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Embolization of a uterine arteriovenous malformation followed by a twin pregnancy
Embolization of a Uterine Arteriovenous Malformation Followed by a Twin Pregnancy Manish Gopal, MD, Jay Goldberg, MD, Thomas A. Klein, MD, and Gregory T. Fossum, MD Department of Obstetrics and Gynecology, Thomas Jefferson University Hospital, Philadelphia, Pennsylvania
BACKGROUND: Uterine arteriovenous malformation is a lesion that can cause recurrent pregnancy loss. Very few successful pregnancies have been reported after selective embolization of an arteriovenous malformation. CASE: A 42-year-old gravida 6 para 0 with a history of four first-trimester spontaneous abortions was found to have a uterine arteriovenous malformation by hysterosalpingogram, which was confirmed by magnetic resonance angiography. She underwent selective transcatheter embolization of the arteriovenous malformation. A twin gestation was later conceived via in vitro fertilization. The twins were delivered by cesarean at term. CONCLUSION: Uterine arteriovenous malformation can be successfully treated with selective embolization, with good obstetric outcome. (Obstet Gynecol 2003;102:696 – 8. © 2003 by The American College of Obstetricians and Gynecologists.)
Uterine arteriovenous malformation is a rare cause of recurrent pregnancy wastage. One can hypothesize that this might be attributed to the hypervascularization that occurs in the presence of a uterine arteriovenous malformation resulting in the physical alteration of the embryo’s implantation site. A successful pregnancy after treatment of a uterine arteriovenous malformation is even more uncommon. We report a selective embolization of a large arteriovenous malformation in a patient with recurrent first-trimester pregnancy loss, followed by a successful twin pregnancy.
CASE A 42 year old, gravida 6, para 0, presented with a history of one first-trimester therapeutic abortion and four subAddress reprint requests to: Manish Gopal, MD, Thomas Jefferson University Hospital, Department of Obstetrics and Gynecology, 834 Chestnut Street, Suite 400, Philadelphia, PA 19107; E-mail: [email protected]. The authors thank Joseph Bonn, MD, and Michael Sobel, DO, for providing clinical information and valuable suggestions regarding this case.
696
sequent first-trimester pregnancy losses. The patient’s first loss was an incomplete abortion, and subsequent losses were due to embryonic demise. Each of the four spontaneous losses was followed by dilation and curettage. She had normal, regular menses. A diagnostic evaluation was initiated, including a normal thrombophilia and karyotype evaluation. An endometrial biopsy showed simple hyperplasia without atypia. Her uterine cavity was then evaluated by a hysterosalpingogram, which revealed a large, left-sided uterine cavity deformity, initially thought to be secondary to a uterine leiomyoma, along with vascular intravasation (Figure 1). The left fallopian tube was patent, despite appearing continuous with the uterine malformation. On magnetic resonance imaging and magnetic resonance angiography, the left upper uterine body and fundus were found to be occupied by a heterogeneous transmural and submucosal mass, measuring 4.9 cm ⫻ 4.3 cm ⫻ 4.0 cm. The prominent flow void and arterial enhancement were consistent with an arteriovenous malformation (Figure 2). The patient underwent a selective transcatheter embolization of the uterine arteriovenous malformation. The arteriovenous malformation was supplied solely by the left uterine artery, which was embolized with 500 – 700- Embosphere particles (Biosphere Medical, Rockland, MA), followed by a gelfoam suspension. After failing to conceive by means of intrauterine insemination and ovarian hyperstimulation after the embolization, hysteroscopy was performed along with a diagnostic laparoscopy to further evaluate her infertility. The hysteroscopy showed that the uterine cavity was normal in appearance, without any signs of a defect from the embolized arteriovenous malformation. The diagnostic laparoscopy was unremarkable. After completion of multiple cycles of ovulation induction with clomiphene citrate and subsequently with exogenous gonadotropins with intrauterine insemination without conception, she underwent in vitro fertilization with uterine embryo transfer. Luteal-phase pituitary down regulation with a gonadotropin-releasing hormone agonist was done, followed by 10 days of gonadotropin ovarian hyperstimulation. Ovum retrieval was timed to occur 34 hours after intramuscular administration of 10,000 IU of human chorionic gonadotropin. Ten oocytes were aspirated. Intracytoplasmic sperm injection was performed and yielded six fertilized eggs. A total of four embryos were transferred after 3 days of culture. Cleaved embryos (two eight-cell and two seven-cell) were transferred under transabdominal ultrasound guidance. The patient subsequently was found to have a diamniotic dichorionic twin intrauterine gestation. The patient’s antenatal course was uncomplicated. She underwent monthly ultrasound examination for
VOL. 102, NO. 4, OCTOBER 2003 © 2003 by The American College of Obstetricians and Gynecologists. Published by Elsevier.
0029-7844/03/$30.00 doi:10.1016/S0029-7844(03)00714-2
Figure 1. Hysterosalpingogram depicting the point of dye intravasation (arrow) and the area of uterine wall irregularity (double arrow). Gopal. Uterine AVM Embolization Followed by Twin Pregnancy. Obstet Gynecol 2003.
growth, which were appropriate for gestational age. At 37 weeks’ gestation, she underwent a low transverse cesarean delivery after a failed induction of labor. The twins, a 3081-g male and a 2935-g female, had Apgar scores of 5 and 9 and 9 and 9, respectively, at 1 and 5 minutes. Normal adnexa and uterus were noted, with no apparent residual from the arteriovenous malformation. Excessive bleeding did not occur at the time of cesarean. The patient and the twins did well, being discharged to home on postoperative day 4. COMMENT An arteriovenous malformation consists of a proliferation of arterial and venous channels of varying sizes with fistula formation along with small capillary-like channels. This aberrant vasculature has also been referred to as a pulsating angioma, arteriovenous aneurysm or fistula, cirsoid or racemose aneurysm, and a cavernous hemangioma. The causes of arteriovenous malformations are varied. They can be divided into two main categories: congenital or acquired. Whereas congenital arteriovenous malformations result from arrest in the development of the vasculature during embryologic develop-
VOL. 102, NO. 4, OCTOBER 2003
ment, the putative causes of acquired arteriovenous malformations are many. These include malignancies, such as endometrial carcinoma and gestational trophoblastic disease, trauma from repeated dilation and curettage, surgery, infection, and diethylstilbestrol exposure.1 Clinically, uterine arteriovenous malformation presents with menorrhagia or metrorrhagia, which at times can be severe. The diagnosis might also be suspected in any patient with a history of repeated pregnancy losses and prolonged bleeding after dilation and curettage.2 Typically, arteriovenous malformations are found in women of childbearing age. However, other uncommon presentations include postmenopausal bleeding and congestive heart failure as a result of vascular steal syndrome.3 The “gold standard” of diagnosis is pelvic angiography; however, ultrasound, Doppler studies, hysterosalpingogram, computed tomography, and magnetic resonance imaging have also been described.4 Historically, the treatment of choice for uterine arteriovenous malformation was abdominal hysterectomy. With the advancement of interventional radiology, selective embolization of the arteriovenous malformation has become the preferred therapy.5 Successful pregnancy outcomes despite the presence of uterine arterio-
Gopal et al
Uterine AVM Embolization Followed by Twin Pregnancy
697
munication in the vasculature during the prior pregnancy losses.6 This is a rare case of a successful twin birth after embolization of a uterine arteriovenous malformation. Notably, the twins were born without any evidence of growth restriction. This is contrary to what one might potentially expect in a pregnancy with a multiple gestation after an embolization of a uterine arteriovenous malformation. When a patient with arteriovenous malformation desires future fertility, selective uterine embolization should be considered.
Figure 2. Magnetic resonance angiography showing a 4.9 cm ⫻ 4.3 cm ⫻ 4.0 cm heterogeneous mass (arrow), representing an arteriovenous malformation. Gopal. Uterine AVM Embolization Followed by Twin Pregnancy. Obstet Gynecol 2003.
venous malformations have occurred with expectant management and ergotamine therapy.6,7 Five successful pregnancies after selective embolization of a uterine arteriovenous malformation have also been reported.8 There are no data available to guide recommendations as to how long pregnancy attempts should be deferred after embolization. In our case, it is believed that the patient’s arteriovenous malformation was congenital or the result of prior dilation and curettage procedures. Although unlikely, it is also possible that the malformation could have resulted from chorionic villi implantation causing com-
698
Gopal et al
Uterine AVM Embolization Followed by Twin Pregnancy
REFERENCES 1. Soberon FA, Loret de Mola JR, Goldberg RS, Tureck RW. Uterine arteriovenous malformation in a patient with recurrent pregnancy loss and a bicornuate uterus. J Reprod Med 1997;42:239–43. 2. Fleming H, Ostor AG, Pickel H, Fortune DW. Arteriovenous malformations of the uterus. Obstet Gynecol 1989; 73:209–13. 3. Hoffman MK, Meilstrup JW, Shackelford DP, Kaminski PF. Arteriovenous malformations of the uterus: An uncommon cause of vaginal bleeding. Obstet Gynecol Surv 1997; 52:736–40. 4. Chow TWP, Nwosu EC, Gould DA, Richmond DH. Pregnancy following successful embolisation of a uterine vascular malformation. Br J Obstet Gynaecol 1995;102:166–8. 5. Wilms GE, Favril A, Baert AL, Poppe W, Van Assche FA. Transcatheter embolization of uterine arteriovenous malformations. Cardiovasc Intervent Radiol 1986;9:61–4. 6. Goldberg RP, Flynn MK. Pregnancy after medical management of a uterine arteriovenous malformation. J Reprod Med 2000;45:961–3. 7. Onoyama I, Fukuhara M, Okuma A, Watanabe Y, Nakanura G. Successful pregnancy after the noninvasive management of uterine arteriovenous malformation. Acta Obstet Gynecol Scand 2001;80:1148–9. 8. Kelly FW. Forceps delivery after molar malignancy in a woman with arteriovenous malformation. J Reprod Med 2001;46:1013–6. Received March 31, 2003. Received in revised form May 22, 2003. Accepted June 5, 2003.
OBSTETRICS & GYNECOLOGY
BACKGROUND: Uterine arteriovenous malformation is a lesion that can cause recurrent pregnancy loss. Very few successful pregnancies have been reported after selective embolization of an arteriovenous malformation. CASE: A 42-year-old gravida 6 para 0 with a history of four first-trimester spontaneous abortions was found to have a uterine arteriovenous malformation by hysterosalpingogram, which was confirmed by magnetic resonance angiography. She underwent selective transcatheter embolization of the arteriovenous malformation. A twin gestation was later conceived via in vitro fertilization. The twins were delivered by cesarean at term. CONCLUSION: Uterine arteriovenous malformation can be successfully treated with selective embolization, with good obstetric outcome. (Obstet Gynecol 2003;102:696 – 8. © 2003 by The American College of Obstetricians and Gynecologists.)
Uterine arteriovenous malformation is a rare cause of recurrent pregnancy wastage. One can hypothesize that this might be attributed to the hypervascularization that occurs in the presence of a uterine arteriovenous malformation resulting in the physical alteration of the embryo’s implantation site. A successful pregnancy after treatment of a uterine arteriovenous malformation is even more uncommon. We report a selective embolization of a large arteriovenous malformation in a patient with recurrent first-trimester pregnancy loss, followed by a successful twin pregnancy.
CASE A 42 year old, gravida 6, para 0, presented with a history of one first-trimester therapeutic abortion and four subAddress reprint requests to: Manish Gopal, MD, Thomas Jefferson University Hospital, Department of Obstetrics and Gynecology, 834 Chestnut Street, Suite 400, Philadelphia, PA 19107; E-mail: [email protected]. The authors thank Joseph Bonn, MD, and Michael Sobel, DO, for providing clinical information and valuable suggestions regarding this case.
696
sequent first-trimester pregnancy losses. The patient’s first loss was an incomplete abortion, and subsequent losses were due to embryonic demise. Each of the four spontaneous losses was followed by dilation and curettage. She had normal, regular menses. A diagnostic evaluation was initiated, including a normal thrombophilia and karyotype evaluation. An endometrial biopsy showed simple hyperplasia without atypia. Her uterine cavity was then evaluated by a hysterosalpingogram, which revealed a large, left-sided uterine cavity deformity, initially thought to be secondary to a uterine leiomyoma, along with vascular intravasation (Figure 1). The left fallopian tube was patent, despite appearing continuous with the uterine malformation. On magnetic resonance imaging and magnetic resonance angiography, the left upper uterine body and fundus were found to be occupied by a heterogeneous transmural and submucosal mass, measuring 4.9 cm ⫻ 4.3 cm ⫻ 4.0 cm. The prominent flow void and arterial enhancement were consistent with an arteriovenous malformation (Figure 2). The patient underwent a selective transcatheter embolization of the uterine arteriovenous malformation. The arteriovenous malformation was supplied solely by the left uterine artery, which was embolized with 500 – 700- Embosphere particles (Biosphere Medical, Rockland, MA), followed by a gelfoam suspension. After failing to conceive by means of intrauterine insemination and ovarian hyperstimulation after the embolization, hysteroscopy was performed along with a diagnostic laparoscopy to further evaluate her infertility. The hysteroscopy showed that the uterine cavity was normal in appearance, without any signs of a defect from the embolized arteriovenous malformation. The diagnostic laparoscopy was unremarkable. After completion of multiple cycles of ovulation induction with clomiphene citrate and subsequently with exogenous gonadotropins with intrauterine insemination without conception, she underwent in vitro fertilization with uterine embryo transfer. Luteal-phase pituitary down regulation with a gonadotropin-releasing hormone agonist was done, followed by 10 days of gonadotropin ovarian hyperstimulation. Ovum retrieval was timed to occur 34 hours after intramuscular administration of 10,000 IU of human chorionic gonadotropin. Ten oocytes were aspirated. Intracytoplasmic sperm injection was performed and yielded six fertilized eggs. A total of four embryos were transferred after 3 days of culture. Cleaved embryos (two eight-cell and two seven-cell) were transferred under transabdominal ultrasound guidance. The patient subsequently was found to have a diamniotic dichorionic twin intrauterine gestation. The patient’s antenatal course was uncomplicated. She underwent monthly ultrasound examination for
VOL. 102, NO. 4, OCTOBER 2003 © 2003 by The American College of Obstetricians and Gynecologists. Published by Elsevier.
0029-7844/03/$30.00 doi:10.1016/S0029-7844(03)00714-2
Figure 1. Hysterosalpingogram depicting the point of dye intravasation (arrow) and the area of uterine wall irregularity (double arrow). Gopal. Uterine AVM Embolization Followed by Twin Pregnancy. Obstet Gynecol 2003.
growth, which were appropriate for gestational age. At 37 weeks’ gestation, she underwent a low transverse cesarean delivery after a failed induction of labor. The twins, a 3081-g male and a 2935-g female, had Apgar scores of 5 and 9 and 9 and 9, respectively, at 1 and 5 minutes. Normal adnexa and uterus were noted, with no apparent residual from the arteriovenous malformation. Excessive bleeding did not occur at the time of cesarean. The patient and the twins did well, being discharged to home on postoperative day 4. COMMENT An arteriovenous malformation consists of a proliferation of arterial and venous channels of varying sizes with fistula formation along with small capillary-like channels. This aberrant vasculature has also been referred to as a pulsating angioma, arteriovenous aneurysm or fistula, cirsoid or racemose aneurysm, and a cavernous hemangioma. The causes of arteriovenous malformations are varied. They can be divided into two main categories: congenital or acquired. Whereas congenital arteriovenous malformations result from arrest in the development of the vasculature during embryologic develop-
VOL. 102, NO. 4, OCTOBER 2003
ment, the putative causes of acquired arteriovenous malformations are many. These include malignancies, such as endometrial carcinoma and gestational trophoblastic disease, trauma from repeated dilation and curettage, surgery, infection, and diethylstilbestrol exposure.1 Clinically, uterine arteriovenous malformation presents with menorrhagia or metrorrhagia, which at times can be severe. The diagnosis might also be suspected in any patient with a history of repeated pregnancy losses and prolonged bleeding after dilation and curettage.2 Typically, arteriovenous malformations are found in women of childbearing age. However, other uncommon presentations include postmenopausal bleeding and congestive heart failure as a result of vascular steal syndrome.3 The “gold standard” of diagnosis is pelvic angiography; however, ultrasound, Doppler studies, hysterosalpingogram, computed tomography, and magnetic resonance imaging have also been described.4 Historically, the treatment of choice for uterine arteriovenous malformation was abdominal hysterectomy. With the advancement of interventional radiology, selective embolization of the arteriovenous malformation has become the preferred therapy.5 Successful pregnancy outcomes despite the presence of uterine arterio-
Gopal et al
Uterine AVM Embolization Followed by Twin Pregnancy
697
munication in the vasculature during the prior pregnancy losses.6 This is a rare case of a successful twin birth after embolization of a uterine arteriovenous malformation. Notably, the twins were born without any evidence of growth restriction. This is contrary to what one might potentially expect in a pregnancy with a multiple gestation after an embolization of a uterine arteriovenous malformation. When a patient with arteriovenous malformation desires future fertility, selective uterine embolization should be considered.
Figure 2. Magnetic resonance angiography showing a 4.9 cm ⫻ 4.3 cm ⫻ 4.0 cm heterogeneous mass (arrow), representing an arteriovenous malformation. Gopal. Uterine AVM Embolization Followed by Twin Pregnancy. Obstet Gynecol 2003.
venous malformations have occurred with expectant management and ergotamine therapy.6,7 Five successful pregnancies after selective embolization of a uterine arteriovenous malformation have also been reported.8 There are no data available to guide recommendations as to how long pregnancy attempts should be deferred after embolization. In our case, it is believed that the patient’s arteriovenous malformation was congenital or the result of prior dilation and curettage procedures. Although unlikely, it is also possible that the malformation could have resulted from chorionic villi implantation causing com-
698
Gopal et al
Uterine AVM Embolization Followed by Twin Pregnancy
REFERENCES 1. Soberon FA, Loret de Mola JR, Goldberg RS, Tureck RW. Uterine arteriovenous malformation in a patient with recurrent pregnancy loss and a bicornuate uterus. J Reprod Med 1997;42:239–43. 2. Fleming H, Ostor AG, Pickel H, Fortune DW. Arteriovenous malformations of the uterus. Obstet Gynecol 1989; 73:209–13. 3. Hoffman MK, Meilstrup JW, Shackelford DP, Kaminski PF. Arteriovenous malformations of the uterus: An uncommon cause of vaginal bleeding. Obstet Gynecol Surv 1997; 52:736–40. 4. Chow TWP, Nwosu EC, Gould DA, Richmond DH. Pregnancy following successful embolisation of a uterine vascular malformation. Br J Obstet Gynaecol 1995;102:166–8. 5. Wilms GE, Favril A, Baert AL, Poppe W, Van Assche FA. Transcatheter embolization of uterine arteriovenous malformations. Cardiovasc Intervent Radiol 1986;9:61–4. 6. Goldberg RP, Flynn MK. Pregnancy after medical management of a uterine arteriovenous malformation. J Reprod Med 2000;45:961–3. 7. Onoyama I, Fukuhara M, Okuma A, Watanabe Y, Nakanura G. Successful pregnancy after the noninvasive management of uterine arteriovenous malformation. Acta Obstet Gynecol Scand 2001;80:1148–9. 8. Kelly FW. Forceps delivery after molar malignancy in a woman with arteriovenous malformation. J Reprod Med 2001;46:1013–6. Received March 31, 2003. Received in revised form May 22, 2003. Accepted June 5, 2003.
OBSTETRICS & GYNECOLOGY