Rates and Technique for Oophoropexy in Pediatric Ovarian Torsion: A Single-Institution Case Series

Accepted Manuscript Rates and Technique for Oophoropexy in Pediatric Ovarian Torsion: A single institution case series I.M. Comeau, MD, N. Hubner, MD, S.L. Kives, MD, L.M. Allen, MD PII:

S1083-3188(16)30278-9

DOI:

10.1016/j.jpag.2016.11.006

Reference:

PEDADO 2068

To appear in:

Journal of Pediatric and Adolescent Gynecology

Received Date: 10 May 2016 Revised Date:

14 November 2016

Accepted Date: 14 November 2016

Please cite this article as: Comeau IM, Hubner N, Kives SL, Allen LM, Rates and Technique for Oophoropexy in Pediatric Ovarian Torsion: A single institution case series, Journal of Pediatric and Adolescent Gynecology (2016), doi: 10.1016/j.jpag.2016.11.006. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.

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I M Comeau, MD, N Hubner, MD, S L Kives, MD, L M Allen, MD Toronto, ON, Canada

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Rates and Technique for Oophoropexy in Pediatric Ovarian Torsion: A single institution case series

Section of Paediatric Gynaecology, Division of Endocrinology, Hospital for Sick Children

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Department of Obstetrics and Gynecology, University of Toronto

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Author for Reprints: L M Allen 555 University Avenue 7th Floor, Black Wing

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Toronto, ON M5G 1X8

Author for correspondence concerning manuscript:

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I M Comeau

T: (514) 260-8951, F: (514) 481-7704

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[email protected] St Mary’s Hospital

3830 Ave Lacombe

Montreal, QC H3T 1M5

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STRUCTURED ABSTRACT Study Objective

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Ovarian torsion (OT) is uncommon, but can result in loss of reproductive function. Traditionally managed by adnexectomy, torsed adnexae are now being conserved, increasing the potential for recurrent OT. As a result, some experts suggest oophoropexy (OP) to prevent recurrence. We present here a series of 11

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patients who underwent OP.

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Design, Setting, Participants.

A retrospective case series was conducted from 2004 to 2013 identifying patients under age 18 with OT. From this, patients with OP were extracted for detailed review. Results

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97 patients with OT were identified, 6/97 (6.2%) had recurrent OT. The rate of recurrence was higher (14.8%) in the group with torsion without an adnexal mass. 11/97 (11.3%) underwent OP. The mean age of patients with OP was 8.8 years. 9 patients had normal adnexae at initial torsion. 5/11 had OP during

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their initial procedure. OP was most commonly performed for long uteroovarian ligaments (6), recurrence (4), or bilateral OT (2). 8/8 patients with follow-up ultrasounds post torsion demonstrated at

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least one marker of normal ovarian function. 1/11 (9%) patients had a recurrence of OT of an oophoropexied ovary. There were no complications due to the OP portion of the procedure. Conclusion

In our series of OT, a small percentage of cases underwent prophylactic OP. Recurrence may still occur after OP. It seems reasonable to offer OP to patients at higher risk of recurrent OT although level one evidence is lacking. Future research should focus on techniques and long-term outcomes of OP.

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KEYWORDS

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Adolescent, pediatric, adnexal torsion, oophoropexy, conservative surgery, laparoscopy

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INTRODUCTION Ovarian torsion (OT) is relatively uncommon with an incidence in the pediatric population between 4.9

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in 100 0001 and 20 in 100 0002. In a series of 3772 adult patients from 1985 OT represents 2.7% of those requiring emergency gynaecologic surgery3. Unfortunately, despite its rarity, if OT is not recognized and

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managed in a timely and appropriate manner, it can result in loss of reproductive potential.

Until recent years, OT was managed by adnexectomy4-7. With recognition of the viability and capacity for

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preserved ovarian function even in the case of a blue-black appearing torsed ovary8-10, a trend towards ovarian preservation rather than oophorectomy has gained acceptance as the most appropriate initial management of pediatric and adolescent OT.

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The adoption of a conservative approach to ovarian torsion has resulted in oophorectomy rates in some institutions reported as low as 13.6 – 30%11-13. As a direct result there is an increasing number of young women at potential risk of recurrent ovarian torsion as more ovaries are maintained. Based on 2 large

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case series, recurrent OT occurs in approximately 5% of cases14,15. The rate of recurrence can be even higher if the OT occurred in the absence of underlying adnexal pathology14. Oophoropexy (OP) has been

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proposed as a means of decreasing future reproductive harm by decreasing the risk of recurrent OT16. The exact role of OP remains unclear. Some have proposed a theoretical negative impact of OP on future fertility due to alterations in anatomy17,18 and not surprisingly some authors have discouraged it’s routine use19. OP does not guarantee that a future torsion will be prevented as recurrences of OT after OP have been documented20.

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The objective of the current study is to describe the indications for, technique of and short term outcomes associated with, a case series of 11 girls in a single, Canadian centre who underwent OP to prevent recurrent OT. Rates of recurrent OT following OP will be contrasted with rates of recurrence of

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OT in an overall cohort of OT.

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METHODS

With institutional review board authorization, a retrospective chart review was conducted on all female

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patient less than 18 years of age with surgically confirmed OT, with or without tubal torsion who were treated within our institution. For the purpose of this case series, those patients with recurrent OT and/or on whom OP was performed between January 1, 2004 through October 15, 2013 were included. Patient data was extracted from the charts including: demographics (age, menarchal status,

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pubarchal/adrenarchal status), operative findings and procedures (detorsion, oophoropexy – including technique and suture material used), operative and non-operative complications and finally follow-up, which included whether recurrent episodes of torsion developed during the study period, any

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subsequent operative procedures that were required and documentation of ovarian function by ultrasound, defined as either presence of follicles or Doppler flow. The presumed indications for OP

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were extracted from the operative record by a single reviewer. If not explicitly stated the operative findings for the procedure were reviewed and the most likely indication or indications for oophoropexy from within that list were assigned by a single reviewer. Indications for OP could be multiple. Ooophoropexy was performed at the discretion of the most responsible surgeon. Standard 3 or 4 port laparoscopy technique using either a veress needle or open entry technique was employed. For those patients who underwent laparotomy, a standard pfannestiel technique was used as a surgical incision.

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Following thorough inspection of the pelvis and abdomen, detorsion was performed and where necessary one of two techniques for oophoropexy. The ovarian stroma was fixed to the ipsilateral uterosacral ligament (9 patients) using either absorbable (1 patients) or non-absorbable suture (8

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patients) at the surgeon’s discretion (fixation technique). Alternatively, the utero-ovarian ligament was shortened by plication with a PDS endoloop (1 patient (non-fixation)). A single patient had fixation to

absorbable suture.

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the uterosacral ligament in addition to plication of the utero-ovarian ligament, both with delayed

Patient characteristics were analyzed using descriptive statistics. Discrete variables are described with

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means, medians and ranges (non-normally distributed). RESULTS

Ninety-seven patients were identified with surgically confirmed OT from 2004 – 2013. The overall

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recurrence rate of OT was 6.2% (6/97 patients). Of the recurrences, 4 occurred in cases where the torsion was previously idiopathic and 2 in individuals with an adnexal mass at their initial surgery. The recurrence rates were 14.8% (4/27) for idiopathic OT and 2.9% (2/70) for OT associated with a discrete

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ovarian or adnexal mass. The average time to recurrence was 10.1 months (range: 2-22 months). One patient of 97 (1.0 %) had a second recurrence.

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The rate of OP was 11.3% (11/97) which included all 6 patients with recurrent torsion. The patient’s age, timing of OP and operative procedures are summarized below in Table 1. TABLE 1 – Summary of patient’s procedures The mean age of patients undergoing OP was 8.8 years (median: 8, range: 3-16). For the 10 patients with documented pubertal status, 50% were pre-pubertal (5/10). 27% of the OP cohort was post-menarchal (3/11).

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Eighty-three percent (9/11) had surgery completed by laparoscopy (or laparoscopies) and the remainder by laparotomy. Of the 11 patients that underwent OP, 9 patients had no discrete ovarian or adnexal mass at their initial torsion (82%). This is notably different from the overall group of patients with OT

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where only 28% (27/97) of the patients had normal ovaries at the time of identified torsion.

Forty-five percent (5/11) had OP during the procedure for management of their initial OT, 36.4% (4/11) had OP at time of recurrent OT and 18.2% (2/11) had OP during a 3rd procedure. The presumed

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indications for OP were long utero-ovarian ligaments or ovarian pedicles (6/11), recurrent OT (4/11), bilateral OT (2/11), electively on a contralateral viable ovary (1/11) and contralateral torsion following

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previous oophorectomy for torsion (1/11). Patients may have had more than one indication for OP. Overall 9 patients had detorsion and simultaneous OP of the torsed ovary and 2 patients prophylactically underwent OP of the non-torsed contralateral ovary. Of these 9 patients 8 had followup imaging where ovarian activity could be assessed. Ultrasounds are performed at the discretion of the

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attending physician for assessment of ovarian function or to rule out the persistence of an ovarian mass following surgical management of an ovarian torsion. 8/8 patients, of those where imaging was available, had normal size and function on ultrasound of the previously torsed ovary. The average time

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to ultrasound following OT was 11 weeks (median 9, range 5-23 weeks).

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Nine of the eleven OP procedures (82%) were fixations of the ovary to the uterosacral ligament. 2/11 were plications of the utero-ovarian ligament. In 73% (8/11) a non-absorbable suture material was used during OP.

One patient or 9% (1/11) had a recurrence of OT following shortening of the uteroovarian ligament with a delayed absorbable loop. This recurrence was attributed to a 2.2cm mature cystic teratoma, which was not excised until a third surgery.

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There were no surgical complications associated directly with the OP procedure. One patient required a partial oophorectomy due to trauma to the torsed ovary causing bleeding during detorsion. Two patients had early (< 48 hours) post-operative fevers. One patient experienced a rash due to a post-

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operative medication. Three patients presented to clinic or the emergency room with abdominal pain in the weeks following their procedure, none of whom required admission to hospital or surgical re-

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exploration. It was felt that their pain was not directly related to the oophoropexy.

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DISCUSSION

Given that the management of OT has become increasingly conservative, the risk of recurrent or asynchronous bilateral OT is becoming more of a concern as more potentially at risk ovaries are left in situ. Health care providers are now left with the question of whether or not to offer OP, when to offer it

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and which type of OP to perform.

The recurrence risk of OT varies with the etiology of the initial torsion event. Beaunoyer reviewed cases of torsion where 39 of 76 girls had simple OT (no leading mass or cyst), 4 of these patients went on to

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have asynchronous bilateral torsion (representing 11 % of patients with normal ovaries), whereas none of the girls with a leading mass or cyst developed asynchronous bilateral torsion14. The recurrence rate

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after pediatric and adolescent OT in our institution was 6.2% overall, 2.9% of patients with OT associated with a discrete ovarian or adnexal mass and 14.8% of patients with idiopathic OT, similar to the reported rates of Beaunoyer. A number of theories have been proposed to explain the torsion events in normal adnexa, these include abnormally long tubes, mesovarium or mesosalpinx leading to increase ovary mobility21; long uteroovarian ligaments18; as well as impeded venous return with stasis and congestion resulting in a heavier

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ovary22. Given the theories of long ligaments and ovarian or adnexal mobility, OP has been proposed and utilized as a potential solution to prevent the occurrence or recurrence of OT.

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Various techniques for OP have been proposed. These usually fall into 2 broad categories: fixation of the adexae or plication of adnexal ligaments. The first method described was to fix the ovary to the uterine serosa23. Guileyardo describes fixation of the ovary to the lateral part of the broad ligament24. Others have recommended the use of permanent suture to fix the ovary to the pelvic side wall25.

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Germain was the first to describe decreasing ovarian mobility by shortening the uteroovarian ligament using a non-absorbable suture18. They theorize that this method may have less impact on future fertility

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than fixation to the pelvic sidewall or round ligament as it is less likely to impair tubal function. Weitzman described a similar technique, shortening the utero-ovarian ligament by laparoscopy with an endoloop26. In our case series, 82% of OP were fixations of the adnexae. The location chosen in all cases was the uterosacral ligament. 9 of 11 OP were performed at the time of a torsion event. It was generally

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felt in our center that fixation of a heavy edematous ovary in a dependent position would be less likely to fail than fixation to the pelvic side wall or shortening of adnexal ligaments. Even with OP the risk of recurrent OT is not eliminated. In our series 1 of 11 patients (9%) had a

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recurrence of OT following their OP, with a delayed absorbable loop for ligament plication. Other groups have documented similar rates – Fuchs had a rate of 1/6 (17%) in 2010 and Tsafrir 2/21 (9.5%) in

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201212,20. A case of unilateral, isolated tubal torsion has also been reported in a girl 2 years after bilateral OP27. Similar to our recurrence after OP, the single recurrence in the cohort by Fuchs et al also occurred when an absorbable suture was utilized, but with fixation to the pelvic sidewall rather than ligament plication. The authors have recommended permanent suture material for OP as a result of this documented failure20. Table 2 – Summary of recurrence of OT following OP in known case series

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Theoretical risks, including the potential impact on future fertility, have been proposed by a number of groups. Breech et al have suggested interference with tubal blood supply, tubal function or tuboovarian communication – all which may impact on future fertility17. Other groups have suggested that

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having the fallopian tube on stretch will also interfere with its function, as may be the case in fixation of the ovary to the uterus or pelvic sidewall18.

To date to our knowledge, no direct comparative study on either the method of OP nor the type of

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suture material have been completed, nor has there been any study documenting the potential long

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term negative impact of OP.

There remain varying opinions on the role of OP in OT. Many authors advocate for OP of the affected side, or more commonly bilaterally10,16,28,29 at the time of an initial OT. Others recommend OP only in the case of recurrence30. Some have recommended interval OP rather than performing detorsion and OP in the same procedure citing reasons such as allowing in-depth discussion with patients and families,

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as well as allowing edema from acute torsion to diminish prior to placing a suture in the ovary20. Currently in our institution, oophoropexy is performed at the discretion of the attending surgeon and

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has been performed selectively. The recurrence rate after OP in our institution was 9%, similar to our overall recurrence rate in the entire OT cohort of 6.2%, but lower than the recurrence rate for ovaries

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without a leading mass (14.8%). Given the lack of long-term data on any ill effects on future fertility, perhaps the current role of OP remains for select cases where risk of recurrence or potential for severe consequences from OT may be higher. OP may have the greatest benefit in OT of a normal ovary with long infundibulopelvic ligaments, bilateral OT, recurrent OT and for the contralateral ovary in OT that results in oophorectomy. The limitation of this study and of the current literature remains the small number of cases of OP performed and the lack of long term follow up to determine which strategy is superior for preserving

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future reproductive capacity. As this study was a retrospective case series, patients may have recurred and had care provided at another health care institution, hence leading to an underestimation of the

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risks of recurrence of OT with or without OP. CONCLUSION

Based on our review it seems very reasonable to offer oophoropexy to high risk patients such as those

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who have had a recurrence of ovarian torsion, those with bilateral OT, or idiopathic torsion. Despite OP, recurrences may still occur. Permanent suture material may be reasonable to consider.

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Future research should be considered to look at the efficacy of OP on a much larger scale as the current literature is limited by small case numbers, reliance on expert opinion and lack of comparative studies. Future studies should focus on recurrence rates after OP, long term outcomes (including fertility), and comparing the two most common methods of OP: plication of the utero-ovarian ligament (non-fixation

1.

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Table 1 Summary of Patient’s Procedures

LOT

DT, LO OP

2

14

LOT

DT, bivalve**

3

7

ROT DT

4

6

LOT

5

12

Partial oopherectomy, DT, bivalve, LO OP BOT DT, bivalve**, bil OP

6

8

ROT DT

7

3

ROT DT, RO OP

8

5

9

21

LOT

DT, LO OP

5

Normal ovary, no torsion

LO OP*

ROT

ROT DT

6

BOT

11

ROT DT

5

ROT

10

7

ROT RSO

20

11

8

LOT

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2

LOT

Time to 3rd procedure (Wks)

Diagnosis at 3rd procedure

Procedure 3

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16

Procedure 2

DT

3

Cystadenoma, no torsion

Cystectomy, RO OP

BDT, Bil OP DT, RO OP

11

ROT, Dermoid

DT, cystectomy

DT, LO OP

Oophoropexy Technique

US fixation, absorbable suture US fixation, nonabsorbable suture US fixation, nonabsorbable suture US fixation & UO plication, absorbable suture

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1

Time Diagnosis at 2nd nd to 2 procedure procedure (Wks)

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Procedure 1

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No Age Dx

US fixation, nonabsorbable suture US fixation, nonabsorbable suture US fixation, nonabsorbable Suture US fixation, nonabsorbable suture UO plication, absorbable suture US fixation, nonabsorbable suture US fixation, nonabsorbable suture

LSO (significantly enlarged ovary) RO OP LOT, left ovarian torsion; ROT, right ovarian torsion; BOT, bilateral ovarian torsion; LO, left ovary; RO, right ovary; DT, detorsion; US, uterosacral ligament; UO, utero-ovarian ligament * Atresia of RO on Ultrasound during follow-up. She underwent elective LO OP. **An incision made into the ovarian stroma sometimes done to assess return of circulation and to relieve pressure from edema on the tissue

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Table 2 Summary of recurrence of OT following OP in known case series

Fuchs N et al. J Minim Invasive Gynecol. 2010;17:205-2087 *

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Recurrent OT after OP %

Study authors and journal citation

16.7 (1/6)

9.5 (2/21)

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Tsafrir Z et al. Eur J Obstet Gynecol Reprod Biol. 2012;162:203-2058 **

9 (1/11)

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Current case series