Journal of Pediatric Surgery (2005) 40, E17 – E20
www.elsevier.com/locate/jpedsurg
Ovarian torsion associated with appendicitis in a 5-year-old girl: a case report and review of the literature Brian A. Hoeya,*, Stanislaw P. Stawickib, William S. Hoffa, Ravi K. Veeramasunenic, Heather Kovichc, Michael D. Grossmana a
Division of Trauma and Critical Care, University of Pennsylvania Trauma Network, Philadelphia, PA 19122, USA Department of Surgery, St. Luke’s Hospital and Health Network, Bethlehem, PA 18015, USA c Division of Plastic Surgery, Department of Surgery, Temple University School of Medicine, Philadelphia, PA 19122, USA b
Index words: Appendicitis; Ovarian torsion
Abstract Acute ovarian torsion is an uncommon cause of abdominal pain in female children and is often difficult to differentiate from other conditions causing lower abdominal pain. Acute adnexal pathology associated with appendicitis is very rare, with only a handful of reports available in the literature. Reported is a case of ovarian torsion associated with appendicitis in a 5-year-old girl along with a comprehensive literature review. D 2005 Elsevier Inc. All rights reserved.
1. Case report A 5-year-old girl presented to the emergency department with 3-day history of lower abdominal pain. The pain was of gradual onset and initially most intense just superior to her pubis. She had been started on antibiotics 1 day before the current admission for a presumed urinary tract infection. Her symptoms gradually worsened, with increasing pain, anorexia, nausea, and vomiting. Her parents reported an oral temperature of 1018F on the eve of admission. On presentation, she was afebrile with normal vital signs but she appeared ill. Her abdomen was flat and soft, with mild tenderness in the right lower quadrant (RLQ) and right suprapubic region without guarding or rebound. The remainder of her physical exam was unremarkable. Initial T Corresponding author. Division of Trauma and Surgical Critical Care, St. Luke’s Hospital and Health Network, Bethlehem, PA 18015, USA. Tel.: +1 610 954 2202; fax: +1 610 9542 220. E-mail address:
[email protected] (B.A. Hoey). 0022-3468/$ – see front matter D 2005 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2005.05.068
laboratory studies included a white blood cell count of 11.1 103 with no left shift. Serum electrolytes were within normal limits. Her urinalysis was unremarkable. A computed tomography (CT) scan of her abdomen and pelvis with oral and intravenous contrast demonstrated inflammatory changes in her RLQ with an bassociated abnormal collectionQ (Fig. 1). An ultrasound was performed to better characterize this collection. It demonstrated a normal appendix with a heterogeneous mass just inferior to the appendiceal tip, which was thought to represent an enlarged right ovary. A color Doppler study showed no blood flow within the mass, but there was blood flow present around the mass. The patient was taken to the operating room for an exploratory laparotomy with a presumed diagnosis of right ovarian torsion (OT). Operative findings included a torsed right ovary twisted around the distal third of the patient’s appendix. The distal appendix was edematous and partially necrosed. The patient underwent an appendectomy and a right salpingo-oophorectomy.
E18
B.A. Hoey et al.
2. Discussion
Fig. 1 A preoperative CT scan of the patient’s abdomen demonstrating RLQ inflammatory changes and the enlarged right ovary.
Postoperatively she progressed well, tolerated her diet, and was discharged home on the first postoperative day. She was well at the 4-week follow-up examination, having completely recovered from her surgery. The final pathological findings included an infarcted right ovary and distal fallopian tube with ischemic hemorrhagic necrosis. The appendix was noted to be inflamed distally with full thickness necrosis at the tip. Table 1
Ovarian torsion is an infrequent cause of abdominal pain in the pediatric female population. Ovarian torsion and concomitant appendicitis are exceedingly rare. Medlinereferenced and cross-referenced sources demonstrated only 6 reported cases [1-6] (Table 1). The present case was added to the cases collected from the literature. The patients in these reports range in age from 5 months to 43 years. Diagnostic modalities used included physical exam, plain radiographs, CT, and ultrasonography. An open operative approach was used in all of these cases. All required both an appendectomy and oophorectomy with or without a salpingectomy, and outcomes were good in all patients. Pathological analysis revealed a cystic ovarian lesion in 5 of 7 cases, and ovarian neoplasm in 1 of 7. The present case was singular in the fact that no coexisting ovarian pathology was identified. In general, OT occurs more frequently on the right side and often poses a diagnostic dilemma because of the difficulty encountered in differentiating it from acute appendicitis [7-9]. Two Russian studies found that 4.2% to 4.6% of female patients with a clinical diagnosis of acute appendicitis had an acute gynecological condition [10,11]. Table 2 shows the differential diagnosis of acute abdomen in a female child. Ovarian torsion most commonly occurs in the presence of ovarian tumors and cysts [7]. As exemplified by the current case and others, it can also occur in normal ovarian tissue [7]. Tumors involved in OT are usually benign [7,12,13]. Young children tend to have either torsion of a mature cystic teratoma or torsion with no underlying
Case reports of simultaneous appendiceal and ovarian operative pathology
Cases
Age
Diagnostic evaluation
Procedure
Pathology
Nikolaev [1]
43 y
Physical examination
Left oopherectomy, appendectomy
Pidoprigora et al [2]
10 y
Physical examination
Left oopherectomy, appendectomy
Kokoszka et al [3]
42 y
Physical examination
Right salpingo-oopherectomy, appendectomy
Gavrilenko et al [4]
17 y
Physical examination
Appendectomy, left salpingooopherectomy
Kalinets et al [5]
5 mo
Physical examination, plain abdominal x-rays
Right salpingo-oopherectomy, appendectomy
Krupinska et al [6]
3y
Current case
5y
Physical examination, ultrasound Physical examination, 2CT of abdomen and pelvis, pelvic ultrasound with duplex imaging
Laparotomy, left salpingooophorectomy, appendectomy Right salpingo-oophorectomy, appendectomy
Phlegmonous appendicitis Torsed left ovary with paraovarian cyst Acute phlegmonous appendicitis Torsed left ovary with a cyst Acute gangrenous appendicitis Torsed right ovary with a cyst Gangrenous, perforated appendicitis Hemorrhagic cyst of left ovary Phlegmonous appendicitis Follicular cyst of right ovary with mural hematoma Phlegmonous appendicitis Left ovarian teratoma Gangrenous appendicitis Ischemic ovarian necrosis
Ovarian torsion associated with appendicitis in a 5-year-old girl Table 2 Differential diagnosis of an acute abdomen in the female patient Nongenitourinary
Genitourinary
Appendicitis Gastroenteritis Mesenteric adenitis Intussusception Henoch-Schonlein purpura Crohn’s disease Meckel’s diverticulitis Duodenal ulcer
Ovarian torsion Congenital biliary dilatation Genital anomalies Pelvic inflammatory disease Ovarian cyst Urinary tract disease
pathology, whereas older children are more likely to have torsion associated with a cyst [7]. The causes of OT with normal adnexa are unclear [14]. Because OT often causes hemorrhagic infarction and significant edema of the ovary, it can be difficult to ascertain on pathological examination whether a cyst was present before the event [14]. Of interest, a significant number of patients with OT in one large series had an underlying solid or cystic mass that may have served as a lead point for torsion [9]. Because of hormone stimulation, functional ovarian cysts in children occur most frequently during the first year of life and around menarche. The cases compiled in this literature review are consistent with these trends. The children who suffered torsion of an ovarian cyst were 5 months of age and 10 years of age. The other young children in the series underwent torsion of a teratoma (3 years of age) and torsion of benign-appearing ovarian tissue (present case, 5 years of age). When OT and acute appendicitis occur concomitantly, it is often not evident which is the primary event. In our case, it appeared that the ovary had torsed around the appendix and may have contributed to the focal distal appendicitis. Conversely, the inflammatory response set off by an acute appendicitis might have affected the hemodynamics of the ovarian blood supply. Of interest, tortuous veins are thought to be among the predisposing factors in OT [14]. It is difficult to find a precedent to help elucidate the causative relationship, and conflicting opinions have been put forth in previous literature reports [10,15]. Diagnosis of OT is often confirmed with CT and sonography [7-9,16,17]. Ultrasonic signs of OT include solid, cystic, or complex pelvic masses with or without associated fluid in the Pouch of Douglas, and multiple follicles in the cortical portions of a unilaterally enlarged ovary [16]. In this case, duplex imaging demonstrated no flow to the torsed ovary. In fact, ultrasonography with color Doppler can be helpful for differentiating acute OT from appendicitis [3]. Treatment of OT is surgical. Attempts should be made to salvage the ovary whenever possible [9,18]. Preservation of future fertility is crucial [18]. If the ovary appears viable and no tumor is present, surgical options include detorsion of the ovary and ovarian cystectomy in cases of ovarian cysts [9,18,19]. In fact, Cohen et al [18] reported that laparo-
E19
scopic detorsion of the twisted, ischemic, hemorrhagic adnexa is a viable option with minimal morbidity and can result in full recovery of ovarian function. After an OT episode, children may be at increased risk for a repeat event [20]. After unilateral ovarian loss, the contralateral ovary may be at risk for future torsion. Consequently, some advocate oophoropexy in both retained detorsed and contralateral ovaries [20]. Ovarian biopsy should be considered whenever suspicion of malignancy is entertained. Treatment for pediatric ovarian malignancies involves salpingo-oophorectomy, and complete staging with evaluation of omentum, retroperitoneal lymph nodes, contralateral ovary, and peritoneal surfaces [9]. Unilateral salpingo-oopherectomy is indicated in cases where the ovary is not salvageable or if tumor involvement is present [9]. If a tumor is suspected, preoperative tumor markers including alpha-fetoproteins and beta-human chorionic gonadotropin may aid in postoperative treatment [7]. Several studies have reported on the use of a laparoscopic approach not only diagnostically but also therapeutically, for detorsion with or without fixation, ovarian cystectomy, and treatment of associated surgical lesions [17]. Laparoscopic surgery for pediatric uterine adnexal torsion has been described to be safe and result in short hospitalizations, good cosmetic results, and rapid return to baseline activity [21]. Ovarian pathology coincidental with acute appendicitis is rare and an indication for surgery in pediatric female patients. Ovarian sparing should be performed whenever possible, although torsed ovary is seldom salvageable. Diagnosis consists of physical examination, CT scan of abdomen and pelvis, and ultrasonic study with Doppler color flow. Treatment is operative and often requires salpingo-oopherectomy along with an appendectomy.
Acknowledgment We acknowledge the generous assistance of Leslaw J Stawicki, MA, with Polish, Russian, and Ukrainian journal article translations.
References [1] Nikolaev VI. Phlegmonous appendicitis in association with a twisted left paraovarian cyst and rupture of the left ovary. Vestn Khir Im Il Grekova 1971;106:141. [2] Pidoprigora AP, Kukliuk VI. Case of a combination of torsion of the crus of an ovarian cyst and acute phlegmonous appendicitis. Pediatr Akush Ginekol 1975;4:60. [3] Kokoszka J, Budzynska-Dobek W. Pregnancy complicated by torsion of an ovarian cyst and gangrenous appendicitis. Pol Tyg Lek 1978;28:2023 - 4. [4] Gavrilenko BG, Chernyshenko GA, Chernega VI. Destructive appendicitis in combination with torsion of a left ovarian cyst. Klin Khir 1986;4:65 - 6. [5] Kalinets OS, Kruk IuE, Semeniuta NM. Torsion of an ovarian cyst combined with phlegmonous appendicitis in a 5-month-old girl. Klin Khir 1988;6:75.
E20 [6] Krupinska E, Klimanek-Sygnet M, Chilarski A. Rare coexistence of phlegmonous appendicitis and tumor of a twisted left ovary. Ginekol Pol 2003;74(4):307 - 11. [7] Kokoska ER, Keller MS, Weber TR. Acute ovarian torsion in children. Am J Surg 2000;180:462 - 5. [8] Neinstein LS, Braud BJ. Coincident acute appendicitis and hemorrhagic corpus luteal cyst. J Adolesc Health Care 1984;5:137 - 8. [9] Cass DL, Hawkins E, Brandt ML, et al. Surgery for ovarian masses in infants, children, and adolescents: 102 consecutive patients treated in a 15-year period. J Pediatr Surg 2001;36:693 - 9. [10] Lutsenko NS. Appendicular-genital syndrome in urgent surgery. Klin Khir 1979;4:16 - 9. [11] Seleznev EK, Soloviev NA, Pugleeva VP. Differential diagnosis of acute appendicitis and gynecological diseases. Klin Med 1972;50: 148 - 51. [12] Sommerville M, Grimes DA, Loonings PP, et al. Ovarian neoplasms and the risk of adnexal torsion. Am J Obstet Gynecol 1991;164: 577 - 8. [13] Petersen WF, Prevost EC, Edmunds FT, et al. Benign cystic teratomas of the ovary. A clinico-statistical study of 1007 cases with a review of literature. Am J Obstet Gynecol 1955;70:368 - 82.
B.A. Hoey et al. [14] Dunnihoo DR, Wolff J. Bilateral torsion of the adnexa: a case report and a review of the world literature. Obstet Gynecol 1984; 64(3 Suppl):55S - 9S. [15] Tanaka T. Ruptured corpus luteum cyst of the ovary following nonperforated acute appendicitis. Three case reports. Clin Exp Obstet Gynecol 1998;25:81 - 2. [16] Shust NM, Hendricksen DK. Ovarian torsion: an unusual cause of abdominal pain in a young girl. Am J Emerg Med 1995;13:307 - 9. [17] Heloury Y, Guibertreau V, Sagot P, et al. Laparoscopy in adnexal pathology in the child: a study of 28 cases. Eur J Pediatr Surg 1993;3:75 - 8. [18] Cohen SB, Oelsner G, Seidman DS, et al. Laparoscopic detorsion allows sparing of the twisted ischemic adnexa. J Am Assoc Gynecol Laparosc 1999;6:139 - 43. [19] Oelsner G, Bider D, Goldenberg M, et al. Long-term follow-up of the twisted ischemic adnexa managed by detorsion. Fertil Steril 1993; 60(6):976 - 9. [20] Abes M, Sarihan H. Oophoropexy in children with ovarian torsion. Eur J Pediatr Surg 2004;14:168 - 71. [21] Cohen Z, Shinhar D, Kopernik G, et al. The laparoscopic approach to uterine adnexal torsion in childhood. J Pediatr Surg 1996;31:1557 - 9.