Asynchronous bilateral adnexal torsion in a prepubertal girl: A case report and review of the world literature

Adolesc Pediatr Gynecol (1990) 3:197-200

Adolescent and Pediatric Gynecology © 1990 Springer-Verlag New York Inc.

Asynchronous Bilateral Adnexal Torsion in a Prepubertal Girl: A Case Report and Review of the World Literature Michael Pearl, M.D., Carol Major, M.D., and Bonnie Coyne, M.D. Department of Obstetrics, Gynecology. and Reproductive Sciences, University of California, San Francisco, San Francisco General Hospital. San Francisco, California

Abstract. A 7-year-old girl who presented with progressive abdominal pain associated with nausea, vomiting, and fever underwent laparotomy for a presumed peri appendiceal abscess. Exploration revealed a torsed left adnexa with hemorrhagic necrosis and a normal appendix, The adnexa was removed along with the appendix. Two years later, the patient presented with increasing right lower abdominal pain, nausea, and fever. Physical examination revealed a tender, soft, midpelvic mass that produced pain upon palpation. Adnexal torsion was diagnosed at laparotomy and the right adnexa was twisted 720 in a clockwise fashion. A right salpingo-oophorectomy was performed and the uterus was retained. The patient did well postoperatively. 0

Key Words. Adnexal torsion-Adnexal mass-Prepubertal girl

Introduction Adnexal torsion is an uncommon disorder that may afflict adolescent girls. The etiology includes adnexal masses, congenital or acquired defects, and tubal dis-+ ease. The diagnosis is difficult, often leading to delay in treatment. Physicians treating young girls must have a high index of suspicion if the adnexa is to be salvaged. In addition, they should be aware that subsequent torsion of the contralateral adnexa may occur. Case Report A 7-year-old oriental girl presented in 1986 with a 4-day history of mild, progressive abdominal pain Address reprint requests to: Michael Pearl, M.D., Department of

Obstetrics, Gynecology and Reproductive Sciences, San Francisco General Hospital. Ward 6D, 1001 Potrero Ave .. San Francisco, CA 94110, USA.

associated with nausea and vomiting. A diagnosis of constipation prompted enema therapy and temporary symptomatic relief. She returned with worsening abdominal pain and fever. She denied any further episodes of nausea or vomiting and had a normal appetite. Her temperature was 38.7°C and her pulse was 144 beats per minute. She was Tanner stage I for breast and pubic hair. She had diffuse lower abdominal tenderness with peritoneal irritation signs. Her white blood count was 20,700 per mm' with a left shift. Ultrasound showed free fluid in both pericolic gutters and a complex mass in the pelvis. She underwent laparotomy for a presumed periappendiceal abscess. Exploration revealed a torsed left adnexa undergoing hemorrhagic necrosis, and a normal appendix; the adnexa was removed along with the appendix. The direction of torsion and number of twists were not recorded. The right ovary was noted to be slightly enlarged for a prepubertal girl and contained mUltiple small follicular cysts. Plication of the right ovary was not performed. The remainder of the pelvis and the abdomen were normal. Her postoperative course was unremarkable. Evaluation for malignancy, including ovarian tumor markers. was negative. Histopathologic evaluation revealed diffuse ovarian parenchymal hemorrhage and occasional primordial follicles. The patient did well until 2 years later, when she presented with 24 hours of right lower abdominal pain and nausea. Her examination was unremarkable and she was treated conservatively for gastroenteritis. She returned the following evening with fever and increasingly severe. well-localized right lower quadrant pain. Gynecologic consultation was sought. Her temperature was ~9.~oC and her pulse was 120 beats per minute. She remained Tanner stage 1 for breasts and pubie hair. Bowel sounds were normal. She had exquisite point tenderness in the right lower quadrant with peritoneal irritation signs. Rectal examination revealed a tender, soft. midpelvie mass that. on palpation, reproduced her pain. White

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Pearl et al.:

Asynchronous Bilateral Adnexal Torsion

Fig. 1. Longitudinal sonogram shows complex mass (arrows) measuring 8 cm x 3 cm posterior to the uterus (u) representing the torsed right adnexa. Free fluid (f) was present in thc cul-de-sac.

blood count was 12,400 per mm 3 with a left shift. Ultrasound (Fig. 1) demonstrated a greatly enlarged right ovary with multiple small cysts. The uterus was normal and premenarchal. There was a small amount of free fluid in the cul-de-sac. A presumptive diagnosis of adnexal torsion was made. At laparotomy, the right adnexa was twisted 720° in a clockwise fashion (Fig. 2). A right salpingooophorectomy was performed as it was felt the adnexa did not contain any viable tissue. The uterus was retained. Postoperatively, she did well. Histopathology demonstrated hemorrhagic infarction of both ovary and fallopian tube. Discussion

Table 1 summarizes the reported cases of asynchronous bilateral adnexal torsion. Six of the nine patients were prepubertal, one was postmenopausal

Fig. 2. Torsed right tube (T) and ovary (0) measuring lJ x 6 cm.

when she presented with her second torsion. and two patients were in their reproductive years with both episodes. It seems, therefore, that this condition is not limited to any age group. Acute appendicitis was the most commonly reported preoperative diagnosis with initial presentation. Possibly due to a heightened sensitivity, the second episode was correctly

Pearl et al.:

Asynchronous Bilateral Adnexal Torsion

1<)1)

Table 1. Reported Cases of Asynchronous Bilateral Adnexal Torsion Age at first surgery (yrs. )

Interval between surgeries

Preopera tive diagnoses (chronologie)

Postoperative diagnoses (chronologic)

37

2 weeks

7

2 years

Acute appendiciti s: nol reported Acute appendicitis ; adnexal torsi Oil Acute appendicitis; ovarian torsion Not reported ; nol reported Not reported; not reported Acute appendicitis; adnexal torsion Ovarian torsion ; ovarian torsion Acute abdome n; adnexal torsion Acute appendicitis; adnexal torsion

Torscd bilateral pyosalpinx Torscd normal adnexa Torscd bilateral hydrosa lpinx Torsed normal adnexa Torsed Ilormal adnexa Torsed normal adnexa Torsed normal adne xa To)'sed normal andexa Torscd normal adnexa

3~

15 years

12

6 weeks

3

3 years

7

1 year

29

7 years

6

2 years

7

2 years

diagnosed as adnexal torsion in all cases with a recQrded preoperative diagnosis. The etiology of adnexal torsion appcars to be multifactorial. Predisposing factors that have been postulated include 1) ovarian enlargement, both malignant and benign, 2) diseases of the fallopian tube, 3) congenital abnormalities of the tune, 4) postsurgical or traumatic changes of the tube, and 5) tubal tortuosity or congestion. 2- 4 .6 Overall, the single most frequent cause of torsion is a benign cystic ovarian tumor. In a recent review of adnexal torsion, approximately 48% of cases involved ovarian neoplasms, 30% of which were dermoids Y The average age was 26 years. The risk of a torsion containing a malignancy ranges from 015 % ,'1.10 This variation may be due to the percentage of post-menopausal women in a given series. Y On the other hand, malignant adnexa may be less mobile due to adhesions and tumor invasion. I() Tubal enlargement can result in pendulum-like movement, leading to torsion. In a report of nonsimultaneous adnexal torsion, 28% of the patients had hydrosalpinx or pyosalpinx.7 Hydrosalpinx or hematosalpinx complicated 70% of cases of unilateral torsion of the tube . 3 An unrecognized ectopic pregnancy and secondary hematosalpinx can lead to torsion. 6 Though identification of villi by the pathologist may be difficult after hemorrhagic necrosis takes place. the usc of a sensitive l3-hCG test may aid in diagnosis. Tubal anomalies such as excessive length or a spiral course have been presented as etiologic faetors. 7. 11 Congenital absence of the mesosalpinx allows the tube to rotate excessively in either direction , predisposing towards torsion. Embryological rem-

Reference

Pozzi' Hawn ' Hansen 1 McCrea" Bower and Adkins' Dunnihoo and Wolff"

lI agay ct al. 7 Worthington-Kirsch t:t ,II ..' Prcst:nt cast:

nants such as hydatid cysts of Morgagni may also lead to torsion. Tubal ligation or other surgical procedures may be associated with torsion by interrupting tubal continuity and providing a pivot point for rotation. In a recent report of patients with unilateral tubal torsion, 8 of 11 had undergone prior sterilization. most frequently by electrocoagulation. I I Circulatory changes may result in edema with subsequent increase in weight of the fimbriated e nd of the fallopian tube. Similarly. as the veins of the mesosalpinx are longer and more tortuous than the arteries, pelvic venous congestion may increase the degree of twist along their length. Several authors believe that this syndrome may be responsible for unilateral torsion associated with pregnancy. 7.12 The clinical presentation of adnexal torsion is nonspecific. Most patients will experience some degree of pain , as well as associated nausea and vomiting. A low-grade fever and tachycardia arc common. Signs of peritoneal irritation may be prese nt, often with a palpable adnexal mass . A mild leukocytosis is usual. Culdocentesis may yield serosanguinous fluid. It is interesting that torsion occurs more frequently (3:2) on the right side . Given that the sigmoid occupies the left side, it may be that there is more room on the right for torsion to occur. The difference in venous drainage may also re~iUlt in rightsided predominance . J.l Radiologic studies are of greatest value in excluding other clinical conditions. Ultrasonographic findings are nonspecific. Examination may reveal an enlarged , diffusely hypoechogenie ovary. Free fluid is a late manifestation that correlates with non viability

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Asynchronous Bilateral Adnexal Torsion

of the adnexa. R In all reported cases of torsion with visualization of free fluid, including this case, the ovary was excised; in three cases with salvagable ovaries, no free fluid was seen. R Laparoscopy has proven useful in excluding torsion as a diagnosisY·ll Accurate preoperative diagnosis is difficult. Lomano et al. 12 correctly diagnosed 18% (8/44) of their patients. Possibly because all their patients presented with abdominal pain, Koonings and Grimes lO were accurate in 42% (8/19). In Hibbard's'} review, the diagnostic accuracy among patients with proven torsion was 66% (85/128). When patients with an incorrect preoperative diagnosis of torsion were included, accuracy fell to 37.8% (85/225).9 Often, surgical exploration is delayed, which may account for the frequent findings of nonviable adnexa. As systemic embolization following release of a thrombotic pedicle is widely feared, salpingo-oophorectomy is the most common procedure. As most patients are young and well able to tolerate surgery, the major morbidity is loss of an ovary. Controversy exists over whether oopexy should be performed on the contralateral ovary at the time of surgery for torsion. In males presenting with testicular torsion, orchiopexy is performed routinely. In females, however, plication may alter the relationship between the ovary and fallopian tube, lowering fertility. Further study is needed before making a recommendation. In summary, adnexal torsion is the fifth most common cause of emergency gynecologic surgery, with approximately 15% of these cases occurring during childhood. 9 However, this clinical entity remains poorly understood and frequently misdiagnosed. Bilateral adnexal torsion occurs rarely, with fewer than 20 reported cases in the world literature. Of these, torsion has occurred asynchronously in nine cases.

Our case serves to emphasize the difficulty in making the diagnosis of adnexal torsion and to alert physicians that torsion may recur in the contralateral adnexa. References 1. Pozzi MS: Note Sur Quatre Nouveaux cas de la Trompe Kystique. Compt rend de la Soc d'Obst de Gyn et de Paed de Paris 1900; 2:95 2. Baron C: Torsion of the normal ovary. JAMA 1934; 102: 1675 3. Hansen OH: Isolated torsion of the fallopian tube. Acta Obstet Gynecol Scand 1970; 49:3 4. McCrea RS: Uterine adnexal torsion with subsequent contralateral recurrence. J Repro Med 1980; 25: 123 5. Bower RJ, Adkins JC: Surgical ovarian lesions in children. Am J Surgery 1980; 47:474 6. Dunnihoo DR, Wolff J: Bilateral torsion of the adnexa: a case report and review of the world literature. Obstet Gynecol 1980; 64:55 7. Hagay ZJ, Mazor M, Katz M, et al: Bilateral uterine adnexal torsion: case report and review of the literature. [sr J Med Sci 1986; 22:54 8. Worthington-Kirsch RL, Raptopoulos V, Cohen IT: Sequential bilateral torsion of normal ovaries in a child. J Ultrasound Med 1986; 5:663 9. Hibbard LT: Adenexal torsion. Am J Obstet Gynecol 1986; 152:456 10. Koonings PP, Grimes DA: Adnexal torsion in postmenopausal women. Obstet Gynecol 1989; 73: 11 11. Bernardus RE, Van Der Slikke JW, Roex AJM, et al: Torsion of the fallopian tube: some considerations on its etiology. Obstet Gynecol 1984; 64:675 12. Lomano JM, Trelford JD, Ullery JC: Torsion of the uterine adnexa causing an acute abdomen. Obstet Gynecol 1970; 35:221 13. Kanbour AJ, Salazar H, Tobon H: Massive ovarian edema. Arch Pathol Lab Med 1970; 103:42.