Journal of Obstetrics and Gynaecology, 2015; Early Online: 1–2 © 2015 Informa UK, Ltd. ISSN 0144-3615 print/ISSN 1364-6893 online
CASE REPORT
Torsed large prenatally detected foetal ovarian cyst
necrosis and the follicular cyst (Figure 1). There was no suspicion of malignancy.
Discussion N. Antonakopoulos, D. Agrapidis, G. Karras, K. Stefanidis & D. Loutradis Department of Obstetrics and Gynecology, University of Athens Medical School, Alexandra Maternity Hospital, Athens, Greece
J Obstet Gynaecol Downloaded from informahealthcare.com by Nyu Medical Center on 06/25/15 For personal use only.
DOI: 10.3109/01443615.2015.1009877 Correspondence: Antonakopoulos Nikolaos, 50-52 Mikras Asias Str, Vyronas 16231, Athens, Greece. E-mail: [email protected]
Introduction The incidence of foetal ovarian cysts detected in utero has increased due to excessive use of ultrasound. Ovarian torsion represents the most common complication. Early diagnosis of ovarian torsion is critical in avoiding complications and planning management. Magnetic resonance imaging is a better imaging modality to evaluate to arrive at a more definitive diagnosis. We present a case of a foetus with a large ovarian cyst detected during prenatal examinations at 24 weeks’ gestation.
Case report A 34-year-old G3P2 pregnant woman at 31 weeks’ gestation was admitted to Alexandra maternity hospital with the diagnosis of a large foetal ovarian cyst. At 24 weeks’ gestation sonographic examinations revealed a cyst measuring 8 ⫻ 7 ⫻ 4.6 cm. The differential diagnosis was mesenteric or ovarian cyst. Magnetic resonance imaging (MRI) advocated for an ovarian cyst (Figure 1). Our monitoring strategy employed ultrasound (US) scans every week, until 28 weeks’ gestation, and twice a week, along with non-stress test (NST), later on. At 33 weeks’ gestation the pregnant woman underwent an emergency caesarean section, because cyst’s size was found to be 1.5 times larger than the one of initial diagnosis, during regular US scans, and there was a suspicion of torsion. Caesarean section was preferred in order to avoid dystocia due to cyst size or rupture of the cyst during vaginal delivery. Cervix evaluation anticipated a poor response to induction of labour (Bishop score ⬍ 6), so induction was not preferred under the suspicion of torsion. Corticosteroids were administered before the caesarean section. On the day of birth the cyst of the right ovary was surgically removed under general anaesthesia. A large ovarian cyst of the right ovary was identified with haemorrhagic fluid provoking ovarian torsion. Unilateral oophorectomy was performed. Histopathological examination revealed an extensive haemorrhagic
Female foetuses frequently present with abdominal cystic formations without urinary or intestinal findings. These tend to have a frequency of 1/500 up to 1/1000 live births with a prevalence of ovarian cysts (Chandler and Gauderer 2004). Functional cysts have a benign origin that classifies them into follicular, corpus luteum and theca lutein types (Bryant and Laufer 2004). Rarely tumours in foetal ovaries such as benign cystic teratomas and lymphangiomas have been reported, whereas malignancies with ovarian carcinoma have been reported only once (Park et al. 2005). In most cases these functional cysts can be easily diagnosed during the third trimester of pregnancy by a typical US scan. Foetal ovarian cysts are pathologically classified into uncomplicated functional cysts, haemorrhagic functional cysts, torsed functional cysts and others (Monnery-Noché et al. 2008). Nussbaum et al. sonographically divided them into two groups. Simple cysts have thin walls and an anechoic content, while complex ones acknowledge with specific findings such as partially anechoic with debris or retracting blood clot or with sediments or solid (Nussbaum et al. 1988). In our case a haemorrhagic functional, follicular type, cyst was diagnosed and both MRI and US scan showed a literally thin wall, partially anechoic with debris and lack of vascularisation. Follicular cysts increase with advanced gestational age and grow in size under the continuous influence of maternal oestrogens, beta human chorionic gonadotropin and in coexistence with complicated pregnancies. Moreover, preterm neonates have immature gonadostat mechanisms that hyperstimulate their ovaries. Congenital adrenal hyperplasia leads to an excess of adrenal androgens that either increase follicle-stimulating hormone receptors or interfere with the cyclicity of gonadotropin release (Zampieri et al. 2008). Surprisingly our patient presented none of the above risk factors, not to mention the fact that the newborn was screened for endocrinology disorders without any abnormal findings. The differential diagnosis involves a broad spectrum of various cystic formations: omental, mesenteric, urachal, choledochal, intestinal and renal cysts, cystic meconium peritonitis, duodenal atresia and anterior meningocele (Kwak et al. 2006). Simple cysts ⬍ 5 cm generally do not require medical assistance, as they tend to withdraw early in neonatal life. Some cysts grow and acquire complex features leading either to haemorrhage or ovarian torsion in postnatal life. Such infants present with pain, fever, vomit, difficulty in feeding and abdominal distention. Anaemia due to intracystic haemorrhage, tachycardia and peritonitis may occur. Large cysts can be responsible for intestinal and urinary obstruction along with pressure phenomena to neighbour organs, causing pulmonary
Figure 1. The foetal ovarian cyst MRI imaging and the histopathology view.
J Obstet Gynaecol Downloaded from informahealthcare.com by Nyu Medical Center on 06/25/15 For personal use only.
2
N. Antonakopoulos et al.
hypoplasia and polyhydramnios (Zampieri et al. 2008). The most common complication of a large simple cyst is ovarian torsion. This leads to necrosis and intracystic haemorrhage or vice versa. After birth if a cyst does not regress, surgical intervention is suggested to avoid torsion and haemorrhage. Mode of delivery regardless of the size of a simple cyst is vaginal, unless an obstetric indication is present (Shimada et al. 2008). In some centres US-guided cyst aspiration is offered to reduce the size of simple cysts ⬎ 4 cm (Kessler et al. 2006). In our case due to the size of the cyst and with the potential of ovarian torsion in mind, as it was depicted in the US scans afterbirth showing intracystic haemorrhage signs, a surgical intervention was offered with mother’s consent. Complex cysts always require surgical inquiry because they are highly suggestive of torsion. The ovarian torsion leads to further complications such as rupture and haemoperitoneum, ascites, intestinal and urinary obstruction, compression of vena cava, autoamputation and finally ovarian loss (Schenkman et al. 2008). MRI can aid in the proper diagnosis. Doppler is considered by many as the method of choice for imaging of adnexal structures for blood flow. It is also true that it is less expensive than MRI. But, although it has a high positive predictive value, its negative predictive value is very low. This was the case in our patient. Doppler was performed, blood flow was not noticed and proceeded to MRI for more information. Mode of delivery is preterm birth after ensuring lung maturity (Conforti et al. 2009). Surgical approach generally divides into mini laparotomy procedures such as cystectomy, salpingo-oophorectomy and laparoscopy. The latter has all the advantages of a minimally invasion procedure, allowing aspiration, stripping and decapsulation of the cyst (Kuebler and Ure 2011). Unfortunately, in our case, due to lack of experience and sheer size a mini laparotomy was conducted in order to perform the oophorectomy. In conclusion, foetal abdominal cysts can provoke life-threatening complications. Early diagnosis of ovarian torsion is critical in avoiding complications and planning management. MRI is a better imaging modality to arrive at a more definitive diagnosis. Medical
role is to ensure the foetus well-being by either monitoring the cysts or intervening surgically in order to protect the ovaries and to ensure fertility. We propose weekly scans until 28 weeks’ gestation and twice weekly along with NST later on. Declaration of interest: The authors report no declarations of interest. The authors alone are responsible for the content and writing of the paper.
References Bryant AE, Laufer MR. 2004. Fetal ovarian cysts: incidence, diagnosis and management. Journal of Reproductive Medicine 49:329–337. Chandler JC, Gauderer MW. 2004. The neonate with an abdominal mass. Pediatric Clinics of North America 51:979–997. Conforti A, Giorlandino C, Bagolan P. 2009. Fetal ovarian cysts management and ovarian prognosis: a report of 82 cases. Journal of Pediatric Surgery 44:868; author reply 868–869. Kessler A, Nagar H, Graif M et al. 2006. Percutaneous drainage as the treatment of choice for neonatal ovarian cysts. Pediatric Radiology 36:954–958. Kuebler JF, Ure BM. 2011. Minimally invasive surgery in the neonate. Seminars in Fetal & Neonatal Medicine 16:151–156. Kwak DW, Sohn YS, Kim SK et al. 2006. Clinical experiences of fetal ovarian cyst: diagnosis and consequence. Journal of Korean Medical Science 21: 690–694. Monnery-Noché ME, Auber F, Jouannic JM et al. 2008. Fetal and neonatal ovarian cysts: is surgery indicated? Prenatal Diagnosis 28:15–20. Nussbaum AR, Sanders RC, Hartman DS et al. 1988. Neonatal ovarian cysts: sonographic-pathologic correlation. Radiology 168:817–821. Park C, Lee JW, Kim SJ et al. 2005. Sonographic findings of prenatal torsion of ovarian lymphangioma. Journal of Clinical Ultrasound 33:421–423. Schenkman L, Weiner TM, Phillips JD. 2008. Evolution of the surgical management of neonatal ovarian cysts: laparoscopic-assisted transumbilical extracorporeal ovarian cystectomy (LATEC). Journal Laparoendoscopic & Advanced Surgical Techniques Part A. 18:635–640. Shimada T, Miura K, Gotoh H et al. 2008. Management of prenatal ovarian cysts. Early Human Development 84:417–420. Zampieri N, Borruto F, Zamboni C et al. 2008. Foetal and neonatal ovarian cysts: a 5-year experience. Archives of Gynecology & Obstetrics 277:303–306.
CASE REPORT
Torsed large prenatally detected foetal ovarian cyst
necrosis and the follicular cyst (Figure 1). There was no suspicion of malignancy.
Discussion N. Antonakopoulos, D. Agrapidis, G. Karras, K. Stefanidis & D. Loutradis Department of Obstetrics and Gynecology, University of Athens Medical School, Alexandra Maternity Hospital, Athens, Greece
J Obstet Gynaecol Downloaded from informahealthcare.com by Nyu Medical Center on 06/25/15 For personal use only.
DOI: 10.3109/01443615.2015.1009877 Correspondence: Antonakopoulos Nikolaos, 50-52 Mikras Asias Str, Vyronas 16231, Athens, Greece. E-mail: [email protected]
Introduction The incidence of foetal ovarian cysts detected in utero has increased due to excessive use of ultrasound. Ovarian torsion represents the most common complication. Early diagnosis of ovarian torsion is critical in avoiding complications and planning management. Magnetic resonance imaging is a better imaging modality to evaluate to arrive at a more definitive diagnosis. We present a case of a foetus with a large ovarian cyst detected during prenatal examinations at 24 weeks’ gestation.
Case report A 34-year-old G3P2 pregnant woman at 31 weeks’ gestation was admitted to Alexandra maternity hospital with the diagnosis of a large foetal ovarian cyst. At 24 weeks’ gestation sonographic examinations revealed a cyst measuring 8 ⫻ 7 ⫻ 4.6 cm. The differential diagnosis was mesenteric or ovarian cyst. Magnetic resonance imaging (MRI) advocated for an ovarian cyst (Figure 1). Our monitoring strategy employed ultrasound (US) scans every week, until 28 weeks’ gestation, and twice a week, along with non-stress test (NST), later on. At 33 weeks’ gestation the pregnant woman underwent an emergency caesarean section, because cyst’s size was found to be 1.5 times larger than the one of initial diagnosis, during regular US scans, and there was a suspicion of torsion. Caesarean section was preferred in order to avoid dystocia due to cyst size or rupture of the cyst during vaginal delivery. Cervix evaluation anticipated a poor response to induction of labour (Bishop score ⬍ 6), so induction was not preferred under the suspicion of torsion. Corticosteroids were administered before the caesarean section. On the day of birth the cyst of the right ovary was surgically removed under general anaesthesia. A large ovarian cyst of the right ovary was identified with haemorrhagic fluid provoking ovarian torsion. Unilateral oophorectomy was performed. Histopathological examination revealed an extensive haemorrhagic
Female foetuses frequently present with abdominal cystic formations without urinary or intestinal findings. These tend to have a frequency of 1/500 up to 1/1000 live births with a prevalence of ovarian cysts (Chandler and Gauderer 2004). Functional cysts have a benign origin that classifies them into follicular, corpus luteum and theca lutein types (Bryant and Laufer 2004). Rarely tumours in foetal ovaries such as benign cystic teratomas and lymphangiomas have been reported, whereas malignancies with ovarian carcinoma have been reported only once (Park et al. 2005). In most cases these functional cysts can be easily diagnosed during the third trimester of pregnancy by a typical US scan. Foetal ovarian cysts are pathologically classified into uncomplicated functional cysts, haemorrhagic functional cysts, torsed functional cysts and others (Monnery-Noché et al. 2008). Nussbaum et al. sonographically divided them into two groups. Simple cysts have thin walls and an anechoic content, while complex ones acknowledge with specific findings such as partially anechoic with debris or retracting blood clot or with sediments or solid (Nussbaum et al. 1988). In our case a haemorrhagic functional, follicular type, cyst was diagnosed and both MRI and US scan showed a literally thin wall, partially anechoic with debris and lack of vascularisation. Follicular cysts increase with advanced gestational age and grow in size under the continuous influence of maternal oestrogens, beta human chorionic gonadotropin and in coexistence with complicated pregnancies. Moreover, preterm neonates have immature gonadostat mechanisms that hyperstimulate their ovaries. Congenital adrenal hyperplasia leads to an excess of adrenal androgens that either increase follicle-stimulating hormone receptors or interfere with the cyclicity of gonadotropin release (Zampieri et al. 2008). Surprisingly our patient presented none of the above risk factors, not to mention the fact that the newborn was screened for endocrinology disorders without any abnormal findings. The differential diagnosis involves a broad spectrum of various cystic formations: omental, mesenteric, urachal, choledochal, intestinal and renal cysts, cystic meconium peritonitis, duodenal atresia and anterior meningocele (Kwak et al. 2006). Simple cysts ⬍ 5 cm generally do not require medical assistance, as they tend to withdraw early in neonatal life. Some cysts grow and acquire complex features leading either to haemorrhage or ovarian torsion in postnatal life. Such infants present with pain, fever, vomit, difficulty in feeding and abdominal distention. Anaemia due to intracystic haemorrhage, tachycardia and peritonitis may occur. Large cysts can be responsible for intestinal and urinary obstruction along with pressure phenomena to neighbour organs, causing pulmonary
Figure 1. The foetal ovarian cyst MRI imaging and the histopathology view.
J Obstet Gynaecol Downloaded from informahealthcare.com by Nyu Medical Center on 06/25/15 For personal use only.
2
N. Antonakopoulos et al.
hypoplasia and polyhydramnios (Zampieri et al. 2008). The most common complication of a large simple cyst is ovarian torsion. This leads to necrosis and intracystic haemorrhage or vice versa. After birth if a cyst does not regress, surgical intervention is suggested to avoid torsion and haemorrhage. Mode of delivery regardless of the size of a simple cyst is vaginal, unless an obstetric indication is present (Shimada et al. 2008). In some centres US-guided cyst aspiration is offered to reduce the size of simple cysts ⬎ 4 cm (Kessler et al. 2006). In our case due to the size of the cyst and with the potential of ovarian torsion in mind, as it was depicted in the US scans afterbirth showing intracystic haemorrhage signs, a surgical intervention was offered with mother’s consent. Complex cysts always require surgical inquiry because they are highly suggestive of torsion. The ovarian torsion leads to further complications such as rupture and haemoperitoneum, ascites, intestinal and urinary obstruction, compression of vena cava, autoamputation and finally ovarian loss (Schenkman et al. 2008). MRI can aid in the proper diagnosis. Doppler is considered by many as the method of choice for imaging of adnexal structures for blood flow. It is also true that it is less expensive than MRI. But, although it has a high positive predictive value, its negative predictive value is very low. This was the case in our patient. Doppler was performed, blood flow was not noticed and proceeded to MRI for more information. Mode of delivery is preterm birth after ensuring lung maturity (Conforti et al. 2009). Surgical approach generally divides into mini laparotomy procedures such as cystectomy, salpingo-oophorectomy and laparoscopy. The latter has all the advantages of a minimally invasion procedure, allowing aspiration, stripping and decapsulation of the cyst (Kuebler and Ure 2011). Unfortunately, in our case, due to lack of experience and sheer size a mini laparotomy was conducted in order to perform the oophorectomy. In conclusion, foetal abdominal cysts can provoke life-threatening complications. Early diagnosis of ovarian torsion is critical in avoiding complications and planning management. MRI is a better imaging modality to arrive at a more definitive diagnosis. Medical
role is to ensure the foetus well-being by either monitoring the cysts or intervening surgically in order to protect the ovaries and to ensure fertility. We propose weekly scans until 28 weeks’ gestation and twice weekly along with NST later on. Declaration of interest: The authors report no declarations of interest. The authors alone are responsible for the content and writing of the paper.
References Bryant AE, Laufer MR. 2004. Fetal ovarian cysts: incidence, diagnosis and management. Journal of Reproductive Medicine 49:329–337. Chandler JC, Gauderer MW. 2004. The neonate with an abdominal mass. Pediatric Clinics of North America 51:979–997. Conforti A, Giorlandino C, Bagolan P. 2009. Fetal ovarian cysts management and ovarian prognosis: a report of 82 cases. Journal of Pediatric Surgery 44:868; author reply 868–869. Kessler A, Nagar H, Graif M et al. 2006. Percutaneous drainage as the treatment of choice for neonatal ovarian cysts. Pediatric Radiology 36:954–958. Kuebler JF, Ure BM. 2011. Minimally invasive surgery in the neonate. Seminars in Fetal & Neonatal Medicine 16:151–156. Kwak DW, Sohn YS, Kim SK et al. 2006. Clinical experiences of fetal ovarian cyst: diagnosis and consequence. Journal of Korean Medical Science 21: 690–694. Monnery-Noché ME, Auber F, Jouannic JM et al. 2008. Fetal and neonatal ovarian cysts: is surgery indicated? Prenatal Diagnosis 28:15–20. Nussbaum AR, Sanders RC, Hartman DS et al. 1988. Neonatal ovarian cysts: sonographic-pathologic correlation. Radiology 168:817–821. Park C, Lee JW, Kim SJ et al. 2005. Sonographic findings of prenatal torsion of ovarian lymphangioma. Journal of Clinical Ultrasound 33:421–423. Schenkman L, Weiner TM, Phillips JD. 2008. Evolution of the surgical management of neonatal ovarian cysts: laparoscopic-assisted transumbilical extracorporeal ovarian cystectomy (LATEC). Journal Laparoendoscopic & Advanced Surgical Techniques Part A. 18:635–640. Shimada T, Miura K, Gotoh H et al. 2008. Management of prenatal ovarian cysts. Early Human Development 84:417–420. Zampieri N, Borruto F, Zamboni C et al. 2008. Foetal and neonatal ovarian cysts: a 5-year experience. Archives of Gynecology & Obstetrics 277:303–306.
