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Adnexal torsion in children may have a catastrophic sequel: Asynchronous bilateral torsion
Adnexal Torsion in Children May Have a Catastrophic Sequel: Asynchronous Bilateral Torsion ¨ zcan, Ahmet C¸elı˙k, Geylani O ¨ zok, Ata Erdener, and Erol Balık By Cos¸kun O ˙Izmir, Turkey
Background/Purpose: Adnexal torsion is a serious condition that frequently may result in ovarian removal, and there always is a risk of castration if the contralateral ovary undergo torsion as well. In this study, the authors present their experience with adnexal torsion in 15 children and describe a catastrophic event, asynchronous bilateral adnexal torsion, with review of the literature.
necessitated adnexal resection. In only 3 cases preservation of the adnexa was possible. Asynchronous adnexal torsion occurred in 2 patients in the time course. Both were treated by laparotomy and adnexal untwisting and fixation by permanent multiple interrupted sutures. In their final evaluation at 40 and 8 months after the operation, they were found to have good ovarian function.
Methods: Between November 1993 and November 2000, 15 children under 15 years of age who had undergone operation because of torsion of uterine adnexal structures were evaluated. Two illustrative cases with asynchronous bilateral adnexal torsion are presented.
Conclusion: Considering the risk of subsequent contralateral torsion and its impact on future fertility, the authors believe that conservative management (untwisting the ovary and pexing, both retained detorsed and contralateral, ovaries) should be considered in cases of ovarian torsion in children. J Pediatr Surg 37:1617-1620. Copyright 2002, Elsevier Science (USA). All rights reserved.
Results: Fourteen cases were associated with additional adnexal pathology, whereas in 1 case the torsion was of normal uterine adnexa. Sonographic studies improved the preoperative diagnosis. Hemorrhagic necrosis of the adnexa secondary to the torsion was found in all cases except 3 and
T
HE CLINICAL DIAGNOSIS of adnexal torsion in children often is uncertain, and delay in surgical intervention frequently may cause the necrosis of adnexal structures necessitating resection.1 Once a girl has lost one ovary because of torsion, she is at risk of being castrated should the contralateral organ undergo torsion as well; this is extremely rare, but for the affected girl it represents a catastrophic event.2,3 In this study, we present our experience with adnexal torsion in 15 children and describe this catastrophic event, asynchronous bilateral adnexal torsion, which we encountered in 2 of our cases, with review of the literature. MATERIALS AND METHODS Between November 1993 and November 2000, 15 children under 15 years of age who had undergone operation because of torsion of uterine adnexal structures were evaluated. Two illustrative cases with asynchronous bilateral adnexal torsion are presented.
INDEX WORDS: Ovarian torsion, adnexal torsion, asynchronous ovarian torsion, subsequent ovarian torsion.
oophorectomy was performed. Left ovary had multiple microcysts but was of normal size. Seven months after her initial presentation, she presented again with a 2-day history of abdominal pain in her left lower quadrant. Ultrasound scan showed the left ovary to be 38 ⫻ 28 mm with a number of follicles and with evidence of either venous or arterial blood flow on Doppler imaging. The patient was observed closely, her symptoms persisted, and repeat ultrasound scan 12 hours later suggested left ovarian torsion; the size of ovary had increased to 47 ⫻ 38 mm, and there was no evidence of blood flow on Doppler imaging. An emergency laparotomy found an edematous ovary with a torsion of 720°. The torsion was untwisted; however, no sign of reperfusion was observed. A wedge biopsy was performed, the residual gonad was pexed to the lateral pelvic side wall with multiple nonabsorbable sutures. Pathologic examination of the biopsy material found no malignancy. Ultrasound scan showed the ovary to be 40 ⫻ 43 mm without evidence of blood flow on Doppler examination on postoperative day 10. Two months later, laboratory evaluation showed a decreased level of estradiol and luteinizing hormone (LH) and an increased level of follicle-stimulating hormone (FSH). After 3 years postoperatively, during which serial ultrasound studies and laboratory tests showed ovarian unfunction, the patient menstruated and estradiol, LH, and FSH levels were found to become normal.
CASE REPORTS
Case 1 A 9-year-old girl was admitted to our department because of a 20-day history of intermittent abdominal pain. Physical examination found tenderness in the right lower quadrant and a pelvic mass. An abdominal ultrasound study showed a 50- ⫻ 30-mm pelvic mass. At abdominal exploration, an edematous, hemorrhagic right ovary with a 720° torsion of the pedicle was found. The torsion was untwisted; however, the ovary remained edematous and dark. A right salpingo-
From the Ege University Faculty of Medicine, Department of Pediatric Surgery, I˙zmir, Turkey. ¨ zcan, MD, Assistant Professor Address reprint requests to Cos¸kun O of Pediatric Surgery, Ege University Faculty of Medicine, Department of Pediatric Surgery 35100, Bornova I˙zmir, Turkey. Copyright 2002, Elsevier Science (USA). All rights reserved. 0022-3468/02/3711-0020$35.00/0 doi:10.1053/jpsu.2002.36195
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Ultrasound study showed the left ovary to be 22 ⫻ 18 mm with 2 large cysts. Laparoscopic exploration found the vital left ovary and the cysts were evacuated.
Case 2 A premenarchal, 12-year-old girl presented to our department with a one-day history of lower abdominal pain and vomiting. Examination of the child found tenderness to deep palpation in the right lower quadrant. An ultrasound scan confirmed a 70- ⫻ 50-mm pelvic cystic mass. Exploratory laparotomy found a ruptured necrotic right ovary with a 720° torsion of pedicle (Fig 1). A right salpingo-oophorectomy was performed. The left ovary was found to have multiple microcysts but was of normal size. Five months after her previous surgery, the patient presented again with a 5-hour history of abdominal pain in the left lower quadrant. A Doppler sonogram found a 48- ⫻ 45-mm pelvic multicystic mass without blood flow. At laparotomy, a large, edematous ovary with a torsion of 720° was found. The torsion was untwisted, and oophoropexy was performed by fixing the gonad to the lateral pelvic wall with multiple interrupted nonabsorbable sutures. Ultrasound study performed 24 hours postoperatively found a left ovary with good blood flow. One month after the operation, estradiol, LH, and FSH levels were found to be normal, and repeat ultrasound studies and laboratory tests performed on postoperative months 2 and 8 found similar findings with results of early postoperative findings confirming ovarian function.
RESULTS
The ages of patients varied from 4 to 15 years, with a mean age of 10.7 years. The mean time from onset of symptoms to hospital admission was 3.8 days (range, 5 hours to 22 days). All patients presented with lower abdominal pain. Onset of pain was sudden in 9 of 15 cases, and 6 of them reported similar previous episodes. Nausea and vomiting were present in 73% of cases, and
Fig 1. case 2.
Operative view of the ruptured necrotic right ovary in
Table 1. Pathologies of Torsed Adnexa Finding
Torsion Torsion Torsion Torsion Torsion Total
No. of Patients
of of of of of
a cystic ovary a benign cystic teratoma an ovarian cyst a tubal arteriovenous malformation normal adnexa
6 4 3 1 1 15
abdominal or pelvic mass in abdominal or rectal examination was palpable in 33% of cases. Abdominal ultrasound, obtained in all cases, confirmed the presence of a mass in all except one case. Sonographic findings included solid or cystic masses as well as the presence of fluid in the pouch of Douglas. Doppler sonography could be performed in 8 of 17 torsions. Blood flow to the ovary was found to be normal in one torsion and decreased or absent in 7. Eight of the lesions occurred on the right, whereas 7 of those occurred on the left side. Fourteen were torsion of diseased uterine adnexa, one of normal adnexa (Table 1). The torsion resulted in hemorrhagic necrosis, and adnexectomy had to be carried out in 12 patients. In the remaining 3, detorsion resulting in recovery of vascularization of ovary was possible. The mean time from the onset of symptoms to operation in those 3 patients and in the remaining 12 patients was 12.6 and 138.8 hours, respectively. Subsequent adnexal torsion in 2 patients occurred 7 and 5 months after the first operation. DISCUSSION
The normal tube and ovary are extremely mobile and are capable of rotation of 90° without giving rise to symptoms.4 However, associated ovarian pathologies, 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 may be a predisposing cause of adnexal twisting during these peripubertal years.2 Although excess rotation of the adnexa can be explained readily to be caused by large masses of associated ovarian pathology, torsion of the normal uterine adnexa is less comprehendible. A few possible mechanisms have been suggested5,6: (1) excess mobility of the adnexa caused by abnormally long tube, mesosalpinx, or mesoovarium; (2) adnexal venous congestion as in premenarchal activity, and (3) jarring movement of the body. Preoperative diagnosis of adnexal torsion often is difficult.1,2,7-9 Persistent abdominal pain may be the only complaint in the pediatric population.2,10 Radiologic surveys of the abdomen will not reveal the condition but may be useful to exclude other causes of abdominal
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pain.5 In view of these problems ultrasonographic studies are recommended in prompt diagnosis.7 Conventional ultrasonography usually confirms a pelvic mass in cases of adnexal torsion but may not establish the diagnosis.10,11 However, Doppler sonography was reported to be useful in the diagnosis of ovarian torsion. However, when normal flow is detected by Doppler sonography, it does not exclude an ovarian torsion, in fact, torsion may be missed in these cases resulting a delay in diagnosis and surgical intervention.12,13 Possible explanation for a normal flow on Doppler sonography in cases of ovarian torsion include (1) venous thrombosis leads to ovarian necrosis before arterial thrombosis occurs, (2) continuing arterial flow to ovary from the branches of uterine artery, and (3) intermittent ovarian torsion: the results of Doppler sonography varies whether the adnexa is twisted at the time the study is performed.12-14 In agreement with these reports, surgical intervention was found to be delayed in one of our patients because normal blood flow was found by Doppler sonography. However, in the remaining 7 torsions, Doppler sonography accurately diagnosed the ovarian torsion. In all cases except 3, the torsion caused hemorrhagic necrosis, and adnexectomy was carried out. Detorsion with recovery of vascularization and excision of ovarian cyst was possible in 3 cases. In these patients, the mean time between first symptom to operation was 12.6 hours (range, 8 to 19 hours), considerably less than that of remaining patients in whom the ovary could not be saved (mean, 138.8 hours, range, 30 to 533 hours). Although the condition is most frequently unilateral,
cases of bilateral synchronous or asynchronous adnexal torsion also have been reported in the literature. Bilateral asynchronous adnexal torsion in childhood was first described by Baron in 1934.15 After a review of the English-language literature, we were able to document 17 such cases in childhood (including our 2 cases, Table 2).2,3,8-10,15-24 The age of patients ranged from 3 to 12 years on first presentation, and the age ranged from 6 to 17 years on second presentation. The second presentation time owing to contralateral torsion ranged from 6 weeks to 9 years after first presentation. A right-sided predominance on first presentation exists with a 11:6 ratio. In 8 cases, a second salpingo-oophorectomy was performed leaving the patient agonadal. However, detorsion with oophoropexy or plication of the utero-ovarian ligament resulted in the salvage of ovarian function in 9 patients in recent years. Although traditional treatment advocates removal of the twisted adnexa, more recent literature contains cases of conservative therapy. Detorsion and oophoropexy in unilateral torsion in children even in delayed diagnosis had been recommended by Templeman et al25 with a good clinical and ultrasonographic results. Untwisting the torsed adnexa, resection of necrotic tissue, and pexing any residual ovarian tissue without salpingooophorectomy also were advocated by Dolgin et al.23 Although the most commonly encountered complication of conservative management is postoperative fever, this can be managed by antipyretics and resolves spontaneously in a few days after the operation.25,26 Another particular concern with conservative management is the
Table 2. Reported Cases of Asynchronous Bilateral Adnexal Torsion in Children
Case No. and Year
115 1934 216 1980 317 1981 418 1984 519 1986 620 1987 79 1989 821 1990 92 1990 103 1993 118 1996 1422 1997 1223 2000 1310 2000 1524 2000 16 (current case) 17 (current case)
Age at Time of First Torsion (yr)
Age at Time of Second Torsion (yr)
Interval Between Surgery
Affected Side/ Surgical Procedure at Time of First Torsion
7 12 3 7 6 7 6.5 3.5 8.5 10 10 UK 8 4.5 UK 9 12
9 12 6 8 8 9 10.5 10.5 9.5 11 12 13 17 6 9 10 12
2 yr 3 mo 6 wk 3 yr 2 yr 2 yr 2 yr 4 yr 7 yr 1 yr 8 mo 2 yr UK 9 yr 17 mo UK 7 mo 5 mo
R/SOP R/SOP R/SOP R/SOP L/SOP R/SOP L/SOP R/SOP R/SOP R/SOP L/SOP L/INCD R/SOP L/SOP L/INCD R/SOP R/SOP
Affected Side/ Surgical Procedure at Time of Second Torsion
L/SOP L/SOP L/SOP L/SOP R/DETORSION L/SOP R/SOP L/SOP L/SOP L/DETORSION ⫹ OPXY R/DETORSION ⫹ PLICA.* R/DETORSION ⫹ PLICA.* L/DETORSION ⫹ OPXY R/DETORSION ⫹ OPXY R/DETORSION* L/DETORSION ⫹ OPXY L/DETORSION ⫹ OPXY
Castration
Yes Yes Yes Yes No Yes Yes Yes Yes No No No No No No No No
Abbreviations: R, right; SOP, salpingo-oophorectomy; L, left; OPXY, oophoropexy; PLICA, plication of the utero-ovarian ligament; UK, unknown; INCD, incidentally found. *Done by laparoscopy.
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possibility of leaving a malignancy in situ.25 However, if there is no tumor seen at exploration, the ovary can be left in place, or biopsies can be performed if there is any suspicious macroscopic appearance of a tumor. Additionally, most of the ovarian malignancies have been reported to occur in adult woman and not in children.1,27 Reasonably, some investigators advocate performing an ultrasound examination 6 weeks postoperatively in cases of ovarian torsion treated by conservative theraphy.6 Although there is a theoretical concern in the literature about a potential risk of thromboembolism caused by untwisting of the pedicle,2,6,28 there are hundreds of cases in the literature of untwisting adnexal pedicles without evidence of thromboembolic sequele.26,29 In 2 of our patients, the ovaries were saved, and oophoropexy was performed at the time of second torsion without any postoperative complication. Forty and 8 months postoperatively, we documented the viability of the ovaries in
these 2 patients using ultrasonographic examination demonstrating blood flow, normal estradiol, LH, and FSH levels, and by visualization of the ovary at laparoscopy. Early diagnosis may lead to more frequent salvage of torsioned adnexa and maximize the success of conservative therapy. Although the detection of normal flow by Doppler sonography does not exclude an ovarian torsion, it still is the most useful noninvasive diagnostic modality, which could lead to early operative intervention. In the current study, subsequent contralateral adnexal torsion occurred in 2 patients. In these 2 patients, the ovaries were saved, and adnexal pexing was performed. Final evaluation showed they had good ovarian function. Based on the observations in this report and others concerning ovarian preservation in adnexal torsion, we believe that untwisting the ovary and pexing both the retained detorsed and contralateral ovaries should be performed.
REFERENCES 1. Spigland N, Ducharme JC, Yazbeck S: Adnexal torsion in children. J Pediatr Surg 24:974-976, 1989 2. Davis AJ, Feins NR: Subsequent asynchronous torsion of normal adnexa in children. J Pediatr Surg 25:687-689, 1990 3. Grunewald B, Keating J, Brown S: Asynchronous ovarian torsion-the case for prophylactic oophoropexy. Postgrad Med J 69:318319, 1993 4. Downer IG, Brines OA: Torsion of the undiseased uterine adnexa. Am J Obstet Gynecol 21:665, 1931, in Evans JP: Torsion of the normal uterine adnexa in premenarchal girls. J Pediatr Surg Apr; 13:195-196, 1978 5. Evans JP: Torsion of the normal uterine adnexa in premenarchal girls. J Pediatr Surg 13:195-196, 1978 6. James DF, Barber HRK, Graber EA: Torsion of normal uterine adnexa in children. Report of three cases. Obstet Gynecol 35:226-230, 1970 7. Mordehai J, Mares AJ, Barki Y, et al: Torsion of uterine adnexa in neonates and children: A report of 20 cases. J Pediatr Surg 26:11951199, 1991 8. Germain M, Rarick T, Robins E: Management of intermittent ovarian torsion by laparoscopic oophoropexy. Obstet Gynecol 88:715717, 1996 9. Wakamatsu M, Wolf P, Benirschke K: Bilateral torsion of the normal ovary and oviduct in a young girl. J Famil Pract 28:101-102, 1989 10. Eckler K, Laufer MR, Perlman SE: Conservative management of bilateral asynchronous adnexal torsion with necrosis in a prepubescent girl. J Pediatr Surg 35:1248-1251, 2000 11. Quillin SP, Siegel MJ: Transabdominal color Doppler ultrasonography of the painful adolescent ovary. J Ultrasound Med 13:549555, 1994 12. Pen˜ a JE, Ufberg D, Cooney N, et al: Usefulness of Doppler sonography in the diagnosis of ovarian torsion. Fertil Steril 73:10471050, 2000 13. Lee EJ, Kwon HC, Joo HJ, et al: Diagnosis of ovarian torsion with color Doppler sonography: Depiction of twisted vascular pedicle. J Ultrasound Med 17:83-89, 1998 14. Rosado WM, Trambert MA, Gosink BB, et al: Adnexal torsion:
diagnosis by using Doppler sonography. AJR Am J Roentgenol 159: 1251-1253, 1992 15. Baron C: Torsion of the normal ovary. JAMA 102:1675-1676, 1934 16. McRea RS: Uterine adnexal torsion with subsequent contralateral recurrence. J Reprod Med 25:123-124, 1980 17. Bower RJ, Adkins JC: Surgical ovarian lesions in children. Am Surg 47:474-478, 1981 18. Dunnihoo DR, Wolff J: Bilateral torsion of the adnexa: A case report and a review of the world literature. Obstet Gynecol 64:55S-59S, 1984 19. Worthington-Kirsch R, Raptopoulos V, Cohen IT: Sequential bilateral torsion of normal ovaries in a child. J Ultrasound Med 5:663-664, 1986 20. Buss JG, Lee RA: Sequential torsion of the uterine adnexa. Mayo Clin Proc 62:623-625, 1987 21. Shun A: Unilateral childhood ovarian loss: An indication for contralateral oophoropexy? Aust N Z J Surg 60:791-794, 1990 22. Nagel TC, Sebastian J, Malo JW: Oophoropexy to prevent sequential or recurrent torsion. J Am Assoc Gynecol Laparosc 4:495498, 1997 23. Dolgin SE, Lublin M, Shlasko E: Maximizing ovarian salvage when treating idiopathic adnexal torsion. J Pediatr Surg 35:624-626, 2000 24. Abargel A, Pansky M, Neeman O, et al: Torsion of single normal adnexa in a premenarchal girl. J Am Assoc Gynecol Laparosc 7:421-422, 2000 25. Templeman C, Hertweck SP, Fallat ME: The clinical course of unresected ovarian torsion. J Pediatr Surg 35:1385-1387, 2000 26. Oelsner G, Bider D, Goldenberg M, et al: Long-term follow-up of the twisted ischemic adnexa managed by detorsion. Fertil Steril 60:976-979, 1993 27. Quint EH, Smith YR: Ovarian surgery in premenarchal girls. J Pediatr Adolesc Gynecol 12:27-29, 1999 28. Hibbard LT: Adnexal torsion. Am J Obstet Gynecol 152:456461, 1985 29. Bayer AI, Wiskind AK: Adnexal torsion: can the adnexa be saved? Am J Obstet Gynecol 171:1506-1511, 1994
Background/Purpose: Adnexal torsion is a serious condition that frequently may result in ovarian removal, and there always is a risk of castration if the contralateral ovary undergo torsion as well. In this study, the authors present their experience with adnexal torsion in 15 children and describe a catastrophic event, asynchronous bilateral adnexal torsion, with review of the literature.
necessitated adnexal resection. In only 3 cases preservation of the adnexa was possible. Asynchronous adnexal torsion occurred in 2 patients in the time course. Both were treated by laparotomy and adnexal untwisting and fixation by permanent multiple interrupted sutures. In their final evaluation at 40 and 8 months after the operation, they were found to have good ovarian function.
Methods: Between November 1993 and November 2000, 15 children under 15 years of age who had undergone operation because of torsion of uterine adnexal structures were evaluated. Two illustrative cases with asynchronous bilateral adnexal torsion are presented.
Conclusion: Considering the risk of subsequent contralateral torsion and its impact on future fertility, the authors believe that conservative management (untwisting the ovary and pexing, both retained detorsed and contralateral, ovaries) should be considered in cases of ovarian torsion in children. J Pediatr Surg 37:1617-1620. Copyright 2002, Elsevier Science (USA). All rights reserved.
Results: Fourteen cases were associated with additional adnexal pathology, whereas in 1 case the torsion was of normal uterine adnexa. Sonographic studies improved the preoperative diagnosis. Hemorrhagic necrosis of the adnexa secondary to the torsion was found in all cases except 3 and
T
HE CLINICAL DIAGNOSIS of adnexal torsion in children often is uncertain, and delay in surgical intervention frequently may cause the necrosis of adnexal structures necessitating resection.1 Once a girl has lost one ovary because of torsion, she is at risk of being castrated should the contralateral organ undergo torsion as well; this is extremely rare, but for the affected girl it represents a catastrophic event.2,3 In this study, we present our experience with adnexal torsion in 15 children and describe this catastrophic event, asynchronous bilateral adnexal torsion, which we encountered in 2 of our cases, with review of the literature. MATERIALS AND METHODS Between November 1993 and November 2000, 15 children under 15 years of age who had undergone operation because of torsion of uterine adnexal structures were evaluated. Two illustrative cases with asynchronous bilateral adnexal torsion are presented.
INDEX WORDS: Ovarian torsion, adnexal torsion, asynchronous ovarian torsion, subsequent ovarian torsion.
oophorectomy was performed. Left ovary had multiple microcysts but was of normal size. Seven months after her initial presentation, she presented again with a 2-day history of abdominal pain in her left lower quadrant. Ultrasound scan showed the left ovary to be 38 ⫻ 28 mm with a number of follicles and with evidence of either venous or arterial blood flow on Doppler imaging. The patient was observed closely, her symptoms persisted, and repeat ultrasound scan 12 hours later suggested left ovarian torsion; the size of ovary had increased to 47 ⫻ 38 mm, and there was no evidence of blood flow on Doppler imaging. An emergency laparotomy found an edematous ovary with a torsion of 720°. The torsion was untwisted; however, no sign of reperfusion was observed. A wedge biopsy was performed, the residual gonad was pexed to the lateral pelvic side wall with multiple nonabsorbable sutures. Pathologic examination of the biopsy material found no malignancy. Ultrasound scan showed the ovary to be 40 ⫻ 43 mm without evidence of blood flow on Doppler examination on postoperative day 10. Two months later, laboratory evaluation showed a decreased level of estradiol and luteinizing hormone (LH) and an increased level of follicle-stimulating hormone (FSH). After 3 years postoperatively, during which serial ultrasound studies and laboratory tests showed ovarian unfunction, the patient menstruated and estradiol, LH, and FSH levels were found to become normal.
CASE REPORTS
Case 1 A 9-year-old girl was admitted to our department because of a 20-day history of intermittent abdominal pain. Physical examination found tenderness in the right lower quadrant and a pelvic mass. An abdominal ultrasound study showed a 50- ⫻ 30-mm pelvic mass. At abdominal exploration, an edematous, hemorrhagic right ovary with a 720° torsion of the pedicle was found. The torsion was untwisted; however, the ovary remained edematous and dark. A right salpingo-
From the Ege University Faculty of Medicine, Department of Pediatric Surgery, I˙zmir, Turkey. ¨ zcan, MD, Assistant Professor Address reprint requests to Cos¸kun O of Pediatric Surgery, Ege University Faculty of Medicine, Department of Pediatric Surgery 35100, Bornova I˙zmir, Turkey. Copyright 2002, Elsevier Science (USA). All rights reserved. 0022-3468/02/3711-0020$35.00/0 doi:10.1053/jpsu.2002.36195
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Ultrasound study showed the left ovary to be 22 ⫻ 18 mm with 2 large cysts. Laparoscopic exploration found the vital left ovary and the cysts were evacuated.
Case 2 A premenarchal, 12-year-old girl presented to our department with a one-day history of lower abdominal pain and vomiting. Examination of the child found tenderness to deep palpation in the right lower quadrant. An ultrasound scan confirmed a 70- ⫻ 50-mm pelvic cystic mass. Exploratory laparotomy found a ruptured necrotic right ovary with a 720° torsion of pedicle (Fig 1). A right salpingo-oophorectomy was performed. The left ovary was found to have multiple microcysts but was of normal size. Five months after her previous surgery, the patient presented again with a 5-hour history of abdominal pain in the left lower quadrant. A Doppler sonogram found a 48- ⫻ 45-mm pelvic multicystic mass without blood flow. At laparotomy, a large, edematous ovary with a torsion of 720° was found. The torsion was untwisted, and oophoropexy was performed by fixing the gonad to the lateral pelvic wall with multiple interrupted nonabsorbable sutures. Ultrasound study performed 24 hours postoperatively found a left ovary with good blood flow. One month after the operation, estradiol, LH, and FSH levels were found to be normal, and repeat ultrasound studies and laboratory tests performed on postoperative months 2 and 8 found similar findings with results of early postoperative findings confirming ovarian function.
RESULTS
The ages of patients varied from 4 to 15 years, with a mean age of 10.7 years. The mean time from onset of symptoms to hospital admission was 3.8 days (range, 5 hours to 22 days). All patients presented with lower abdominal pain. Onset of pain was sudden in 9 of 15 cases, and 6 of them reported similar previous episodes. Nausea and vomiting were present in 73% of cases, and
Fig 1. case 2.
Operative view of the ruptured necrotic right ovary in
Table 1. Pathologies of Torsed Adnexa Finding
Torsion Torsion Torsion Torsion Torsion Total
No. of Patients
of of of of of
a cystic ovary a benign cystic teratoma an ovarian cyst a tubal arteriovenous malformation normal adnexa
6 4 3 1 1 15
abdominal or pelvic mass in abdominal or rectal examination was palpable in 33% of cases. Abdominal ultrasound, obtained in all cases, confirmed the presence of a mass in all except one case. Sonographic findings included solid or cystic masses as well as the presence of fluid in the pouch of Douglas. Doppler sonography could be performed in 8 of 17 torsions. Blood flow to the ovary was found to be normal in one torsion and decreased or absent in 7. Eight of the lesions occurred on the right, whereas 7 of those occurred on the left side. Fourteen were torsion of diseased uterine adnexa, one of normal adnexa (Table 1). The torsion resulted in hemorrhagic necrosis, and adnexectomy had to be carried out in 12 patients. In the remaining 3, detorsion resulting in recovery of vascularization of ovary was possible. The mean time from the onset of symptoms to operation in those 3 patients and in the remaining 12 patients was 12.6 and 138.8 hours, respectively. Subsequent adnexal torsion in 2 patients occurred 7 and 5 months after the first operation. DISCUSSION
The normal tube and ovary are extremely mobile and are capable of rotation of 90° without giving rise to symptoms.4 However, associated ovarian pathologies, 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 may be a predisposing cause of adnexal twisting during these peripubertal years.2 Although excess rotation of the adnexa can be explained readily to be caused by large masses of associated ovarian pathology, torsion of the normal uterine adnexa is less comprehendible. A few possible mechanisms have been suggested5,6: (1) excess mobility of the adnexa caused by abnormally long tube, mesosalpinx, or mesoovarium; (2) adnexal venous congestion as in premenarchal activity, and (3) jarring movement of the body. Preoperative diagnosis of adnexal torsion often is difficult.1,2,7-9 Persistent abdominal pain may be the only complaint in the pediatric population.2,10 Radiologic surveys of the abdomen will not reveal the condition but may be useful to exclude other causes of abdominal
ADNEXAL TORSION
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pain.5 In view of these problems ultrasonographic studies are recommended in prompt diagnosis.7 Conventional ultrasonography usually confirms a pelvic mass in cases of adnexal torsion but may not establish the diagnosis.10,11 However, Doppler sonography was reported to be useful in the diagnosis of ovarian torsion. However, when normal flow is detected by Doppler sonography, it does not exclude an ovarian torsion, in fact, torsion may be missed in these cases resulting a delay in diagnosis and surgical intervention.12,13 Possible explanation for a normal flow on Doppler sonography in cases of ovarian torsion include (1) venous thrombosis leads to ovarian necrosis before arterial thrombosis occurs, (2) continuing arterial flow to ovary from the branches of uterine artery, and (3) intermittent ovarian torsion: the results of Doppler sonography varies whether the adnexa is twisted at the time the study is performed.12-14 In agreement with these reports, surgical intervention was found to be delayed in one of our patients because normal blood flow was found by Doppler sonography. However, in the remaining 7 torsions, Doppler sonography accurately diagnosed the ovarian torsion. In all cases except 3, the torsion caused hemorrhagic necrosis, and adnexectomy was carried out. Detorsion with recovery of vascularization and excision of ovarian cyst was possible in 3 cases. In these patients, the mean time between first symptom to operation was 12.6 hours (range, 8 to 19 hours), considerably less than that of remaining patients in whom the ovary could not be saved (mean, 138.8 hours, range, 30 to 533 hours). Although the condition is most frequently unilateral,
cases of bilateral synchronous or asynchronous adnexal torsion also have been reported in the literature. Bilateral asynchronous adnexal torsion in childhood was first described by Baron in 1934.15 After a review of the English-language literature, we were able to document 17 such cases in childhood (including our 2 cases, Table 2).2,3,8-10,15-24 The age of patients ranged from 3 to 12 years on first presentation, and the age ranged from 6 to 17 years on second presentation. The second presentation time owing to contralateral torsion ranged from 6 weeks to 9 years after first presentation. A right-sided predominance on first presentation exists with a 11:6 ratio. In 8 cases, a second salpingo-oophorectomy was performed leaving the patient agonadal. However, detorsion with oophoropexy or plication of the utero-ovarian ligament resulted in the salvage of ovarian function in 9 patients in recent years. Although traditional treatment advocates removal of the twisted adnexa, more recent literature contains cases of conservative therapy. Detorsion and oophoropexy in unilateral torsion in children even in delayed diagnosis had been recommended by Templeman et al25 with a good clinical and ultrasonographic results. Untwisting the torsed adnexa, resection of necrotic tissue, and pexing any residual ovarian tissue without salpingooophorectomy also were advocated by Dolgin et al.23 Although the most commonly encountered complication of conservative management is postoperative fever, this can be managed by antipyretics and resolves spontaneously in a few days after the operation.25,26 Another particular concern with conservative management is the
Table 2. Reported Cases of Asynchronous Bilateral Adnexal Torsion in Children
Case No. and Year
115 1934 216 1980 317 1981 418 1984 519 1986 620 1987 79 1989 821 1990 92 1990 103 1993 118 1996 1422 1997 1223 2000 1310 2000 1524 2000 16 (current case) 17 (current case)
Age at Time of First Torsion (yr)
Age at Time of Second Torsion (yr)
Interval Between Surgery
Affected Side/ Surgical Procedure at Time of First Torsion
7 12 3 7 6 7 6.5 3.5 8.5 10 10 UK 8 4.5 UK 9 12
9 12 6 8 8 9 10.5 10.5 9.5 11 12 13 17 6 9 10 12
2 yr 3 mo 6 wk 3 yr 2 yr 2 yr 2 yr 4 yr 7 yr 1 yr 8 mo 2 yr UK 9 yr 17 mo UK 7 mo 5 mo
R/SOP R/SOP R/SOP R/SOP L/SOP R/SOP L/SOP R/SOP R/SOP R/SOP L/SOP L/INCD R/SOP L/SOP L/INCD R/SOP R/SOP
Affected Side/ Surgical Procedure at Time of Second Torsion
L/SOP L/SOP L/SOP L/SOP R/DETORSION L/SOP R/SOP L/SOP L/SOP L/DETORSION ⫹ OPXY R/DETORSION ⫹ PLICA.* R/DETORSION ⫹ PLICA.* L/DETORSION ⫹ OPXY R/DETORSION ⫹ OPXY R/DETORSION* L/DETORSION ⫹ OPXY L/DETORSION ⫹ OPXY
Castration
Yes Yes Yes Yes No Yes Yes Yes Yes No No No No No No No No
Abbreviations: R, right; SOP, salpingo-oophorectomy; L, left; OPXY, oophoropexy; PLICA, plication of the utero-ovarian ligament; UK, unknown; INCD, incidentally found. *Done by laparoscopy.
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possibility of leaving a malignancy in situ.25 However, if there is no tumor seen at exploration, the ovary can be left in place, or biopsies can be performed if there is any suspicious macroscopic appearance of a tumor. Additionally, most of the ovarian malignancies have been reported to occur in adult woman and not in children.1,27 Reasonably, some investigators advocate performing an ultrasound examination 6 weeks postoperatively in cases of ovarian torsion treated by conservative theraphy.6 Although there is a theoretical concern in the literature about a potential risk of thromboembolism caused by untwisting of the pedicle,2,6,28 there are hundreds of cases in the literature of untwisting adnexal pedicles without evidence of thromboembolic sequele.26,29 In 2 of our patients, the ovaries were saved, and oophoropexy was performed at the time of second torsion without any postoperative complication. Forty and 8 months postoperatively, we documented the viability of the ovaries in
these 2 patients using ultrasonographic examination demonstrating blood flow, normal estradiol, LH, and FSH levels, and by visualization of the ovary at laparoscopy. Early diagnosis may lead to more frequent salvage of torsioned adnexa and maximize the success of conservative therapy. Although the detection of normal flow by Doppler sonography does not exclude an ovarian torsion, it still is the most useful noninvasive diagnostic modality, which could lead to early operative intervention. In the current study, subsequent contralateral adnexal torsion occurred in 2 patients. In these 2 patients, the ovaries were saved, and adnexal pexing was performed. Final evaluation showed they had good ovarian function. Based on the observations in this report and others concerning ovarian preservation in adnexal torsion, we believe that untwisting the ovary and pexing both the retained detorsed and contralateral ovaries should be performed.
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diagnosis by using Doppler sonography. AJR Am J Roentgenol 159: 1251-1253, 1992 15. Baron C: Torsion of the normal ovary. JAMA 102:1675-1676, 1934 16. McRea RS: Uterine adnexal torsion with subsequent contralateral recurrence. J Reprod Med 25:123-124, 1980 17. Bower RJ, Adkins JC: Surgical ovarian lesions in children. Am Surg 47:474-478, 1981 18. Dunnihoo DR, Wolff J: Bilateral torsion of the adnexa: A case report and a review of the world literature. Obstet Gynecol 64:55S-59S, 1984 19. Worthington-Kirsch R, Raptopoulos V, Cohen IT: Sequential bilateral torsion of normal ovaries in a child. J Ultrasound Med 5:663-664, 1986 20. Buss JG, Lee RA: Sequential torsion of the uterine adnexa. Mayo Clin Proc 62:623-625, 1987 21. Shun A: Unilateral childhood ovarian loss: An indication for contralateral oophoropexy? Aust N Z J Surg 60:791-794, 1990 22. Nagel TC, Sebastian J, Malo JW: Oophoropexy to prevent sequential or recurrent torsion. J Am Assoc Gynecol Laparosc 4:495498, 1997 23. Dolgin SE, Lublin M, Shlasko E: Maximizing ovarian salvage when treating idiopathic adnexal torsion. J Pediatr Surg 35:624-626, 2000 24. Abargel A, Pansky M, Neeman O, et al: Torsion of single normal adnexa in a premenarchal girl. J Am Assoc Gynecol Laparosc 7:421-422, 2000 25. Templeman C, Hertweck SP, Fallat ME: The clinical course of unresected ovarian torsion. J Pediatr Surg 35:1385-1387, 2000 26. Oelsner G, Bider D, Goldenberg M, et al: Long-term follow-up of the twisted ischemic adnexa managed by detorsion. Fertil Steril 60:976-979, 1993 27. Quint EH, Smith YR: Ovarian surgery in premenarchal girls. J Pediatr Adolesc Gynecol 12:27-29, 1999 28. Hibbard LT: Adnexal torsion. Am J Obstet Gynecol 152:456461, 1985 29. Bayer AI, Wiskind AK: Adnexal torsion: can the adnexa be saved? Am J Obstet Gynecol 171:1506-1511, 1994