Long-term results of conservative management of adnexal torsion in children

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Long-term results of conservative management of adnexaltorsion in children

Ahmet Celik*, Orkan Ergun, Hakan Aldemir, CosSkun Ozcan, Geylani Ozok,Ata Erdener, Erol Balyk

Department of Pediatric Surgery, Ege University Faculty of Medicine, 35100 Izmir, Turkey

0022-3468/05/4004-0016$30.00/0 D 200

doi:10.1016/j.jpedsurg.2005.01.008

T Corresponding author. Tel.: +90 2

21 42.

E-mail address: [email protected]

Index words:Adnexal torsion;

Child;

Conservative

management;

Detorsion;

Oophoropexy

AbstractBackground/Purpose: Adnexal torsion is a condition that may result in serious morbidity including

adnexal removal. However, conservative management with preserving the torsed adnexa is not justified,

and long-term outcomes remain unclear.

Methods: The records of 14 girls with ovarian torsion whose adnexal structures were preserved after

detorsion were reviewed to evaluate the long-term results of conservative management. Data including

age, previous history, duration of complaints, surgical findings and type of intervention, color Doppler

ultrasound findings performed in the early and late postoperative periods, and final outcomes

were collected.

Results: Mean age of patients was 11.5 F 2.8 (range 6 to 15) years. Time interval between the onset

of pain to surgery was 46.78 F 35.5 (range 12 to 126) hours. Seven patients had a benign solitary

cyst as an underlying cause for adnexal torsion. The intervention performed by open surgery in 9 and

by laparoscopy in 5 patients included detorsion, simple cyst aspiration, unroofing and/or cystectomy

in 7 patients with ovarian cysts, and oophoropexy in 9 of 14 patients. Follow-up ranged from 3 to 66

(mean 21.9 F 20.1) months. Thirteen patients resumed normal size and folliculogenesis, whereas in 1

patient, the involved ovary atrophied. No recurrence or contralateral adnexal torsion was observed on

follow-up.

Conclusions: Conservative management with untwisting the ovary and pexing both retained detorsed

and contralateral ovaries especially in idiopathic torsions should be considered in cases of ovarian

torsion in children.

D 2005 Elsevier Inc. All rights reserved.

Torsion of normal ovaries more commonly occurs in

young and adolescent girls than in women [1]. The signs

and symptoms of ovarian torsion are often similar to those

of acute appendicitis; therefore, ovarian torsion is often

5 Elsevier Inc. All rights reserved.

32 388 14 12; fax: +90 232 342

.tr (A. Celik).

misdiagnosed, especially in young girls. A number of

theories on the etiology of torsion of normal ovaries have

been proposed, including impeded venous return causing

vascular stasis and adnexal congestion, excessive mobility

of the adnexa because of long fallopian tubes and

mesosalpinx, and long utero-ovarian ligaments [2,3].

Traditional management has been oophorectomy for

hemorrhagic ovary representing nonviable tissue and the

Journal of Pediatric Surgery (2005) 40, 704–708

Long-term results of conservative management of adnexal torsion in children 705

fear of future malignant transformation if left in place [4,5].

We have previously reported 2 cases whose torsed ovaries

were removed and later presented with metachronous

contralateral torsions; they were treated with detorsion and

fixation after which folliculogenesis and development of

secondary sex characters were observed in the long-term

follow-up [6]. The purpose of this study was to evaluate our

experience with adnexal torsion in 14 children managed

conservatively. To the best of our knowledge, this is the

second report with a larger number of patients to address the

long-term outcome of conservative management of adnexal

torsion in children.

1. Materials and methods

After approval of the institutional review board, records

of patients who had undergone surgery because of torsion of

uterine adnexal structures between January 1997 and

December 2003 were reviewed. Those with a malignant

cause as an underlying etiology that required oophorectomy

were excluded. Fourteen patients managed conservatively

were included in the study, and their long-term results were

assessed. The conservative management in the present study

referred to preserving adnexal structures after surgical

detorsion and fixation of adnexa. The adnexal pexy was

performed by fixing the gonad to the lateral pelvic wall with

interrupted nonabsorbable sutures.

The preoperative diagnosis of adnexal torsion was made

based upon patients’ symptoms, physical examination, and

imaging studies when and as required. Color Doppler

ultrasonography (CDU) was used to verify the viability of

the ovarian tissue, especially in the follow-up. Age, duration

of symptoms before admission, side of involvement, type of

the surgical procedure, and outcome were evaluated.

Table 1 Perioperative and postoperative properties of patients

No Age/

side

Macroscopic

appearance

Degree

of torsion

Additional

pathology

Surgical

procedure

Postopera

course

1 13/L black-blue 9008 SC DET/CE N/S

2 9/L black-blue 7208 DET-FIX N/S

3 15/L blue 2708 SC DET/CE N/S

4 14/R blue 1808 SC DET/CE N/S


6 11/L black-blue 10808 DET-FIX Fever

7 14/R black-blue 5408 SC CE/DET-FIX N/S


9 11/R black-blue 3608 DET-FIX N/S


11 12/R black-blue 7208 SC CE/DET-FIX Fever

12 10/R black-blue 9008 DET-FIX Fever

13 6/R black-blue 5408 DET-FIX N/S


AS indicates arterial signals; CE, cyst excision; DET, detorsion; FIX, fixation; H

lutein cyst; N/S, not significant; SC, solitary cyst; SCA, serous cyst adenoma.

Operative approach was made by laparotomy (9 cases) or

laparoscopy (5 cases). The procedures included detorsion of

adnexa, resection of additional pathological structures

(ie, ovarian cysts) that seemingly lead to torsion, and oophor-

opexy of torsed and contralateral adnexa in idiopathic

torsions or suspected anatomic predisposition.

Early postoperative follow-up included CDU on first

postoperative day and routine clinical parameters. Patients

were seen at the clinic at postoperative weeks 2 and 4 and

then every 3 months.

2. Results

Mean age of the patients was 11.5 F 2.87 (range 6-15)

years. Time elapsed from the initial onset of symptoms to

hospital admission was 42.5 F 35.2 (range 9-120) hours.

Two patients had the history of previous contralateral

salpingo-oophorectomy because of idiopathic adnexal tor-

sion in our institution [6] and presented with metachronous

torsions. One patient had open cardiac surgery because of

atrial septal defect, and another patient had Von Willebrand

disease diagnosed 2 years ago.

All patients presented with lower abdominal pain. Onset

of pain was abrupt in all cases, and 4 of them reported

similar previous episodes. Nausea and vomiting were

present in 71% of cases, and in 5 patients, a pelvic mass

was palpable in abdominal or rectal examination. Fever was

present in only 1 patient. Laboratory data were noncontrib-

utory, and the white blood cell count ranged from 6000 to

20.300/mm3, with a mean count of 11.985 F 4275/mm3.

Plain abdominal radiograms obtained in all patients were

also nonspecific.

Abdominal-pelvic ultrasonography obtained in all cases

confirmed the presence of a mass in all but 1 patient.

tive Pathologic

result

Early

postoperative

CDU (day 1)

Late

postoperative

CDU

Follow-up

(months)

Final

outcome

LC Min AS Normal 6 Good

HI No AS Normal 66 Good

SCA Min AS Normal 60 Good

LC Min AS Normal 6 Good

– Min AS Normal 36 Good


IO Min AS Normal 22 Good




– Min AS Atrophy 14 Atrophy


HI No AS Min AS 3 Good

HC Min AS Normal 3 Good

C, hemorrhagic cyst; HI, hemorrhagic infarction; IO, ischemic ovary; LC,

A. Celik et al.706

Sonographic findings included solid or cystic masses with

heterogenic density as well as the presence of fluid in the

pouch of Douglas. Preoperative CDUwas performed in 10 of

14 patients, and there was only 1 false-negative interpreta-

tion: blood flow to the involved ovary was found to be normal

in 1 patient and decreased or absent in 9 patients.

Torsion was slightly more common on the right side (left:

6 patients vs right: 8 patients). Seven (50%) of the torsions

were associated with a solitary cyst as an underlying cause.

The degree of the torsions ranged from 1808 to 10808. Thecysts were managed by simple cyst aspiration and unroofing

and/or cystectomy in 7 patients. Oophoropexy was per-

formed in 9 patients after adnexal detorsion by using

nonabsorbable material to the pelvic sidewall. Ipsilateral

ovarian biopsy was performed in 10 children. The postop-

erative courses of the patients were uneventful except for

transient temperature elevation in 3 patients.

Table 1 summarizes the demographic data of the 14

patients in the series. Mean follow-up were 21.9 F 20.1

months ranging from 3 to 66 months. On the postoperative

day 1 studies, CDU was not able to demonstrate any kind of

arterial signal in the ovarian tissue in 2 patients; however,

long-term results of these patients are good. No recurrence

or contralateral metachronous torsion was observed on the

follow-up of these patients. In 1 patient, ultrasound follow-

ups revealed atrophy of the effected ovary. In 12 patients,

affected ovaries were normal in size with follicular

development at the postoperative month 3. One patient,

who had undergone contralateral salpingo-oophorectomy

previously and presented with metachronous torsion, had no

evidence of revascularization in the ovarian tissue until 3

years postoperatively; however, she menstruated and

showed follicular development detected by sonograms after

this long period (patient 2). She required consecutive

laparoscopic interventions because of persistent large

follicular cysts.

3. Discussion

Timely diagnosis of adnexal torsion in young girls

sometimes can be challenging because the differential

diagnosis includes many of the pathologies that cause

abdominal pain [4,7-11]. Delay in surgical intervention may

cause the necrosis of adnexal structures necessitating

resection [7,9,10]. More recently, to improve the diagnostic

accuracy, CDU has been used for the assessment of ovarian

blood supply. Although CDU may play a confirmatory role

in the diagnosis of ovarian torsion, the decision of whether

to undertake a conservative or extirpative procedure is

challenging and not always clinically straightforward.

Potentially salvageable ovaries may be removed if CDU is

used as a guide [4,5,8,10].

The normal tube and ovary are extremely mobile and are

capable of rotation of 908 without giving rise to symptoms

[12]. Excess mobility of the adnexa because of an

abnormally long tube, mesosalpinx, or mesovarium, adnexal

venous congestion as in premenarchal activity, and jarring

movement of the body are possible mechanisms that have

been suggested in torsion of normal uterine adnexa

[2,3,13,14]. On the other hand, associated ovarian pathol-

ogies, such as cysts and tumors, may lead to excessive

rotation resulting in torsion and ischemia of the adnexa.

Adnexa appear to be particularly prone to torsion in the

early pubertal years. Functional ovarian cysts are very

common during these peripubertal years and may be a

predisposing cause of adnexal twisting [15]. Younger

children more commonly have either a mature cystic

teratoma or torsion with no underlying abnormality as an

etiology, compared with torsion in older children that is

more likely to result from either a follicular or corpus

luteum cyst [10]. In the present series, 50% of patients had a

solitary cyst (luteal, hemorrhagic, or follicular cysts) as an

underlying cause of adnexal torsion.

Torsion of the ovarian blood supply will result in venous

congestion, hemorrhage, and eventually, necrosis of ovarian

tissue [4,5]. Ideally, the diagnosis needs to be made before

tissue necrosis occurs as this allows for conservative

management [4]. However, the duration of symptoms is

not indicative for necrosis; intraoperative appearance and

viability tests are not unique and specific of the degree of

ischemia, and patients with preoperative leukocytosis or

fever are at 2-fold risk for adnexectomy [4,5,16,17]. Early

postoperative CDU imaging is not predictive of the end

result of adnexal blood flow [4,6]. Therefore, the decision

to perform salpingo-oophorectomy should not be based on

the color and consistency of the adnexa; dark hemorrhagic

edematous appearance of the torsed adnexa is the result of

ovarian engorgement, secondary to venous stasis. Com-

plete arterial obstruction probably does not occur in most

cases [5]. In an experimental study performed in rats, a

complete histological resuscitation was observed after

reperfusion of ischemic ovaries, even after an ischemic

period of 24 hours [18].

The dominance of affected side is controversial

[9,7,13,14]. In our relatively small series, the torsion of

the right side was slightly more common. Although the

condition is most frequently unilateral, cases of bilateral

synchronous or asynchronous adnexal torsion have also

been reported in literature since 1934. Bilateral asynchro-

nous adnexal torsion in childhood was first described by

Baron in 1934 [19]. After a review of English literature, we

were able to document 17 such cases in childhood [6]. In

8 cases, a second salpingo-oophorectomy was performed

leaving the patient agonadal. However, detorsion and

conservative management with oophoropexy by laparotomy

or laparoscopy resulted in the salvage of ovarian function in

9 patients in recent years [6].

Although traditional treatment advocates removal of the

twisted adnexa, more recent literature contains cases of

conservative therapy [3-6,8,11,16,17,20-24]. A large litera-

ture review reported successful conservative treatment

Long-term results of conservative management of adnexal torsion in children 707

without serious complications in the adult population of 214

cases with adnexal torsion [16]. Vancaillie and Schmidt [20]

first reported the untwisting of the torsed adnexa of a

19-year-old girl with no complications. Shalev and Peleg

[21] reported on the use of laparoscopy to manage torsion in

an 11-year-old girl without any postoperative complication.

Detorsion and oophoropexy in unilateral torsion in children,

even in delayed diagnosis, have been recommended by

Templeman et al [4] and in adults by Oelsner et al [5] with

good clinical and ultrasonographic results. Untwisting the

torsed adnexa, resection of necrotic tissue, and pexing any

residual ovarian tissue without salpingo-oophorectomy were

also advocated by Dolgin et al [8].

Although the most commonly encountered complication

of conservative management is postoperative fever, this can

be managed by medication and resolves spontaneously

within a few days of operation [4,5,11]. Lower abdominal

discomfort has also been reported in these patients [4].

Three patients in the present series experienced postopera-

tive subfebrile fever and abdominal discomfort especially on

affected side, and these patients were managed symptom-

atically with nonsteroidal medications.

Another particular concern with conservative manage-

ment is the possibility of leaving a malignancy in situ [4].

However, if there is no tumor seen at exploration, the ovary

can be left in place or biopsies can be performed if there is

suspicious macroscopic appearance [5,8]. Most of ovarian

malignancies have been reported to occur in adult woman

and not in children [6,7,25]. Reasonably, some authors

advocate performing an ultrasound examination 6 weeks

postoperatively in cases of ovarian torsion treated by

conservative therapy [5]. Biopsy of the contralateral ovary

is controversial [10]. In our series, ipsilateral ovarian biopsy

was performed in 10 children. All biopsies revealed ischemic

ovarian tissue and/or solitary cyst, and no malignant

degeneration was detected on histopathological examination.

Although there is a theoretical concern quoted in the

literature about a potential risk of thromboembolism caused

by untwisting of the pedicle [5,13,15,16,26], there are

hundreds of cases in the literature of untwisting adnexal

pedicles without evidence of thromboembolic complication

[3,5,8,11,16,17,20-22,27,28], except for 1 adult patient with

concurrent pulmonary embolism and ovarian vein throm-

bosis by adnexal torsion [16]. Therefore, the procedure is

reasonably safe. Long-term follow-up in women treated

conservatively has been shown to result in follicular activity

on ultrasound scan [5,20-22].

It is clear that early diagnosis is beneficial in terms of a

higher possibility to salvage the torsed adnexa and maximize

the success of conservative therapy [6,7,29-31]. Although the

detection of normal flow by CDU does not exclude ovarian

torsion, it is still the most useful noninvasive diagnostic

modality [7] to expedite early operative intervention. On

early postoperative days, absence of venous or arterial flows

on CDU is not indicative of a critically ischemic or nonviable

adnexa [4,7] as was observed in 2 of our patients. Return of

normal blood flow could take 2 to 6 months [4,20]; in 1 of our

patients, this period was 3 years. Adnexal size also reduces to

normal in this time interval [4,20]. The immediate febrile

morbidity, lower abdominal discomfort, and slow resolution

of ovarian enlargement are the main postoperative problems

in patients with detorsed ischemic adnexa.

The benefit of pexing of the gonads is controversial.

Some series make no mention of it at all, ignoring the

possibility of future contralateral or ipsilateral torsion [8,11].

But this event can lead to a catastrophic consequence,

resulting in an agonadal patient [6]. It is mostly recom-

mended in cases of idiopathic adnexal torsion [6,8-

11,15,20,29]. Oophoropexy of the contralateral ovary in a

child with ovarian torsion is also controversial [4,24,29]. We

were able to save the ovaries with detorsion, and oophor-

opexy was performed without any postoperative complica-

tion. We were also able to document the viability of the

ovaries as proven by CDU demonstration of blood flow,

folliculogenesis, and normal hormone levels in 2 patients

who had previous contralateral salpingo-oophorectomy.

Given this report and others concerning ovarian preser-

vation in adnexal torsion, we believe that conservative

management with untwisting of the ovary and pexing both

retained detorsed and contralateral ovaries, especially in

idiopathic adnexal torsion, should be performed in cases of

ovarian torsion in children. Recurrence of ipsilateral and/or

contralateral torsion could be prevented by this approach.

Because malignant tumors have not been reported com-

monly to occur in association with torsion in children, we

encourage this approach because future reproductive func-

tion is of concern. To exclude a neoplasmic etiology and/or

potential malignant degeneration, serial CDU studies are

recommended for follow-up.

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Long-term results of conservative management of adnexal torsion in children