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Decidualized juvenile cystic adenomyoma mimicking a cornual pregnancy
Decidualized juvenile cystic adenomyoma mimicking a cornual pregnancy Huseyin Kiyak, M.D.,a Kerem Doga Seckin, M.D.,a Lale Karakis, M.D.,b Tolga Karacan, M.D.,c Eser Sefik Ozyurek, M.D.,c and Mehmet Resit Asoglu, M.D.b a
Department of Obstetrics and Gynecology, Kanuni Sultan Suleyman Research and Education Hospital, University of Health Sciences; b Department of Obstetrics and Gynecology, Bahceci Woman Health Center Reproductive Endocrinology and Infertility; and c Department of Obstetrics and Gynecology, Bagcilar Research and Education Hospital, University of Health Sciences, Istanbul, Turkey
Objective: To demonstrate the laparoscopic excision technique of a juvenile cystic adenomyoma and show how the decidualization of ectopic endometrial tissue can lead to the misdiagnosis of a focus of ectopic pregnancy. Design: Description and step-by-step demonstration of the surgical procedure using a video recording (Canadian Task Force Classification 3). Setting: Teaching and research hospital. Patient(s): A 27-year-old gravida 2 parity 1 patient with one previous caesarean delivery presented to the emergency department with symptoms of pelvic pain and delayed menses. Her beta-human chorionic gonadotropin level was 2,161 mIU/mL. On transvaginal ultrasonography the uterine cavity appeared empty without any signs of a gestational sac, and a 4245 mm heterogeneous mass was observed on the right cornual area. An 1821 mm cystic area was observed within the mass. A diagnosis of cornual pregnancy was made and two doses of systemic methotrexate treatment were administered. On the 12th day following medical treatment, the patient reported increasing abdominal pain and free fluid was observed in the pouch of Douglas on ultrasonography. The decision to perform laparoscopic cornual excision was made. Main Outcomes and Measure(s): On laparoscopic exploration a tubal ectopic pregnancy was observed within the left fallopian tube. The presence of two simultaneous ectopic pregnancies, located in the left fallopian tube and the right cornual area, was suspected. However, upon careful inspection, the right fallopian tube appeared normal and the mass initially thought to be a right cornual pregnancy appeared more like a degenerated fibroid. A left salpingectomy was performed and the excision of the mass in the right cornual area was planned. An incision was made over the mass and the cystic inner area containing chocolate-brown colored fluid was drained. As there was no pseudo capsule surrounding the mass, the diagnosis of focal adenomyosis instead of degenerated fibroid was made. No endometriotic foci were observed within the pelvis. Different from the enucleation of a fibroid, the mass was dissected from the middle into two halves until healthy myometrium was reached on the floor of the mass. The two halves of the mass were resected totally by dissecting the adenomyotic tissue from the myometrium starting from the caudal end towards the cranial end. The first layer of the remaining myometrial defect was sutured extracorporeally with No.1 polyglactan sutures. The second and third layers were sutured intracorporeally with V-loc sutures. The resected left fallopian tube containing the ectopic pregnancy and the adenomyotic mass were externalized through a posterior colpotomy incision. Result(s): The patient was discharged 24 hours postoperatively without any complications. A diagnosis of juvenile cystic adenoma was made upon histopathological examination. The patient reported subsiding of her dysmenorrhea on the postoperative third month. Conclusion(s): Juvenile cystic adenomyosis (JCA), is a rare form of focal adenomyosis which is usually located in close proximity to the uterine insertion of the round ligament, contains a cystic inner area larger than 1 cm and is encountered before the age of 30 years. Some authors reported JCA to be an accessory and cavitated uterine mass (ACUM) anomaly developing as a result of gubernaculum dysfunction. The only difference between the two conditions is reported to be the presence of a denser area of adenomyosis surrounding the cystic area lined with endometrium in JCA than in ACUM. This case has shown that decidual changes observed in ectopic endometrial tissue within an adenomyotic area may be misdiagnosed as a focus of ectopic pregnancy. Atypical endometriomas demonstrating decidual changes may also be misdiagnosed as ovarian malignancies. In
Received August 31, 2019; revised October 11, 2019; accepted October 12, 2019. H.K. has nothing to disclose. K.D.S. has nothing to disclose. L.K. has nothing to disclose. T.K., has nothing to disclose. E.S.O. has nothing to disclose. M.R.A. has nothing to disclose. Correspondence: Tolga Karacan, M.D. Department of Obstetrics and Gynecology, Bagcilar Research and Education Hospital, University of Health Sciences, Istanbul, Turkey (E-mail: [email protected]). Fertility and Sterility® Vol. 113, No. 2, February 2020 0015-0282/$36.00 Copyright ©2019 American Society for Reproductive Medicine, Published by Elsevier Inc. https://doi.org/10.1016/j.fertnstert.2019.10.026 VOL. 113 NO. 2 / FEBRUARY 2020
463
VIDEO
non-emergency situations, waiting for the decidualization effect of ectopic endometrium to subside can help in the definitive diagnosis of such cases. Our technique for JCA excision is different from enucleation of a fibroid and may aid in the total resection and dissection of the adenomyotic tissue from healthy myometrium. (Fertil SterilÒ 2020;113:463-5. Ó2019 by American Society for Reproductive Medicine.) El resumen está disponible en Español al final del artículo. Key Words: Juvenile cystic adenomyosis, ectopic pregnancy, laparoscopy, decidualization, accessory and cavitated uterine mass Discuss: You can discuss this article with its authors and other readers at https://www. fertstertdialog.com/users/16110-fertility-and-sterility/posts/54734-28891
AVAILABLE ON YOUTUBE https://youtu.be/H0yJla4sq_I
3.
SUGGESTED READING 1.
2.
464
Takeuchi H, Kitade M, Kikuchi I, Kumakiri J, Kuroda K, Jinushi M. Diagnosis, laparoscopic management, and histopathologic findings of juvenile cystic adenomyoma: a review of nine cases. Fertil Steril 2010;94:862–8. Aci en P, Bataller A, Fernandez F, Acien MI, Rodríguez JM, Mayol MJ. New cases of accessory and cavitated uterine masses (ACUM): a significant cause
4.
Use your smartphone to scan this QR code and connect to the video for this article now.*
* Download a free QR code scanner by searching for “QR scanner” in your smartphone’s app store or app marketplace.
of severe dysmenorrhea and recurrent pelvic pain in young women. Hum Reprod 2012;27:683–94. Chun SS, Hong DG, Seong WJ, Choi MH, Lee TH. Juvenile cystic adenomyoma in a 19-year-old woman: a case report with a proposal for new diagnostic criteria. J Laparoendosc Adv Surg Tech A 2011;21: 771–4. Louise HT, Thumuluru KM, Woodruff W, Karen M, Anil T, Simon BM. Decidualisation of ovarian endometriomas in pregnancy: a management dilemma. A case report and review of the literature. Arch Gynecol Obstet 2015;291: 961–8.
VOL. 113 NO. 2 / FEBRUARY 2020
Fertility and Sterility® Decidualized juvenile cystic adenomyoma mimicking a cornual pregnancy Conclusion(es): La JCA es una forma poco frecuente de adenomiosis focal que suele localizarse muy cerca de la inserci on uterina del ligamento redondo, contiene un area interna quística >1 cm3 y se encuentra antes de los 30 a~ nos de edad (1). Algunos autores han informado de que la JCA es una anomalía de la masa uterina accesoria y cavitada (ACUM) que se desarrolla como resultado de la disnica diferencia entre las dos condiciones es la presencia de un area mas densa de adenomiosis alrefunci on del gubernaculum (2). La u dedor del area quística revestida de endometrio en la JCA que en la ACUM (3). El presente caso muestra que los cambios deciduales observados en el tejido endometrial ect opico dentro de una zona adenomi otica pueden ser diagnosticados err oneamente como un foco de embarazo ect opico. Los endometriomas atípicos que demuestran cambios deciduales tambien pueden ser mal diagnosticados como malignidades ovaricas (4). En situaciones que no son de emergencia, esperar a que el efecto de decidualizaci on del endometrio ect opico disminuya puede ayudar en el diagn ostico definitivo de tales casos. Nuestra tecnica para la escisi on de JCA es diferente de la enucleaci on de un fibroma y puede ayudar en la resecci on y disecci on total del tejido adenomi otico del miometrio sano.
VOL. 113 NO. 2 / FEBRUARY 2020
465
Department of Obstetrics and Gynecology, Kanuni Sultan Suleyman Research and Education Hospital, University of Health Sciences; b Department of Obstetrics and Gynecology, Bahceci Woman Health Center Reproductive Endocrinology and Infertility; and c Department of Obstetrics and Gynecology, Bagcilar Research and Education Hospital, University of Health Sciences, Istanbul, Turkey
Objective: To demonstrate the laparoscopic excision technique of a juvenile cystic adenomyoma and show how the decidualization of ectopic endometrial tissue can lead to the misdiagnosis of a focus of ectopic pregnancy. Design: Description and step-by-step demonstration of the surgical procedure using a video recording (Canadian Task Force Classification 3). Setting: Teaching and research hospital. Patient(s): A 27-year-old gravida 2 parity 1 patient with one previous caesarean delivery presented to the emergency department with symptoms of pelvic pain and delayed menses. Her beta-human chorionic gonadotropin level was 2,161 mIU/mL. On transvaginal ultrasonography the uterine cavity appeared empty without any signs of a gestational sac, and a 4245 mm heterogeneous mass was observed on the right cornual area. An 1821 mm cystic area was observed within the mass. A diagnosis of cornual pregnancy was made and two doses of systemic methotrexate treatment were administered. On the 12th day following medical treatment, the patient reported increasing abdominal pain and free fluid was observed in the pouch of Douglas on ultrasonography. The decision to perform laparoscopic cornual excision was made. Main Outcomes and Measure(s): On laparoscopic exploration a tubal ectopic pregnancy was observed within the left fallopian tube. The presence of two simultaneous ectopic pregnancies, located in the left fallopian tube and the right cornual area, was suspected. However, upon careful inspection, the right fallopian tube appeared normal and the mass initially thought to be a right cornual pregnancy appeared more like a degenerated fibroid. A left salpingectomy was performed and the excision of the mass in the right cornual area was planned. An incision was made over the mass and the cystic inner area containing chocolate-brown colored fluid was drained. As there was no pseudo capsule surrounding the mass, the diagnosis of focal adenomyosis instead of degenerated fibroid was made. No endometriotic foci were observed within the pelvis. Different from the enucleation of a fibroid, the mass was dissected from the middle into two halves until healthy myometrium was reached on the floor of the mass. The two halves of the mass were resected totally by dissecting the adenomyotic tissue from the myometrium starting from the caudal end towards the cranial end. The first layer of the remaining myometrial defect was sutured extracorporeally with No.1 polyglactan sutures. The second and third layers were sutured intracorporeally with V-loc sutures. The resected left fallopian tube containing the ectopic pregnancy and the adenomyotic mass were externalized through a posterior colpotomy incision. Result(s): The patient was discharged 24 hours postoperatively without any complications. A diagnosis of juvenile cystic adenoma was made upon histopathological examination. The patient reported subsiding of her dysmenorrhea on the postoperative third month. Conclusion(s): Juvenile cystic adenomyosis (JCA), is a rare form of focal adenomyosis which is usually located in close proximity to the uterine insertion of the round ligament, contains a cystic inner area larger than 1 cm and is encountered before the age of 30 years. Some authors reported JCA to be an accessory and cavitated uterine mass (ACUM) anomaly developing as a result of gubernaculum dysfunction. The only difference between the two conditions is reported to be the presence of a denser area of adenomyosis surrounding the cystic area lined with endometrium in JCA than in ACUM. This case has shown that decidual changes observed in ectopic endometrial tissue within an adenomyotic area may be misdiagnosed as a focus of ectopic pregnancy. Atypical endometriomas demonstrating decidual changes may also be misdiagnosed as ovarian malignancies. In
Received August 31, 2019; revised October 11, 2019; accepted October 12, 2019. H.K. has nothing to disclose. K.D.S. has nothing to disclose. L.K. has nothing to disclose. T.K., has nothing to disclose. E.S.O. has nothing to disclose. M.R.A. has nothing to disclose. Correspondence: Tolga Karacan, M.D. Department of Obstetrics and Gynecology, Bagcilar Research and Education Hospital, University of Health Sciences, Istanbul, Turkey (E-mail: [email protected]). Fertility and Sterility® Vol. 113, No. 2, February 2020 0015-0282/$36.00 Copyright ©2019 American Society for Reproductive Medicine, Published by Elsevier Inc. https://doi.org/10.1016/j.fertnstert.2019.10.026 VOL. 113 NO. 2 / FEBRUARY 2020
463
VIDEO
non-emergency situations, waiting for the decidualization effect of ectopic endometrium to subside can help in the definitive diagnosis of such cases. Our technique for JCA excision is different from enucleation of a fibroid and may aid in the total resection and dissection of the adenomyotic tissue from healthy myometrium. (Fertil SterilÒ 2020;113:463-5. Ó2019 by American Society for Reproductive Medicine.) El resumen está disponible en Español al final del artículo. Key Words: Juvenile cystic adenomyosis, ectopic pregnancy, laparoscopy, decidualization, accessory and cavitated uterine mass Discuss: You can discuss this article with its authors and other readers at https://www. fertstertdialog.com/users/16110-fertility-and-sterility/posts/54734-28891
AVAILABLE ON YOUTUBE https://youtu.be/H0yJla4sq_I
3.
SUGGESTED READING 1.
2.
464
Takeuchi H, Kitade M, Kikuchi I, Kumakiri J, Kuroda K, Jinushi M. Diagnosis, laparoscopic management, and histopathologic findings of juvenile cystic adenomyoma: a review of nine cases. Fertil Steril 2010;94:862–8. Aci en P, Bataller A, Fernandez F, Acien MI, Rodríguez JM, Mayol MJ. New cases of accessory and cavitated uterine masses (ACUM): a significant cause
4.
Use your smartphone to scan this QR code and connect to the video for this article now.*
* Download a free QR code scanner by searching for “QR scanner” in your smartphone’s app store or app marketplace.
of severe dysmenorrhea and recurrent pelvic pain in young women. Hum Reprod 2012;27:683–94. Chun SS, Hong DG, Seong WJ, Choi MH, Lee TH. Juvenile cystic adenomyoma in a 19-year-old woman: a case report with a proposal for new diagnostic criteria. J Laparoendosc Adv Surg Tech A 2011;21: 771–4. Louise HT, Thumuluru KM, Woodruff W, Karen M, Anil T, Simon BM. Decidualisation of ovarian endometriomas in pregnancy: a management dilemma. A case report and review of the literature. Arch Gynecol Obstet 2015;291: 961–8.
VOL. 113 NO. 2 / FEBRUARY 2020
Fertility and Sterility® Decidualized juvenile cystic adenomyoma mimicking a cornual pregnancy Conclusion(es): La JCA es una forma poco frecuente de adenomiosis focal que suele localizarse muy cerca de la inserci on uterina del ligamento redondo, contiene un area interna quística >1 cm3 y se encuentra antes de los 30 a~ nos de edad (1). Algunos autores han informado de que la JCA es una anomalía de la masa uterina accesoria y cavitada (ACUM) que se desarrolla como resultado de la disnica diferencia entre las dos condiciones es la presencia de un area mas densa de adenomiosis alrefunci on del gubernaculum (2). La u dedor del area quística revestida de endometrio en la JCA que en la ACUM (3). El presente caso muestra que los cambios deciduales observados en el tejido endometrial ect opico dentro de una zona adenomi otica pueden ser diagnosticados err oneamente como un foco de embarazo ect opico. Los endometriomas atípicos que demuestran cambios deciduales tambien pueden ser mal diagnosticados como malignidades ovaricas (4). En situaciones que no son de emergencia, esperar a que el efecto de decidualizaci on del endometrio ect opico disminuya puede ayudar en el diagn ostico definitivo de tales casos. Nuestra tecnica para la escisi on de JCA es diferente de la enucleaci on de un fibroma y puede ayudar en la resecci on y disecci on total del tejido adenomi otico del miometrio sano.
VOL. 113 NO. 2 / FEBRUARY 2020
465