it is possible that the diagnosis is made more frequently. 13 Fallopian tube torsion with hydrosalpinx is a very rare condi- tion in adolescent girls with no history of pelvic inflammatory disease or surgical intervention. The presumed aetiology of events in our patient is that hydrosalpinx was either a congen- ital abnormality or was a consequence of the fallopian tube torsion. In a case series of 13 patients, Boukaidi et al reported on cases comparable with our case. 13 In this series, the mean age of the patients was 13.5 years (range 11–18 years) and all of the patients were sexually inactive with no previous medical history. It is presumed that activation of the ovarian and tubal function caused by high FSH levels before the onset of the menses may unveil a previously asymptomatic distal occlusion of the tube, causing hydrosalpinx. Possible explanations for the distal occlu- sion may be an episode of asymptomatic pelvic inflammation during childhood or asymptomatic torsion of the hydatid of Morgagni. 13 There are several therapeutic options described for the treat- ment of isolated fallopian tube torsion, all with the common goal of salvaging adnexal blood supply. Emergency detorsion of the tube is the first choice of treatment. Complete or partial salpingectomy might be indicated in cases of ischaemic, irreversible changes of the tubal wall. Lapa - roscopic tubal clip occlusion has been described as an alterna- tive to salpingectomy. 14 Most of the published cases of fallopian tube torsion with hydrosalpinx underwent salpingectomy. In the series of Boukaidi et al, 33% of the patients underwent salpingectomy as initial treatment. Eventually, 83% of the patients underwent a salpingectomy because of recurrent torsion after the conservative treatment. The mean recurrence period was 68 days (43–83 days). Boukaidi et al proposed a conser - vative surgical management flow chart. The first step in this flow chart is surgical detorsion and puncture of the hydrosal - pinx. After 3 weeks, a second-look laparoscopy and salpingos- copy should be performed to evaluate the tubal function and the potential indication for conservative management, salpingos- tomy or salpingectomy. 13 The largest cohort study, performed by Bertozzi et al, reported on the management of isolated fallo - pian tube torsion with hydrosalpinx in a paediatric population of 21 cases. Bertozzi et al concluded that diagnostic laparoscopy should be performed as soon as possible to preserve the fallopian tube. Based on the results of this cohort study, Bertozzi et al stated that conservative management is to be preferred whenever possible, in order to provide the best outcome for future fertility for the patients. 14 Concerning future conception capacity in patients with one-sided hydrosalpinx, there is no consensus on the type of treatment in the literature. Fair chances of spontaneous preg- nancy and successful IVF treatment after tube-preserving surgery opposed to salpingectomy have been reported.15 However, there is evidence that laparoscopic salpingectomy or tubal occlusion of unilateral hydrosalpinx increases the potential for spontaneous pregnancy and improves outcomes of IVF treatment compared with no surgical intervention. 16 17 Unfortunately, no information about fertility outcome in patients with prior treatment of torsion of the fallopian tube with hydrosalpinx is described in these patients. 13 18 However, future adverse impact on fertility due to pathology of the remaining tube is always unpredictable and has to be kept in mind. Therefore, we believe that tubal conservation should be favoured when possible. We believe that a second-look laparoscopy, as advised by Boukaidi et al 13 in their flow chart, is not always indicated. It can be considered based on the clin - ical symptoms and findings on ultrasound at the postoperative visit. In our case, we decided not to perform a second-look lapa- roscopy, and with this conservative management we prevented another operative procedure for our patient. Concerning future conception, we advised our patient to pursue the chance of spontaneous conception for 1 year . If conception does not occur within 1 year , she will be attending the fertility clinic for further analysis. In case of persistent hydrosalpinx, it may be indicated to perform a salpingectomy to increase the changes of spontaneous conception or pregnancy after in vitro fertilisation. Protected by copyright. on 24 October 2025 at Universiteit van Amsterdam.http://casereports.bmj.com/ BMJ Case Reports: first published as 10.1136/bcr-2018-224671 on 20 August 2018. Downloaded from 4 Zaat  tr , et al. BMJ Case Rep 2018. doi:10.1136/bcr-2018-224671 r are disease In order to gain knowledge about the rare condition of isolated fallopian tube torsion, its optimal treatment and influence on future fertility, it is necessary to perform more well-designed prospective studies, with long-term follow-ups. l earning points ► Isolated torsion of the fallopian tube with hydrosalpinx is a rare condition but should be considered as a cause of lower abdominal pain in women. ► The clinical presentation of torsion of the fallopian tube is non-specific and resembles other , more common causes of lower abdominal pain. ► Preoperative diagnosis of isolated fallopian tube torsion is difficult. The key to improving diagnosis based on imaging modalities, such as ultrasound, is increasing awareness of this rare disorder among clinicians. ► Diagnostic laparoscopic surgery needs to be performed when a women of reproductive age presents herself with acute abdominal pain. ► Therapeutic options in relation to future conception capacity after fallopian tube torsion with hydrosalpinx include tube- preserving surgery and salpingectomy. There is no consensus on the treatment options yet. Contributors a ll authors have made a significant contribution to the manuscript. t hey have read and approved the fi nal version. t Z contributed to patient care, conducting manuscript, reporting, conception and design. MB contributed to patient care, conducting manuscript, analysis of data and literature. e K contributed to patient care, analysis of data and literature, revising and supervision. LW p contributed to patient care, analysis of data and literature, revising and supervision. f unding the authors have not declared a speci fi c grant for this research from any funding agency in the public, commercial or not-for-pro fi t sectors. Competing interests None declared. patient consent o btained. p rovenance and peer review Not commissioned; externally peer reviewed. Refe Rences 1 Hansen o H. Isolated torsion of the Fallopian tube. Acta Obstet Gynecol Scand 1970;49:3–6. 2 Comerci G , Colombo FM, s tefanetti M, et al. Isolated fallopian tube torsion: a rare but important event for women of reproductive age. Fertil Steril 2008;90:1198.e23–5. 3 v an der Zanden M, Nap a , van Kints M. Isolated torsion of the fallopian tube: a case report and review of the literature. Eur J Pediatr 2011;170:1329–32. 4 Lo LM, Chang s D, Lee CL, et al. Clinical manifestations in women with isolated fallopian tubal torsion; a rare but important entity. Aust N Z J Obstet Gynaecol 2011;51:244–7. 5 s un Lt, Ning C p, Guo XJ, et al. r ole of ultrasound in diagnosing isolated torsion of fallopian tube. J Obstet Gynaecol Res 2014;40:208–14. 6 r aban o, Zilber H, Hadar e , et al. Isolated fallopian tube torsion: a unique ultrasound identity or a serial copycat? J Ultrasound Med 2018. doi: 10.1002/jum.14595. [ e pub ahead of print 24 Mar 2018]. 7 Baumgartel p B, Fleischer aC , Cullinan J a , et al. Color Doppler sonography of tubal torsion. Ultrasound Obstet Gynecol 1996;7:367–70. 8 V ijayaraghavan s B, s enthil s. Isolated torsion of the fallopian tube: the sonographic whirlpool sign. J Ultrasound Med 2009;28:657–62. 9 Harmon JC , Binkovitz L a , Binkovitz L e . Isolated fallopian tube torsion: sonographic and C t features. Pediatr Radiol 2008;38:175–9. 10 r ezvani M, s haaban a M. Fallopian tube disease in the nonpregnant patient. Radiographics 2011;31:527–48. 11 s aloum NMI, Hunt HW, Fadl sa . Core curriculum case illustration: a rare case of an isolated fallopian tube torsion. Emerg Radiol 2017. 12 s akuragi M, Kido a , Himoto Y , et al. M r I fi ndings of isolated tubal torsions: case series of 12 patients: M r I fi ndings suggesting isolated tubal torsions, correlating with surgical fi ndings. Clin Imaging 2017;41:28–32. 13 Boukaidi sa , Delotte J, s teyaert H, et al. thirteen cases of isolated tubal torsions associated with hydrosalpinx in children and adolescents , proposal for conservative management: retrospective review and literature survey. J Pediatr Surg 2011;46:1425–31. 14 Bertozzi M, Magrini e , r iccioni s, et al. Isolated fallopian tube torsion with hydrosalpinx: r eview of a debated management in a pediatric population. J Pediatr Surg 2017;52:1553–60. 15 Chanelles o, Ducarme G, s ifer C, et al. Hydrosalpinx and infertility: what about conservative surgical management? Eur J Obstet Gynecol Reprod Biol 2011;159:122–6. 16 Johnson N p, Mak W, s owter MC. s urgical treatment for tubal disease in women due to undergo in vitro fertilisation. Cochrane Database Syst Rev 2004;3:CD002125. 17 s agoskin aW , Lessey B a , Mottla GL, et al. s alpingectomy or proximal tubal occlusion of unilateral hydrosalpinx increases the potential for spontaneous pregnancy. Hum Reprod 2003;18:2634–7. 18 p ampal a , a tac GK, Nazli Z s, et al. a rare cause of acute abdominal pain in adolescence: hydrosalpinx leading to isolated torsion of fallopian tube. J Pediatr Surg 2012;47:e31–e34. Copyright 2018 BMJ publishing Group. a ll rights reserved. For permission to reuse any of this content visit http://group.bmj.com/group/rights-licensing/permissions. BMJ Case r eport Fellows may re-use this article for personal use and teaching without any further permission. Become a Fellow of BMJ Case r eports today and you can: ► s ubmit as many cases as you like ► e njoy fast sympathetic peer review and rapid publication of accepted articles ► a ccess all the published articles ► r e-use any of the published material for personal use and teaching without further permission For information on Institutional Fellowships contact [email protected] Visit casereports.bmj.com for more articles like this and to become a Fellow Protected by copyright. on 24 October 2025 at Universiteit van Amsterdam.http://casereports.bmj.com/ BMJ Case Reports: first published as 10.1136/bcr-2018-224671 on 20 August 2018. Downloaded from