Factors Associated With Delay in Care for Children and Adolescents With Adnexal Torsion

Failing to recognize adnexal torsion in a timely fashion can lead to the need for unilateral oophorectomy, salpingectomy, and impaired future fertility.1 Fifty-two percent of adnexal torsions occur in people 9 to 14 years of age2 and affect approximately 5 of 100 000 female individuals aged 1 to 20 years.3,4 Females older than 10 years of age are at a particularly increased risk of torsion because of the hormonal influences and gonadal growth that can cause physiologic and pathologic adnexal masses.5,6 Unfortunately, there is well-documented evidence of delays in the diagnosis of adnexal torsion in children and adolescents, leading to adverse outcomes.7,8 In this issue of Pediatrics, Rich et al explore the patient as well as system factors that contribute to delays in the diagnosis of adnexal torsion.9 It is useful to explore these factors as well as review the challenges of diagnosing adnexal torsion, debunk myths surrounding the appropriate surgical approach, and discuss potential strategies to improve effective and timely management.The timely diagnosis of adnexal torsion is challenging because the differential diagnosis of an adolescent with abdominal pain is broad and the presentation of adnexal torsion is nonspecific.8,10 The most common clinical symptom of torsion is sudden onset of abdominal pain that is intermittent, nonradiating, and associated with nausea and vomiting. Clinical signs of adnexal torsion include abdominal tenderness, which is reported in up to 88% of patients. Rebound and peritoneal signs are reported in just 12% to 27% of patients.11,12 A bimanual examination to assess for a palpable adnexal mass is generally not necessary or tolerated.13 Adnexal torsion is more common on the right side (64%), presumably due to the somewhat longer mesosalpinx and utero-ovarian ligament on the right side2 and the protective nature of the descending colon on the left side.14 In contrast to adnexal torsion in adults, adnexal torsion in pediatric and adolescent patients involves an ovary without an associated mass or cyst in as many as 46% of cases.10Diagnosis of adnexal torsion is challenging not only due to the nonspecific clinical presentation but also because of often inconclusive imaging results. Transabdominal ultrasonography remains the modality of choice for diagnosing adnexal torsion in the adolescent and pediatric population, with a sensitivity of 92% and specificity of 96%.15 Ultrasonography findings suggestive of ovarian torsion include (1) unilateral ovarian enlargement; (2) ovarian edema characterized by a hyperechogenic ovary with peripherally displaced follicles and echogenic stroma; (3) free fluid; and (4) a coiled vascular pedicle (referred to as the “whirlpool sign”).16,17 However, there are no definitive clinical or imaging criteria sufficient to confirm the preoperative diagnosis of adnexal torsion.16 Doppler flow alone should not guide clinical decision making due to its low sensitivity and operator dependency. One study reported normal Doppler arterial flow in as many as 60% of surgically confirmed cases of adnexal torsion.10 Preservation of Doppler arterial flow can be explained by intermittent torsion, or collateral blood supply from either the utero-ovarian or infundibulopelvic vessels. If ovarian torsion is suspected, diagnostic laparoscopy is indicated to preserve ovarian function, pubertal development, and future fertility.Torsion can be treated with minimally invasive surgery for detorsion of the ovary or, if necessary, oophorectomy.18 An unfounded myth contributing to unnecessary oophorectomy is that a black or blue ovary suggests necrosis and thus should be removed. The appearance of the ovary at surgery is not a reliable indicator of ovarian viability. Future ovarian function may be preserved despite a grossly ischemic appearance at the time of surgery.19–27 A second unfounded myth is that embolic phenomena will occur after untwisting. No cases of venous thromboembolism after detorsion have been reported in the literature.4,18 Many obstetrician-gynecologists also cite the fear of leaving behind a potential malignancy as a reason to perform oophorectomy. Although more than one half of cases of pediatric and adolescent adnexal torsion occur in the setting of an adnexal mass, cancer in this age group rarely presents as adnexal torsion.24,28 The rate of malignancy among premenarchal adolescents with ovarian torsion ranges from 0.4% to 5%.13,18,28 For torsion management, the American College of Obstetricians and Gynecologist recommends “a minimally invasive surgical approach with detorsion and preservation of the adnexal structures regardless of the appearance of the ovary. Surgeons should not remove a torsed ovary unless oophorectomy is unavoidable, such as when a severely necrotic ovary falls apart.”16According to the American College of Obstetricians and Gynecologist, cystectomy need not be performed at the time of detorsion and can be determined on a case-by-case basis.16 These guidelines also state that if a cyst is identified, it is likely benign and therefore reasonable to proceed with a concomitant cystectomy.16 Some may opt to proceed with a cystectomy to avoid retorsion if adnexal torsion is not apparent during surgery, but intermittent torsion is suspected. However, the twisted adnexa are occasionally severely edematous and friable, and in such cases, attempts at cystectomy can result in ovarian tissue damage and bleeding that may lead to oophorectomy. If a cystectomy is not performed, a surgeon may consider incision and drainage for large cysts. Ultrasonography to reevaluate the cyst at 6 to 12 weeks is recommended, although reevaluation should be done sooner if there is concern for malignancy.19,24Delays in diagnosis of adnexal torsion are well documented in the pediatric and adolescent population.7,8 Among female patients aged 1 to 20 years, the estimated incidence of ovarian torsion is 4.9 per 100 000,4 and it accounts for 4% of all consultations for acute abdominal pain in females in the pediatric emergency department (ED).29 Michelson et al report a delayed diagnosis incidence of more than 15% for 1460 ovarian torsion patients under 18 years old from 2015 to 2019 in a recently published retrospective analysis.8Children are at particular risk of diagnostic delays for several reasons. Serious illnesses can be more subtle in children and certain developmental stages may render children less able to communicate symptoms. Many emergency physicians have little experience with adnexal torsion in children. Michelson et al found EDs with fewer pediatric encounters were associated with higher rates of delayed diagnoses.8 EDs with high pediatric volume are also more likely to have access to high-quality imaging modalities, diagnostic tools, and specialty consultants.Rich et al sought to identify what other factors may be contributing to the diagnostic delays in pediatric and adolescent patients with adnexal torsion. They performed a multi-institutional retrospective analysis of female patients aged 5 and 18 years with confirmed operative adnexal torsion between 2013 and 2022. Delay to care was defined as (1) prior ED discharge within 7 days of operation and/or (2) hospital admission without initial plan for operative intervention. The Area Deprivation Index was used by the authors as a proxy for social drivers of health but was originally created as an index of socioeconomic deprivation based on 17 measures of education, employment housing, and poverty, with the most disadvantaged neighborhoods having the highest score.30 The authors noted a 30% delayed diagnosis rate in the 862 patients who were included from 10 children’s hospitals. The authors found that patients with delay were less likely to present with emesis or fever, less likely to have initial pediatric surgery consultation, and less likely to have typical ultrasound findings of torsion. They also found the odds of delay to be increased by 1.3% for every unit increase in Area Deprivation Index. Patients living more than 30 miles away from the hospital had 81% greater odds of having a delay of diagnosis and treatment for adnexal torsion compared with those patients living within 1 to 10 miles away.These findings highlight how the variable clinical and radiographic presentation of ovarian torsion contributes to delays in the diagnosis of adnexal torsion. This work also parallels that of Michelson et al in demonstrating an increased odds of delayed diagnoses for patients in areas with limited resources.In the pursuit of improving timely diagnoses, several strategies should be considered. First, diagnostic expertise in gynecological surgical problems should be available in all EDs, either in person or via telemedicine. Stronger education efforts for low-volume emergency care providers on adnexal torsion detection and management could also be beneficial.7Adnexal torsion is a clinical diagnosis and a surgical emergency. The key to torsion management is timely recognition, yet the diagnosis of adnexal torsion remains a challenge in the pediatric and adolescent population given its variable clinical and radiological findings. Treatment entails minimally invasive surgical intervention, adnexal detorsion, ovarian preservation, and an individualized approach to ovarian cyst management. Future efforts in this area should strive to provide EDs with the tools necessary to provide timely diagnoses and management for pediatric and adolescent patients with ovarian torsion. Ongoing multispecialty collaboration among ED providers, pediatricians, pediatric surgeons, gynecologists, and pertinent family practice providers is crucial to optimize care and to ensure that minimally invasive detorsion with ovarian preservation is the standard treatment provided going forward.