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doi:10.1111/jog.12382

J. Obstet. Gynaecol. Res. Vol. 40, No. 6: 1689–1694, June 2014

New myomectomy technique for diffuse uterine leiomyomatosis Masato Nishida, Ryota Ichikawa, Yuko Arai, Miyako Sakanaka and Yasuo Otsubo Department of Obstetrics and Gynecology, National Hospital Organization, Kasumigaura Medical Center, Ibaraki, Japan

Abstract Aim: The aim of this study was to determine the effects of a new myomectomy technique for diffuse uterine leiomyomatosis. Material and Methods: To enucleate multiple myomas, we developed a novel myomectomy technique involving longitudinal dissection of the uterus in the midline. On each side of the anterior and posterior walls, the uterine wall was further divided into two pieces from the incision site. Myomas were separated into serosal and mucosal sides, and then enucleated for removal from the thinned myometrium. Results: This procedure was applied for seven patients with diffuse leiomyomatosis. The mean number of myomas enucleated from each patient was 117 (range, 74–226). Mean total weight of enucleated myomas was 147.6 g (range, 59–360 g). Mean operative time was 284 min (range, 212–407 min). Mean blood loss was 1614 g (range, 428–4421 g), with three patients requiring blood transfusion. Anemia due to menorrhagia improved in all women. Three patients became pregnant, with two undergoing cesarean section after a normal course of pregnancy and giving birth to healthy babies. The third pregnancy ended in miscarriage. We noticed intraoperatively that myoma nodules were connected to each other in every case. Conclusions: This procedure should be considered as a therapeutic option in women suffering from symptoms of diffuse uterine leiomyomatosis who wish to avoid hysterectomy. Key words: conservative surgery, myomectomy, therapy, uterine diffuse leiomyomatosis.

Introduction Diffuse leiomyomatosis is a rare disorder in which numerous small myomas occupy the myometrium. According to our search of the literature, only 36 cases have been reported to date.1–13 The uterus is symmetrical and smooth-surfaced, but myomas protrude into the uterine cavity. This results in menorrhagia, which can be a cause of infertility in younger women.3 The youngest patient reported was 16 years old.12 The diameter of each myoma in diffuse leiomyomatosis is small (0.5–3.0 cm), with myomas distributed throughout all areas of the uterus except the cervix. Preservation of the uterus is thus difficult.2,3 To

date, conservative surgery has been reported in only a small number of cases.5,6,9,10 We describe herein our application of a novel enucleatic myomectomy technique in seven patients with diffuse leiomyomatosis.

Methods Between October 2007 and June 2011, we performed surgery in seven patients with diffuse leiomyomatosis, which had been clinically diagnosed preoperatively based on findings from magnetic resonance imaging (MRI) (Fig. 1a). In all cases, the uterus was enlarged and the myometrium displayed numerous round

Received: September 30 2013. Accepted: December 13 2013. Reprint request to: Dr Masato Nishida, Department of Obstetrics and Gynecology, National Hospital Organization, Kasumigaura Medical Center, 2-7-14 Shimotakatsu, Tsuchiura, Ibaraki 300-8585, Japan. Email: [email protected]

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

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(a)

(b)

Figure 1 (a) Preoperative sagittal T2-weighted imaging shows multiple intramural and submucosal myoma nodules. (b) Postoperative sagittal T2-weighted imaging of the same patient (Patient 7).

lesions of different sizes. The uterine surface was smooth, with no subserosal myomas; all were submucosal myomas protruding into the uterine cavity. Mean age at the time of surgery was 32.4 years (range, 22–37 years). Five patients had previously undergone myomectomy. In all patients, the chief complaint was menorrhagia. Six patients had never been pregnant, and one had experienced a single miscarriage 3 years previously. All seven women desired preservation of the uterus. Preoperatively, three women who had received therapy using gonadotrophin-releasing hormone agonists (GnRHa) in the previous hospital continued that therapy, while the remaining four patients received no preoperative treatment. This surgery was developed as a way of complying with the wishes of individual patients expressing a strong desire to avoid hysterectomy. We therefore did not obtain institutional review board approval. A transverse incision was made in the lower abdomen, and the uterus was injected with a 100-fold dilution of vasopressin. The uterus was dissected longitudinally in the midline using a high-frequency electric surgical knife (spear-type electrode; Honest Medical) at 124 W. The dissection extended to just above the internal os (Fig. 2a). From the incision site, each side of the anterior and posterior walls was divided into two layers: a serosal side and a mucosal side. At this time, dissection of intramural myomas was a feature of the technique. Care was taken not to injure the interstitial portion of the fallopian tube or cut the uterine artery (Fig. 2b,c). During this procedure, the myomas, together with the uterine wall, can be thinly cut, and for many patients, the cut surfaces of the myomas were exposed to the dissection plane. The myomas were easily enucleated for removal from the thinned myometrium. Myomas were separated into serosal and mucosal sides, and most could be enucleated from the dissected area. Myomas facing the mucosa were enucleated from the mucosal side using a

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method similar to a conventional procedure. The spaces created by myoma enucleation were sutured and closed with 3-0 absorbable suture. The same procedure was performed on each side of the left and right anterior and posterior walls of the uterus, so myoma enucleation was performed in four locations. When enucleation was completed, the myometrium divided into two pieces was first sutured and closed (Fig. 2d). The incision wound through the anterior and posterior walls was then sutured and closed in two layers (Fig. 2e,f). The myometrium was then sutured using 1-0 absorbable suture. Antibiotics were given for 2 days after surgery. MRI was performed 3 months postoperatively to evaluate the presence or absence of menorrhagia and anemia, and pregnancy was permitted thereafter.

Results The mean number of myomas enucleated from each patient was 117 (range, 74–226). Mean total weight of enucleated myomas was 147.6 g (range, 59–360 g). Mean weight of a single myoma in each case was about 1 g. Mean operative time was 284 min (range, 212– 407 min). Mean blood loss was 1614 g (range, 428– 4421 g), and three patients required blood transfusion (Table 1). Mean duration of postoperative follow-up was 44.7 months (range, 27–70 months). Postoperatively, three patients became pregnant. Two of these women (Patients 1 and 4) achieved natural pregnancies at 12 and 16 months postoperatively, respectively. Courses of pregnancy were smooth, with births by cesarean section at 36 weeks and 38 weeks. Patient 1 is currently in her second pregnancy. The other woman (Patient 3) became pregnant by in vitro fertilization, but experienced a miscarriage. Postoperative MRI showed findings suspected to represent remaining myomas in each patient (Fig. 1b).

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

Myomectomy for diffuse leiomyomatosis

(b)

(c)

(e)

(f)

(a)

(d)

Figure 2 (a) Dissection of the uterus. (b) Second dissection of the anterior uterine wall. (c) Second dissection of the opposite side of the anterior uterine wall. (d) Suture of the lesion. (e) Closure of the wound in two layers. (f) Rejoining of the uterus.

Table 1 Patient characteristics No. 1 2 3 4 5 6 7

Age

G/P

PM

GnRHa†

TW (g)

NM (pieces)

OT (min)

EBL (g)

BT

Follow-up (months)

Hb (g/dL)‡

PAM

CB

22 37 33 31 36 32 36

0/0 0/0 0/0 0/0 0/0 1/0 0/0

N Y Y N Y Y Y

Y N N Y Y N N

80 187 93 59 136 360 138

74 70 90 62 147 226 150

212 320 210 288 280 407 275

428 2070 450 570 2230 4421 1130

N Y N N Y Y N

70 66 51 40 32 27 27

12.4 9.0 12.2 13.6 11.7 12.6 12.6

Y N Y Y N N N

Y N N Y N N N

†GnRHa before surgery. ‡Hb (g/dL) after surgery. BT, blood transfusion; CB, childbirth; EBL, estimated blood loss; GnRHa, gonadotrophinreleasing hormone agonists; G/P, gravidity/parity; Hb, hemoglobin; NM, number of myoma nodules; OT, operative time; PAM, pregnancy after myomectomy; PM, previous myomectomy; TW, total weight.

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

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While menorrhagia has recurred in one woman (Patient 2) to date, anemia has improved in the remaining patients. During these surgeries, a new observation was that the myomas were not isolated, but rather merged together. The pattern of merging varied. In some cases, a few myomas joined successively one after another (Fig. 3). In other cases, thin stalks were spread throughout the myometrium, like a network. The enucleated myomas were thus not spherical, but irregularly shaped.

Discussion In diffuse leiomyomatosis, because multiple myomas are present in the middle of the uterus, conventional myomectomy with an external approach is very disadvantageous as a surgical procedure. Fedele et al.3 reported attempting myomectomy by laparotomy, but total hysterectomy ended up being performed in all cases. Grignon et al.5 were the first to report pregnancy after conservative surgery. Their patient achieved preg-

Figure 3 A merged myoma.

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nancy and delivered a live birth after enucleation, but total hysterectomy was performed postpartum due to placenta accreta. Conservative treatments for diffuse leiomyomatosis other than laparotomy include hormone therapy, uterine artery embolization (UAE), and transcervical resection (TCR). Purohit et al.7 reported a patient with a natural pregnancy after GnRHa therapy. On examination of the uterine cavity by hysteroscopy after GnRHa therapy, no protrusion of the myomas into the uterine cavity was observed. If myomas protruding into the uterine cavity disappear with GnRHa therapy, then pregnancy with GnRHa therapy may be anticipated. Koh et al.8 reported a patient who became pregnant after treatment with UAE. If UAE is effective in patients with diffuse leiomyomatosis who wish to avoid hysterectomy, this may be the most rational treatment. However, when pregnancy is desired after treatment, pregnancy after UAE is controversial.14 Yen et al.9 performed hysteroscopic myomectomy in five patients with diffuse leiomyomatosis, and three patients who wished to conceive achieved pregnancy and live births. Because a key cause of infertility in patients with diffuse leiomyomatosis is thought to be multiple submucosal myomas, pregnancy may be possible following resection of the myomas. Shimizu et al.10 also reported a natural pregnancy in a patient who underwent hysteroscopic myomectomy after GnRHa therapy. Although pregnancy after hysteroscopic myomectomy has been reported, Fedele et al.3 reported patients who did not become pregnant after hysteroscopic surgery. Hysteroscopic myomectomy had also been performed in two of our seven patients, but neither achieved pregnancy and the myomas recurred. The effectiveness of hysteroscopic surgery in diffuse leiomyomatosis is thus limited, but the procedure may be effective if the submucosal myomas are small and few in number. In the present series, we performed a novel myomectomy technique for conservative treatment of diffuse leiomyomatosis. This procedure is similar to a conservative technique for diffuse adenomyosis that we previously reported15 from the standpoint that the thickened muscularis is divided into two layers, mucosal and serosal, and then the lesion is resected. However, the big difference lies in the fact that in diffuse adenomyosis, the endometrium is left intact, whereas the main purpose of the new technique is to achieve resection of the submucosal myomas. This was performed because in all cases except Patient 4, pregnancy had not been achieved despite

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

Myomectomy for diffuse leiomyomatosis

previous GnRHa therapy, myomectomy by laparotomy, or hysteroscopic resection. Postoperatively, three patients became pregnant and two patients delivered live births. The surgical procedure was performed in a similar manner in all patients. This was the first surgery for both women who gave birth. This is because diffuse leiomyomatosis is always associated with submucosal myomas, and repeated surgery can unfavorably affect the endometrium for pregnancy. We believe this demonstrates that our technique may represent a useful option for conservative treatment. Because of the numerous myomas in diffuse leiomyomatosis, resection of all myomas was not feasible. With the procedure we performed, although at least all myomas that were visualized or palpated could be removed, the operative time was long (mean, 284 min) and blood loss was large, with three patients requiring blood transfusion. As shown in Table 1, operative time and blood loss were strongly correlated. This means that shortening the operative time is extremely useful for reducing blood loss. To reduce the operative time, preoperative GnRHa administration may be useful to reduce uterine volume. We also believe that focusing on enucleation of myomas on the mucosal side, which would affect pregnancy, may be effective, whereas some myomas on the serosal side could safely be ignored. In one patient, menorrhagia has recurred. The reason may have been due to the site of recurrence. Menorrhagia is associated with recurrence of submucosal myomas, whereas recurrence of intramural myomas may be asymptomatic. In terms of the invasiveness of conservative treatment methods, myomectomy by laparotomy is the most highly invasive. Therefore, for conservative management of diffuse leiomyomatosis, treatment should begin with GnRHa. If pregnancy is not achieved, but the submucosal myomas are small and few in number, hysteroscopic resection should be performed. If pregnancy is not achieved despite the above methods, and the patient wishes to continue conservative treatment, the myomectomy technique described herein may represent a useful option. During enucleation of the uterine myomas, we noticed that the myomas were interconnected. This is probably why innumerable small myomas are scattered throughout the muscularis of the uterus. Baschinsky et al.11 performed tumor clonality analysis in diffuse leiomyomatosis and found that just like more common myomas, each myoma developed from different clones. This suggests the development of multiple

independent myomas even in diffuse leiomyomatosis, growing like buds or with creation of a stalk. This growth mechanism may be the reason myomas are absent from the uterine surface. In conclusion, we performed a novel myomectomy technique in seven patients with diffuse leiomyomatosis. Although this procedure may not be the initial choice for conservative treatment, we demonstrated the effectiveness of this approach for resection of at least those myomas that can be visualized and palpated in diffuse leiomyomatosis. This procedure may become an option for conservative treatment of diffuse leiomyomatosis.

Acknowledgment We wish to express our gratitude to Dr Keisuke Koyama, who drew the wonderful illustration (Fig. 2).

Disclosure No authors have any financial support or relationship that may pose a potential conflict of interest.

References 1. Murray HL, Glynn E. A case of complete fibromatosis of the corpus uteri. J Obstet Gynaecol Br Emp 1924; 31: 398–401. 2. Lapan B, Solomon L. Diffuse leiomyomatosis of the uterus precluding myomectomy. Obstet Gynecol 1979; 53 (Suppl): 82–85. 3. Fedele L, Zamberletti D, Carinelli S, Motta T, Candiani GB. Diffuse uterine leiomyomatosis. Acta Eur Fertil 1982; 13: 125– 131. 4. Clement PB, Young RH. Diffuse leiomyomatosis of the uterus: A report of four cases. Int J Gynecol Pathol 1987; 6: 322–330. 5. Grignon DJ, Carey MR, Kirk ME, Robinson ML. Diffuse uterine leiomyomatosis: A case study with pregnancy complicated by intrapartum hemorrhage. Obstet Gynecol 1987; 69: 477–480. 6. Fedele L, Bianchi S, Zanconato G, Carinelli S, Berlanda N. Conservative treatment of diffuse uterine leiomyomatosis. Fertil Steril 2004; 82: 450–453. 7. Purohit R, Sharma JG, Singh S. A case of diffuse uterine leiomyomatosis who had two successful pregnancies after medical management. Fertil Steril 2011; 95: 2434; e5-6. 8. Koh J, Kim MD, Jung DC et al. Uterine artery embolization (UAE) for diffuse leiomyomatosis of the uterus: Clinical and imaging results. Eur J Radiol 2012; 81: 2726–2729. 9. Yen CF, Lee CL, Wang CJ, Soong YK, Phil M, Arici A. Successful pregnancies in women with diffuse uterine leiomyomatosis after hysteroscopic management. Fertil Steril 2007; 88: 1667–1673. 10. Shimizu Y, Yomo H, Kita N, Takahashi K. Successful pregnancy after gonadotropin-releasing hormone analogue and

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hysteroscopic myomectomy in a woman with diffuse uterine leiomyomatosis. Arch Gynecol Obstet 2009; 280: 145–147. 11. Baschinsky DY, Isa A, Niemann TH, Prior TW, Lucas JG, Frankel WL. Diffuse leiomyomatosis of the uterus: A case report with clonality analysis. Hum Pathol 2000; 31: 1429– 1432. 12. Pai D, Coletti MC, Elkins M, Landino-Torres M, Caoili E. Diffuse uterine leiomyomatosis in a child. Pediatr Radiol 2012; 42: 124–128.

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13. Scheurig C, Islam T, Zimmermann E, Hamm B, Kroencke TJ. Uterine artery embolization in patients with symptomatic diffuse leiomyomatosis of the uterus. J Vasc Interv Radiol 2008; 19: 279–284. 14. Lefebvre G, Vilos G, Allaire C et al. The management of uterine leiomyomas. J Obstet Gynaecol Can 2003; 25: 396–418. 15. Nishida M, Takano K, Arai Y, Ozone H, Ichikawa R. Conservative surgical management for diffuse uterine adenomyosis. Fertil Steril 2010; 94: 715–719.

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

New myomectomy technique for diffuse uterine leiomyomatosis.

The aim of this study was to determine the effects of a new myomectomy technique for diffuse uterine leiomyomatosis...
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