| Original Full Text | Citation: Psilopatis, I.; Damaskos, C.;Garmpis, N.; Vrettou, K.; Garmpi, A.;Antoniou, E.A.; Chionis, A.;Nikolettos, K.; Kontzoglou, K.;Dimitroulis, D. Ovarian Torsion inPolycystic Ovary Syndrome: APotential Threat? Biomedicines 2023,11, 2503. https://doi.org/10.3390/biomedicines11092503Academic Editor: Ebrahim AsadiReceived: 16 August 2023Revised: 3 September 2023Accepted: 9 September 2023Published: 10 September 2023Copyright: © 2023 by the authors.Licensee MDPI, Basel, Switzerland.This article is an open access articledistributed under the terms andconditions of the Creative CommonsAttribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).biomedicinesReviewOvarian Torsion in Polycystic Ovary Syndrome: APotential Threat?Iason Psilopatis 1 , Christos Damaskos 2,3,4,* , Nikolaos Garmpis 2,3 , Kleio Vrettou 5, Anna Garmpi 6,Efstathios A. Antoniou 2,3, Athanasios Chionis 6, Konstantinos Nikolettos 7, Konstantinos Kontzoglou 2,3and Dimitrios Dimitroulis 2,31 Department of Obstetrics and Gynecology, University Erlangen, Universitaetsstrasse 21-23,91054 Erlangen, Germany; iason.psilopatis@alumni.charite.de2 Second Department of Propedeutic Surgery, Laiko General Hospital, Medical School, National andKapodistrian University of Athens, 11527 Athens, Greece; nikosg22@hotmail.com (N.G.)3 Nikolaos Christeas Laboratory of Experimental Surgery and Surgical Research, Medical School, National andKapodistrian University of Athens, 11527 Athens, Greece4 Renal Transplantation Unit, Laiko General Hospital, 11527 Athens, Greece5 Department of Cytopathology, Sismanogleio General Hospital, 15126 Athens, Greece; kliovr1@gmail.com6 Department of Gynecology, Laiko General Hospital, Medical School, National and Kapodistrian University ofAthens, 11527 Athens, Greece; annagar@windowslive.com (A.G.)7 Obstetric and Gynecologic Clinic, Medical School, Democritus University of Thrace,68110 Alexandroupolis, Greece* Correspondence: x_damaskos@yahoo.grAbstract: Polycystic ovary syndrome (PCOS) constitutes the most prevalent endocrine disorder inwomen of reproductive age worldwide. Given the increased risk of ovarian torsion in the presence oflarge ovarian cysts, polycystic ovarian syndrome could be regarded as one of the most significantrisk factors for ovarian and/or adnexal torsion in cases of significantly enlarged ovaries. Theaim of the present review is to investigate, for the first time, the association between polycysticovarian syndrome and ovarian torsion. We performed a review of the literature using the MEDLINEand LIVIVO databases in order to find relevant studies. By using the search terms “polycysticovarian syndrome” and “ovarian torsion”, we were able to identify 14 studies published between1995 and 2019. The present work constitutes the most up-to-date, comprehensive literature reviewfocusing on the risk of ovarian/adnexal torsion in patients with polycystic ovaries. Ovarian/adnexaltorsion seems to be a feared complication in patients with polycystic ovary syndrome. Acute lowerabdominal pain in patients with known polycystic ovaries represents the most common symptom,while diagnostic assessment almost always incorporates transvaginal ultrasound and computertomography or magnetic resonance tomography scans. In case of suspected torsion, emergencylaparoscopy with ovarian or adnexal detorsion seems to be the standard therapeutic approach witha view to restitute the interrupted blood supply. In cases of repeated ovarian/adnexal torsions,ovariopexy or ovariectomy/adnexectomy had to be discussed with the patient in the context of riskrecurrence minimization.Keywords: polycystic; ovary; syndrome; adnexal; ovarian; torsion; emergency1. IntroductionThe most prevalent endocrine disorder in women of reproductive age worldwide ispolycystic ovary syndrome (PCOS) [1]. Symptoms typically begin during adolescence andrange from insulin resistance and related health issues to irregular menstrual cycles andskin conditions [2]. After ruling out other endocrinological conditions, PCOS is diagnosedin adults depending on the fulfilment of at least two of the Rotterdam criteria. The pres-ence of two of the following is required by the Rotterdam criteria: hyperandrogenism,oligo-ovulation and/or anovulation, or ovarian volume 10 mL (enlarged) and/or presenceBiomedicines 2023, 11, 2503. https://doi.org/10.3390/biomedicines11092503 https://www.mdpi.com/journal/biomedicinesBiomedicines 2023, 11, 2503 2 of 11of multiple cystic follicles in one or both ovaries (polycystic ovary) on ultrasound [3].Laboratory tests are crucial for confirming hyperandrogenism and ruling out alternativeendocrinological conditions like hyperprolactinemia or thyroid dysfunction in additionto a thorough medical history and scholastic clinical examination [4]. The evaluation ofmental health and quality of life, as well as metabolic screening and monitoring, shouldoccur both at the initial visit and at regular intervals because women with PCOS are at ahigh risk of developing serious comorbidities [5]. Modifications to the patient’s lifestyleshould always be a part of the standard therapeutic protocol for PCOS patients. Morespecifically, the foundation of non-pharmacological approaches is made up of weight loss,caloric restriction, specialized diets, and physical activity [5–7]. The primary goals ofthe pharmaceutical approach for PCOS patients who do not want to become pregnantare the control of menstrual cycle irregularities and hyperandrogenism, the handling ofcomorbidities, and the enhancement of quality of life. Biguanides may be supplementarilyapplied in addition to combined oral contraceptives and lifestyle changes with a view toameliorating menstrual cycle abnormalities, weight, and metabolic outcomes [8]. Com-bined oral contraceptives or progestins are the first-line medications of choice for menstrualirregularities and/or hyperandrogenism. Antiandrogens are used to treat hirsutism andalopecia in women who cannot tolerate combined oral contraceptives [9]. Letrozole orclomiphene should be administered to PCOS patients who intend to become pregnant inorder to trigger ovulation [10]. Notably, it has been also demonstrated in the literature howsupplementation of vitamin D, inositols, or alpha lipoic acid could improve reproductiveoutcomes in PCOS patients [11–13]. Due to the ovulatory dysfunction associated withPCOS, affected patients usually also develop luteinized unruptured follicle syndrome [14].This particular category of patients usually needs to resort to in vitro fertilization treat-ment methods to get pregnant, alongside the psychosocial implication in their infertilepathway [15].When the ovary twists over the ligaments that support it in the adnexa, the processknown as ovarian torsion takes place [16]. Adnexal torsion is the term used when thefallopian tube frequently twists along with the ovary [16]. An ovarian mass with a diameterof 5 cm or more is the main risk factor for ovarian torsion [17]. The mass makes it more likelythat the ovary will rotate around the axes of the two ligaments that keep it suspended [18].This torsion prevents arterial inflow and, eventually, venous outflow [18]. Females of allages can experience torsion but childbearing-age women are more likely to experienceit [16]. The infundibulopelvic and utero-ovarian ligaments, which serve as the ovary’ssupporting ligaments, are torn when the ovary twists over them. Swelling and bloodflow obstruction result from this. Initial obstruction of the venous outflow is followed byinterruption of the arterial inflow as a result of increased swelling, which results in ovariannecrosis, infarction, hemorrhage, and possibly peritonitis [19]. Because the sigmoid colonis located in the left pelvis, the right side has been seen more frequently than the left; this isthought to be a result of the increased space in the right pelvis [18].A complete metabolic panel, a serum human Chorionic Gonadotropin (hCG), and acomplete blood count should all be performed in terms of laboratory testing. If the torsionis causing hemorrhage, leukocytosis or anemia may be profound. Given that pregnancyincreases the risk of torsion, hCG is particularly significant. The lab values will typically benormal in torsion despite these generalized laboratory abnormalities [16]. The preferredimaging study is ultrasound with Doppler. It is necessary to perform both transvaginal andtransabdominal ultrasounds. The sensitivity of ultrasound for ovarian torsion dependson a variety of elements, including the expertise of the physician and the anatomy ofthe patient. Free fluid or the whirlpool sign, which is thought to be caused by vascularpedicle twisting in the cross-section, may also be present. It is important to measure bloodflow in comparison with the ovary on the opposite side. Because the ovaries have twoblood supplies, a complete lack of flow is not required for symptoms to exist [20]. Becauseovarian torsion may not be present at the time of ultrasound, this test alone cannot excludeit. Although they are frequently used to rule out other abdominal pathologies like acuteBiomedicines 2023, 11, 2503 3 of 11appendicitis, computer tomography (CT) and magnetic resonance tomography (MRI) arenot typically used to diagnose ovarian torsion [21,22].Direct observation of a rotated ovary during surgery allows for the final determinationof the diagnosis of ovarian torsion. For this reason, the patient must always undergosurgical evaluation if clinical suspicion persists, despite relatively normal lab results andultrasound imaging. The surgeon must determine whether the ovary is viable beforeattempting to salvage it in females who are of reproductive age [16]. Surgery shouldtypically be performed laparoscopically and with direct visualization of the twisted ovary.Visualization is primarily used to assess viability. Blood flow may be compromised inan enlarged, dark ovary with hemorrhagic lesions; however, the organ is frequently stillsalvageable [18]. More than 90% of patients who underwent detorsion had functionalovaries after the procedure. This was determined by the way the adnexa appeared onultrasound, including the development of follicles on the ovaries. So, the preferred course oftreatment is surgery with adnexal sparing [23]. Rarely, the surgeon may decide to performa salpingo-oophorectomy if the ovary appears necrotic and gelatinous beyond repair. If abenign cyst is present, the surgeon may also perform a cystectomy. Salpingo-oophorectomyis the recommended course of treatment if the cyst appears to be cancerous or if the patientis postmenopausal [16].Given the increased risk of ovarian torsion in the presence of large ovarian cysts,polycystic ovarian syndrome could be regarded as one of the most significant risk factorsin cases of significantly enlarged ovaries (Figure 1). However, so far, no review article hasbeen published on the association of polycystic ovarian syndrome and ovarian torsion.Thus, the aim of the present work is to investigate, for the first time, the association betweenpolycystic ovarian syndrome and ovarian torsion.Biomedicines 2023, 11, x FOR PEER REVIEW 4 of 13 Thus, the aim of the present work is to investigate, for the first time, the association be-tween polycystic ovarian syndrome and ovarian torsion. Figure 1. Ovarian torsion in polycystic ovary syndrome. 2. Literature Research We performed the literature review by using the MEDLINE and LIVIVO databases. Original research articles written in the English language that clearly reported on the as-sociation between polycystic ovarian syndrome and ovarian torsion were included in the data analysis. Studies centering purely on the role of ovarian cysts in the absence of poly-cystic ovarian syndrome or that did not explicitly specify the presence of polycystic ovar-ian syndrome were excluded. By employing the search terms “polycystic ovarian syn-drome” and “ovarian torsion”, we identified a total of 114 (duplicate records removed) articles published between 1966 and 2023. After the abstract review, 58 records were dis-carded in the initial selection process. The full texts of the remaining 56 publications were assessed. A total of 14 relevant case reports meeting the inclusion criteria and published between 1995 and 2019 were selected for the final literature review. The aforementioned selection process is schematically depicted in Figure 2. Figure 1. Ovarian torsion in polycystic ovary syndrome.Biomedicines 2023, 11, 2503 4 of 112. Literature ResearchWe performed the literature review by using the MEDLINE and LIVIVO databases.Original research articles written in the English language that clearly reported on theassociation between polycystic ovarian syndrome and ovarian torsion were included inthe data analysis. Studies centering purely on the role of ovarian cysts in the absence ofpolycystic ovarian syndrome or that did not explicitly specify the presence of polycysticovarian syndrome were excluded. By employing the search terms “polycystic ovariansyndrome” and “ovarian torsion”, we identified a total of 114 (duplicate records removed)articles published between 1966 and 2023. After the abstract review, 58 records werediscarded in the initial selection process. The full texts of the remaining 56 publications wereassessed. A total of 14 relevant case reports meeting the inclusion criteria and publishedbetween 1995 and 2019 were selected for the final literature review. The aforementionedselection process is schematically depicted in Figure 2.Biomedicines 2023, 11, x FOR PEER REVIEW 5 of 13 Figure 2. PRISMA flow diagram visually summarizing the screening process. 3. Ovarian Torsion in Polycystic Ovary Syndrome By scholastically searching the literature, we were to identify a total of twelve cases of adnexal torsion in patients with polycystic ovaries aged between 8 and 37 years old (mean age at diagnosis: 25 years old). Asch et al. commented that torsion can be difficult to diagnose in patients with poly-cystic ovary syndrome who undergo ovarian stimulation, given the already enlarged ova-ries at baseline [24]. Giulini et al. presented the case of a 31-year-old woman with polycystic ovary syn-drome and right adnexal torsion during pregnancy after an oocyte in vitro maturation and intracytoplasmic sperm injection cycle. Two days after embryo transfer, the patient presented with right lower abdominal pain and leukocytosis. Transvaginal ultrasound re-vealed an enlarged right ovary, alongside normal bilateral ovarian vascular flow. Due to symptom deterioration, an explorative laparoscopy was preformed, identifying a twisted right adnexa with an ischemic ovary. After successful laparoscopic detorsion with preser-vation of adnexa, the patient had an uneventful postoperative course and delivered a healthy infant at 40 weeks of gestation [25]. Furthermore, Gonçalves et al. reported the case of a 31-year-old woman with poly-cystic ovary syndrome and recurrent right adnexal torsion. The patient first presented with acute right pelvic pain and the sonographic constellation of acute adnexal torsion. After emergency laparoscopy, the enlarged polycystic right ovary was successfully de-torsed and the ovary and tube regained their normal colors. Three months later, the pa-tient experienced a similar set of symptoms, with the radiologic results being once again consistent with recurrent torsion of the right adnexa. The diagnosis was confirmed by a second laparoscopic exploration, and the utero-ovarian ligament was sutured with a non-absorbable suture. The patient started having milder, more persistent right abdominal discomfort two months after the second surgery. This time, MRI revealed an enlarged, Figure 2. PRISMA flow diagram visually summarizing the screening process.3. Ovarian Torsion in Polycystic Ovary SyndromeBy scholastically searching the literature, we were to identify a total of twelve cases ofadnexal torsion in patients with polycystic ovaries aged between 8 and 37 years old (meanage at diagnosis: 25 years old).Asch et al. commented that torsion can be difficult to diagnose in patients withpolycystic ovary syndrome who undergo ovarian stimulation, given the already enlargedovaries at baseline [24].Giulini et al. presented the case of a 31-year-old woman with polycystic ovary syn-drome and right adnexal torsion during pregnancy after an oocyte in vitro maturationand intracytoplasmic sperm injection cycle. Two days after embryo transfer, the patientpresented with right lower abdominal pain and leukocytosis. Transvaginal ultrasoundrevealed an enlarged right ovary, alongside normal bilateral ovarian vascular flow. Due toBiomedicines 2023, 11, 2503 5 of 11symptom deterioration, an explorative laparoscopy was preformed, identifying a twistedright adnexa with an ischemic ovary. After successful laparoscopic detorsion with preserva-tion of adnexa, the patient had an uneventful postoperative course and delivered a healthyinfant at 40 weeks of gestation [25].Furthermore, Gonçalves et al. reported the case of a 31-year-old woman with polycys-tic ovary syndrome and recurrent right adnexal torsion. The patient first presented withacute right pelvic pain and the sonographic constellation of acute adnexal torsion. Afteremergency laparoscopy, the enlarged polycystic right ovary was successfully detorsed andthe ovary and tube regained their normal colors. Three months later, the patient experi-enced a similar set of symptoms, with the radiologic results being once again consistentwith recurrent torsion of the right adnexa. The diagnosis was confirmed by a second la-paroscopic exploration, and the utero-ovarian ligament was sutured with a non-absorbablesuture. The patient started having milder, more persistent right abdominal discomfort twomonths after the second surgery. This time, MRI revealed an enlarged, edematous rightovary with ipsilateral abnormal ovarian enhancement, suggesting a subacute process. Thesecond recurrence of right adnexal torsion was confirmed by a subsequent laparoscopy.Two fixation points were used to perform a different oophoropexy between the right adnexaand the ipsilateral round ligament. The right adnexa torsion returned after the patienthad been followed up for a full year without experiencing any pertinent symptoms. Thedecision to perform a unilateral right adnexectomy was met after thorough explanationand the patient’s informed consent. The procedure went smoothly, and the recovery timewas typical. An ovary that was necrotic and hemorrhagic was found during anatomopatho-logical analysis. Consequently, the patient exhibited no further signs of a recurrence ofcontralateral ovarian torsion [26].Moreover, Hiei et al. described the case of a 22-year-old patient with polycystic ovariesand a six-hour history of right lower abdominal pain upon hospital admission. MRIrevealed bilateral enlarged ovaries with a right twisted and thickened peduncle, a findingthat ultimately led to the decision to perform an exploratory laparotomy with detorsion ofthe twisted ovary and drilling of the bilateral ovaries [27].Additionally, Tay et al. announced the case of a 31-year-old woman with polycysticovarian syndrome who experienced chronic, severe pelvic pain and presented with asonographically suspected hydrosalpinx and a small amount of free fluid. An exploratorylaparoscopy revealed that the woman had isolated left fallopian tube torsion. Followingsuccessful detorsion, the patient reported symptom absence at the six-week follow-up [28].Matsuoka et al. presented a case of polycystic ovaries with bilateral adnexal torsionoccurring asynchronously during a natural cycle of a 37-year-old woman suffering fromlower left abdominal pain. Left ovarian edematous swelling, alongside ventral movementto the uterus, was detected on ultrasound and MRI, thus rendering an urgent laparoscopicadnexectomy necessary. Nine months after this primary operation, she began to experienceright lower abdominal pain. The patient underwent a second emergency laparotomy, aftersuspected right adnexal torsion was discovered by ultrasonography. The right ovary hadpartial polycystic changes, was 7 cm in size, and was twisted 540 degrees counterclockwise.After the right adnexectomy, the patient had an uneventful postoperative course andreceived hormone replacement therapy [29].Furthermore, Murakami et al. published their case report on a 21-year-old nulliparouswoman with polycystic ovaries who complained of right lower abdominal pain and wasdiagnosed with ischemic edema of the right ovary by MRI. A necrotic right ovary thatwas purplish-black in color and had undergone a 540◦ torsion around the utero-ovarianligament was discovered during an emergency laparoscopy, justifying the decision toconduct a successful right salpingo-oophorectomy [30].Moreover, Obut et al. announced the impressive case of left ovarian torsion of a21-year-old female patient with polycystic ovarian syndrome for the seventh time in a row.At the fifth and sixth laparoscopic surgeries, ovarian fixation was attempted in additionto detorsion but failed. The authors used a different technique: folding the utero-ovarianBiomedicines 2023, 11, 2503 6 of 11ligament, which had folded on itself due to the recurrence of ovarian torsion followingovarian fixation. The ovary and the proximal portion of the round ligament, which wasnext to the uterus, were both fixed to the distal portion of the utero-ovarian ligament. Thepostoperative period was uneventful, with the blood flow to both ovaries remaining normalat follow-up [31].In addition, Pryor et al. reported on a 29-year-old patient with polycystic ovarysyndrome who developed ovarian hyperstimulation syndrome during an IVF-ET cycleand experienced a torsion of the right tube and ovarian pedicle. Cyst drainage wasperformed, and a pelviscopic technique was used in a detorsion attempt; however, alaparotomy was ultimately necessary to complete the detorsion. The distal tube’s colorreturned to normal after the pedicle had been rotated 360 degrees. Although the tube wasedematous, no palpable thrombus was present. Following surgery, the patient experiencedsymptoms of ileus. About 40 h after the initial procedure, an exploratory laparotomy wasconducted. Non-purulent ascitic fluid and a necrotic right ovary without recurrent torsionwere discovered. During the right salpingo-oophorectomy, a thrombus was seen in thevessels as the pedicle was being cut [32].Shah et al. suggested screening premenarchal girls with ovarian torsion, who donot show obvious ovarian pathology, for ultrasound and biochemical signs of polycysticovary syndrome. In cases where polycystic ovary syndrome is present, oral contraceptivesmay be used to reduce ovarian volume, taking into account the patient’s age and pubertaldevelopment [33].Sheizaf et al. reported the case of an 8-year-old girl with polycystic ovaries thatunderwent a laparoscopic untwisting after initially presenting with right adnexal torsion.Four additional laparoscopies were required over the course of the next three years to treatleft adnexal torsions. Torsion returned despite twice undergoing bilateral utero-ovarianligament plication. The left ovary was ultimately fixed to the sidewall just below the pelvicbrim [34].Shi et al. investigated a 34-year-old woman with polycystic ovarian syndrome and ahistory of infertility who presented with abdominal pain and persistently enlarged ovaries.Ultrasonography and serum tests were used to assess the enlarged ovaries. The patientwas treated by diagnostic laparoscopic surgery with detorsion and drainage followedby Gonadotropin-Releasing Hormone agonist (GnRH-a) therapy. Five days after theprocedure, the patient was released from the hospital without any notable complications.After receiving GnRH-a injections every month for three consecutive months, both ovarieswere nearly back to normal [35].Shukunami et al. published the case of a 19-year-old nulligravida with polycysticovaries who was diagnosed with twisted right adnexa and an unexplained anemia withsubsequent shock. Emergent laparotomy revealed torsion of a right paraovarian tumor to-gether with a right polycystic ovary. The finding of necrotic adnexa justified the conductionof a right salpingo-oophorectomy. After an uneventful recovery without blood transfusion,the patient was discharged in a stable condition [36].Simsek et al. announced the case of a 20-year-old woman with polycystic ovaries whopresented with right adnexal torsion for the sixth time. In the third and fifth laparoscopies,she experienced two unsuccessful ovarian fixation attempts. At elective surgery one monthafter the last detorsion operation, a combined ovarian fixation method was employed inorder to fix the ovary to the pelvic side wall and shorten the utero-ovarian ligament. Atfollow-up, there was no evidence of recurrent adnexal torsion [37].Table 1 summarizes the characteristics of the aforementioned cases.Biomedicines 2023, 11, 2503 7 of 11Table 1. Cases of patients with polycystic ovary syndrome and ovarian torsion.Study Age(Years)TorsionSide Symptoms Diagnostic Evaluation Therapy RemarksGiulini et al.[25] 31 Right ovaryTenderness in theright lowerabdominalquadrant.Leukocytosis.Transvaginal ultrasound:enlarged right ovarywithin coexistent mass andsmall amount of fluidin the pouch of Douglas.Laparoscopic detorsionwith recoloration anda decrease in size ofadnexal edema.First report ofadnexaltorsion after anin vitromaturation cycle.Gonçalveset al. [26] 31 Right ovarySudden rightpelvic pain.Ultrasound and MRI of theright ovary: enlargededematous rightovary with ipsilateralabnormal ovarianenhancement color sign.Laparoscopic detorsionwith recoloration anda decrease in size ofadnexal edema.Plication of theutero-ovarian ligament.Oophoropexy to theround ligament.Ultimately, unilateralright adnexectomy.Recurrent rightadnexal torsion.Hiei et al.[27] 22 Right ovaryReboundtenderness in therightlower abdomen.MRI: bilateral enlargedovaries with a right twistedand thickened peduncle.Detorsion of the twistedovary and drilling of thebilateral ovaries vialaparotomy.Ultrasound wasconsistent withtheMRI findings ofpolycystic ovarysyndrome butfailed to detectthe stalkconditions.Tay et al. [28] 31LeftfallopiantubeRecurrentepisodes ofsevere pelvicpain.Ultrasound: suspectedhydrosalpinx and a smallamount of free fluid.Diagnostic laparoscopy.Isolated fallopiantube torsion in apatient withknown polycysticovariansyndrome.Matsuokaet al. [29] 37Bothovaries1. Leftlower abdominalpain;2. Right lowerabdominal pain.1. Plain CT:ovarianswelling,MRI: leftovary exhibited edematousswelling locatedsuperior–anterior to the uteruswith partial cystic changes;2. CT:mass with uneven internalabsorptionanterior to the uterus,1. Laparoscopic leftadnexectomy;2. Right adnexectomy vialaparotomy.Ultrasonographycould not identifyeither of theovaries.Bilateral ovariantorsion.Murakamiet al. [30] 21 Right ovaryRightlower abdominalpain.ultrasonography:enlarged right ovary,MRI: ischemicedema of the right ovary.Emergency laparoscopywith rightsalpingo-oophorectomy.Ovarian torsionassociated withcessation ofhormonaltreatment forpolycysticovariansyndrome.Obut et al.[31] 21 Left ovaryPain in the lowerleft quadrant ofthe abdomen.Ultrasound: enlarged leftovary with diminished bloodperfusion.Laparoscopy with foldingand fixation of theutero-ovarian ligament tothe round ligament.Seventhrecurrence of leftovarian torsion.Biomedicines 2023, 11, 2503 8 of 11Table 1. Cont.Study Age(Years)TorsionSide Symptoms Diagnostic Evaluation Therapy RemarksPryor et al.[32] 29 Right ovaryNausea, vomiting,and rightlower quadrantpain.Positive serumpregnancy test, leukocytosis,and enlarged right ovary inultrasound.Cyst drainageand detorsion bypelviscopic techniqueand subsequentlaparotomy with rightsalpingo-oophorectomy.Adnexalinfarction afterconservativesurgicalmanagementof torsion of ahyperstimulatedovary in apregnant patient.Sheizaf et al.[34] 81. Rightovary;2. Leftovary;3. Leftovary;4. Leftovary;5. Leftovary.1. Cramping rightabdominal painand vomiting;2. Left abdominalpain;3. Left abdominalpain;4. Left abdominalpain;5. Left abdominalpain.1. Ultrasound: abundant freefluid and a cysticmass in the pelvis,CT scan: suggestive of rightovarian torsion;4. Ultrasound:enlarged left ovary withno significant blood flow.1. Laparoscopic detorsion;2. Laparoscopic detorsion;3. Laparoscopic detorsionwith bilateral plication;4. Laparoscopic detorsionwith bilateral plication;5. Laparoscopic detorsionwith oophoropexy.Recurrence aftertwooophoropexiesin a prepubertalgirl.Shi et al. [35] 34 Left ovaryLeft lowerquadrantabdominal pain.Leukocytosis, ultrasoundimaging, and CT:enlarged ovaries withmultiple follicles.Laparoscopic detorsion.Persistentmegalocysticovaries afterovarianhyperstimulationsyndrome in apostpartumpatient.Shukunamiet al. [36] 19 Right ovaryAcute abdomenand hemorrhagicshock.Ultrasound: cystic mass in theright side of the uterus.Emergency laparotomywith rightsalpingo-oophorectomy.Twistedparaovariancyst together withan ipsilateralpolycystic ovary.Simsek et al.[37] 20 Right ovaryRight lowerquadranttenderness andreboundtenderness.Ultrasonography: enlargedright ovary with minimalpelvic fluid;Dopplerinvestigation: absence ofblood flow to the right ovary.Laparoscopic detorsionand ovariopexy.Repeatedovariopexyfailure inrecurrent adnexaltorsion.4. DiscussionComplete or partial rotation of the adnexal supporting organ with ischemia is referredto as ovarian torsion, to which females of all ages are susceptible. In between 2% and 15% ofpatients who undergo surgical treatment for adnexal masses, ovarian torsion develops [16].An ovarian mass is the main risk in the context of ovarian torsion. According to datafrom the World Health Organization (WHO), polycystic ovary syndrome affects about 116million women worldwide [38]. As such, enlarged polycystic ovaries could be regardedas one of the major threatening factors leading to ovarian torsion and potentially adnexalnecrosis. Nonetheless, no review article to date has been published on the correlationof polycystic ovary syndrome and ovarian torsion. The present work represents, to thebest of our knowledge, the most up-to-date comprehensive review of the literature on theoccurrence of ovarian/adnexal torsion in patients with polycystic ovaries.The majority of ovarian torsion cases occurs in women of reproductive age, whereaspostmenopausal or premenarchal women are less likely to experience it. In the presentreview, we present the cases of adnexal torsion in twelve patients with polycystic ovariesaged between 8 and 37 years old (mean age at diagnosis: 25 years old). Interestingly, of thetwelve reported cases, only one patient was premenarchal and had experienced a total ofBiomedicines 2023, 11, 2503 9 of 11five adnexal torsions bilaterally, while there was no reported case of ovarian or adnexaltorsion in postmenopausal women with polycystic ovaries.As far as symptomatology is concerned, most women seem to present with severelower abdominal pain and even signs of acute abdomen. The pain is mostly acute and mayeven lead to vegetative symptoms such as nausea or vomiting. In cases of left-sided acutepain, diverticulitis always needs to be excluded, whereas appendicitis constitutes the mostimportant exclusion diagnosis in the context of acute right-sided lower abdominal painattacks.For diagnostic purposes, laboratory testing is always required in order to exclude acuteconditions such as bleeding or fulminant infections. However, in most cases of ovariantorsion and polycystic ovary syndrome, values seem to be within normal ranges. Imagingalways includes a first sonographic evaluation of the ovarian size and blood supply. Ifinconclusive, abdominal CT scans and/or pelvic MRI scans may endorse the diagnosticevaluation process and help exclude ovarian torsion. In cases of diagnostic uncertainty,diagnostic laparoscopy or even laparotomy need to be performed with a view to directlyvisualize the polycystic ovaries and identify a feared adnexal or ovarian torsion.Emergent laparoscopy with ovarian detorsion and eventual bilateral ovariopexy incase of feared recurrence is the gold standard in the context of ovarian and/or adnexal tor-sion in polycystic ovary syndrome. Surgical detorsion of the ovary should be performed assoon as possible; otherwise, ovarian necrosis may occur, rendering salpingo-oophorectomyinevitable. Laparotomy for ovarian/adnexal detorsion and/or salpingo-oophorectomyneeds to be performed only in cases that do not allow for a laparoscopic and, hence,less-invasive, therapeutic approach.Interestingly enough, recurrent or even habitual ovarian torsions seem to represent amajor threat in patients with polycystic ovaries. Such conditions undoubtedly representa challenge for the operating surgeon given the complexity of each case and the lack ofstandardized surgical approaches to this matter. Most importantly, each recurrence may beassociated with severe symptomatology; complicate the procedure; and, in the worst-casescenario, even lead to permanent dysfunction and consecutive loss of the ovary and/or theadnexa.The nonsystematic methodology regarding the study selection is one limitation ofthe current review. Even though the strategy of systematic literature reviews is the mostaccurate in terms of relevant study detection, because of the strict rules and standards,this approach demands a specific research question that fails to cover broader topics. Theeventual evidence selection bias, arising from publication bias, represents an additionallimitation, given that data from statistically significant studies usually succeed in reachingpublication. Furthermore, a single person performed the literature analysis, which was,in turn, conducted employing solely two databases. Lastly, eventually relevant originalresearch articles not written in the English language were not included in the data analysis.5. ConclusionsTaken altogether, ovarian/adnexal torsion can occur in patients with polycystic ovarysyndrome. Acute lower abdominal pain in patients with known polycystic ovaries shouldnot be underestimated and diagnostic assessment including transvaginal ultrasound andCT or MRI scans should be performed without delay. In case of suspected torsion, emer-gency laparoscopy with ovarian or adnexal detorsion needs to be performed in order tosave the organ. In cases of repeated ovarian/adnexal torsions, ovariopexy or ovariec-tomy/adnexectomy need to be discussed with the patient in order to reduce the risk ofrecurrence.Author Contributions: Literature analysis and conceptualization, I.P., N.G. and C.D.; original draftpreparation and writing, I.P., N.G. and C.D.; artwork, N.G.; review and supervision, N.G., A.G., K.V.,E.A.A., D.D., K.K., K.N., A.C. and C.D. All authors have read and agreed to the published version ofthe manuscript.Biomedicines 2023, 11, 2503 10 of 11Funding: This research received no external funding.Institutional Review Board Statement: Not applicable.Informed Consent Statement: Not applicable.Data Availability Statement: Not applicable.Conflicts of Interest: The authors declare no conflict of interest.References1. McGowan, M.P. Polycystic ovary syndrome: A common endocrine disorder and risk factor for vascular disease. Curr. Treat.Options Cardiovasc. Med. 2011, 13, 289–301. [CrossRef]2. Rasquin, L.I.; Anastasopoulou, C.; Mayrin, J.V. Polycystic Ovarian Disease. 15 November 2022; In StatPearls [Internet]; StatPearlsPublishing: Treasure Island, FL, USA, 2023.3. Gleicher, N.; Darmon, S.; Patrizio, P.; Barad, D.H. Reconsidering the Polycystic Ovary Syndrome (PCOS). Biomedicines 2022, 10,1505. [CrossRef]4. Al Wattar, B.H.; Fisher, M.; Bevington, L.; Talaulikar, V.; Davies, M.; Conway, G.; Yasmin, E. Clinical Practice Guidelines onthe Diagnosis and Management of Polycystic Ovary Syndrome: A Systematic Review and Quality Assessment Study. J. Clin.Endocrinol. Metab. 2021, 106, 2436–2446. [CrossRef] [PubMed]5. Legro, R.S.; Arslanian, S.A.; Ehrmann, D.A.; Hoeger, K.M.; Murad, M.H.; Pasquali, R.; Welt, C.K.; Endocrine, S. Diagnosis andtreatment of polycystic ovary syndrome: An Endocrine Society clinical practice guideline. J. Clin. Endocrinol. Metab. 2013, 98,4565–4592. [CrossRef]6. Hakimi, O.; Cameron, L.C. Effect of Exercise on Ovulation: A Systematic Review. Sports Med. 2017, 47, 1555–1567. [CrossRef]7. Brennan, L.; Teede, H.; Skouteris, H.; Linardon, J.; Hill, B.; Moran, L. Lifestyle and Behavioral Management of Polycystic OvarySyndrome. J. Womens Health 2017, 26, 836–848. [CrossRef]8. Sadeghi, H.M.; Adeli, I.; Calina, D.; Docea, A.O.; Mousavi, T.; Daniali, M.; Nikfar, S.; Tsatsakis, A.; Abdollahi, M. PolycysticOvary Syndrome: A Comprehensive Review of Pathogenesis, Management, and Drug Repurposing. Int. J. Mol. Sci. 2022, 23, 583.[CrossRef]9. Psilopatis, I.; Vrettou, K.; Nousiopoulou, E.; Palamaris, K.; Theocharis, S. The Role of Peroxisome Proliferator-Activated Receptorsin Polycystic Ovary Syndrome. J. Clin. Med. 2023, 12, 2912. [CrossRef]10. Hoeger, K.M.; Dokras, A.; Piltonen, T. Update on PCOS: Consequences, Challenges, and Guiding Treatment. J. Clin. Endocrinol.Metab. 2021, 106, e1071–e1083. [CrossRef]11. Menichini, D.; Forte, G.; Orru, B.; Gullo, G.; Unfer, V.; Facchinetti, F. The role of vitamin D in metabolic and reproductivedisturbances of polycystic ovary syndrome: A narrative mini-review. Int. J. Vitam. Nutr. Res. 2022, 92, 126–133. [CrossRef]12. Lagana, A.S.; Monti, N.; Fedeli, V.; Gullo, G.; Bizzarri, M. Does Alpha-lipoic acid improve effects on polycystic ovary syndrome?Eur. Rev. Med. Pharmacol. Sci. 2022, 26, 1241–1247. [CrossRef]13. Coldebella, D.; Buzzaccarini, G.; Ferrari, J.; Sleiman, Z.; D’Alterio, M.N.; Corte, L.D.; Cucinella, G.; Gullo, G. Inositols administra-tion: Further insights on their biological role. Ital. JOG 2023, 35, 30–36. [CrossRef]14. Etrusco, A.; Buzzaccarini, G.; Cucinella, G.; Agrusa, A.; Di Buono, G.; Noventa, M.; Lagana, A.S.; Chiantera, V.; Gullo, G.Luteinised unruptured follicle syndrome: Pathophysiological background and new target therapy in assisted reproductivetreatments. J. Obstet. Gynaecol. 2022, 42, 3424–3428. [CrossRef]15. Burgio, S.; Polizzi, C.; Buzzaccarini, G.; Lagana, A.S.; Gullo, G.; Perricone, G.; Perino, A.; Cucinella, G.; Alesi, M. Psychologicalvariables in medically assisted reproduction: A systematic review. Prz. Menopauzalny 2022, 21, 47–63. [CrossRef]16. Huang, C.; Hong, M.K.; Ding, D.C. A review of ovary torsion. Ci Ji Yi Xue Za Zhi 2017, 29, 143–147. [CrossRef]17. Pansky, M.; Smorgick, N.; Herman, A.; Schneider, D.; Halperin, R. Torsion of normal adnexa in postmenarchal women and risk ofrecurrence. Obstet. Gynecol. 2007, 109, 355–359. [CrossRef]18. Guile, S.L.; Mathai, J.K. Ovarian Torsion. 17 July 2023; In StatPearls [Internet]; StatPearls Publishing: Treasure Island, FL, USA,2023.19. Chang-Patel, E.J.; Palacios-Helgeson, L.K.; Gould, C.H. Adnexal torsion: A review of diagnosis and management strategies. Curr.Opin. Obstet. Gynecol. 2022, 34, 196–203. [CrossRef]20. Rostamzadeh, A.; Mirfendereski, S.; Rezaie, M.J.; Rezaei, S. Diagnostic efficacy of sonography for diagnosis of ovarian torsion.Pak. J. Med. Sci. 2014, 30, 413–416. [CrossRef]21. Singh, T.; Prabhakar, N.; Singla, V.; Bagga, R.; Khandelwal, N. Spectrum of magnetic resonance imaging findings in ovariantorsion. Pol. J. Radiol. 2018, 83, e588–e599. [CrossRef]22. Singh, S.; Sasmal, P.K.; Nagarajan, K. CT Imaging in Predicting Ovarian Torsion: Report of Two Cases, With and WithoutInfarction. Cureus 2021, 13, e17082. [CrossRef]23. Winton, C.; Yamoah, K. Ovarian torsion and laparoscopy in the paediatric and adolescent population. BMJ Case Rep. 2020, 13,e232610. [CrossRef] [PubMed]24. Asch, E.; Wei, J.; Mortele, K.J.; Humm, K.; Thornton, K.; Levine, D. Magnetic resonance imaging performance for diagnosis ofovarian torsion in pregnant women with stimulated ovaries. Fertil. Res. Pract. 2017, 3, 13. [CrossRef] [PubMed]Biomedicines 2023, 11, 2503 11 of 1125. Giulini, S.; Dante, G.; Xella, S.; La Marca, A.; Marsella, T.; Volpe, A. Adnexal Torsion during Pregnancy after Oocyte In VitroMaturation and Intracytoplasmic Sperm Injection Cycle. Case Rep. Med. 2010, 2010, 141875. [CrossRef]26. Goncalves, I.S.; Sampaio, J.; Felix, J.; Silva, A.C.; Fornelos, G.; Silva, P.T. Oophoropexy to the Round Ligament after RecurrentAdnexal Torsion. Rev. Bras. Ginecol. Obstet. 2018, 40, 726–730. [CrossRef]27. Hiei, K.; Takagi, H.; Matsunami, K.; Imai, A. Ovarian torsion; early diagnosis by MRI to prevent irreversible damage. Clin. Exp.Obstet. Gynecol. 2010, 37, 233–234.28. Tay, J.; Parker, H.; Dhange, P.; Paton-Forrester, C.; Atiomo, W. Isolated torsion of the fallopian tube in a patient with polycysticovarian syndrome (PCOS). Eur. J. Obstet. Gynecol. Reprod. Biol. 2010, 150, 218–219. [CrossRef]29. Matsuoka, S.; Kobayashi, T.; Kusunoki, S.; Ogishima, D. Polycystic ovary syndrome with asynchronous bilateral adnexal torsionin a natural cycle. BMJ Case Rep. 2017, 2017, bcr2016218880. [CrossRef]30. Murakami, M.; Takiguchi, E.; Hayashi, S.; Nakagawa, Y.; Iwasa, T. Ovarian torsion associated with cessation of hormonaltreatment for polycystic ovarian syndrome: A case report. Clin. Exp. Obstet. Gynecol. 2013, 40, 609–611. [PubMed]31. Obut, M.; Deger, U. A New Technique of Oophoropexy: Folding and Fixating of Utero-Ovarian Ligament to Round Ligament in aPatient with Reccurrent Ovarian Torsion. Case Rep. Obstet. Gynecol. 2019, 2019, 7647091. [CrossRef]32. Pryor, R.A.; Wiczyk, H.P.; O’Shea, D.L. Adnexal infarction after conservative surgical management of torsion of a hyperstimulatedovary. Fertil. Steril. 1995, 63, 1344–1346. [CrossRef]33. Shah, A.A.; Likes, C.E.; Price, T.M. Early polycystic ovary syndrome as a possible etiology of unexplained premenarcheal ovariantorsion. J. Pediatr. Adolesc. Gynecol. 2009, 22, 265–269. [CrossRef] [PubMed]34. Sheizaf, B.; Ohana, E.; Weintraub, A.Y. “Habitual adnexal torsions”—recurrence after two oophoropexies in a prepubertal girl: Acase report and review of the literature. J. Pediatr. Adolesc. Gynecol. 2013, 26, e81–e84. [CrossRef] [PubMed]35. Shi, J.; Ren, X.; Tian, Q.; Sun, A.; Chen, R. Persistent megalocystic ovaries after ovarian hyperstimulation syndrome in apostpartum patient with polycystic ovarian syndrome: A case report. J. Ovarian Res. 2018, 11, 79. [CrossRef]36. Shukunami, K.; Nishijima, K.; Orisaka, M.; Yoshida, Y.; Kotsuji, F. Acute abdomen in a Jehovah’s witness with chronic anemia.Am. J. Emerg. Med. 2004, 22, 242–243. [CrossRef] [PubMed]37. Simsek, E.; Kilicdag, E.; Kalayci, H.; Yuksel Simsek, S.; Parlakgumus, A. Repeated ovariopexy failure in recurrent adnexal torsion:Combined approach and review of the literature. Eur. J. Obstet. Gynecol. Reprod. Biol. 2013, 170, 305–308. [CrossRef]38. Jabeen, A.; Yamini, V.; Rahman Amberina, A.; Dinesh Eshwar, M.; Vadakedath, S.; Begum, G.S.; Kandi, V. Polycystic OvarianSyndrome: Prevalence, Predisposing Factors, and Awareness Among Adolescent and Young Girls of South India. Cureus 2022, 14,e27943. [CrossRef] [PubMed]Disclaimer/Publisher’s Note: The statements, opinions and data contained in all publications are solely those of the individualauthor(s) and contributor(s) and not of MDPI and/or the editor(s). MDPI and/or the editor(s) disclaim responsibility for any injury topeople or property resulting from any ideas, methods, instructions or products referred to in the content. |
|---|