Anda di halaman 1dari 5

Pe d i a t r i c I m a g i n g O r i g i n a l R e s e a r c h

Ozcan et al.
Fetal-Neonatal Ovarian Cysts

Downloaded from www.ajronline.org by 111.223.255.18 on 08/31/16 from IP address 111.223.255.18. Copyright ARRS. For personal use only; all rights reserved

Pediatric Imaging
Original Research

Imaging Findings of Fetal-Neonatal


Ovarian Cysts Complicated
With Ovarian Torsion
and Autoamputation
H. Nursun Ozcan1
Serife Balci1
Saniye Ekinci2
Altan Gunes1
Berna Oguz1
Arbay Ozden Ciftci2
Mithat Haliloglu1
Ozcan HN, Balci S, Ekinci S, et al.

OBJECTIVE. Large nonresolving neonatal ovarian cysts may be a risk factor for complications such as torsion, mass effect, rupture, intracystic hemorrhage, and autoamputation.
Torsed cysts and autoamputated cysts can cause a diagnostic dilemma. The objective of our
study was to correlate the imaging findings of intrauterine ovarian torsion and autoamputated
ovaries with their pathologic findings.
MATERIALS AND METHODS. We retrospectively analyzed the pre- and postnatal
medical records, sonographic findings, operation notes, and pathologic reports of 15 patients
with ovarian torsion. All patients had complex cysts noted on postnatal sonographic examination. A complex heterogeneous ovarian cyst was defined by the presence of a fluid-debris
level indicating hemorrhage within the cyst, a retracting clot, septations with or without internal echoes, calcification, and a solid component.
RESULTS. On ultrasound examination, four cysts had solid components, and 11 were
heterogeneous and had a fluid-debris level. Calcifications were seen in two patients. The mean
patient age at the time of surgery was 3.9 months. Exploratory laparotomy was performed on
all patients. Torsed ovaries were identified in five patients. Ten patients had ovaries that were
floating free in the peritoneal cavity at the time of surgery. Histopathologic evaluation revealed that 11 of the cysts consisted of extensive hemorrhagic, necrotic autolytic tissue with
dystrophic calcification. None of the cysts contained any ovarian tissue.
CONCLUSION. A complex heterogeneous ovarian cyst with a fluid-debris level indicating hemorrhage is a significant sonographic hallmark for the diagnosis of ovarian torsion. A
calcified abdominal mass, with or without wandering, can be an autoamputated ovary.

Keywords: intrauterine ovarian torsion, ultrasonography


DOI:10.2214/AJR.14.13426
Received July 12, 2014; accepted after revision
November 13, 2014.
1
Department of Radiology, Hacettepe University Medical
School 06100 Ankara, Turkey. Address correspondence
to H. N. Ozcan (drhnozcan@yahoo.com).
2
Department of Pediatric Surgery, Hacettepe University
Medical School, Ankara, Turkey.

AJR 2015; 205:185189


0361803X/15/2051185
American Roentgen Ray Society

etal ovaries are subject to the development of cysts owing to the


hormonal environment associated with gestation. Increased detection of ovarian cysts during the antenatal
period has resulted from improvements in
imaging techniques. Most fetal ovarian cysts
are small, simple cysts that resolve spontaneously after birth, when the influence of maternal hormones gradually declines. However, larger cysts, which we have defined as
nonresolving cysts measuring more than 5
cm, are at risk for complications that include
torsion, mass effect, rupture, intracystic
hemorrhage, and autoamputation. Torsed
cysts can cause a diagnostic dilemma and
possible complications such as intestinal obstruction [1]. Therefore, imaging evaluation
is very important in the differential diagnosis of ovarian cysts. In the literature, intrauterine ovarian torsion has been reported
from clinical or surgical perspectives but

rarely from the perspective of imaging findings [26].


The aim of this study was to review patients with intrauterine ovarian torsion who
were followed at our hospital. We have correlated their postnatal sonographic findings
with their pathologic findings.
Materials and Methods
Patients
We retrospectively analyzed the pre- and postnatal medical records, postnatal sonographic examination findings, operation notes, and pathologic reports of infants with intrauterine ovarian
torsion who were seen at our hospital between
2004 and 2014. A total of 15 infants who had a
prenatal diagnosis of ovarian cyst were included
in our study. For all infants, postnatal sonographic
examination identified the cyst as complex. At the
time of the antenatal ultrasound examination that
resulted in the initial diagnosis of ovarian cyst, the
mean gestational age was 32.9 weeks (range, 27

AJR:205, July 2015 185

Ozcan et al.
37 weeks). Soon after birth, the infants underwent
surgery performed by a pediatric surgeon.

Downloaded from www.ajronline.org by 111.223.255.18 on 08/31/16 from IP address 111.223.255.18. Copyright ARRS. For personal use only; all rights reserved

Sonography
Prenatal ultrasound examinations were performed by obstetricians according to protocol,
which included routine first-, second-, and thirdtrimester studies. All postnatal ultrasound studies
occurred when patients were between 1 day and
6 months old, and all were performed by radiologists using a sonographic machine with a 7.5-MHz
transducer (Sonoline Elegra, Siemens Healthcare).
Ultrasound studies included a complete evaluation of the abdomen and measurements of the
cyst. A complex heterogeneous ovarian cyst was
defined by the presence of a fluid-debris level indicating hemorrhage within the cyst, cysts with a
retracting clot, septations with or without internal echoes, calcification, and a solid component.
Ultrasound images and reports were reviewed by
three pediatric radiologists.

Results
The demographic characteristics and imaging and pathologic findings of the 15 patients are summarized in Table 1. None of
the patients had a history of maternal diabetes, fetal hypothyroidism, or congenital malformations. All patients had complex ovarian
cysts. Follow-up postnatal ultrasound examinations were performed before surgery and
showed neither spontaneous regression nor
any change in the characteristics of the cysts.
Cyst size ranged from 30 to 70 mm at the
time of prenatal diagnosis and from 25 to 70
mm by the time of surgery.
On follow-up ultrasound examination,
four cysts were found to have solid components (Fig. 1), and the other 11 cysts were
found to have a heterogeneous pattern and
a fluid-debris level (Fig. 2). Color Doppler
imaging showed vascularity in four cysts,
which also had solid components. Calcifications were seen in two patients (Fig. 3).
Intraabdominal free fluid was not identified
in any of the patients. At preoperative ultrasound examination, in addition to evaluation for the presence of complex cysts, the
uterus and contralateral ovaries were also
assessed, and findings were found to be normal for all patients.
One patient underwent MRI examination
before surgery. Ultrasound examination of
that same patient revealed peripheral calcifications in a cyst located in the right side
of the abdomen. The subsequent MR image showed a complex cyst with a fluid-level; however, because the cyst was seen in the

186

Discussion
The majority of fetal-neonatal ovarian
cysts are functional or, less commonly, benign tumors. Hence, it is important to note
that complex cysts in infants are almost
never associated with malignant neoplasm.
However, concern about complications, such
as torsion, plays a significant role in the management of fetal-neonatal ovarian cyst, because such complications can be an indication for surgical intervention [1].
Ovarian torsion and necrosis are followed
by intracystic hemorrhage. Ovarian torsion

can be suggested when ultrasound examination detects changes in the size and characteristic of cysts, resulting in the reclassification of simple cysts as complex cysts.
Ovarian torsion is more commonly seen in
cysts that are 5 cm or greater [7]. Most ovarian torsions occur during and after the antenatal period; they are very rarely seen postnatally [8]. Nussbaum et al. [9] classified the
ultrasound patterns of ovarian cysts as simple or complex on the basis of histopathologic
findings for a series of infants. Simple cysts
are completely anechoic, homogeneous, and
thin walled; are frequently unilocular; and
are located unilaterally. Complex cysts are
typically thick walled, have a solid structure
and septa, and contain blood clots and debris. According to these criteria, all our patients had complex cysts with a fluid-debris
level indicating hemorrhage. Because it was
seen in all our patients, the fluid-debris level
seems to be a significant hallmark for ovarian torsion on ultrasound examination.
Postnatal management of prenatally detected ovarian cysts is controversial. Generally, a wait-and-see policy is preferred
because most cysts resolve spontaneously after birth. On the other hand, early surgery may allow preservation of ovarian tissue if detorsion is possible [1014]. Enrquez
et al. [15] reported involution of 11 clinically
asymptomatic complex fetal-neonatal ovarian cysts, which were conservatively managed and monitored by ultrasound. In their
study, the mean age at which involution of
cysts occurred was 1 year in most cases.
In our series of patients, both symptomatic and asymptomatic complex cysts were
seen. Eight patients had clinical symptoms
caused by the cystic mass, and seven patients
did not have any symptoms before the surgery. The maximum duration of symptoms
during the period from birth to surgery was

Fig. 110-day-old girl with complex ovarian cyst.


Ultrasound image shows heterogeneous solid component
in left ovary (arrow).

Fig. 27-day-old girl with large complex ovarian


cyst. Ultrasound image shows ovarian cyst with fluiddebris level and internal echogenicity.

left side of the abdomen, autoamputation was


suggested (Fig. 4).
The mean patient age at the time of surgery
was 3.9 months (range, 110 months). Presurgical diagnoses included intrauterine ovarian
torsion for 10 patients, benign cystic teratoma
for three patients, and wandering abdominal
mass suggestive of autoamputation for two patients. Eight of the patients were symptomatic
and presented with vomiting, failure to thrive,
and constipation. The other seven patients did
not have any symptoms before surgery.
Exploratory laparotomy was performed
for all patients. Ovarian cysts originated
from the left ovary in seven cases and from
the right ovary in eight cases. Torsed ovaries
were identified in five patients at the time
of surgery. In 10 patients, the ovaries were
brown and fibrotic and were floating free in
the peritoneal cavity. Normal findings were
noted for the contralateral fallopian tube and
ovary in all patients. Histopathologic analysis revealed that, of the 15 cysts that consisted of extensive hemorrhagic necrotic autolytic tissue, 11 were seen with dystrophic
calcification and four were seen without calcification. None of the cysts contained any
ovarian tissue. No complications occurred
during or after the surgery.

AJR:205, July 2015

Gestational Age
at Time of
Prenatal
Ultrasound (wk)

30

28

32

36

37

34

34

32

34

36

35

32

30

37

27

Patient

10

11

12

13

14

15

0/10

0/2

0/10

0/7

1/0

2/0

6/0

0/4

0/3

5/0

0/10

6/0

0/7

0/1

2/0

Postnatal Age at
Diagnosis (mo/d)

51 37

70 60

68 42

60 50

40 30

63 42

62 45

30 25

47 30

60 40

42 30

48 40

50 44

47 35

40 31

Cyst Size (mm)

Fluid-debris level

Fluid-debris level

Fluid-debris level, solid


component

Fluid-debris level, solid


component

Fluid-debris level

Fluid-debris level

Fluid-debris level,
hemorrhage

Solid component,
septation

Fluid-debris level

Fluid-debris level

Solid component

Fluid-debris level,
calcification

Fluid-debris level,
internal echogenicity

Fluid-debris level,
calcification

Fluid-debris level, thick


wall

Postnatal Sonographic
Features

No

No

No

No

No

No

No

No

Yes

No

No

No

No

Yes

No

Wandering
Abdominal
Mass

TABLE 1: Demographic Characteristics and Imaging Findings of the Patients

Torsed ovary

Torsed ovary

Torsed ovary

Torsed ovary

Torsed ovary

Torsed ovary

Torsed ovary

Dermoid

Autoamputated
ovary

Torsed ovary

Torsed ovary

Dermoid

Teratoma

Autoamputated
ovary

Torsed ovary

Preoperative
Diagnosis

Hemorrhagic
infarct, necrosis,
calcification

Hemorrhagic
infarct

Necrosis and
fibrotic tissue
with calcification

Hemorrhagic
infarct, necrosis,
calcification

Hemorrhagic
necrosis,
calcification

Hemorrhagic
necrosis,
calcification

Necrosis with
calcification

Necrosis with
calcification

Torsed ovary

Hemorrhagic
necrosis

Torsed ovary with brownish Hemorrhagic


appearance
necrosis

Torsed ovary with brownish Hemorrhagic


appearance
infarction

Free-floating ovary with a


thin fibrous septa that
attached to the colon

Free-floating ovary

Free-floating ovary

Free-floating ovary

Wandering torsed ovary


Necrosis and
with brownish appearance autolysis with
calcification

Wandering torsed ovary


Necrosis and
with brownish appearance autolysis with
calcification

Wandering torsed ovary

Torsed ovary with brownish Hemorrhagic


appearance
necrosis,
calcification

Free-floating ovary

No

No

No

Yes

Yes

Yes

Yes

Yes

Yes

Yes

No

Yes

No

Yes

Yes

Pathologic Findings Autoamputation

Torsed ovary with brownish Hemorrhagic


appearance
necrosis,
calcification

Free-floating ovary with


brownish appearance

Free-floating ovary

Surgical Findings

Downloaded from www.ajronline.org by 111.223.255.18 on 08/31/16 from IP address 111.223.255.18. Copyright ARRS. For personal use only; all rights reserved

Fetal-Neonatal Ovarian Cysts

AJR:205, July 2015 187

Downloaded from www.ajronline.org by 111.223.255.18 on 08/31/16 from IP address 111.223.255.18. Copyright ARRS. For personal use only; all rights reserved

Ozcan et al.

Fig. 36-month-old-girl with complex ovarian cyst.


A and B, Transverse (A) and sagittal (B) ultrasound images show cyst with fluid-debris level and echogenic areas
with posterior shadowing that correspond to calcification (arrows).

Fig. 44-month-old girl with ovarian cyst.


AC, Axial (A), coronal (B), and sagittal (C) T2-weighted images show large cystic mass with fluid-debris level
located superior and to left of bladder.

188

10 months, and the mean patient age at the


time of surgery was 3.9 months. None of the
cysts showed shrinkage on follow-up ultrasound examination. Pathologic examination
of the excised ovaries revealed a necrotic
cystic mass in all patients, and none of the
patients had viable ovarian tissue.
The presence of vascular flow on Doppler imaging does not exclude ovarian torsion. Preoperative Doppler ultrasound examination showed vascularization of the torsed
ovaries in four patients. Pathologic examination of these patients revealed vascular proliferation in the fibrotic walls of the cysts and
congestion of the fibrotic tissues surrounding
the cyst wall. These findings may have led
to the vascular flow shown on Doppler ultrasound imaging. Thus, sonographically visualized vascularity does not exclude the diagnosis of ovarian torsion.
Another misleading imaging finding is
calcification. The presence of a calcified solid part in an ovarian mass raises the suspicion for teratoma. MRI is used when there is
an equivocal diagnosis or suspicion for malignancy. Of the two patients in our series
who had calcification noted on ultrasound
examination, one underwent MRI examination because malignancy was suspected.
The other patient had autoamputation of the
ovary diagnosed after ultrasound examination showed a change in position compared
with the position noted during a previous
ultrasound examination. Pathologic examination revealed that 11 patients had calcifications in the cyst wall, whereas preoperative ultrasound examination showed such
calcifications in only two patients. For 10
of the 11 patients who had calcifications in
the wall of the cysts, pathologic examination also found calcifications on the ovaries
and identified the ovaries as autoamputated.
In 10 patients, ovaries were floating free in
the peritoneal cavity; however, only two of
the ovaries were found to be wandering on
ultrasound examination. Hence, we suggest
that the presence of a cystic lesion with calcifications, with or without wandering of the
ovary, may suggest the diagnosis of autoamputation in a newborn.
It has been postulated that torsion occurs
more frequently in the right ovary than in the
left ovary because of the close anatomic association of the sigmoid colon with the left
ovary [16]. Similarly, in eight of our patients,
torsion was detected in the right ovary. Torsion of the ovary can cause circulatory impairment that leads to hemorrhagic infarction,

AJR:205, July 2015

Downloaded from www.ajronline.org by 111.223.255.18 on 08/31/16 from IP address 111.223.255.18. Copyright ARRS. For personal use only; all rights reserved

Fetal-Neonatal Ovarian Cysts


rupture resulting in hemoperitoneum, and ascites resulting from transudation or autoamputation of the ovary. In five of our patients,
torsed ovaries were identified at the time of
surgery, and histopathologic examination revealed hemorrhagic and necrotic tissue without the presence of any ovarian tissue.
Autoamputation of the ovary is thought
to result from devascularization caused by
chronic ovarian torsion. Autoamputation of
the ovary in infants younger than 1 year is extremely rare and was first reported by Kennedy et al. [17]. Because autoamputation is
quite rare, the pertinent literature lacks consensus regarding its diagnosis and treatment.
The characteristic feature of an autoamputated ovary is the presence of a free-floating mobile mass or wandering tumor in the abdominal cavity [18]. A change in position of the
ovary, compared with that noted on the prior
examination, indicates a wandering or freefloating ovary, which is an important imaging feature for autoamputation. The diagnosis
can be made during surgery by detection of a
wandering or free-floating ovary in a location
other than the typical anatomic location.
In 10 of the patients in our series, the ovaries were floating freely in the peritoneal
cavity, and in one patient the ovary was attached to the colon with thin fibrous septa,
as seen during surgery. However, only two
of these patients had been given a preoperative diagnosis of autoamputated ovary. For
these patients, the key to diagnosis was identification of the cystic mass in different positions on postnatal ultrasound examination
compared with prenatal ultrasound examination. The cystic masses may also undergo resorption or calcification, and may present as
a mobile calcified abdominal mass.
Ushakov et al. [19] reviewed 23 cases of
teratoma of the greater omentum, and some
of these cases involved autoamputated ovarian teratomas that became reimplanted as
omental masses. The authors identified au-

toamputation and reimplantation of an ovarian dermoid cyst as the most common causes
of omental teratomas. This finding suggests
that autoamputated ovarian cysts might
evolve into omental masses and have malignant potential in the future. Moreover, there
is some concern that a calcified remnant may
result in an increased risk of bowel obstruction or adhesions. In light of these findings,
surgical removal of an autoamputated ovary is preferable to spontaneous regression, as
observed in our patients.
In conclusion, ovarian torsion is the most
frequent complication of fetal-neonatal ovarian cysts. On ultrasound examination, the
presence of a complex heterogeneous ovarian cyst with a fluid-debris level indicating
hemorrhage is a significant hallmark for the
early diagnosis of ovarian torsion. It should
be kept in mind that, with or without wandering, a calcified abdominal mass might be an
autoamputated ovary.
References
1. Brandt ML, Helmrath MA. Ovarian cysts in infants and children. Semin Pediatr Surg 2005;
14:7885
2. Brandt ML, Luks FI, Filiatrault D, et al. Surgical
indications in antenatally diagnosed ovarian
cysts. J Pediatr Surg 1991; 26:276282
3. Bagolan P, Giorlandino C, Nahom A, et al. The
management of fatal ovarian cysts. J Pediatr Surg
2002; 37:2530
4. Koike Y, Inoure M, Uchida K, et al. Ovarian autoamputation in a neonate: a case report with literature review. Pediatr Surg Int 2009; 25:655658
5. Esposito C, Garipoli V, Di Matteo G, et al. Laparoscopic management of ovarian cysts in newborns. Surg Endosc 1998; 12:11521154
6.
Ozyuncu O, Canpolat FE, Ciftci AO, Yurdakok M, Onderoglu LS, Deren O. Perinatal outcomes of fetal abdominal cysts and comparison of
prenatal and postnatal diagnoses. Fetal Diagn
Ther 2010; 28:153159
7. Chiaramonte C, Piscopo A, Cataliotti F. Ovarian

cysts in newborns. Pediatr Surg Int 2001; 17:171174


8. Akn MA, Akn L, zbek S, et al. Fetal-neonatal
ovarian cysts: their monitoring and managementretrospective evaluation of 20 cases and
review of the literature. J Clin Res Pediatr Endocrinol 2010; 2:2833
9. Nussbaum AR, Sanders RC, Hartman DS, Dudgeon DL, Parmley TH. Neonatal ovarian cysts:
sonographic-pathologic correlation. Radiology
1988; 168:817821
10. Croitoru DP, Aaron LE, Laberge JM, Neilson IR,
Guttman FM. Management of complex ovarian
cysts presenting in the first year of life. J Pediatr
Surg 1991; 26:13661368
11. Dolgin SE. Ovarian masses in the newborn. Semin Pediatr Surg 2000; 9:121127
12. Louis-Borrione C, Delarue A, Petit P, Sabiani F,
Guys JM. Antenatal diagnosis of ovarian cyst: perinatal management. Arch Pediatr 2002; 9:417421
13. Vogtlnder MF, Rijntjes-Jacobs EGJ, Van den
Hoonaard TNL, Versteegh FHA. Neonatal ovarian cysts. Acta Paediatr 2003; 92:498501
14. Suita S, Sakaguchi T, Ikeda K, Nakano H. Therapeutic dilemmas associated with antenatally detected ovarian cysts. Surg Gynecol Obstet 1990;
171:502508
15. Enrquez G, Duran C, Toran N, et al. Conservative versus surgical treatment for complex neonatal ovarian cysts: outcomes study. AJR 2005;
185:501508
16. Currarino G, Rutledge JC. Ovarian torsion and amputation resulting in partially calcified, pedunculated cystic mass. Pediatr Radiol 1989; 19:395399
17. Kennedy LA, Pinckney LE, Currarino G, Votteler
TP. Amputated calcified ovaries in children. Radiology 1981; 141:8386
18. Avni EF, Godart S, Israel C, Schmitz C. Ovarian
torsion cyst presenting as a wandering tumour in a
newborn: antenatal diagnosis and postnatal assessment. Pediatr Radiol 1983; 13:169171
19. Ushakov FB, Meirow D, Prus D, Libson E, BenShushan A, Rojansky N. Parasitic ovarian dermoid
tumor of the omentum-A review of the literature
and report of two new cases. Eur J Obstet Gynecol
Reprod Biol 1998; 81:7782

AJR:205, July 2015 189