Fallopian Tube Torsion in the Pediatric Age Group

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1 CASE SERIES Fallopian Tube Torsion in the Pediatric Age Group Radiologic Evaluation Srikala Narayanan, MD, Anjum Bandarkar, MD, Dorothy I. Bulas, MD Fallopian tube torsion is a rare but important cause of acute pelvic pain in young adolescent girls. It is a surgical emergency treated with either detorsion or salpingectomy. The imaging findings can be nonspecific and challenging. However, an accurate early diagnosis is essential for prompt surgical treatment. Our objective was to review whether imaging findings can be specific enough to suggest the diagnosis of tubal torsion prospectively in the appropriate clinical setting. An Institutional Review Board approved retrospective review of our imaging database from 2005 to 2012 revealed 10 surgically proven cases of fallopian tube torsion. All cases had sonography performed; 5 cases had additional multidetector computed tomography. All 10 patients (9 17 years) presented with acute pelvic pain. Sonographic findings included dilated tubular structures in 6 of 10 cases: adjacent to a normal ipsilateral ovary in 5 of 6 and adjacent to a benign ovarian teratoma in 1. In 4 cases, no dilated tube was identified; 3 of 4 had a cystic mass separate from the ovaries, and 1 had the imaging appearance of a multicystic ovary. Computed tomographic findings in the 5 cases that underwent multidetector computed tomography included a dilated tubular structure in 3 of 5; 2 of 5 had a cystic adnexal mass identified. Although rare, tubal torsion should be considered in female adolescents with acute pelvic pain. Sonography should be the first imaging choice. When a tubular structure or a midline cystic mass associated with a normal ipsilateral ovary is noted, tubal torsion should be considered in the differential diagnosis. Key Words acute pelvic pain; adnexa; children; computed tomography; pediatric ultrasound; sonography; tubal torsion Received November 11, 2013, from the Division of Diagnostic Imaging and Radiology, Children s National Medical Center, Washington, DC USA. Dr Narayanan is currently with the Department of Radiology, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania USA. Revision requested December 6, Revised manuscript accepted for publication January 9, Address correspondence to Dorothy I. Bulas, MD, Division of Diagnostic Imaging and Radiology, Children's National Medical Center, 111 Michigan Ave NW, Washington, DC USA. dbulas@childrensnational.org Abbreviations MDCT, multidetector computed tomography doi: /ultra F allopian tube torsion is a rare but important cause of acute pelvic pain in young adolescent girls. Bland-Sutton first described this entity in Isolated fallopian tube torsion has a reported incidence of 1 per 1.5 million. 2,3 Clinical examination findings may mimic appendicitis, pyosalpinx, a complex adnexal cyst, or a neoplasm. The imaging findings can be nonspecific and challenging. Treatment remains controversial, and detorsion is favored based on gross examination of the tube. 4 The exact correlation between the duration of torsion and nonviability is unclear. The outcome of recovery of torsed tubes from severe ischemia is unknown. It has been proposed that laparoscopic detorsion is a more conservative approach than conventional treatment by salpingectomy and may prove to have a better impact on repro by the American Institute of Ultrasound in Medicine J Ultrasound Med 2014; 33:

2 ductive potential. 5 Thus, accurate early diagnosis with imaging would be helpful for guiding appropriate therapy. Our objective was to review whether imaging findings can be specific enough to suggest the diagnosis prospectively in the appropriate clinical setting. Materials and Methods An Institutional Review Board approved and Health Insurance Portability and Accountability Act compliant study was performed by retrospective review of cases from 2005 to 2012 using electronic medical records and an intrahospital radiology imaging database (Montage Healthcare Solutions, Inc, Philadelphia, PA) search engine. Ten surgically proven cases of fallopian tube torsion were identified, and the imaging findings were reviewed along with final pathologic reports, when present. All 10 cases had pelvic sonography performed before surgery. One pelvic sonographic examination performed both transabdominally and transvaginally (transvaginal imaging provided superior resolution); the remaining cases had only transabdominal scans due to the young age and sexual inactivity of the patients (LOGIQ E9; GE Healthcare, Milwaukee, WI). In 5 of 10 cases, multidetector computed tomography (MDCT) was also performed before surgery (64-slice MDCT; Discovery PET/CT 690; GE Healthcare; or 64-slice MDCT; Brilliance CT; Philips Healthcare, Andover, MA). In 2 cases, MDCT was performed before sonography; in the remaining 3 cases, MDCT was performed after sonography. Results Clinical Data Patient s ages ranged from 9 to 17 years; 1 was premenarcheal, and the others were postmenarcheal. All patients presented to the emergency department with acute lower abdominal or pelvic pain. Four of 10 patients had acute pain for 24 hours, whereas the remaining patients had pain lasting from 3 days to 3 weeks with worsening pain on presentation to the emergency department. No patient had ongoing symptoms of dysuria or a documented history of sexually transmitted disease on review of medical records. Table 1 summarizes the clinical histories, imaging findings, and management. Sonographic Findings Sonographic findings included dilated tubular structures in 6 of 10 cases: 5 of 6 had a dilated tube adjacent to a normal ipsilateral ovary (Figure 1), and 1 had a benign ovarian teratoma. In 4 cases, no discrete dilated tube was identified; 3 of 4 had a cystic mass separate from normalappearing ovaries. On histopathologic examination, 2 of these turned out to be paratubal cysts (Figure 2), whereas the midline cystic mass was a torsed tube (Figure 3). The remaining case was noted to have an enlarged multicystic-appearing ovary that was initially thought to be an ovarian mass or possible ovarian torsion. At surgery, the cystic mass was actually a torsed tube adjacent to a normal ovary (Figure 4). On sonography, 5 of 10 cases had normal ovaries bilaterally, whereas the remaining 5 had ovarian abnormalities. Two ovaries were noted to have hemorrhagic cysts at surgery, whereas 1 contained a benign teratoma. The remaining 2 cases with abnormal-appearing ovaries on sonography were actually normal at surgery. In 1 case, the torsed tube adjacent to the normal ovary mimicked a multicystic ovarian mass (Figure 4). In the second case, a mesovarian cyst was noted to be adjacent to a normal ovary at surgery, which acted as a lead point for the fallopian tube torsion. Three of 10 cases had well-defined simple cystic masses separate from normal-appearing bilateral ovaries, without a discrete tubular structure, as described above. Six of 10 cases showed the classic fusiform dilated tubular structure, with internal echogenicity in 4 and the beak sign, which refers to tapered ends of the fallopian tube, in 3. Two cases had clear fluid within the dilated tube. Secondary findings included a small amount of simple free fluid in 5 of 10 cases. Multidetector Computed Tomographic Findings Of the 5 MDCT scans performed, 3 showed a dilated tubular structure (Figures 1c and 5). In 2 cases, a cystic adnexal mass was identified. At surgery, 1 of the cystic adnexal masses was a torsed tube with a paratubal cyst and an adjacent viable ovary (Figure 2, c and d), whereas the other was a torsed tube with an adjacent mesovarian cyst (Figure 6). In these 2 cases, MDCT did not separately identify normal ovaries. Of note, in 1 of these cases, sonography was better than MDCT in showing a normal ovary adjacent to the cystic mass. Secondary MDCT findings included a small amount of simple free fluid in 3 of 5 cases and peritubular fat stranding, vascular congestion, and thickening of the broad ligament in 1 case (Figure 1c). Management Salpingectomy was performed in 3 cases. The durations of pain in these 3 cases were 2 days, 5 days, and 3 weeks, respectively. In 1 case, detorsion was performed initially, followed by salpingectomy when the tube was found to be blind ending. Laparoscopic detorsion was performed in 1698 J Ultrasound Med 2014; 33:

the remaining 6 cases, accompanied by other procedures, including ovarian cyst drainage in 3, marsupialization of a mesovarian cyst in 1, and removal of a benign teratoma in 1 (Table 1).

3 the remaining 6 cases, accompanied by other procedures, including ovarian cyst drainage in 3, marsupialization of a mesovarian cyst in 1, and removal of a benign teratoma in 1 (Table 1). The basis for decisions to perform salpingectomy versus detorsion was unclear in our series and likely reflected the gross surgical findings. All patients with pain for less than 24 hours underwent successful detorsion, suggesting that early diagnosis improves the chances for successful conservative management. Discussion Fallopian tube torsion is an exceedingly rare but treatable cause of acute pelvic pain. Reported risk factors include a long mesosalpinx, immediate premenstrual congestion of the tube, prior tubal ligation, adnexal cysts, hydrosalpinx, pelvic inflammatory disease, hypermobility of the fallopian tube, and trauma. 6 9 Pelvic congestion secondary to obstruction of adnexal veins and lymphatic vessels is a pro- Figure 1. Case 1: 13-year-old patient with acute pelvic pain for 2 days. a, Transabdominal pelvic sonogram of a dilated tubular structure (large arrow) with internal debris and a tapering end (beak sign). b, Normal ipsilateral ovary. c, Axial MDCT showing a dilated tubular structure with layering of debris (large arrow). Secondary signs of peritubular fat stranding, vascular congestion, and thickening of the broad ligament (small arrows) are shown. J Ultrasound Med 2014; 33:

b, Oblique transabdominal sonogram showing a cystic midline structure adjacent to a normal-appearing right ovary (arrow).

4 Figure 2. Case 2: 14-year-old patient with worsening right lower quadrant pain for 5 days. a, Transverse transabdominal pelvic sonogram showing a cystic midline structure connected to a dilated tubular structure (arrow). b, Oblique transabdominal sonogram showing a cystic midline structure adjacent to a normal-appearing right ovary (arrow). c, Coronal MDCT showing a cystic midline structure connected to a dilated tubular structure (arrow). The right ovary was difficult to separately visualize on CT. d, Axial MDCT showing a cystic midline structure connected to a dilated tubular structure on the left with a normal left ovary (arrow). This structure was a paratubal cyst on histopathologic examination in this patient with left-sided tubal torsion. Figure 3. Case 3: 13-year-old patient with right lower quadrant and pelvic pain for 3 weeks. a, Cystic midline structure with normal bilateral ovaries (b, right; c, left; arrows) in this patient with a torsed tube mimicking a cyst J Ultrasound Med 2014; 33:

posed mechanism of tubal torsion. 10 12 Adnexal cysts (either paratubal or paraovarian) have been reported in the pediatric age group and may act as lead points.

the broad ligament between the ovary and tube. 6 In our series, 2 cases had a presumed lead point (benign teratoma and mesovarian cyst), and another 2 had associated benign paratubal cysts.

frequent imaging evaluations of right-sided pain to exclude appendicitis. 3,7,9,11,14,15 However, in our series, equal incidence was noted (5 right and 5 left).

5 posed mechanism of tubal torsion Adnexal cysts (either paratubal or paraovarian) have been reported in the pediatric age group and may act as lead points. 3,6,13,14 Paratubal cysts or hydatid cysts of Morgagni are unilocular thin-walled cysts attached to the fimbriated end of the fallopian tube, whereas paraovarian cysts arise from the paraovarium of the broad ligament between the ovary and tube. 6 In our series, 2 cases had a presumed lead point (benign teratoma and mesovarian cyst), and another 2 had associated benign paratubal cysts. It has been reported that torsion of the right fallopian tube is more common, related to fixation of the left tube by the sigmoid colon and mesentery, limiting excessive movement, or due to more frequent imaging evaluations of right-sided pain to exclude appendicitis. 3,7,9,11,14,15 However, in our series, equal incidence was noted (5 right and 5 left). Presentation typically is in ovulating women, with only a few reported cases in premenarcheal girls. 6,16,17 In our series, 1 girl was premenarcheal. Figure 4. Case 6: 13-year-old patient with right lower quadrant pain for 24 hours. a, Torsed tube mimicking a multicystic ovary. b, The longitudinal tubular appearance was not recognized preoperatively. On surgery, a torsed tube adjacent to a normal ovary was found. Figure 5. Case 10: 15-year-old patient with left lower quadrant pain for 1 day. Axial MDCT showing a coiled tubular structure anterior to the bladder (arrow), surgically proven to be a torsed fallopian tube. Figure 6. Case 9: 14-year-old patient with acute left lower quadrant pain. Axial MDCT image showing a multicystic left adnexal mass (long arrow), surgically proven to be a mesovarian cyst with associated fallopian tube torsion. The normal right ovary is marked by the short arrow; the left ovary could not be identified separately on MDCT but was normal at surgery. J Ultrasound Med 2014; 33:

6 Specific findings include a normal-appearing uterus and normal-appearing ovaries with normal flow, with a fusiform dilated tube representing a torsed tube. These findings were noted in 6 of our cases. 6,9,17 21 The dilated tube can have thickened echogenic walls with internal debris or hemorrhage. These findings were noted in 4 of our cases. Another specific sign is the beak sign: the tapered end of a dilated tube secondary to twisting. 9,11 This sign was seen in 3 of our cases (Figure 1a). Sonographic findings may be nonspecific. A torsed tube can mimic a cyst similar to a fluid-filled balloon knotted at one end (Figure 3) or can mimic a multicystic mass (Figure 4). A fluid-filled tube coursing posterior to the uterus could mimic free fluid in the cul-de-sac (Figure 7). Thus, fallopian tube torsion should be considered in young adolescent girls with acute pelvic pain, normal-appearing ovaries, and cystic collections. 14,17 20 Table 1. Summary of Clinical Histories, Imaging, and Management in 10 Patients With Proven Tubal Torsion Preoperative Age, Imaging Surgical and Case y Clinical History US CT Imaging Diagnosis Diagnosis Management Pathologic Findings 1 13 Acute pelvic pain + + Fallopian tube torsion Correct Salpingectomy Left fallopian tube with vomiting and torsion, necrotic diarrhea for 2 d 2 14 Worsening RLQ pain + + Hydrosalpinx, Correct Salpingectomy Right fallopian tube and nausea for 5 d possible torsion torsion with paratubal cyst 3 13 RLQ and pelvic pain + Cystic ovarian Correct Salpingectomy Right fallopian tube for 3 wk neoplasm vs torsion paraovarian cyst, torsion could not be excluded 4 15 Left flank pain with + + Torsion of fallopian Correct Detorsion, Left fallopian tube vomiting and nausea tube with associated parafallopian and ovary torsion, for 5 d adnexal cyst cystectomy, viable ovary with hemorrhagic hemorrhagic cyst, ovarian paratubal cyst cystectomy 5 11 LLQ pain for 3 d with + Torsed fallopian tube Correct Detorsion with Left fallopian tube acute worsening vs hydrosalpinx and/or paratubal cyst torsion, paratubal cyst paraovarian cyst drainage 6 13 RLQ pain for 24 h + Ovarian torsion Incorrect Detorsion Right fallopian tube torsion, normal ovaries 7 9 Abdominal pain + Right hydrosalpinx Correct Detorsion Right fallopian tube (epigastric migrating vs fallopian torsion, torsion, hydrosalpinx to suprapubic area) right ovarian cyst with vomiting and diarrhea for 24 h 8 17 Intermittent RLQ pain + Torsion with lead Correct Detorsion with Right fallopian tube with vomiting for 1 wk point such as a removal of mass torsion, right ovarian dermoid dermoid 9 14 LLQ pain, acute onset + + Multiseptated left Correct Detorsion, Left fallopian tube with nausea and adnexal mass, marsupialization, torsion, normal vomiting for 24 h differential diagnosis: biopsy of ovaries, left hemorrhagic cyst vs mesovarian cyst mesovarian cyst primary ovarian neoplasm vs torsion Correct Detorsion of left Left fallopian tube History of right ovarian + + Hydrosalpinx vs adnexa followed by torsion, right torsion in 2007 and left fallopian tube torsion left salpingectomy, hemorrhagic cyst, ovarian torsion in 2009, right ovarian drainage of right normal left ovary acute LLQ pain for 24 h hemorrhagic cyst ovarian hemorrhagic cyst LLQ indicates left lower quadrant; RLQ, right lower quadrant; and US, sonography J Ultrasound Med 2014; 33:

Figure 7. Case 5: 11-year-old patient with worsening left lower quadrant pain for 3 days. A dilated tubular structure (arrow) posterior to the uterus mimicking free fluid in cul-de-sac is shown.

7 Figure 7. Case 5: 11-year-old patient with worsening left lower quadrant pain for 3 days. A dilated tubular structure (arrow) posterior to the uterus mimicking free fluid in cul-de-sac is shown. At surgery, a torsed fallopian tube was found. On Doppler imaging, high impedance reflecting a higher resistive index due to decreased blood flow and thinner fallopian tube walls, 22 absence of vascular flow in the tube, and the whirlpool sign have been described. 11,22,23 Doppler evaluation was limited in our series, with no whirlpool signs identified. Previous reported MDCT findings of isolated tubal torsion include an adnexal mass, a twisted appearance to the fallopian tube, a dilated tube greater than 15 mm, a thickened and enhancing tubal wall, and luminal MDCT attenuation greater than 50 Hounsfield units, consistent with hemorrhage. 9,11,21 Rezvani and Shaaban 9 suggested that multiplanar reformatted images could be helpful in visualizing the tubular shape and tapered ends. Secondary signs on MDCT, including free fluid, peritubular fat stranding, enhancement and thickening of the broad ligament, and a focal reactive ileus, have been described in previous studies. 7,9 In our series, a dilated tube was noted in 3 cases and a cystic complex adnexal mass in the remaining 2 cases. Multidetector computed tomography was not as sensitive as sonography in separating the ovary from the adjacent tube in 1 case. Magnetic resonance imaging may have a future role in the assessment of fallopian tube torsion but was not evaluated in our series. On review of the literature, hemato salpinx has been suggested as an associated finding with fallopian tube torsion and could be detected on magnetic resonance imaging due to different signal changes from blood products. 7 Absence of enhancement of the tube could suggest infarction on magnetic resonance imaging as well. 24 On sonography and MDCT, 9 of 10 cases had the diagnosis of tubal torsion correctly suggested prospectively. One case was misdiagnosed as a torsed multicystic ovary (Figure 4). We found sonography to be most useful and should be the initial modality of choice in evaluation acute pelvic pain. 25 The limitations of this series included its retrospective nature and the small number of cases. Doppler evaluation was limited. Because of the limited number of cases, the sensitivity and specificity of findings such as the beak sign could not be derived. In conclusion, whereas rare, tubal torsion is a diagnosis to be considered in adolescent girls with acute pelvic pain. Sonography with Doppler imaging should be the first imaging choice. As clinical examination is nonspecific, it is important to consider tubal torsion in the differential diagnosis when a tubular structure or a midline cystic mass associated with a normal ipsilateral ovary is noted in the appropriate clinical setting. Early and accurate diagnosis may help in tube-sparing surgery. References 1. Bland-Sutton J. Salpingitis and some of its effects. Lancet 1890; 2: Ferrera PC, Kass LE, Verdile VP. Torsion of the fallopian tube. Am J Emerg Med 1995; 13: Raziel A, Mordechai E, Friedler S, Schachter M, Pansky M, Ron-El R. Isolated recurrent torsion of the fallopian tube: case report. Hum Reprod 1999; 14: J Ultrasound Med 2014; 33:

8 4. Casey RK, Damle LF, Gomez-Lobo V. Isolated fallopian tube torsion in pediatric and adolescent females: a retrospective review of 15 cases at a single institution. J Pediatr Adolesc Gynecol 2013; 26: Boukaidi SA, Delotte J, Steyaert 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: Harmon JC, Binkovitz LA, Binkovitz LE. Isolated fallopian tube torsion: sonographic and CT features. Pediatr Radiol 2008; 38: Orazi C, Inserra A, Lucchetti MC, Schingo PM. Isolated tubal torsion: a rare cause of pelvic pain at menarche sonographic and MR findings. Pediatr Radiol 2006; 36: Provost MW. Torsion of the normal fallopian tube. Obstet Gynecol 1972; 39: Rezvani M, Shaaban AM. Fallopian tube disease in the nonpregnant patient. Radiographics 2011; 31: Bernardus RE, Van der Slikke JW, Roex AJ, Dijkhuizen GH, Stolk JG. Torsion of the fallopian tube: some considerations on its etiology. Obstet Gynecol 1984; 64: Gross M, Blumstein SL, Chow LC. Isolated fallopian tube torsion: a rare twist on a common theme. AJR Am J Roentgenol 2005; 185: Merlini L, Anooshiravani M, Vunda A, Borzani I, Napolitano M, Hanquinet S. Noninflammatory fallopian tube pathology in children. Pediatr Radiol 2008; 38: Goktolga U, Ceyhan T, Ozturk H, et al. Isolated torsion of fallopian tube in a premenarcheal 12-year-old girl. J Obstet Gynaecol Res 2007; 33: Rizk DE, Lakshminarasimha B, Joshi S. Torsion of the fallopian tube in an adolescent female: a case report. J Pediatr Adolesc Gynecol 2002; 15: Bondioni MP, McHugh K, Grazioli L. Isolated fallopian tube torsion in an adolescent: CT features. Pediatr Radiol 2002; 32: Adekanmi OA, Barrington JW, Edwards G, Farrell D. Isolated torsion and haemorrhagic infarction of a normal fallopian tube in an eleven year old girl. BJOG 2000; 107: Terada Y, Murakami T, Nakamura S, et al. Isolated torsion of the distal part of the fallopian tube in a premenarcheal 12 year old girl: a case report. Tohoku J Exp Med 2004; 202: Elchalal U, Caspi B, Schachter M, Borenstein R. Isolated tubal torsion: clinical and ultrasonographic correlation. J Ultrasound Med1993; 12: Lineberry TD, Rodriguez H. Isolated torsion of the fallopian tube in an adolescent: a case report. J Pediatr Adolesc Gynecol 2000; 13: Propeck PA, Scanlan KA. Isolated fallopian tube torsion. AJR Am J Roentgenol 1998; 170: Ghossain MA, Buy JN, Bazot M, et al. CT in adnexal torsion with emphasis on tubal findings: correlation with US. J Comput Assist Tomogr 1994; 18: Vijayaraghavan SB, Senthil S. Isolated torsion of the fallopian tube: the sonographic whirlpool sign. J Ultrasound Med 2009; 28: Zalel Y, Soriano D, Lipitz S, Mashiach S, Achiron R. Contribution of color Doppler flow to the ultrasonographic diagnosis of tubal abnormalities. J Ultrasound Med 2000; 19: Bader T, Ranner G, Haberlik A. Torsion of a normal adnexa in a premenarcheal girl: MRI findings. Eur Radiol 1996; 6: Andreotti RF, Harvey SM. Sonographic evaluation of acute pelvic pain. J Ultrasound Med 2012; 31: J Ultrasound Med 2014; 33:

JMSCR Vol 3 Issue 9 Page September 2015

JMSCR Vol 3 Issue 9 Page September 2015 www.jmscr.igmpublication.org Impact Factor 3.79 ISSN (e)-2347-176x DOI: http://dx.doi.org/10.18535/jmscr/v3i9.66 MR Evaluation of Isolated Fallopian Tubal Torsion, Rare Cause of Lower Abdominal Pain in