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Ovarian torsion
ARTICLE in SEMINARS IN PEDIATRIC SURGERY MAY 2005
Impact Factor: 2.22 DOI: 10.1053/j.sempedsurg.2005.01.003
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Darrell Cass
Texas Children's Hospital
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Ovarian torsion
Darrell L. Cass, MD
From the Michael E. DeBakey Department of Surgery, and Department of Pediatrics, Baylor College of Medicine,
Houston, Texas.
INDEX WORDS
Adnexal torsion;
Children;
Management;
Detorsion;
Oophorectomy
Ovarian torsion is a rare problem in the pediatric age group that must be included in the differential
diagnosis of any girl with abdominal pain or a pelvic or abdominal mass. Clinical presentation is
nonspecific, and diagnosis is based on a high index of suspicion. Ultrasound scan remains the most
useful investigation, but blood flow on Doppler examination does not exclude ovarian torsion. Current
recommendations of treatment strongly support ovary conservation, and macroscopic appearance of the
ovary is not a reliable indicator of the degree of necrosis and potential for ovary recovery. For children
with ovarian torsion, laparoscopic detorsion should be performed with strong consideration of oophoropexy. An underlying ovary lesion such as mature teratoma or functional cyst is found in most cases;
however, the risk of cancer in these patients is extremely low. If there is concern of a mass or underlying
pathology, then follow-up ultrasound, resolution of edema and interval laparoscopic treatment may be
required.
2005 Elsevier Inc. All rights reserved.
1055-8586/$ -see front matter 2005 Elsevier Inc. All rights reserved.
doi:10.1053/j.sempedsurg.2005.01.003
Pathophysiology
As in adults, most children with ovarian torsion have pathology in the involved ovary or tube that likely causes the
abnormal twisting.10-12 In adult series, a causative finding
for adnexal torsion is found in 64% to 82% of cases.10-14 In
large pediatric series, the incidence of underlying ovarian
pathology ranges from 51% to 84%.3,4,8,9 The most frequent
pathologic findings in children with adnexal torsion are
benign cystic teratomas, or hemorrhagic or follicular cysts;
Cass
Ovarian Torsion
87
whereas the rate for a borderline tumor was 9.7, and a
benign tumor was 14.3.18 Indeed, 3% to 16% of patients
with mature teratoma of the ovary present with torsion, and
in one series 21% had torsion.20,21 In our experience, 15 of
34 (44%) children who had resection for ovarian torsion had
a mature teratoma in the involved ovary.8 Whether malignant tumors have a lower rate of torsion because they have
more tendency toward local invasion and inflammation, and
thus adherence to surrounding structures, remains speculative.18
Torsion is also a common complication of ovarian cysts
(Figure 2).4 Functional ovarian cysts in children usually
develop as a result of perturbed hormonal stimulation. This
usually occurs during two peak periods: the fetal period and
first year of life, and around the time of menarche. Ovarian
cyst formation in the latter period is associated with changes
in gonadotropin release, and during the initiation of menstruation up to 20% of girls may have multicystic and
enlarged ovaries.22 These functional (follicular and corpus
luteal) cysts enlarge during the menstrual cycle, but usually
resolve within several months. Most data suggest that the
risk of torsion correlates with the size of an ovarian cyst,
and the risk is higher for cysts greater than 4 to 5 cm in
size.23-27 Some authors have suggested that the risk of
torsion correlates better with hypermobility of the adnexa,
and the length and laxity of the ovarian suspensory ligaments.24,28 However, this prognostic factor is not helpful in
deciding the role for surgery in these patients, since it is a
finding that can only be made intraoperatively. The treatment of a fetus, neonate or child with an ovarian cyst has not
been clearly defined (discussed by Drs. Brandt and
Helmrath in this edition of the Journal). It is clear that
operation is indicated for those children with symptoms
related to the ovarian pathology, or those that have a persistent lesion that does not resolve on follow-up ultrasound.
88
It is possible that laparoscopic or image-guided aspiration,
or laparoscopic fenestration or cystectomy may help to
minimize the risk of torsion in girls with large cysts or
persistent cysts. Recently, Bagolan has shown that for cysts
5 cm prenatal aspiration is safe and increases the rate of
spontaneous resolution, and thus may decrease the risk of
torsion.24 However, in this study no data were presented to
demonstrate long-term function of the involved ovary. The
role of aspiration and treatment of a fetus, infant or child
with an ovarian cyst merits further study with a prospective
randomized design.
Adnexal torsion may occur in girls and women with no
underlying adnexal pathology. There are several theories
that have been proposed to explain this process: (1) excess
mobility of the adnexa due to an abnormally long fallopian
tube, mesosalpinx, or mesoovarium; (2) adnexal venous
congestion due to constipation, sigmoid distention, pregnancy, or premenarchal hormonal activity; and (3) jarring
movement of the body in the presence of a small infantile
uterus and a relatively large ovary.29,30 Adnexal torsion is
more common on the right side than on the left, with a ratio
of about 3:2.30,31 The increased risk of right-sided torsion
may be explained by the increased amount of space on the
right side compared with the sigmoid-filled left pelvis, hypermobility of the cecum and ileum on the right, and the
increased likelihood that a patient with right lower quadrant
pain will have more thorough investigation due to concerns
about appendicitis.32
Spontaneous adnexal torsion may occur prenatally or in
the neonatal period. In many instances this may be due to an
underlying follicular cyst or tumor, but in some instances
the underlying adnexa may have no apparent lesion. When
this process occurs it may be asymptomatic and lead to
congenital absence or spontaneous disappearance of the
adnexa. In addition, autoamputation may occur in which
there is a free floating, often partially calcified cystic mass
in the abdomen and a missing ovary on one side.24,33
Asynchronous bilateral ovarian torsion has been described, and occurs even in children with no underlying
adnexa pathology.3,34-40 It is likely that these patients have
underlying anatomic variations that put them at increased
risk of ovarian torsion. Oophoropexy should be strongly
considered in patients with adnexal torsion and no underlying pathologic abnormality, particularly in those that
present with left-sided torsion as the first event.3
Cass
Ovarian Torsion
Management
The optimal management of ovarian torsion has evolved in
the last 10 years. Whereas early diagnosis and prompt surgical evaluation remain key to optimizing long-term ovary
function, there has been a trend toward increased use of
89
laparoscopy, and significant evidence to support detorsion
and ovary conservation, regardless of the appearance of the
ovary at operation.2,3,5,9,53-59
Classic treatment of adnexal torsion for all age groups
has been resection of an ischemic appearing ovary and/or
fallopian tube. Support for this approach has been: (1) the
concern of malignancy associated with torsed ovaries, (2)
the theoretical risk of thromboembolism from untwisting
torsed ovarian veins, and (3) the belief that a grossly blue
black and hemorrhagic adnexa is irreversibly damaged. Indeed, in older pediatric and adult series and in some current
series, the ovary salvage rate is 0% to 16%.3,4,6,7,10
Way provided one of the earliest descriptions of ovary
detorsion.41 In his paper, he clearly described 2 patients
(among others) that were treated with detorsion of extremely ischemic adnexa. In both, there was evidence of
ovary recovery at salpinography or subsequent direct examination at laparotomy. He states, even unfavourable-looking tumors may be amenable to conservative surgery. In
the last 17 years there has been increasing support for this
hypothesis. In 1987, Vancaillie described a 19-year-old
woman who had detorsion despite a dark blue and markedly
congested appearing gonad.60 The patient also had partial
cystectomy and then oophoropexy by plication of the ovarian ligament. At second look laparoscopy the ovary was
smaller than the contralateral side, but subsequent ultrasound examinations showed normal follicle development.
In 1989, Shalev and colleagues from Chaim Sheba Medical
Center and the Sackler School of Medicine in Tel Aviv
reported 19 patients between 2.5 and 35 years of age that
had detorsion of torsed ovaries despite a purplish black
and nonviable appearance.61 Surprisingly, 15 of 16 patients showed normal ovaries with follicle formation on
follow-up ultrasound examinations. In this series there were
no ovarian neoplasms and no immediate or long-term complications. In 1999, Cohen, from this same institution, reviewed 58 consecutive patients (mean age of 25 years;
including 3 premenarchal girls) that underwent laparoscopic
detorsion of necrotic or ischemic appearing adnexa characterized by marked edema and a black bluish color.62
Twenty-nine patients had cyst aspiration, 9 had cystectomy,
and 1 had associated oophoropexy, but no patient had oophorectomy. Sonographic evidence of ovarian function was
shown in 39 of 42 patients at 6 weeks, and in 54 of 58
patients (93%) by 3 months. Importantly, cystectomy was
attempted in 3 of the 4 patients that had no evidence of
ovarian function at long-term follow-up. Since then, multiple investigators from multiple other institutions have found
similar results.14,63-67 Recently, Oelsner updated the experience at Sheba Medical Center and reviewed the worlds
literature.68 Of 172 patients that had detorsion of torsed
adnexa, 160 (93%) showed evidence of subsequent ovarian
function either by ultrasound, or in some cases with interval
surgical examination. Similar results are accumulating in
children with ovarian torsion.2,5,9,40,54,55,57
90
The intraoperative use of fluorescein may improve ovary
conservation rates as well. McHutchison and colleagues
treated 11 women with adnexal torsion, the youngest of
which was 15 years of age.64 At operation, fluorescein was
administered as a test dose of 1 mL, followed by a bolus of
4 mL, and the ovary was inspected under ultraviolet light.
Nonfluorescent tissue was considered nonviable and removed. In this group, 4 ovaries were untwisted and preserved, 4 had wedge excision of nonfluorescent areas, and 3
were removed resulting in a conservation rate of 72%.
In light of the increased evidence in support of detorsion,
there has been reevaluation of the initial theoretical concerns. Thus far there has been no report of a torsed cancer
that has been treated with detorsion. In addition, there have
been no cases of thromboembolism from a pelvic vein in
these patients. McGovern examined the risk of thromboembolism in patients with adnexal torsion and found 2 proven
cases of pulmonary embolism since 1900, for an incidence
of 0.2%. Interestingly, both cases occurred among 672 patients treated with adnexal resection, and no cases were
found among 309 patients treated with detorsion.
It has become clear that the macroscopic appearance of
the ovary at operation does not correlate well with subsequent ovary recovery and function. Kruger reviewed 31
patients with adnexal torsion, of which 9 were less than 18
years of age.42 Twenty-eight of 31 patients had adnexal
resection, and 3 had detorsion with cystectomy. When the
histology of the specimens was reviewed, 19 patients had
50% tissue necrosis, but 12 patients had no evidence of
necrosis in resected specimens. In this study the presence of
fever preoperatively was associated with an increased risk
of necrosis. In addition, there was a trend toward necrosis
with longer symptom duration. Recently, we have reviewed
the pathology findings of all children who underwent adnexal resection for ovary torsion in the last 20 years.15 In all
cases, the ovary was removed because of a necrotic appearance at the time of surgery. Histologic sections from each
specimen were re-evaluated and a viability index was
assessed based on the percentage of viable tissue (1 0%,
2 1-25%, 3 26-75%, 4 75%). Not surprisingly,
61% showed viable ovarian tissue, and 47% had a viability
index of 3 or 4. Similarly, of ovaries described as black
intraoperatively, 38% showed viable ovarian tissue, and
21% showed an index of 4. Of interest, when the patients
were divided into 2 groups based on the presence or absence
of viable ovarian tissue microscopically, we could find no
differences with regard to the presence of fever, admission
white blood cell count, or the time of symptom onset to
surgical resection.
The ability of ischemic ovarian tissues to recover has
been examined experimentally. Taskin reported a rat model
of ovarian torsion in which adnexa were either twisted three
times and fixed to the pelvic sidewall, or rendered ischemic
by clamps on the ovary vessels.69 Tissues were then examined at different time points following 4- to 36-hour ischemia times. At sacrifice all ischemic adnexa were black
Cass
Ovarian Torsion
through the mid-portion of the ligament in the opposite direction, and then back through the ligament at its uterine origin.35
Other methods include: fixation of the ovary to the pelvic
sidewall,37,73 plication of the utero-ovarian ligament to the
round ligament,74 plication of the utero-ovarian ligament,36,71
and fixation of the ovary to the uterine serosa.30 In cases of
massive edema of the ovary, wedge resection has been recommended.75 Laparoscopic techniques are described.36 It is possible that different techniques should be used in different patients based on the size and relationship of the ovary to the
uterus, fallopian tube and pelvic sidewall. Preservation of the
proximity of the ovary to the fimbriae of the tube should be
considered. The impact of oophoropexy on subsequent fertility
merits further study.
A high index of suspicion is critical to the evaluation and
management of children with ovarian torsion. Lower abdominal pain, nausea and vomiting, a history of previous episodes
of similar pain, low grade fever and tenderness should increase
concern and prompt sonographic evaluation. If the ultrasound
shows abnormalities, such as an enlarged ovary or an enlarged
ovary with reduced blood flow, then serologic tumor markers
(FP, HCG) should be sent to complete the workup. When
there is concern about torsion, these results will not be available preoperatively; however, they may be useful later depending on the operative findings. Patients should then proceed
promptly to operation, and in most cases a laparoscopic procedure is preferred. In all cases the ovary should be detorsed.
If there is no significant edema or ischemia and an underlying
lesion is found, then cystectomy, tumorectomy, or cyst aspiration may be considered. In all cases, even those with very
large teratomata, there is a rim of normal ovary at the hilum
that can be preserved. In patients with no underlying ovarian
pathology or those with abnormally long ovarian ligaments,
oophoropexy should be considered for both the involved and
contralateral ovary. However, if there is significant edema and
hemorrhage within the gonad, then further procedures should
be avoided to minimize the risk of damage to the ovary. If there
is any suspicion of malignancy based on ultrasound or operative findings (theoretically possible, though not reported), if
there is concern about the correct approach, or if the involved
gland is significantly inflamed or ischemic, then there should
be no hesitancy to detorse the ovary alone. The patient should
have tumor markers reviewed and follow-up ultrasound examinations. If the tumor markers are elevated, then the patient
requires reoperation with a cancer approach (described by Dr.
van Allman in this edition of the Journal). If the tumor markers
are normal and a lesion persists on follow-up ultrasound, then
interval laparoscopy and tumorectomy or cystectomy, and possible oophoropexy should be performed after the edema has
resolved in 6 to 8 weeks.
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