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Pediatric synchronous bilateral ovarian torsion: a case report and review of the literature Tania Dumont , Nicolette Caccia, Lisa Allen Division of Endocrinology, Section of Gynecology, Sick Kids Hospital, Toronto, Ontario, Canada M5G 1XB Received 16 May 2011; revised 28 August 2011; accepted 30 August 2011 Key words: Ovary; Torsion; Bilateral; Synchronous; Oophoropexy; Pediatric Abstract Ovarian torsion is a surgical emergency that can present with a variety of symptoms and hence is difficult to diagnose. We present the first case of a pediatric synchronous bilateral ovarian torsion in ovaries without pathology and review its presentation, diagnosis, treatment, outcome, and the associated literature. © 2011 Elsevier Inc. All rights reserved. 1. Background Ovarian torsion, one of the few gynecologic surgical emergencies, has an incidence of 4.9 in 100 000 [1]. In up to 46% of cases of ovarian torsion in children, no underlying ovarian mass is found [2]. Warnek in 1895 reported the first adult case of bilateral ovarian torsion, which remains a rare occurrence to this day [3]. To the best of our knowledge, we report the first confirmed childhood case of synchronous bilateral ovarian torsion in ovaries without an underlying cyst or mass and provide a review of the literature on the topic. 2. Case A previously healthy, premenarchal, 12-year-old girl was referred to the pediatric and adolescent gynecology clinic with a 2-year history of recurrent episodes of right lower quadrant pain with no associated symptoms. A pelvic ultrasound study performed before referral was consistent with polycystic ovaries. Repeat pelvic ultrasound imaging at the time of consultation revealed bilateral bulky ovaries (43 × 32 × 38 mm with volume of 20.2 mL [right] and 56 × 35 × 55 mm with volume of 52.3 mL [left]) and peripheral follicles in keeping with polycystic ovarian morphology (Fig. 1). Normal ovarian volumes in the pediatric population have been described by Garel et al [4], and the accepted range is 2 to 20 mL in the adolescent population with 2 to 4 mL as the norm in the premenarchal population. Four months after initial consultation, she presented to the emergency department with a 30-hour history of periumbi- lical pain associated with nausea and vomiting. On physical examination, the pain was localized to the left lower quadrant and was associated with rebound tenderness. The white blood cell count was normal. Repeat pelvic ultrasound showed a significantly enlarged left ovary on gray scale imaging (61 × 45 × 49 mm, 70 mL) that was more heterogeneous than previously noted, contained peripheral follicles, and had diminished flow on Doppler assessment. The right ovary was unchanged from the previous study (41 × 37 × 28 mm, 22 mL), also contained peripheral follicles, but Corresponding author. Tel.: +1 416 813 4981; fax: +1 416 813 7271. E-mail address: [email protected] (T. Dumont). www.elsevier.com/locate/jpedsurg 0022-3468/$ see front matter © 2011 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2011.08.024 Journal of Pediatric Surgery (2011) 46, E19E23

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www.elsevier.com/locate/jpedsurg

Journal of Pediatric Surgery (2011) 46, E19–E23

Pediatric synchronous bilateral ovarian torsion: a casereport and review of the literatureTania Dumont⁎, Nicolette Caccia, Lisa Allen

Division of Endocrinology, Section of Gynecology, Sick Kids Hospital, Toronto, Ontario, Canada M5G 1XB

Received 16 May 2011; revised 28 August 2011; accepted 30 August 2011

0d

Key words:Ovary;Torsion;Bilateral;Synchronous;Oophoropexy;Pediatric

Abstract Ovarian torsion is a surgical emergency that can present with a variety of symptoms andhence is difficult to diagnose. We present the first case of a pediatric synchronous bilateral ovariantorsion in ovaries without pathology and review its presentation, diagnosis, treatment, outcome, andthe associated literature.© 2011 Elsevier Inc. All rights reserved.

1. Background

Ovarian torsion, one of the few gynecologic surgicalemergencies, has an incidence of 4.9 in 100 000 [1]. In upto 46% of cases of ovarian torsion in children, nounderlying ovarian mass is found [2]. Warnek in 1895reported the first adult case of bilateral ovarian torsion,which remains a rare occurrence to this day [3]. To the bestof our knowledge, we report the first confirmed childhoodcase of synchronous bilateral ovarian torsion in ovarieswithout an underlying cyst or mass and provide a review ofthe literature on the topic.

2. Case

A previously healthy, premenarchal, 12-year-old girl wasreferred to the pediatric and adolescent gynecology clinicwith a 2-year history of recurrent episodes of right lower

⁎ Corresponding author. Tel.: +1 416 813 4981; fax: +1 416 813 7271.E-mail address: [email protected] (T. Dumont).

022-3468/$ – see front matter © 2011 Elsevier Inc. All rights reserved.oi:10.1016/j.jpedsurg.2011.08.024

quadrant pain with no associated symptoms. A pelvicultrasound study performed before referral was consistentwith polycystic ovaries. Repeat pelvic ultrasound imaging atthe time of consultation revealed bilateral bulky ovaries (43 ×32 × 38 mmwith volume of 20.2 mL [right] and 56 × 35 × 55mmwith volume of 52.3 mL [left]) and peripheral follicles inkeeping with polycystic ovarian morphology (Fig. 1).Normal ovarian volumes in the pediatric population havebeen described by Garel et al [4], and the accepted range is 2to 20 mL in the adolescent population with 2 to 4 mL as thenorm in the premenarchal population.

Four months after initial consultation, she presented to theemergency department with a 30-hour history of periumbi-lical pain associated with nausea and vomiting. On physicalexamination, the pain was localized to the left lower quadrantand was associated with rebound tenderness. The whiteblood cell count was normal. Repeat pelvic ultrasoundshowed a significantly enlarged left ovary on gray scaleimaging (61 × 45 × 49 mm, 70 mL) that was moreheterogeneous than previously noted, contained peripheralfollicles, and had diminished flow on Doppler assessment.The right ovary was unchanged from the previous study (41 ×37 × 28 mm, 22 mL), also contained peripheral follicles, but

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Fig. 1 Ultrasound images of case. A, Pretorsion. B, At time of diagnosis of torsion (notice absence of Doppler flow in left ovary). C, Atpostoperative visit.

E20 T. Dumont et al.

had adequate Doppler flow (Fig. 1). The patient underwentlaparoscopy with a presumed diagnosis of left ovariantorsion. However, on inspecting the pelvis, bilateral ovariantorsion of 720° in each ovary was detected (Fig. 2). No focallesions were noted, but both ovaries were enlarged, with theleft ovary larger than the right. The ovaries were detorsed and,as both infundibulopelvic and uteroovarian ligaments werenoted to be abnormally long, an oophoropexy was performed,tacking both ovaries to their respective uterosacral ligaments(Fig. 3). The ovary was pexed to the uterosacral ligament,with a figure of eight 3.0 nonabsorbable suture in the ovariancortex with an intracorporeal knot tying technique. Afteridentifying that the ureter was medial to the uterosacralligament, a single pass through of the uterosacral ligamentwas taken. Given the findings of bilateral torsion, theoophoropexy procedure was repeated on the contralateralside. At her 6-week postoperative visit, she had had nofurther episodes of pain and had returned to normalactivities. Abdominal examination was unremarkable.Postoperative ultrasound study confirmed neither ovary-contained focal lesions; the right ovary measured 54 × 24 ×33 mm (21 mL), and the left, 44 × 33 × 23 mm (17 mL).Both showed multiple follicular cysts and central Doppler

flow (Fig. 1). At 9 months postsurgery, there has been norecurrence of adnexal pain.

3. Comments

Ovarian torsion is a rare entity in which the diagnosis isdifficult to make, particularly in children where torsion mayinvolve a normal ovary without a leading mass. Presentingsymptoms are variable and can include variable abdominalpain (the most common presenting symptom), nausea,vomiting, diarrhea, and fever [5-7]. Clinical findings alsodiffer between patients who may present with an acutesurgical abdomen, a palpable mass, and/or leukocytosis [5-7].The diagnosis of ovarian torsion relies on history, clinicalfindings, and supportive imaging. The most commonultrasound finding is an enlarged ovary [8]. Other findingsinclude absence of Doppler flow, peripheral follicles of 8 to15 mm with fluid debris levels, free intraperitoneal fluid,thickening of cyst wall, and whirlpool sign [5,9-11]. The onlyultrasound abnormality in the current case was an enlargedleft ovary with diminished Doppler flow; in the clinical

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Fig. 2 Synchronous bilateral ovarian torsion of normal ovaries in a 12-year-old premenarchal girl.

E21Pediatric synchronous bilateral ovarian torsion

setting of abdominal pain, vomiting, and peritoneal signs, thisled to a preoperative diagnosis of left ovarian torsion. Thesmaller size and maintenance of Doppler flow to thecontralateral ovary resulted in a failure to identify the bilateralsynchronous ovarian torsion until surgery.

Bilateral ovarian torsion remains a rare entity. Medlinewas searched using the key words bilateral, ovary, ovariandiseases/cysts/neoplasms, and torsion and was limited tohuman subjects. Articles were reviewed, and references werechecked for additional literature. There are 24 cases ofasynchronous torsion in the pediatric population and only 2cases of synchronous bilateral ovarian torsion, both occur-ring with bilateral dermoids. Rogers [12], in 1925, reportedthe first case of a 16-year-old girl with bilateral adnexaltorsion (original article unavailable) [3]. In 2002, Özcan et al[5] reviewed the literature and added 2 of his own cases(summarized in Table 1). As seen in Table 1, there has since

Fig. 3 Laparoscopic view after ovarian detorsion and bilateral outeroovarian ligaments.

been 8 additional cases reported [13-16] of which 2 weresynchronous torsions of bilateral ovarian dermoids [14,17].Our case presents the first child with bilateral synchronouspediatric torsion of ovaries without distinct pathology suchas an ovarian cyst or neoplasm.

Considering that this condition can result in subsequentinfertility because of loss of both ovaries if the diagnosis ismissed, bilateral ovarian torsion should be considered whena young female presents with abdominal pain on alternatingsides or recurrent bilateral abdominal pain with no obviousetiology. In our patient, the initial presenting pain was in theright lower quadrant; her acute episode of pain on the day ofsurgery was localized to the contralateral side. Theawareness of the potential for synchronous bilateral ovariantorsion should be conveyed to all health care providers caringfor infants, children, and adolescents. It has been recognizedin our institution that there is a service-specific discrepancy

ophoropexy to the uterosacral ligaments. Note the elongated

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Table 1 Summary of pediatric bilateral ovarian torsions from review of the literature

Authors Age at firsttorsion

Time intervalbetween torsions

First OR Second OR

Findings Procedure Findings Procedure

Özcan et al2002 [5]

7 2 y, 3 mo RSO LSO12 6 wk RSO LSO3 3 y RSO LSO7 2 y RSO LSO6 2 y LSO Detorsion7 2 y RSO LSO6.5 4 y LSO RSO3.5 7 y RSO LSO8.5 12 mo RSO LSO10 8 mo RSO Detorsion and

oophoropexy10 2 y LSO Detorsion and

oophoropexy? ? Incidental finding

(left side)Detorsion andoophoropexy

8 9 y RSO Detorsion andoophoropexy

4.5 17 mo LSO Detorsion andoophoropexy

? ? Incidental finding(left side)

Detorsion

9 7 mo 720° torsion Rthemorrhagicedematous ovary

RSO 720° torsion of Ltedematous ovary

Detorsion andoophoropexy

12 5 mo 720° torsion of Rtruptured necroticovary

RSO 720° torsion of Ltedematous ovary

Detorsion andoophoropexy

Beaunoyeret al 2004[13]

3 2 y Torsion of Lt ovarywithout pathology

LSO Torsion of Rt ovarywithout pathology

Detorsion andoophoropexy

12 5 mo Torsion of Lt ovarywithout pathology

LSO Torsion of Rt ovarywithout pathology

Detorsion andoophoropexy

12 6 mo Torsion of Lt ovarywithout pathology

LSO Torsion of Rt ovarywithout pathology

Detorsion andoophoropexy

10 18 mo Torsion of Lt ovarywithout pathology

LSO Torsion of Rt ovarywithout pathology

Detorsion andoophoropexy

Karnik et al2005 [15]

11 10 mo Torsion of Rt ovarywith follicular cysts

Laparotomy, RSO,appendectomy

1260° torsion of Ltovary withoutpathology

Laparotomy,detorsion,oophoropexy

Takeda et al2006 [17]

9 SYNCHRONOUS 360° torsion of Ltdermoid cyst

Bilateral detorsionand cystectomy

NA NA

180° torsion of Rtdermoid cyst

Svenssonet all2008 [16]

6 2 y Torsion of Rt ovaryon hemorrhagic cyst

Laparotomy, RSO,appendectomy

720° torsion of Ltovary withoutpathology

Laparoscopy,detorsion, hyperbaricoxygen therapy

Rousseauet al 2008[14]

13 SYNCHRONOUS Torsion of Rtdermoid cyst

RSO NA NA

Torsion of Ltdermoid cyst

Detorsion, Ltcystectomy

Dumontet al 2011

12 SYNCHRONOUS Torsion of Rtadnexa, no cystor masses

Laparoscopy, bilateraldetorsion, and bilateraloophoropexy

NA NA

Torsion of Ltadnexal, no cystor masses

Rt indicates right; Lt, left; RSO, right salpingo-oophorectomy; LSO; left salpingo-oophorectomy; NA, not applicable; blank box, data not available.

E22 T. Dumont et al.

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E23Pediatric synchronous bilateral ovarian torsion

in the preoperative suspicion of torsion between pediatricsurgeons who suspect torsion in 47% of cases compared with94% preoperative diagnosis for gynecologists [18].

The currently accepted treatment of torsion is detorsionwith concurrent or subsequent cystectomy if an ovarian cyst/neoplasm exists. Oophoropexy, however, remains a contro-versial management choice in ovarian torsion. This procedureconsists of stabilizing the ovary to prevent recurrent torsion.Various methods can be used including pexing the ovary to theuterosacral ligament, as described above. After suturing theovary to the uterosacral ligament, it will often reside easily inthe pelvis, and in the case of the acutely torted, friable, blue-black ovary, we feel that this location minimizes the risk of thesuture pulling through the ovarian cortex. Another methodcommonly used is plication of the uteroovarian ligament tominimize the laxity of this elongated structure and reduce therisk of torsion. Plication is completed by suturing the distal andproximal ends of the ligament to each other or by gathering theexcess ligament length within an Endoloop (Ethicon Endo-Surgery, Inc., Cincinnati, OH) [19,20]. Alternatively, the ovarycan be sutured, to either the posterior aspect of the uterus or tothe pelvic sidewall at the level of the pelvic brim, after ensuringadequate localization of the iliac vessels and the ureter [19,22].Permanent, nonabsorbable suture material should be used forall procedures [21]. Theoretical concerns for future fertilityexist for this procedure such as interruption of blood flow to thefallopian tube as well as disruption of the intricate communi-cation between the tube and the ovary [6,20,21]. Controversyexists as well on when to perform the procedure. Performingoophoropexy at the time of torsion may be technically moredifficult given the edema and size of the ovary. It also may notallow for full discussion of the benefits and risks of theprocedure with the family. It does, however, prevent asubsequent surgical procedure [21]. On the other hand,performing the oophoropexy during a second procedurewould allow detailed discussion with patients and familyabout the risks and benefits and would allow for choosing theprocedure that best fits the patient's natural placement of thefallopian tube and ovary when the ovary is neither enlarged noredematous. In our institution, consistent with indications foundin the literature, oophoropexies are performed for recurrenttorsion, obviously abnormal adnexal attachments or, wherethere has been a previous loss of an ovary subsequent toadnexal torsion without a lead point [21]. Because there arecase reports of complete loss of ovarian function in childrenwho have had bilateral oophorectomies performed forasynchronous bilateral ovarian torsions, synchronous bilateralovarian torsion should be considered as a potential indicationfor oophoropexy as was performed in our case [5]. Given thatthe diagnosis of ovarian torsion remains clinical, physiciansand parents should understand that there will be times whenlaparoscopy, performed to ensure ovarian salvage and futurefertility, will not demonstrate evidence of torsion.

We present the first case of a pediatric synchronousbilateral ovarian torsion on ovaries without cysts orneoplasms. If not identified in a timely fashion, this

diagnosis could have led to subsequent bilateral loss ofovarian function. We urge all caregivers to consider thepossibility of ovarian torsion and bilateral torsion as adiagnosis and suggest prompt laparoscopic intervention,reduction of the torsion, and consideration of oophoropexy.

References

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[2] Oltmann SC, Fischer A, Barber R, et al. Cannot exclude torsion—a15-year review. J Pediatr Surg 2009;44:1212.

[3] Dunnihoo DR, Wolff J. Bilateral torsion of the adnexa: a case reportand a review of the world literature. Obstet Gynecol 1984;3:55S.

[4] Garel L, Dubois J, Grignon A, et al. US of the pediatric female pelvis: aclinical perspective. Radiographics 2001;21:1393.

[5] Özcan C, Çelik A, Özok G, et al. Adnexal torsion in children may havea catastrophic sequel: asynchronous bilateral torsion. J Pediatr Surg2002;37:1617.

[6] Breech LL, Hillard PJA. Adnexal torsion in pediatric and adolescentgirls. Curr Opin Obstet Gynecol 2005;17:483.

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[10] Hurh PJ, Meyer JS, Shaaban A. Ultrasound of a torsed ovary:characteristic gray-scale appearance despite normal arterial and venousflow on Doppler. Pediatr Radiol 2002;32:586.

[11] Kiechl-Kohlendorfer U, Maurer K, Unsinn KM, et al. Fluid-debrislevel in follicular cysts: a pathognomonic sign. Pediatr Radiol 2006;36:421.

[12] Rogers L. Torsion of fallopian tubes. Br Med J 1925;1:778.[13] Beaunoyer M, Chapdelaine J, Bouchard S, et al. Asynchronous

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emergency management and conservative surgery of ovarian torsion inchildren: a report of 40 cases. J Pediatr Adolesc Gynecol 2008;21:201.

[15] Karnik AS, Sainani NI, Kamat NN. Case report—sequential bilateraltorsion of normal ovaries in a prepubertal child. J Clin Ultrasound2006;34:33.

[16] Svensson JF, Larsson A, Uusijärvi J, et al. Oophoropexy, hyperbaricoxygen therapy, and contrast-enhanced ultrasound after asynchronousbilateral ovarian torsion. J Pediatr Surg 2008;43:1380.

[17] Takeda A, Manabe S, Mitsui T, et al. Case report—laparoscopicmanagement of mature cystic teratoma of bilateral ovaries withadnexal torsion occurring in a 9-year-old premenarchal girl. J PediatrAdolesc Gynecol 2006;19:403.

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[19] Rollene N, Nunn M, Wilson T, et al. Recurrent ovarian torsion in apremenarchal adolescent girl contemporary surgical management.Obstet Gynecol 2009;114:422.

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[21] Fuchs N, Smorgick N, Tovbin Y, et al. Oophoropexy to preventtorsion: how, when and for whom? J Minim Invasive Gynecol2010;17:205.

[22] Abes M, Sarihan H. Oophoropexy in children with ovarian torsion D.Eur J Pediatr Surg 2004;14:168.