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Asynchronous bilateral adnexal torsion in a 13-year-old adolescent: Our experience of a rare case with review of the literature
Journal of Adolescent Health 37 (2005) 244 –247
Clinical observation
Asynchronous bilateral adnexal torsion in a 13-year-old adolescent: Our experience of a rare case with review of the literature Michail Varras, M.D., Ph.D.a,*, Christodoulos Akrivis, M.D., Ph.D.b, Asymo Demou, M.D.c, and Nikolaos Antoniou, M.D., Ph.D.b a
Second Department of Gynaecology, Anticancer Oncologic Hospital “Agios Savvas,” Athens, First District National Health System, Athens, Greece b Department of Obstetrics and Gynaecology, “G. Chatzikosta” General State Hospital, Ioannina, District National Health System, Ioannina, Epirus, Greece c Department of Pathology, “G. Chatzikosta” General State Hospital, Ioannina, District National Health System, Ioannina, Epirus, Greece Manuscript received April 9, 2004; manuscript accepted October 8, 2004
Abstract
Adnexal torsion is a serious condition and delay in surgical intervention may result in loss of the tube and/or ovary. Children and adolescents who have suffered from uterine adnexal torsion may be at risk for asynchronous torsion of the contralateral uterine adnexa. We report the case of sequential bilateral torsion of uterine adnexa in a 13-year-old girl, resulting in right and subsequently left salpingo-oophorectomy because of gross evidence of total necrosis in both uterine adnexa. After the castration the patient was started on hormone replacement therapy. Families of children who suffered from ovarian torsion and unilateral ovarian loss should be educated about the risk of the contralateral ovary for future torsion and should be encouraged to seek immediate medical help with the recurrence of abdominal pain. © 2005 Society for Adolescent Medicine. All rights reserved.
Keywords:
Ovarian torsion; Adolescent; Children; Asynchronous; Subsequent; Bilateral
J. Blant Sutton described for the first time torsion of adnexa in 1890 [1]. Review of the literature shows that most cases are secondary to adnexal pathology such as ovarian cysts and tumors [2,3]. Adnexal torsion is most frequently unilateral [4]. The first case of bilateral adnexal torsion reported was by Warnek in 1895 [5]. Since then a few cases of bilateral adnexal torsion have been described, usually as simultaneous bilateral torsion of the adnexal structures [4]. Adnexal torsion in children and adolescents is rare. The clinical diagnosis is often uncertain, and delay in surgical intervention frequently leads to the finding of gangrenous adnexal structures at surgery [6]. Once a girl has lost one ovary because of torsion, she is at risk for asynchronous torsion of the contralateral adnexa [7]. Bilateral torsion, for the affected girl and adolescent, represents a catastrophic
*Address correspondence to: Dr. Michail N. Varras, Consultant in Obstetrics and Gynaecology, Platonos 33, Politia Kifisia, Athens 14563, Greece. E-mail address: [email protected]
event from both the reproductive and emotional vantages [7,8]. The purpose of this case report is to describe an extremely rare case of sequential bilateral torsion of uterine adnexae in an adolescent, with review of the literature. Case Report A 13-year-old girl was admitted to the Local General Hospital, Filiates, Epirus, Greece, because of a 12-hour history of intermittent, crampy lower abdominal pain. The pain had acute onset, was located in the right iliac fossa, and radiated down to her right leg. The pain was associated with nausea and she had vomited a number of times. She denied other gastrointestinal or urinary symptoms. The patient’s past medical history was unremarkable. She had no history of similar previous episodes of abdominal pain. She had no prior sexual intercourse. The onset of menarche was at the age of 12 years but she had not had regular cycles yet. On admission, her temperature was 37.8°C and her blood pressure was 90/60 mm Hg. Physical examination found a
1054-139X/05/$ – see front matter © 2005 Society for Adolescent Medicine. All rights reserved. doi:10.1016/j.jadohealth.2004.10.020
M. Varras et al. / Journal of Adolescent Health 37 (2005) 244 –247
Fig. 1. Pelvic ultrasonography of patient’s left adnexal cyst.
nondistended abdomen. Tenderness to deep palpation in the right lower quadrant, without peritoneal signs, was detected. Bowel sounds were present. No masses were palpated. With rectal examination no masses were detected, but mild tenderness in the cul-de-sac was found. A full blood count showed a white cell count of 15,800 cells/mm3 with 84.5% polymorphonuclear cells. The hemoglobin value was 13 g/dl and the hematocrit 38.9%. Serum electrolytes, urea, creatinine, liver function tests, and urinalysis were within normal limits. Abdominal flat and upright roentgenograms disclosed unremarkable findings. The patient was observed closely in the Department of General Surgery; her symptoms persisted and a repeated full blood count 22 hours later showed white cell count of 17,300/mm3 with 88.9% polymorphonuclear cells. The preoperative diagnosis was appendicitis. At abdominal exploration, the patient was found to have a normal appendix with a twisted cyanotic right ovary. The direction of the torsion of the right uterine adnexae was in a clockwise direction. In view of the infarction and nonviability of the right uterine adnexae, a right salpingo-oophorectomy was performed. At that time an appendectomy was also performed. Uterus and left adnexa (ovary and fallopian tube) were of normal size. The patient’s postoperative course was uneventful and she was dismissed on the fourth postoperative day. The fallopian tube measured 0.8 cm in maximum diameter and was 7 cm in length. The right ovary measured 8 ⫻ 4 ⫻ 3.5 cm. On cut section, multiple small cysts with maximum diameter ranging from 0.3 cm to 0.6 cm were recognized. Histologic examination showed hemorrhagic necrosis of the right ovary. Multiple ovarian follicles were recognized. Also, the right fallopian tube showed hemorrhagic necrosis. The appendix was normal. Twenty days after the initial operation, the patient presented to Department of Obstetrics and Gynecology, “G. Chatzikosta” General Hospital, Ioannina, with a 6-hour history of abdominal pain in her left lower quadrant. On arrival
245
her temperature was 37.5°C and her vital signs were normal. On physical examination, tenderness in the left lower quadrant and mild peritoneal signs were present. Bowel sounds were noted to be present. Pelvic ultrasound showed a 5.67 ⫻ 4.84-cm left adnexal cyst (Figure 1). Her laboratory evaluation findings showed white blood count 14,850/mm3 with 91.9% polymorphonuclear cells. Urinalysis had no abnormality. Serum levels of CA-125, AFP, CEA, CA 19-9, and CA 15-3 were within normal values. After discussion with her parents, the decision was made to proceed with an emergency laparotomy. Therefore a 5-cm Pfannenstiel incision was made. The patient was found to have a twisted and cyanotic left fallopian tube and ovary in a clockwise direction. A right salpingo-oophorectomy was perfomed. The patient’s postoperative course was uneventful, and she was dismissed on the fourth postoperative day. At gross examination the left fallopian tube was 7 cm in length and the left ovary measured 14 ⫻ 10 ⫻ 2 cm. At histologic examination the left ovary showed hemorrhagic necrosis (Figure 2). Also, a corpus luteum cyst was found. The left fallopian tube showed hemorrhagic infiltration (Figure 3). No malignancy was found. The patient is treated with hormone replacement therapy. Discussion Adnexal torsion is a gynecologic surgical emergent condition, which occurs at any age but is most common in women of reproductive age [4,9]. In children, adnexal torsion is an infrequent entity [6,10 –14]. Although the adnexal torsion is most frequently unilateral, cases of bilateral synchronous or asynchronous adnexal torsion have been reported. Bilateral asynchronous adnexal torsion in childhood was first described by Baron in 1934 [15]. Özcan et al reviewed the English-language literature in point of asynchronous bilateral adnexal torsion in children and adoles-
Fig. 2. Portion of the left ovary with hemorrhagic necrosis (E & H ⫻ 40).
246
M. Varras et al. / Journal of Adolescent Health 37 (2005) 244 –247
Fig. 3. Cross-section of the left fallopian tube with hemorrhagic infiltration (E & H ⫻ 40).
cents and they were able to document 17 such cases. The age of patients ranged from 3 to 12 years on first presentation, and 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 [7]. Also, Beaunoyer et al reported four cases with asynchronous bilateral adnexal torsion. The mean age at presentation was 10.6 years and the mean interval between the two episodes of ovarian torsion was 15.7 months [16]. The symptomatology and presentation of asynchronous bilateral ovarian torsion in children and adolescents appears to be similar to that of single ovarian torsion [8,16]. The mechanism of adnexal torsion is not known with certainty. Most cases are secondary to adnexal pathology. However, during the peripubertal years, functional ovarian cysts associated with changes in gonadotropin release and failure of follicular involution, seem to be a predisposing cause of adnexal torsion [14,17]. The classic presentation of adnexal torsion is the acute onset of abdominal pain, an adnexal mass, and clinical evidence of peritonitis. However, this triad is usually not present in most patients later found to have adnexal torsion [9]. One aspect that should raise a high index of clinical suspicion of adnexal torsion is a history of previous abdominal pain that is intermittent in nature and probably represents varying degrees of intermittent torsion [6]. The reported frequency of recurrent attacks of pain interspersed with asymptomatic intervals has ranged from 10% to 50% in the literature [6,18,19]. Owing to the intra-abdominal position of the adnexa in a prepubertal child, the diagnosis may be confused with acute appendicitis or another intraperitoneal or gastrointestinal process. This often leads to diagnostic delay and subsequent ovarian loss [17]. The gray-scale ultrasonographic findings of adnexal mass, either cystic, solid or complex, in a child or adolescent presenting with acute or recurrent lower quadrant abdominal pain, nausea and vomiting should raise the possibility of adnexal torsion [20].
Therapy of adnexal torsion remains controversial. Historically, torsion of the adnexa has been managed by excision of the affected structure with extra care taken to avoid untwisting the pedicle because of the fear of dislodging an embolus from the twisted pedicle, and the inability to clinically assess necrosis and tissue viability [9]. However, investigators have found no evidence to support this fear, and now detorsion of the adnexa is advocated, especially in children and young adults who have potentially viable adnexa. If an ovarian or paratubal cyst or neoplasm is found in the twisted adnexa, then a cystectomy or resection of the associated neoplasm should be performed after detorsion of the adnexa. Ovarian function has been shown to recover after detorsion of the adnexa in many reproductive-age women with adnexal torsion [21]. Also, if a torsed ovary appears cyanotic, ovarian bivalving after detorsion has been described as a way to salvage the ovary [22]. If no evidence of tissue perfusion or viability is noted, the adnexa should probably be excised for the theoretical risk of peritonitis from the necrotic tissue, as well as the fact that the resumption of ovarian function is unlikely [9,23]. Adnexal reperfusion with untwisting of the adnexa seems to occur in children as well. Eckler et al reported the case of a child with 72 hours of abdominal pain and necrotic-appearing adnexae who underwent detorsion. Serial postoperative ultrasounds confirmed viability of the adnexa [24]. In addition, Pansky et al advocated the use of laparoscopic detorsion in their series of eight girls and documented ovarian viability in 87% of cases [25]. Complications of conservative management have included postoperative fever and failure, which may necessitate repeat surgery to remove the affected ovary [26]. In cases of ovarian torsion treated by conservative therapy, ultrasound examination should be performed 6 weeks postoperatively. Consideration should also be given to the risk and prevention of future contralateral torsion [21]. Many authors have advocated contralateral oophoropexy to decrease the risk of subsequent torsion [24,27–29]. Oophoropexy can be performed by fixation of the ovary to (a) the lateral part of the pelvic wall, (b) the broad ligament, (c) the posterior wall of the uterus, and (d) the posterior peritoneum just below the bifurcation of the common iliac vessels. Possible risks of this procedure may include adhesions or displacement of the ovary from the fallopian tube with subsequent infertility. Abes and Sarihan presented their experiences with oophoropexy in 10 children with ovarian torsion who underwent oophorectomy and contralateral oophoropexy. The ovary was connected to the peritoneum of the posterior abdominal wall, avoiding a disturbance of the tubo-ovarian anatomic relationship. At follow-up, all pubertal girls had normal menstrual periods [30]. However, no randomized, double-blinded prospective studies of oophoropexy at the time of adnexal torsion have been published to resolve questions regarding success of surgical intervention and impact on fertility [24]. An increased body of literature has documented successful oo-
M. Varras et al. / Journal of Adolescent Health 37 (2005) 244 –247
phoropexy in pediatric and adult patients receiving pelvic radiation for treatment of malignancy [24]. In the case of oophoropexy it is better to use absorbable suture, because permanent suture may predispose to further ovarian injury and bleeding when the ovarian edema resolves and the ovary is retracted from the suture. Conclusion We presented an extremely rare case of asynchronous bilateral adnexal torsion in an adolescent who was surgically castrated because of the gross evidence of total necrosis in both uterine adnexa. Detorsion of the ovary, bivalving, resection of the necrotic tissue, and pexis of any residual ovarian tissue without oophorectomy should be performed for the management of ovarian torsion. References [1] Sutton JB. Salpingitis and some of its effects. Lancet 1890;2:1146. [2] Mordehai J, Mares AJ, Barki Y, et al. Torsion of uterine adnexa in neonates and children: a report of 20 cases. J Pediatr Surg 1991;26: 1195–9. [3] Varras M, Tsikini A, Polyzos D, et al. Uterine adnexal torsion: pathologic and gray-scale ultrasonographic findings. Clin Exp Obstet Gynecol 2004;34 – 8. [4] Buss JG, Lee RA. Sequential torsion of the uterine adnexa. Mayo Clin Proc 1987;62:623–5. [5] Warnek L. Trois cas de tumeurs des trompes compliquees de la torsion du pedicle. N Arch D’ Obstet De Gynec (Paris) 1895;10:81. [6] Spigland N, Ducharme JC, Yazbeck S. Adnexal torsion in children. J Pediatr Surg 1989;24:974 – 6. [7] Özcan C, Çelik, Özok G, Erdener A, Balik E. Adnexal torsion in children may have a catastrophic sequel: asynchronous bilateral torsion. J Pediatr Surg 2002;37:1617–20. [8] Davis AJ, Feins NR. Subsequent asynchronous torsion of normal adnexa in children. J Pediatr Surg 1990;25:687–9. [9] Bayer AI, Wiskind AK. Adnexal torsion: can the adnexa be saved? Am J Obstet Gynecol 1994;171:1506 –10. [10] Lee CH, Raman S, Sivanesaratnam V. Torsion of ovarian tumors: a clinicopathological study. Int J Gynaecol Obstet 1989;28:21–5.
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[11] Brown MF, Hebra A, McGeehin K, Ross AJ 3rd. Ovarian masses in children: a review of 91 cases of malignant and benign masses. J Pediatr Surg 1993;28:930 –3. [12] Comerci JT Jr, Licciardi F, Bergh PA, et al. Mature cystic teratoma: a clinicopathologic evaluation of 517 cases and review of the literature. Obstet Gynecol 1994;84:22– 8. [13] Piipo S, Mustaniemi L, Lenko H, et al. Surgery for ovarian masses during childhood and adolescent: a report of 79 cases. J Pediatr Adolesc Gynecol 1999;12:223–7. [14] Kokoska ER, Keller MS, Weber TR. Acute ovarian torsion in children. Am J Surg 2000;180:462– 4. [15] Baron C. Torsion of the normal ovary. JAMA 1934;102:1675. [16] Beaunoyer M, Chapdelaine J, Ouimet A. Asynchronous bilateral ovarian torsion. J Pediatr Surg 2004;39:736 – 49. [17] Strickland JL. Ovarian cysts in neonates, children and adolescents. Curr Opin Obstet Gynecol 2002;14:459 – 65. [18] Schultz LR, Newton WA, Clatworth HW. Torsion of previously normal tube and ovary in children. N Engl J Med 1963;268:343– 6. [19] Hibbard LT. Adnexal torsion. Am J Obstet Gynecol 1985;152:456 – 61. [20] Warner MA, Fleischer AC, Edell SL, et al. Uterine adnexal torsion: sonographic findings. Radiology 1985;154:773–5. [21] Quint EH, Smith YR. Ovarian surgery in premenarchal girls. J Pediatr Adolesc Gynecol 1999;12:27–9. [22] Styer AK, Laufer MR. Ovarian bivalving after detorsion. Fertil Steril 2002;77:1053–5. [23] Zweizig S, Perron J, Grubb D, Mishell DR Jr. Conservative management of adnexal torsion. Am J Obstet Gynecol 1993;168:1791–5. [24] Eckler K, Laufer MR, Perlman SE. Conservative management of bilateral asynchronous adnexal necrosis in a prepubescent girl. J Pediatr Surg 2000;35:1248 –51. [25] Pansky M, Abargil A, Dreazen E, et al. Conservative management of adnexal torsion in premenarchal girls. J Am Assoc Gynecol Laparosc 2000;7:121– 4. [26] Pryor RA, Wiczyk HP, O’Shea DL. Adnexal infarction after conservative surgical management of torsion of a hyperstimulated ovary. Fertil Steril 1995;63:1344 – 6. [27] Shun A. Unilateral childhood ovarian loss: an indication for contralateral oophoropexy? Aust N Z J Surg 1990;60:791– 4. [28] Grunewald B, Keating J, Brown S. Asynchronous ovarian torsion— the case for prophylactic oophoropexy. Postgrad Med J 1993;69: 318 –9. [29] Germain M, Rarick T, Robins E. Management of intermittent ovarian torsion by laparoscopic oophoropexy. Obstet Gynecol 1996;88:715–7. [30] Abes M, Sarihan H. Oophoropexy in children with ovarian torsion. Eur J Pediatr Surg 2004;14:168 –71.
Clinical observation
Asynchronous bilateral adnexal torsion in a 13-year-old adolescent: Our experience of a rare case with review of the literature Michail Varras, M.D., Ph.D.a,*, Christodoulos Akrivis, M.D., Ph.D.b, Asymo Demou, M.D.c, and Nikolaos Antoniou, M.D., Ph.D.b a
Second Department of Gynaecology, Anticancer Oncologic Hospital “Agios Savvas,” Athens, First District National Health System, Athens, Greece b Department of Obstetrics and Gynaecology, “G. Chatzikosta” General State Hospital, Ioannina, District National Health System, Ioannina, Epirus, Greece c Department of Pathology, “G. Chatzikosta” General State Hospital, Ioannina, District National Health System, Ioannina, Epirus, Greece Manuscript received April 9, 2004; manuscript accepted October 8, 2004
Abstract
Adnexal torsion is a serious condition and delay in surgical intervention may result in loss of the tube and/or ovary. Children and adolescents who have suffered from uterine adnexal torsion may be at risk for asynchronous torsion of the contralateral uterine adnexa. We report the case of sequential bilateral torsion of uterine adnexa in a 13-year-old girl, resulting in right and subsequently left salpingo-oophorectomy because of gross evidence of total necrosis in both uterine adnexa. After the castration the patient was started on hormone replacement therapy. Families of children who suffered from ovarian torsion and unilateral ovarian loss should be educated about the risk of the contralateral ovary for future torsion and should be encouraged to seek immediate medical help with the recurrence of abdominal pain. © 2005 Society for Adolescent Medicine. All rights reserved.
Keywords:
Ovarian torsion; Adolescent; Children; Asynchronous; Subsequent; Bilateral
J. Blant Sutton described for the first time torsion of adnexa in 1890 [1]. Review of the literature shows that most cases are secondary to adnexal pathology such as ovarian cysts and tumors [2,3]. Adnexal torsion is most frequently unilateral [4]. The first case of bilateral adnexal torsion reported was by Warnek in 1895 [5]. Since then a few cases of bilateral adnexal torsion have been described, usually as simultaneous bilateral torsion of the adnexal structures [4]. Adnexal torsion in children and adolescents is rare. The clinical diagnosis is often uncertain, and delay in surgical intervention frequently leads to the finding of gangrenous adnexal structures at surgery [6]. Once a girl has lost one ovary because of torsion, she is at risk for asynchronous torsion of the contralateral adnexa [7]. Bilateral torsion, for the affected girl and adolescent, represents a catastrophic
*Address correspondence to: Dr. Michail N. Varras, Consultant in Obstetrics and Gynaecology, Platonos 33, Politia Kifisia, Athens 14563, Greece. E-mail address: [email protected]
event from both the reproductive and emotional vantages [7,8]. The purpose of this case report is to describe an extremely rare case of sequential bilateral torsion of uterine adnexae in an adolescent, with review of the literature. Case Report A 13-year-old girl was admitted to the Local General Hospital, Filiates, Epirus, Greece, because of a 12-hour history of intermittent, crampy lower abdominal pain. The pain had acute onset, was located in the right iliac fossa, and radiated down to her right leg. The pain was associated with nausea and she had vomited a number of times. She denied other gastrointestinal or urinary symptoms. The patient’s past medical history was unremarkable. She had no history of similar previous episodes of abdominal pain. She had no prior sexual intercourse. The onset of menarche was at the age of 12 years but she had not had regular cycles yet. On admission, her temperature was 37.8°C and her blood pressure was 90/60 mm Hg. Physical examination found a
1054-139X/05/$ – see front matter © 2005 Society for Adolescent Medicine. All rights reserved. doi:10.1016/j.jadohealth.2004.10.020
M. Varras et al. / Journal of Adolescent Health 37 (2005) 244 –247
Fig. 1. Pelvic ultrasonography of patient’s left adnexal cyst.
nondistended abdomen. Tenderness to deep palpation in the right lower quadrant, without peritoneal signs, was detected. Bowel sounds were present. No masses were palpated. With rectal examination no masses were detected, but mild tenderness in the cul-de-sac was found. A full blood count showed a white cell count of 15,800 cells/mm3 with 84.5% polymorphonuclear cells. The hemoglobin value was 13 g/dl and the hematocrit 38.9%. Serum electrolytes, urea, creatinine, liver function tests, and urinalysis were within normal limits. Abdominal flat and upright roentgenograms disclosed unremarkable findings. The patient was observed closely in the Department of General Surgery; her symptoms persisted and a repeated full blood count 22 hours later showed white cell count of 17,300/mm3 with 88.9% polymorphonuclear cells. The preoperative diagnosis was appendicitis. At abdominal exploration, the patient was found to have a normal appendix with a twisted cyanotic right ovary. The direction of the torsion of the right uterine adnexae was in a clockwise direction. In view of the infarction and nonviability of the right uterine adnexae, a right salpingo-oophorectomy was performed. At that time an appendectomy was also performed. Uterus and left adnexa (ovary and fallopian tube) were of normal size. The patient’s postoperative course was uneventful and she was dismissed on the fourth postoperative day. The fallopian tube measured 0.8 cm in maximum diameter and was 7 cm in length. The right ovary measured 8 ⫻ 4 ⫻ 3.5 cm. On cut section, multiple small cysts with maximum diameter ranging from 0.3 cm to 0.6 cm were recognized. Histologic examination showed hemorrhagic necrosis of the right ovary. Multiple ovarian follicles were recognized. Also, the right fallopian tube showed hemorrhagic necrosis. The appendix was normal. Twenty days after the initial operation, the patient presented to Department of Obstetrics and Gynecology, “G. Chatzikosta” General Hospital, Ioannina, with a 6-hour history of abdominal pain in her left lower quadrant. On arrival
245
her temperature was 37.5°C and her vital signs were normal. On physical examination, tenderness in the left lower quadrant and mild peritoneal signs were present. Bowel sounds were noted to be present. Pelvic ultrasound showed a 5.67 ⫻ 4.84-cm left adnexal cyst (Figure 1). Her laboratory evaluation findings showed white blood count 14,850/mm3 with 91.9% polymorphonuclear cells. Urinalysis had no abnormality. Serum levels of CA-125, AFP, CEA, CA 19-9, and CA 15-3 were within normal values. After discussion with her parents, the decision was made to proceed with an emergency laparotomy. Therefore a 5-cm Pfannenstiel incision was made. The patient was found to have a twisted and cyanotic left fallopian tube and ovary in a clockwise direction. A right salpingo-oophorectomy was perfomed. The patient’s postoperative course was uneventful, and she was dismissed on the fourth postoperative day. At gross examination the left fallopian tube was 7 cm in length and the left ovary measured 14 ⫻ 10 ⫻ 2 cm. At histologic examination the left ovary showed hemorrhagic necrosis (Figure 2). Also, a corpus luteum cyst was found. The left fallopian tube showed hemorrhagic infiltration (Figure 3). No malignancy was found. The patient is treated with hormone replacement therapy. Discussion Adnexal torsion is a gynecologic surgical emergent condition, which occurs at any age but is most common in women of reproductive age [4,9]. In children, adnexal torsion is an infrequent entity [6,10 –14]. Although the adnexal torsion is most frequently unilateral, cases of bilateral synchronous or asynchronous adnexal torsion have been reported. Bilateral asynchronous adnexal torsion in childhood was first described by Baron in 1934 [15]. Özcan et al reviewed the English-language literature in point of asynchronous bilateral adnexal torsion in children and adoles-
Fig. 2. Portion of the left ovary with hemorrhagic necrosis (E & H ⫻ 40).
246
M. Varras et al. / Journal of Adolescent Health 37 (2005) 244 –247
Fig. 3. Cross-section of the left fallopian tube with hemorrhagic infiltration (E & H ⫻ 40).
cents and they were able to document 17 such cases. The age of patients ranged from 3 to 12 years on first presentation, and 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 [7]. Also, Beaunoyer et al reported four cases with asynchronous bilateral adnexal torsion. The mean age at presentation was 10.6 years and the mean interval between the two episodes of ovarian torsion was 15.7 months [16]. The symptomatology and presentation of asynchronous bilateral ovarian torsion in children and adolescents appears to be similar to that of single ovarian torsion [8,16]. The mechanism of adnexal torsion is not known with certainty. Most cases are secondary to adnexal pathology. However, during the peripubertal years, functional ovarian cysts associated with changes in gonadotropin release and failure of follicular involution, seem to be a predisposing cause of adnexal torsion [14,17]. The classic presentation of adnexal torsion is the acute onset of abdominal pain, an adnexal mass, and clinical evidence of peritonitis. However, this triad is usually not present in most patients later found to have adnexal torsion [9]. One aspect that should raise a high index of clinical suspicion of adnexal torsion is a history of previous abdominal pain that is intermittent in nature and probably represents varying degrees of intermittent torsion [6]. The reported frequency of recurrent attacks of pain interspersed with asymptomatic intervals has ranged from 10% to 50% in the literature [6,18,19]. Owing to the intra-abdominal position of the adnexa in a prepubertal child, the diagnosis may be confused with acute appendicitis or another intraperitoneal or gastrointestinal process. This often leads to diagnostic delay and subsequent ovarian loss [17]. The gray-scale ultrasonographic findings of adnexal mass, either cystic, solid or complex, in a child or adolescent presenting with acute or recurrent lower quadrant abdominal pain, nausea and vomiting should raise the possibility of adnexal torsion [20].
Therapy of adnexal torsion remains controversial. Historically, torsion of the adnexa has been managed by excision of the affected structure with extra care taken to avoid untwisting the pedicle because of the fear of dislodging an embolus from the twisted pedicle, and the inability to clinically assess necrosis and tissue viability [9]. However, investigators have found no evidence to support this fear, and now detorsion of the adnexa is advocated, especially in children and young adults who have potentially viable adnexa. If an ovarian or paratubal cyst or neoplasm is found in the twisted adnexa, then a cystectomy or resection of the associated neoplasm should be performed after detorsion of the adnexa. Ovarian function has been shown to recover after detorsion of the adnexa in many reproductive-age women with adnexal torsion [21]. Also, if a torsed ovary appears cyanotic, ovarian bivalving after detorsion has been described as a way to salvage the ovary [22]. If no evidence of tissue perfusion or viability is noted, the adnexa should probably be excised for the theoretical risk of peritonitis from the necrotic tissue, as well as the fact that the resumption of ovarian function is unlikely [9,23]. Adnexal reperfusion with untwisting of the adnexa seems to occur in children as well. Eckler et al reported the case of a child with 72 hours of abdominal pain and necrotic-appearing adnexae who underwent detorsion. Serial postoperative ultrasounds confirmed viability of the adnexa [24]. In addition, Pansky et al advocated the use of laparoscopic detorsion in their series of eight girls and documented ovarian viability in 87% of cases [25]. Complications of conservative management have included postoperative fever and failure, which may necessitate repeat surgery to remove the affected ovary [26]. In cases of ovarian torsion treated by conservative therapy, ultrasound examination should be performed 6 weeks postoperatively. Consideration should also be given to the risk and prevention of future contralateral torsion [21]. Many authors have advocated contralateral oophoropexy to decrease the risk of subsequent torsion [24,27–29]. Oophoropexy can be performed by fixation of the ovary to (a) the lateral part of the pelvic wall, (b) the broad ligament, (c) the posterior wall of the uterus, and (d) the posterior peritoneum just below the bifurcation of the common iliac vessels. Possible risks of this procedure may include adhesions or displacement of the ovary from the fallopian tube with subsequent infertility. Abes and Sarihan presented their experiences with oophoropexy in 10 children with ovarian torsion who underwent oophorectomy and contralateral oophoropexy. The ovary was connected to the peritoneum of the posterior abdominal wall, avoiding a disturbance of the tubo-ovarian anatomic relationship. At follow-up, all pubertal girls had normal menstrual periods [30]. However, no randomized, double-blinded prospective studies of oophoropexy at the time of adnexal torsion have been published to resolve questions regarding success of surgical intervention and impact on fertility [24]. An increased body of literature has documented successful oo-
M. Varras et al. / Journal of Adolescent Health 37 (2005) 244 –247
phoropexy in pediatric and adult patients receiving pelvic radiation for treatment of malignancy [24]. In the case of oophoropexy it is better to use absorbable suture, because permanent suture may predispose to further ovarian injury and bleeding when the ovarian edema resolves and the ovary is retracted from the suture. Conclusion We presented an extremely rare case of asynchronous bilateral adnexal torsion in an adolescent who was surgically castrated because of the gross evidence of total necrosis in both uterine adnexa. Detorsion of the ovary, bivalving, resection of the necrotic tissue, and pexis of any residual ovarian tissue without oophorectomy should be performed for the management of ovarian torsion. References [1] Sutton JB. Salpingitis and some of its effects. Lancet 1890;2:1146. [2] Mordehai J, Mares AJ, Barki Y, et al. Torsion of uterine adnexa in neonates and children: a report of 20 cases. J Pediatr Surg 1991;26: 1195–9. [3] Varras M, Tsikini A, Polyzos D, et al. Uterine adnexal torsion: pathologic and gray-scale ultrasonographic findings. Clin Exp Obstet Gynecol 2004;34 – 8. [4] Buss JG, Lee RA. Sequential torsion of the uterine adnexa. Mayo Clin Proc 1987;62:623–5. [5] Warnek L. Trois cas de tumeurs des trompes compliquees de la torsion du pedicle. N Arch D’ Obstet De Gynec (Paris) 1895;10:81. [6] Spigland N, Ducharme JC, Yazbeck S. Adnexal torsion in children. J Pediatr Surg 1989;24:974 – 6. [7] Özcan C, Çelik, Özok G, Erdener A, Balik E. Adnexal torsion in children may have a catastrophic sequel: asynchronous bilateral torsion. J Pediatr Surg 2002;37:1617–20. [8] Davis AJ, Feins NR. Subsequent asynchronous torsion of normal adnexa in children. J Pediatr Surg 1990;25:687–9. [9] Bayer AI, Wiskind AK. Adnexal torsion: can the adnexa be saved? Am J Obstet Gynecol 1994;171:1506 –10. [10] Lee CH, Raman S, Sivanesaratnam V. Torsion of ovarian tumors: a clinicopathological study. Int J Gynaecol Obstet 1989;28:21–5.
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