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The Role of Surgery in Antenatal Ovarian Torsion: Retrospective

Evaluation of 28 Cases and Review of the Literature

Bade Toker Kurtmen , Emre Divarci , Orkan Ergun , Geylani Ozok ,

Ahmet Celik

PII: S1083-3188(21)00268-0
DOI: https://doi.org/10.1016/j.jpag.2021.08.007
Reference: PEDADO 2714

To appear in: Journal of Pediatric and Adolescent Gynecology

Received date: 24 May 2021

Revised date: 30 July 2021
Accepted date: 7 August 2021

Please cite this article as: Bade Toker Kurtmen , Emre Divarci , Orkan Ergun , Geylani Ozok ,
Ahmet Celik , The Role of Surgery in Antenatal Ovarian Torsion: Retrospective Evaluation of 28
Cases and Review of the Literature, Journal of Pediatric and Adolescent Gynecology (2021), doi:
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ORIGINAL ARTICLE

The Role of Surgery in Antenatal Ovarian Torsion: Retrospective

Evaluation of 28 Cases and Review of the Literature

Bade Toker Kurtmen, Emre Divarci, Orkan Ergun, Geylani Ozok, Ahmet Celik

Department of Pediatric Surgery, Ege University Faculty of Medicine, İzmir, Turkey

*Corresponding author at Ege University Faculty of Medicine, Department of Pediatric Surgery, 35100, Bornova, İzmir, Turkey. Tel.:

+90 2323902800; fax: +90 2323902802

E-mail addresses: [email protected] (Celik A)

1
Abstract

Background: Antenatal ovarian torsion (AOT) is rare and requires differentiating from other congenital
cystic masses of the abdomen and pelvis in neonates. In this study, it was aimed to evaluate the prenatal
characteristics and postnatal outcomes of AOT.

Patients and methods: Hospital records of patients (n = 28) with diagnosis of AOT between 2004 and
2020 were reviewed and their prenatal characteristics, postnatal examination, imaging, operative and
histopathological findings were evaluated.

Results: There were 28 patients during the study period. Majority of the patients (89.3%) were
prenatally detected. All were term babies with a mean birth weight of 3010±466.6 grams. Mean
maternal age was 25.75±3.65 years. Prenatal history was event-free in all and none had additional
anomalies. Physical examination revealed mobile intraabdominal cystic lesion in 16 (57.1%) patients.
AOT mimicked other pathologies as intestinal duplication cyst/mesenteric cyst (n:7), complex ovarian
cyst (n:3), mature cystic teratoma (n=3), simple renal cyst (n:1), and ectopic kidney (n:1) in 15 patients
(53.6%) in postnatal ultrasonography. Elective laparoscopic surgery was performed in 26 (92.8%)
patients. Other two patients required emergency open surgery due to presenting with acute symptom.
Ovaries were auto-amputated in 17 (60.7%) patients. Hystopathological examination revealed necrosis
and dystrophic calcification in all specimens. There was neoplastic involvement in two patients (Serous
cystadenoma and gonadoblastoma). No complication occurred in early follow-up period (mean:
73.9±46.8 months).

Conclusion: AOTs can be detected easily during antenatal period. Neoplastic involvement with AOT is
rare but possible. Although AOTs may mimic other cystic pathologies, it should be one of the first
diagnoses to be considered, in the presence of a palpable intra-abdominal mass and complex cystic
lesion in infant girls. It can be efficiently and safely managed by minimal invasive techniques.

Keywords: Fetal ovarian cyst; Antenatal ovarian torsion; Adnexal torsion; Autoamputation

2
Introduction

Ovarian cysts are the most common intraabdominal masses found in female fetuses. Its

incidence including milimetric follicles, is reported 98% at the birth [1]. Because of the surrounding

hormonal environment at the antenatal period, cyst development is commonly seen in fetal ovary. Most

of the simple fetal ovarian cysts regress spontaneously after birth. However, especially the large ones

are less likely to resolve spontaneously, and may cause ovarian torsion in the intrauterine period [2].

Ovarian torsion is the most common complication (25-75%) of fetal ovarian cysts and often

occurs in the antenatal period, rather than postnatally [1]. Antenatal adnexal torsion may result in auto-

amputation due to of ischemia and infarction. It is a very rare complication, and its diagnosis and

treatment have not been clearly defined. The characteristic feature of an autoamputated ovary is the

presence of a free-floating mobile mass in the abdominal cavity. A wandering echogenic cyst without

Doppler evidence of perfusion and the fallopian tube blind stump suggest autoamputation [3–5]. We

reported the management and follow-up data of 28 cases with antenatal ovarian torsion and the review

of the literature.

Material and Methods

We reviewed hospital records of patients with a diagnosis of antenatal ovarian torsion (n=28)

between January 2004 to January 2020. We included patients younger than one year old at the time of

surgery and had an operative diagnosis of ovarian torsion. After IRB-approval, clinical data, including

gestational information, associated medical conditions, clinical manifestation, the management and

pathologic records were reviewed. All private health information for the patients was collected.

Informed consent was obtained from all enrolled parents.

For the literature review, a Medline, Web of Science, Ovid and Google Scholar search was

performed using the search terms “ovarian torsion,” “antenatal,” “auto-amputation” and “adnexal

torsion.” Search criteria were restricted to the English language. Reference lists of relevant articles and

reviews were hand searched for additional reports.

3
Results

The mean gestational age of the patients was 38.5±1.48 weeks and mean birth weight was

3010±466.6 g. Maternal age had ranged from 20 to 33 years (mean, 25.75±3.65 years). Twenty-five

(89.3%) of 28 patients were diagnosed antenatally. The age of antenatal diagnosis ranged between 27

and 38 gestational weeks. Only in four patients’ antenatal US findings were reported as antenatal

ovarian torsion (AOT). Most of the patients (n=11) had unspecified abdominal cystic mass in the US;

seven patients were diagnosed as a hemorrhagic fetal ovarian cyst, two were diagnosed as intestinal

duplication in the antenatal period. The maternal medical histories were not remarkable, and prenatal

history was event-free in all. Except for four patients who had an inguinal hernia, none of them had

additional anomalies.

Twenty-five patients were referred to our clinic because of their antenatal diagnosis. Three cases

did not have any pathological finding in the antenatal ultrasonographic examination. In the postnatal

period, one of them had constipation, one had presented to the emergency department due to falling

down from a sofa, and the other one developed bilateral inguinal swelling due to inguinal hernia. Three

of them had been evaluated for these problems with the abdominal US’s, and they were referred after

the detection of abdominal mass on these sonographic evaluations.

Physical examination revealed a mobile intraabdominal mass in 16 patients (57.1%). Four

patients (%14.3) had a reducible inguinal hernia. All of the patients had normal female external

genitalia. In our institution, AFP and hCG measurements are part of the routine preoperative diagnostic

check-up for all cystic changes of the neonatal ovary. Serum levels of α-fetoprotein (<50.000 ng/ml) and

β-HCG (<1.0 IU/L) were determined in the normal range for age, hematologic and biochemical

investigations were within normal limits, in all.

Initial postnatal sonograms had been obtained between 0-30 days after birth (mean, 14.5 days)

expect for one case. When these sonograms were reviewed, it was seen that AOT mimicked other

pathologies such as intestinal duplication cyst/mesenteric cyst (n:7), complex ovarian cyst (n:3), mature

cystic teratoma (n=3), simple renal cyst (n:1), and ectopic kidney (n:1) in 15 patients (53.6%). Their

4
size ranged from 2.4 to 17 cm (mean, 5.2cm), with intracystic debris (n:19; 67.9%), fluid-fluid level

(n:19; 67.9%) and multiple septation (n:13; 46.4%). Further imagining such as CT / MRI were

performed on 8 (28.6%) patients referred from other centers before their admission. Prenatal

characteristics, postnatal examination, imaging findings of the patients are given in Table 1.

Surgery had been performed in all of the patients at the ages of 3 days to 12 months (mean,

85.54±73.74 days). Elective laparoscopic exploration was performed after the end of the neonatal period

in cases with antenatal diagnosis and no reduction in cyst diameter in postnatal control ultrasounds.

Twenty-six (92.9%) of patients were treated by laparoscopic oophorectomy electively. Two patient

required emergency laparotomy. One of them had respiratory distress due to a giant (17cm) cystic mass,

the other had abdominal discomfort and vomiting due to compression of the autoamputated ovary on

adjacent organs. On exploration, all patients had unilateral ovarian pathology and 15 of 28 patients had

left ovarian involvement. Ovaries were auto-amputated in 17 (60.7%) patients; 13 (76,5%) of those 17

patients had intraabdominal adhesions extending to the liver, bowels and anterior abdominal wall. No

intervention such as fixation to the contralateral ovary was performed in any patient.

Histopathological examination revealed necrosis and dystrophic calcification in all specimens.

There was neoplastic involvement in 2 patients (Serous cystadenoma and gonadoblastoma). The patient

diagnosed as gonadoblastoma had the karyotyping result as 46XX. They did not require any further

treatment. No complication occurred during the follow-up period (mean: 73.9±46.8 months).

Management and histopathological features of the patients are given in Table 2.

Discussion

Ovarian cysts are the most common intraabdominal masses found in female fetuses. The first

description of a fetal ovarian cyst was made by Doran in 1889. He reported bilateral ovarian cysts in a

seven-month premature stillborn. In 1898 Power reported the first infant to be successfully treated with

5
surgery [6] and Bulfamonte reported the first successfully treated newborn in 1942 [7]. The first large

review was by Karrer and Swenson in 1961, and they presented 25 cases of ovarian cysts in stillborn

and infants under 1-year of age [6]. Five of the cases (including their own) had ovarian torsion as well.

According to the reviews about antenatal ovarian torsion, Longino described the first successfully

treated ovarian torsion in a newborn [8,9]. In 1975 D.J DeSa reviewed necropsy records of all female

stillbirths and neonatal deaths at 28 days of age or less, and he found small follicular cysts in 113 (34%)

of 332 cases [10]. The first prenatal detection of a fetal ovarian cyst was by Valenti et al. in 1975 [11].

The incidence of antenatally diagnosed and clinically significant ovarian cysts is almost 1/2500

live births. One very comprehensive series on 28,080 female newborns in 2015 has determined an

incidence of 1/1755 [12]. At birth, 98% of newborn girls have small ovarian cysts by ultrasound [13,14].

There is no consensus regarding the etiopathogenesis of ovarian cysts; most commonly ovarian cysts are

believed to be a consequence of maternal gonadotropin stimulation delivered through the placenta to the

fetus. Elevated fetal gonadotropins, elevated placental human chorionic gonadotropin, and maternal

estrogens stimulate the fetal ovary and cause follicle production and maturation [14]. Fetal ovarian cysts

generally manifest in the third trimester and are thought to be a consequence of exposure to elevated

hormonal levels [10,15,16]. Moreover, preterm neonates have immature gonadostat mechanisms that

hyperstimulate their ovaries. Congenital adrenal hyperplasia leads to an excess of adrenal androgens that

either increase follicle-stimulating hormone receptors or interfere with the cyclicity of gonadotropin

release [17]. These cysts are usually unilateral and can be of sizes up to 5 cm in diameter. Nussbaum et

al. in 1988 categorized ovarian cysts into two groups according to ultrasound criteria as being “simple

(uncomplicated)” and “complicated”. According to these criteria, simple cysts are anechogenic and

usually < 2 cm in diameter, thin-walled, unilocular, intrapelvic, or more often intraabdominal, unilateral

or seldom bilateral. Sonographic features of complicated cysts were being thick-walled, heterogeneous

with hyperechogenic components containing free-floating material and intracystic septations [18].

Bascietto et al. demonstrated that ultrasound appearance and cyst size are the major

determinants of the perinatal outcome in these cases. But they also concluded that some small cysts can

change their appearance during pregnancy or after birth and cause an ovarian loss due to surgical

6
oophorectomy or auto-amputation [19]. Tyraskis et al. have shown that cysts greater than 40 mm, are

significantly less likely to resolve spontaneously [2]. In 2019, Rialon et al. concluded that cyst features

such as debris, fluid–fluid or fluid-debris levels, solid components, calcifications, absence of Doppler

flow are more effective in deciding for surgery, but cyst size is not equally important [20]. In 2020 Chen

et al. conducted a cohort of prenatally diagnosed ovarian cysts, and they concluded that spontaneous

resolution of ovarian cysts is predicted by cyst size and characteristics, whereas likelihood of torsion

cannot be predicted [21].

Large cysts may present with complications, such as ascites due to rupture of cyst [22–25],

mass symptoms such as compression of ureters/vena cava, bowel obstruction and necrosis with

intraabdominal adhesions [26], respiratory compromise and fetal/neonatal ovarian torsion with/without

auto-amputation of the ovary. Because of these complications some surgeons recommend prenatal

aspiration of the large (>4cm) fetal ovarian cysts to prevent prenatal torsion [27]. Intrauterine aspiration

of fetal ovarian cyst first performed by Valenti et al. in 1975 [11]. Bagolan et al. had evaluated the

outcome of fetal simple ovarian cysts measuring ≥5 cm in diameter after prenatal aspiration and

suggests that “in utero” aspiration of ovarian cysts is a safe procedure [28,29]. However, any invasive

prenatal procedure has complications including preterm labor, rupture of membranes, bleeding

chorioamnionitis and fetal injury [30]. Amari et al. analyzed the data of 354 fetuses of intra-abdominal

anomalies detected prenatally; only a few cases (6.8%) had undergone prenatal invasive management.

They concluded that it could not alter the unfavorable course of the pregnancy in more than half of the

treated cases [31]. In a systematic review of fetal ovarian cysts’ outcome, showed that the incidence of

recurrence after prenatal invasive therapy was 37.9% [19]. The role of fetal cyst aspiration is still

controversial. There is not enough evidence based data to approve the effectiveness of prenatal

aspiration, and many physicians are concerned about the potential risk of the procedure to both fetus and

mother.

The differential diagnosis of fetal ovarian cysts involves a broad spectrum of various cystic

formations: renal cyst, ureterocele, urinoma, urachal anomalies, dilated bowel, meconium cyst, enteric

duplication cyst, duodenal atresia, lymphangioma, choledochal cyst, cystic neuroblastoma, and anterior

7
meningocele [27,32]. Except for ovarian cysts and many gastrointestinal anomalies, intra-abdominal

cysts are usually detected around 18–20 weeks’ gestation [33]. The overall accuracy of prenatal US in

identifying intraabdominal cyst’s origin is around to 95%. The highest sensitivity of identifying the

nature of the cystic lesion was in the ovarian and mesenteric cysts [27]. There is a 7.5% false positive

rate of prenatal ultrasound in detecting ovarian cysts [19]. In our series, the origin of abdominal cystic

lesions were not identified by prenatal ultrasound in 48% cases, and only 16% of them were reported as

AOT in prenatal period.

AOT can mimic other intraabdominal pathologies in the postnatal US. Determining the correct

diagnosis may be difficult prenatally; therefore early postnatal ultrasonography needs to be performed

for differential diagnosis. In our series, postnatal US identified antenatal ovarian torsion in 46.4% of

patients. Antenatal adnexal torsion may result in auto-amputation due to of ischemia and infarction [3–

5]. In our series 60.7% of patients with AOT had auto-amputated ovary and 76,5% of them had

intraabdominal adhesions.

Neoplastic masses of the gonad in newborns and infants are exceedingly rare in literature. The

first reported case of malignancy in a neonatal ovarian mass was in 1945 by Ziegler; he reported a case

of bilateral ovarian tumors in a 30-week stillborn. The pathologic diagnosis was undifferentiated

carcinoma [34]. Few authors in the English literature have described ovarian neoplasms such as

teratoma, gonadoblastoma, dermoid cysts in infants. [33-37]. In our series 7.14% of patients had

neoplastic involvement in histopathological examination.

Management of intrauterine torsion and autoamputation of the ovary is not clearly defined.

There is no debate about symptomatic ones, requiring prompt surgical intervention; however, there are

some controversies in the ideal treatment of asymptomatic ones. Some authors recommended non-

surgical management and close follow-up with serial US [36,37] while some conversely recommeded

surgical management due to complications like intestinal obstruction from intestinal adhessions [38–40].

Intraabdominal adhesions were detected 50% of patients in our series. This rate was found to be higher

(76.5%) in our patients with auto-amputated ovary. Combined with the information in the literature, we

8
think that these adhesions need to be concomittantly managed by early intervention for the antenatally

torted ovary before presenting with any other and acute symptoms.

Surgical management of antenatal ovarian torsion can be performed by either laparoscopy or

laparotomy. Minimally invasive tecniques such as laparoscopic and mini-laparotomic procedures are

recommended [41]. Laparoscopic removal of a neonatal ovarian torsion was first described in detail in

1995 [42]. In 2007 Lin et al. described the transumbilical management of neonatal ovarian cysts. They

successfully treated two AOT’s with mini-laparotomy in their series [43]. However, since the first

reported case of laparoscopically treatment, about 50% of the reported cases are still treated by

laparotomy. Except for 2 patients who underwent emergency laparotomy in our series, 92.9% of patients

were treated laparoscopically.

Conclusion

Antenatal ovarian torsion, which is one of the first pathologies to be considered in female

babies with abdominal cystic mass, requires careful follow-up because of the possible complications of

intraabdominal adhesions and the rare risk of ovarian neoplasia. It can be treated safely and effectively

with minimally invasive methods.

Conflict of Interest: The authors declare that they have no conflict of interest.

Ethical approval: The Institutional Review Board approved the study.

Patient Consent Statement: Written informed consent was obtained from the patient for
publication.

9
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13
Table 1. Prenatal characteristics, postnatal examination, imaging findings of cases
Initial Postnatal US
Antenatal GA at Physical Days Fluid
diagnosis diagnosis(w examination* after Max. Multiple -
* ) * Birt Diamete Septatio Intracysti Fluid Ultrasonographi
h r (cm) n c Debris Level c diagnosis***
1 UAM 28 - 30 5 - + + IDC/MC
2 UAM 27 MAM 0 17 + + + AOT
3 FOC 30 MAM 20 3.8 + + + AOT
4 UAM 32 MAM 30 6 + + + AOT
5 UAM 28 - 20 3.3 + + + AOT
6 FOC 28 MAM 7 5.4 + + + AOT
7 UAM 27 MAM 8 2.4 - + + IDC/MC
8 No No MAM 27 4.8 + + + AOT
9 No No - 30 3.4 - - - IDC/MC
1 11
UAM 27 - 3.5 - - - COC
0
1 2
UAM 30 - 6 - - - IDC/MC
1
1 1
FOC 32 - 4.6 - + + AOT
2
1 30
IDC 28 - 3.5 - + + AOT
3
1 0 5.7(L),
UAM 37 - - - - IDC (L), SRC (R)
4 5.5(R)
1 0
IDC 28 - 4 - + + IDC/MC
5
1 30
AOT 27 MAM 7.2 - + + AOT
6
1 28
AOT 31 MAM 3.7 - + + AOT
7
1 10
FOC 36 MAM 3 - + + IDC/MC
8
1 3
UAM 34 MAM 4 - - - EK
9
2 4
FOC 34 MAM 7 + + + AOT
0
2 5
FOC 37 MAM 5.2 - - - AOT
1
2 30
AOT 28 MAM 3.7 + + + COC
2
2 FOC 38 MAM 5 3.8 + + + COC

14
3
2 5
UAM 33 MAM 5.9 + + + MCT
4
2 24
UAM 34 MAM 6 + + + IDC/MC
5
2 14
UAM 32 - 4.5 + - - MCT
6
2 15
AOT 32 - 5.2 + - - MCT
7
2 120
No No - 6.9 - - - AOT
8
*UAM: Unspecified abdominal mass, FOC: Fetal ovarian cyst, IDC: Intestinal duplication cyst, AOT: Antenatal
ovarian torsion
**MAM: Mobile abdominal mass
*** IDC: Intestinal duplication cyst, MC: Mesenteric cyst, AOT: Antenatal ovarian torsion, COC: Complex ovarian
cyst, SRC: Simple renal cyst, EK: Ectopic Kidney, MCT: Mature cystic teratoma

15
Table 2. Management and histopathological features of cases

Auto- Intra-
Age at amputation abdominal
operation(d) Operation Side Procedure adhesions Histology

1 120 Laparoscopy + + Necrosis, dystrophic

Right Oophorectomy calcification

2 8 Urgent - Salpingo- - Necrosis, dystrophic

laparotomy Left oophorectomy calcification

3 28 Laparoscopy + + Necrosis, dystrophic

Left Oophorectomy calcification

4 61 Laparoscopy - - Necrosis, dystrophic

Left Oophorectomy calcification

5 23 Laparoscopy - - Necrosis, dystrophic

Left Oophorectomy calcification

6 87 Laparoscopy - - Necrosis, dystrophic

Right Oophorectomy calcification

7 332 Laparoscopy + + Necrosis, dystrophic

Right Oophorectomy calcification

8 85 Laparoscopy - - Necrosis, dystrophic

Right Oophorectomy calcification

9 150 Laparoscopy + - Necrosis, dystrophic

Left Oophorectomy calcification

10 38 Laparoscopy + - Necrosis, dystrophic

Right Oophorectomy calcification

11 115 Urgent + - Necrosis, dystrophic

laparotomy Right Oophorectomy calcification

12 79 Laparoscopy + - Necrosis, dystrophic

Right Oophorectomy calcification

13 84 Laparoscopy + + Necrosis, dystrophic

Left Oophorectomy calcification

14 6 Laparoscopy + + Gonadoblastoma,
Salpingo- necrosis, dystrophic
Right oophorectomy calcification

15 3 Laparoscopy - - Necrosis, dystrophic

Right Oophorectomy calcification

16 55 Laparoscopy - - Necrosis, dystrophic

Right Oophorectomy calcification

17 113 Laparoscopy - - Necrosis, dystrophic

Right Oophorectomy calcification

18 135 Laparoscopy Left + + Serous cystadenoma,

Salpingo-

16
 oophorectomy necrosis, dystrophic
calcification

19 10 Laparoscopy - Salpingo- - Necrosis, dystrophic

Left oophorectomy calcification

20 56 Laparoscopy + Salpingo- + Necrosis, dystrophic

Left oophorectomy calcification

21 59 Laparoscopy - - Necrosis, dystrophic

Left Oophorectomy calcification

22 115 Laparoscopy + + Necrosis, dystrophic

Right Oophorectomy calcification

23 172 Laparoscopy + Salpingo- + Necrosis, dystrophic

Left oophorectomy calcification

24 105 Laparoscopy + + Necrosis, dystrophic

Left Oophorectomy calcification

25 73 Laparoscopy - Salpingo- + Necrosis, dystrophic

Right oophorectomy calcification

26 97 Laparoscopy + Salpingo- + Necrosis, dystrophic

Left oophorectomy calcification

27 76 Laparoscopy + Salpingo- + Necrosis, dystrophic

Left oophorectomy calcification

28 125 Laparoscopy + + Necrosis, dystrophic

Left Oophorectomy calcification

17