Mariel A Focseneanu1, Kenan Omurtag, Valerie S Ratts, Diane F Merritt. 1. Department of Obstetrics and Gynecology, Divisions of Reproductive Endocrinology and Infertility and Pediatric and Adolescent Gynecology, Washington University, St. Louis, Missouri.
Abstract
STUDY OBJECTIVE: To quantify our experience and that of the literature with diagnosis and management of the auto-amputated adnexa in a pediatric population. DESIGN: Case series and literature review. SETTING: Tertiary care medical center. PARTICIPANTS: Case series of pediatric patients (<18 years of age) with surgically documented adnexal auto-amputation collected from our medical center and the literature. INTERVENTIONS: None. MAIN OUTCOME MEASURE: Auto-amputated adnexa. RESULTS: In addition to the 3 cases discussed from our institution, 91 cases of auto-amputated adnexa were identified in the literature dating back to 1943, for a total of 94 cases. Forty-nine percent (46/94) of the cases involved girls in a pediatric population (<18 years of age). Of these, the majority (n = 26) were identified in a subgroup of girls who were diagnosed with an adnexal cyst by antenatal ultrasound. Most of these neonates were asymptomatic at birth or had a palpable abdominal mass (n = 6) and at the time of surgical exploration were found to have an auto-amputated adnexa. 34 out of 46 cases were analyzed in detail. The right adnexa were involved in 56% of the cases. The most common presenting complaint verbalized by the older girls was pain; however, 8 cases were identified in asymptomatic girls undergoing unrelated diagnostic testing. CONCLUSION: The auto-amputated adnexa is a rare finding in the pediatric population, but it must be considered as a possible explanation for the incidental finding of absence of the fallopian tube or ovary in the subgroup of patients who undergo surgery for any reason. Patients with an antecedent history of pelvic pain either chronic or intermittent in nature may be diagnosed with torsion or less frequently auto-amputation of the adnexa. A fetal "pelvic mass" or "ovarian cyst" may predispose the adnexa to torsion and subsequent auto-amputation either in-utero or post-delivery. Many of these antenatally diagnosed cysts and even subsequent auto-amputations are completely asymptomatic, however, and do not compromise fertility assuming the contralateral adnexa are normal. Thus expectant management is appropriate for small (less than 4 cm), asymptomatic simple cysts and even suspected auto-amputated adnexa in an asymptomatic patient.
STUDY OBJECTIVE: To quantify our experience and that of the literature with diagnosis and management of the auto-amputated adnexa in a pediatric population. DESIGN: Case series and literature review. SETTING: Tertiary care medical center. PARTICIPANTS: Case series of pediatric patients (<18 years of age) with surgically documented adnexal auto-amputation collected from our medical center and the literature. INTERVENTIONS: None. MAIN OUTCOME MEASURE: Auto-amputated adnexa. RESULTS: In addition to the 3 cases discussed from our institution, 91 cases of auto-amputated adnexa were identified in the literature dating back to 1943, for a total of 94 cases. Forty-nine percent (46/94) of the cases involved girls in a pediatric population (<18 years of age). Of these, the majority (n = 26) were identified in a subgroup of girls who were diagnosed with an adnexal cyst by antenatal ultrasound. Most of these neonates were asymptomatic at birth or had a palpable abdominal mass (n = 6) and at the time of surgical exploration were found to have an auto-amputated adnexa. 34 out of 46 cases were analyzed in detail. The right adnexa were involved in 56% of the cases. The most common presenting complaint verbalized by the older girls was pain; however, 8 cases were identified in asymptomatic girls undergoing unrelated diagnostic testing. CONCLUSION: The auto-amputated adnexa is a rare finding in the pediatric population, but it must be considered as a possible explanation for the incidental finding of absence of the fallopian tube or ovary in the subgroup of patients who undergo surgery for any reason. Patients with an antecedent history of pelvic pain either chronic or intermittent in nature may be diagnosed with torsion or less frequently auto-amputation of the adnexa. A fetal "pelvic mass" or "ovarian cyst" may predispose the adnexa to torsion and subsequent auto-amputation either in-utero or post-delivery. Many of these antenatally diagnosed cysts and even subsequent auto-amputations are completely asymptomatic, however, and do not compromise fertility assuming the contralateral adnexa are normal. Thus expectant management is appropriate for small (less than 4 cm), asymptomatic simple cysts and even suspected auto-amputated adnexa in an asymptomatic patient.
Authors: Marjolein Husen; Pauline C Schut; Adriana C H Neven; Nagma Yousoufi; Nanko de Graaf; Cornelius E J Sloots; Alex J Eggink; Titia E Cohen-Overbeek Journal: Fetal Diagn Ther Date: 2019-01-10 Impact factor: 2.587