Arch Gynecol Obstet DOI 10.1007/s00404-014-3309-2
Case Report
Diffuse uterine leiomyomatosis in patient with successful pregnancy following new surgical management Yasuo Otsubo · Masato Nishida · Yuko Arai · Ryota Ichikawa · Miyako Sakanaka
Received: 17 February 2014 / Accepted: 2 June 2014 © Springer-Verlag Berlin Heidelberg 2014
Abstract Diffuse uterine leiomyomatosis is associated with significant infertility and miscarriage complications. Appropriate diagnosis and therapy is essential. A 33-yearold woman was referred to our hospital because of GnRH analogue treatment-resistant multiple myomas and infertility. Following new technical myomectomy, she conceived spontaneously and delivered a 2,470 g healthy baby by cesarean section. New technical myomectomy may become an important conservative treatment option for patients with diffuse uterine leiomyomatosis. Keywords Diffuse uterine leiomyomatosis · Pregnancy · Myomectomy
Introduction Diffuse uterine leiomyomatosis is a relatively uncommon condition [1–12] associated with significant infertility and miscarriage complications [8]. Although an enlarged uterus is often seen in infertile women [5], other diagnoses should not be ruled out if multiple nodules are noted within the uterine myometrium and/or uterine cavity. When those occur, conservative therapy with GnRH analogue (GnRH-a) followed by post-therapeutic reproductive treatment is important [6, 8–10]. However, when the results are not satisfactory, interventional surgical management may be necessary. We present a case of diffuse uterine leiomyomatosis complicated by infertility in a patient who later successfully
Y. Otsubo (*) · M. Nishida · Y. Arai · R. Ichikawa · M. Sakanaka Department of Obstetrics and Gynecology, National Hospital Organization, Kasumigaura Medical Center, 2‑7‑14 Shimotakatsu, Tsuchiura, Ibaraki 300‑8585, Japan e-mail: [email protected]
conceived and delivered after undergoing a new technical myomectomy procedure.
Case report A 33-year-old woman, who previously had a spontaneous abortion after 12 weeks of gestation, was referred to our hospital because of GnRH-a treatment-resistant multiple myomas and infertility. A pelvic examination showed uterine enlargement to a size normally seen after 16 weeks of gestation as well as menorrhagia. Magnetic resonance imaging (MRI) revealed a centered uterine cavity with sub-mucosal myomas, and hypertrophy of the anterior and posterior wall with multiple myomas and a smooth uterine form (Fig. 1). Based on these findings, we diagnosed diffuse uterine leiomyomatosis. To preserve fertility, conservative surgical management was selected. We performed a myomectomy using a new technique, as follows. 1. The uterus was injected with a total of approximately 30 ml of 100-fold diluted vasopressin. 2. Next, the uterus was dissected longitudinally by cutting the center of the uterine cavity and preserving the bilateral uterine arteries. The incision formed a line between the height of the internal ostium and uterine fundus (Fig. 2a). 3. An incision was then made on the myometrium between the uterine endometrium and serosa to half its depth, and a myomectomy was performed as usual on both the right and left sides of the dissected uterus. The gaps created by myoma enucleation were sutured and closed using a 3–0 absorbable suture.
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Arch Gynecol Obstet
of uterine rupture. When the abdomen was opened, the lower uterine segment appeared normal, while the upper segment of the myometrium was quite thin around the midline of the uterus. To avoid uncontrollable profuse hemorrhaging during surgery, bleeding was controlled with a lower segmental transverse incision performed as usual instead of a classical fundal cesarean section. A 2,470-g girl with Apgar scores of 8 at 1 min and 9 at 5 min was delivered. Thereafter, the clinical course was uneventful and the patient was discharged from the hospital with her baby on postnatal day 10.
Discussion
Fig. 1 MRI T2-weighted findings. Multiple myomas were observed in the uterine myometrium and cavity
4. Following the myomectomy, the prior incision was closed using a 2–0 absorbable separate stitch suture, then the same procedure was performed on each side of the left and right anterior and posterior walls of the uterus (Fig. 2b). 5. Finally, the dissected uterus was rejoined using a method similar to Tompkins’s procedure. The endometrium and myometrium were closed using a 2–0 absorbable separate stich suture (Fig. 2c, d), while the uterine serosa was closed with a 2–0 absorbable running suture (Fig. 2e). Moreover, the re-constructed uterus was wrapped with an absorbable adhesion barrier. In the present patient, the number of enucleated myomas was 100 and the total weight of the myomas was 260 g (Fig. 2f). Initial operative bleeding was 900 ml and then subsided. The post-operative course was uneventful and the patient was discharged on post-operative day 8. A pathological study of the resected specimen confirmed diffuse uterine leiomyomatosis. Four months after the myomectomy, the patient spontaneously conceived, and was re-admitted at 24 weeks of gestation because of uterine wall thinness and possible uterine rupture. To avoid such rupture, an intravenous administration of ritodrine hydrochloride was started for tocolysis, with magnesium sulfate given later. The fetus showed normal growth with a normal amount of amniotic fluid, though the placenta was located on the remaining leiomyoma nodules and completely occupied the left uterine wall (Fig. 3). A cesarean section was performed at 35 weeks of gestation due to uncontrollable uterine contraction and for prevention
13
Diffuse uterine leiomyomatosis is a rare condition in which innumerable, ill-defined, and small smooth-muscle nodules have replaced most of the uterine parenchyma, which produces symmetrical enlargement of the uterus [1–12]. Notably, innumerable submucosal myomas usually affect women of reproductive age with menorrhagia [3] and infertility [6, 12]. Although a hysterectomy has been suggested as the standard treatment [1, 12], clinicians do not find that to be an easy choice for women of active reproductive age who wish to preserve fertility. Conservative therapy such as GnRH-a [6], transcervical resection (TCR) [8–10], uterine artery embolization (UAE) [7, 11], and myomectomy are sometimes considered for their fertility preserving effects. However, for patients with diffuse uterine leiomyomatosis, a conventional myomectomy with an external approach is disadvantageous as a surgical procedure because it is difficult to achieve complete clearance [1] or repair of the uniformly involved myometrium. Thus, conservative treatments such as hormonal therapy, UAE, and TCR have been recommended in several published reports [4, 6, 8–10], with GnRH-a considered to be an especially useful agent for diffuse uterine leiomyoma [6, 8–10]. A strategy of repeated GnRH-a or GnRH-a treatments followed by TCR has been reported to lead to conception in previous studies [8–10]. The patient had been already treated with GnRH-a for 6 months, though it was not successful. Since the patient desired pregnancy and requested conventional surgical management, we performed the present new technical myomectomy instead of TCR or UAE with informed consent. In accordance with the concept that myomas with submucosal or intracavitary components are the main cause of impaired fertility, and those with intramural components may restrict uterine expansion during pregnancy, our aim was to achieve a normal looking endometrial cavity by removing the submucosal myomata and provide complete clearance of the intramural myomata on the longitudinal
Arch Gynecol Obstet Fig. 2 a Appearance of longitudinally dissected uterus, cut along the center of the uterine cavity. Multiple submucosal and intramural nodules completely subverting the endometrial cavity can be seen. b Appearance of dissected uterus after myomectomy. Visualized myomas were removed from the endometrium and palpated myomas from the myometrium. c Appearance of uterine endometrium after rejoining 2–0 absorbable suture. d Appearance of uterine myometrium after rejoining 2–0 absorbable suture. e Appearance of rejoined uterus. The uterine serosa was closed with a 2–0 absorbable running suture. f Appearance of resected myomas. The number of enucleated myomas was 100 and their total weight was 260 g. Multiple independent myomas and myomas growing like buds or with stalk can be seen
Fig. 3 Transverse (left) and coronal (right) views of T2-weighted MR imaging at 33 weeks of gestation. Myomas remaining in the bilateral uterine walls are enlarged. The placenta can be observed along with myomas occupying the left uterine wall. The uterine cavity is nearly large enough for the fetus and a normal amount of amniotic fluid is present
13
uterine part as thoroughly as possible. In addition, we sought to leave the other small myomata in place throughout the myometrium and preserve bilateral uterine arterial blood flow. Following her second normal menstrual period after treatment, the patient spontaneously conceived and went through a normal pregnancy that reached 35 weeks. Although longitudinal wall thinness of the uterus and bilateral uterine wall enlargement from the remaining myomas were observed, the uterine cavity was adequately enlarged with normal fetal growth and a normal volume of amniotic fluid. The present method can be complicated by threatened premature delivery, though our concept may be supported by the clinical course of the patient. In addition, we think that this new surgical procedure can be effective for those of active reproductive age with diffuse uterine leiomyomatosis. Although this method may not be the initial choice for conventional treatment, it may become an important conservative treatment option for patients with diffuse uterine leiomyomatosis. Conflict of interest We declare that we have no conflict of interest.
References 1. Lapan B, Solomon L (1979) Diffuse leiomyomatosis of the uterus precluding myomectomy. Obstet Gynecol 53:82S–84S
13
Arch Gynecol Obstet 2. Fedele L, Zamberletti D, Carinelli S, Motta T, Gandiani GB (1982) Diffuse uterine leiomyomatosis. Acta Eur Fertil 13:125–131 3. Clement PB, Young RH (1987) Diffuse leiomyomatosis of the uterus: a report of four cases. Int J Gynecol Pathol 6:322–330 4. Grignon DJ, Carey MR, Kirk ME, Robinson ML (1987) Diffuse leiomyomatosis: a case study with pregnancy complicated by intrapartum hemorrhage. Obstet Gynecol 69:477–480 5. Mantovani MS, Neto B, Philbert PMP et al (1999) Multiple uterine leiomyomas: cytogenetic analysis. Gynecol Oncol 72:71–75 6. Fedele L, Bianchi S, Zanconato G, Carinelli S, Barlanda N (2004) Conservative treatment of diffuse uterine leiomatosis. Fertil Steril 82:450–453 7. Scheurig C, Islam T, Zimmermann E, Hamm B, Kroencke TJ (2008) Uterine artery embolization in patients with symptomatic diffuse leiomyomatosis of the uterus. J Vasc Radiol 19:279–284 8. Yen CF, Lee CL, Wang CJ, Soong YK, Phil M, Arici A (2007) Successful pregnancies in women with diffuse uterine leiomyomatosis after hysteroscopic management. Fertil Steril 88:1667–1673 9. Shimizu Y, Yomo H, Kita N, Takahashi K (2009) Successful pregnancy after gonadotropin releasing hormone analogue and hysteroscopic myomectomy in a woman with diffuse uterine leiomyomatosis. Arch Gynecol Obstet 280:145–147 10. Purohit R, Sharma JG, Singh S (2011) A case of diffuse uterine leiomyomatosis who had two successful pregnancies after medical management. Fertil Steril 95:2434.e5-6 11. Koh J, Kim MD, Jung DC, Lee M, Lee MS, Won JY, Lee DY, Park SI, Lee KH (2012) Uterine artery embolization (UAE) for diffuse leiomyomatosis of the uterus: clinical and imaging results. Eur J Radiol 81:2726–2729 12. Pai D, Coletti MC, Elkins M, Ladino-Torres M, Caoili E (2012) Diffuse uterine leiomyomatosis in a child. Pediatr Radiol 42:124–128
Case Report
Diffuse uterine leiomyomatosis in patient with successful pregnancy following new surgical management Yasuo Otsubo · Masato Nishida · Yuko Arai · Ryota Ichikawa · Miyako Sakanaka
Received: 17 February 2014 / Accepted: 2 June 2014 © Springer-Verlag Berlin Heidelberg 2014
Abstract Diffuse uterine leiomyomatosis is associated with significant infertility and miscarriage complications. Appropriate diagnosis and therapy is essential. A 33-yearold woman was referred to our hospital because of GnRH analogue treatment-resistant multiple myomas and infertility. Following new technical myomectomy, she conceived spontaneously and delivered a 2,470 g healthy baby by cesarean section. New technical myomectomy may become an important conservative treatment option for patients with diffuse uterine leiomyomatosis. Keywords Diffuse uterine leiomyomatosis · Pregnancy · Myomectomy
Introduction Diffuse uterine leiomyomatosis is a relatively uncommon condition [1–12] associated with significant infertility and miscarriage complications [8]. Although an enlarged uterus is often seen in infertile women [5], other diagnoses should not be ruled out if multiple nodules are noted within the uterine myometrium and/or uterine cavity. When those occur, conservative therapy with GnRH analogue (GnRH-a) followed by post-therapeutic reproductive treatment is important [6, 8–10]. However, when the results are not satisfactory, interventional surgical management may be necessary. We present a case of diffuse uterine leiomyomatosis complicated by infertility in a patient who later successfully
Y. Otsubo (*) · M. Nishida · Y. Arai · R. Ichikawa · M. Sakanaka Department of Obstetrics and Gynecology, National Hospital Organization, Kasumigaura Medical Center, 2‑7‑14 Shimotakatsu, Tsuchiura, Ibaraki 300‑8585, Japan e-mail: [email protected]
conceived and delivered after undergoing a new technical myomectomy procedure.
Case report A 33-year-old woman, who previously had a spontaneous abortion after 12 weeks of gestation, was referred to our hospital because of GnRH-a treatment-resistant multiple myomas and infertility. A pelvic examination showed uterine enlargement to a size normally seen after 16 weeks of gestation as well as menorrhagia. Magnetic resonance imaging (MRI) revealed a centered uterine cavity with sub-mucosal myomas, and hypertrophy of the anterior and posterior wall with multiple myomas and a smooth uterine form (Fig. 1). Based on these findings, we diagnosed diffuse uterine leiomyomatosis. To preserve fertility, conservative surgical management was selected. We performed a myomectomy using a new technique, as follows. 1. The uterus was injected with a total of approximately 30 ml of 100-fold diluted vasopressin. 2. Next, the uterus was dissected longitudinally by cutting the center of the uterine cavity and preserving the bilateral uterine arteries. The incision formed a line between the height of the internal ostium and uterine fundus (Fig. 2a). 3. An incision was then made on the myometrium between the uterine endometrium and serosa to half its depth, and a myomectomy was performed as usual on both the right and left sides of the dissected uterus. The gaps created by myoma enucleation were sutured and closed using a 3–0 absorbable suture.
13
Arch Gynecol Obstet
of uterine rupture. When the abdomen was opened, the lower uterine segment appeared normal, while the upper segment of the myometrium was quite thin around the midline of the uterus. To avoid uncontrollable profuse hemorrhaging during surgery, bleeding was controlled with a lower segmental transverse incision performed as usual instead of a classical fundal cesarean section. A 2,470-g girl with Apgar scores of 8 at 1 min and 9 at 5 min was delivered. Thereafter, the clinical course was uneventful and the patient was discharged from the hospital with her baby on postnatal day 10.
Discussion
Fig. 1 MRI T2-weighted findings. Multiple myomas were observed in the uterine myometrium and cavity
4. Following the myomectomy, the prior incision was closed using a 2–0 absorbable separate stitch suture, then the same procedure was performed on each side of the left and right anterior and posterior walls of the uterus (Fig. 2b). 5. Finally, the dissected uterus was rejoined using a method similar to Tompkins’s procedure. The endometrium and myometrium were closed using a 2–0 absorbable separate stich suture (Fig. 2c, d), while the uterine serosa was closed with a 2–0 absorbable running suture (Fig. 2e). Moreover, the re-constructed uterus was wrapped with an absorbable adhesion barrier. In the present patient, the number of enucleated myomas was 100 and the total weight of the myomas was 260 g (Fig. 2f). Initial operative bleeding was 900 ml and then subsided. The post-operative course was uneventful and the patient was discharged on post-operative day 8. A pathological study of the resected specimen confirmed diffuse uterine leiomyomatosis. Four months after the myomectomy, the patient spontaneously conceived, and was re-admitted at 24 weeks of gestation because of uterine wall thinness and possible uterine rupture. To avoid such rupture, an intravenous administration of ritodrine hydrochloride was started for tocolysis, with magnesium sulfate given later. The fetus showed normal growth with a normal amount of amniotic fluid, though the placenta was located on the remaining leiomyoma nodules and completely occupied the left uterine wall (Fig. 3). A cesarean section was performed at 35 weeks of gestation due to uncontrollable uterine contraction and for prevention
13
Diffuse uterine leiomyomatosis is a rare condition in which innumerable, ill-defined, and small smooth-muscle nodules have replaced most of the uterine parenchyma, which produces symmetrical enlargement of the uterus [1–12]. Notably, innumerable submucosal myomas usually affect women of reproductive age with menorrhagia [3] and infertility [6, 12]. Although a hysterectomy has been suggested as the standard treatment [1, 12], clinicians do not find that to be an easy choice for women of active reproductive age who wish to preserve fertility. Conservative therapy such as GnRH-a [6], transcervical resection (TCR) [8–10], uterine artery embolization (UAE) [7, 11], and myomectomy are sometimes considered for their fertility preserving effects. However, for patients with diffuse uterine leiomyomatosis, a conventional myomectomy with an external approach is disadvantageous as a surgical procedure because it is difficult to achieve complete clearance [1] or repair of the uniformly involved myometrium. Thus, conservative treatments such as hormonal therapy, UAE, and TCR have been recommended in several published reports [4, 6, 8–10], with GnRH-a considered to be an especially useful agent for diffuse uterine leiomyoma [6, 8–10]. A strategy of repeated GnRH-a or GnRH-a treatments followed by TCR has been reported to lead to conception in previous studies [8–10]. The patient had been already treated with GnRH-a for 6 months, though it was not successful. Since the patient desired pregnancy and requested conventional surgical management, we performed the present new technical myomectomy instead of TCR or UAE with informed consent. In accordance with the concept that myomas with submucosal or intracavitary components are the main cause of impaired fertility, and those with intramural components may restrict uterine expansion during pregnancy, our aim was to achieve a normal looking endometrial cavity by removing the submucosal myomata and provide complete clearance of the intramural myomata on the longitudinal
Arch Gynecol Obstet Fig. 2 a Appearance of longitudinally dissected uterus, cut along the center of the uterine cavity. Multiple submucosal and intramural nodules completely subverting the endometrial cavity can be seen. b Appearance of dissected uterus after myomectomy. Visualized myomas were removed from the endometrium and palpated myomas from the myometrium. c Appearance of uterine endometrium after rejoining 2–0 absorbable suture. d Appearance of uterine myometrium after rejoining 2–0 absorbable suture. e Appearance of rejoined uterus. The uterine serosa was closed with a 2–0 absorbable running suture. f Appearance of resected myomas. The number of enucleated myomas was 100 and their total weight was 260 g. Multiple independent myomas and myomas growing like buds or with stalk can be seen
Fig. 3 Transverse (left) and coronal (right) views of T2-weighted MR imaging at 33 weeks of gestation. Myomas remaining in the bilateral uterine walls are enlarged. The placenta can be observed along with myomas occupying the left uterine wall. The uterine cavity is nearly large enough for the fetus and a normal amount of amniotic fluid is present
13
uterine part as thoroughly as possible. In addition, we sought to leave the other small myomata in place throughout the myometrium and preserve bilateral uterine arterial blood flow. Following her second normal menstrual period after treatment, the patient spontaneously conceived and went through a normal pregnancy that reached 35 weeks. Although longitudinal wall thinness of the uterus and bilateral uterine wall enlargement from the remaining myomas were observed, the uterine cavity was adequately enlarged with normal fetal growth and a normal volume of amniotic fluid. The present method can be complicated by threatened premature delivery, though our concept may be supported by the clinical course of the patient. In addition, we think that this new surgical procedure can be effective for those of active reproductive age with diffuse uterine leiomyomatosis. Although this method may not be the initial choice for conventional treatment, it may become an important conservative treatment option for patients with diffuse uterine leiomyomatosis. Conflict of interest We declare that we have no conflict of interest.
References 1. Lapan B, Solomon L (1979) Diffuse leiomyomatosis of the uterus precluding myomectomy. Obstet Gynecol 53:82S–84S
13
Arch Gynecol Obstet 2. Fedele L, Zamberletti D, Carinelli S, Motta T, Gandiani GB (1982) Diffuse uterine leiomyomatosis. Acta Eur Fertil 13:125–131 3. Clement PB, Young RH (1987) Diffuse leiomyomatosis of the uterus: a report of four cases. Int J Gynecol Pathol 6:322–330 4. Grignon DJ, Carey MR, Kirk ME, Robinson ML (1987) Diffuse leiomyomatosis: a case study with pregnancy complicated by intrapartum hemorrhage. Obstet Gynecol 69:477–480 5. Mantovani MS, Neto B, Philbert PMP et al (1999) Multiple uterine leiomyomas: cytogenetic analysis. Gynecol Oncol 72:71–75 6. Fedele L, Bianchi S, Zanconato G, Carinelli S, Barlanda N (2004) Conservative treatment of diffuse uterine leiomatosis. Fertil Steril 82:450–453 7. Scheurig C, Islam T, Zimmermann E, Hamm B, Kroencke TJ (2008) Uterine artery embolization in patients with symptomatic diffuse leiomyomatosis of the uterus. J Vasc Radiol 19:279–284 8. Yen CF, Lee CL, Wang CJ, Soong YK, Phil M, Arici A (2007) Successful pregnancies in women with diffuse uterine leiomyomatosis after hysteroscopic management. Fertil Steril 88:1667–1673 9. Shimizu Y, Yomo H, Kita N, Takahashi K (2009) Successful pregnancy after gonadotropin releasing hormone analogue and hysteroscopic myomectomy in a woman with diffuse uterine leiomyomatosis. Arch Gynecol Obstet 280:145–147 10. Purohit R, Sharma JG, Singh S (2011) A case of diffuse uterine leiomyomatosis who had two successful pregnancies after medical management. Fertil Steril 95:2434.e5-6 11. Koh J, Kim MD, Jung DC, Lee M, Lee MS, Won JY, Lee DY, Park SI, Lee KH (2012) Uterine artery embolization (UAE) for diffuse leiomyomatosis of the uterus: clinical and imaging results. Eur J Radiol 81:2726–2729 12. Pai D, Coletti MC, Elkins M, Ladino-Torres M, Caoili E (2012) Diffuse uterine leiomyomatosis in a child. Pediatr Radiol 42:124–128
