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The hysteroscopic management of endometrial leiomyomatosis
February ]997, Vol. 4, No. 2
TheJournal of the American Association of Gynecologic Laparoscopists
The HysteroscopicManagement of Endometrial Leiomyomatosis Milton H. Goldrath, M.D., and Mujtaba Husain, M.D.
Abstract Uterine myomata are frequently the cause of abnormal uterine bleeding. They may be removed successfully by hysteroscopy. As a rule, the myomata are single, although on occasion several pedunculated myomas may be present. Three women were diagnosed with extremely large numbers of submucous leiomyomata. Many of the lesions were removed by hysteroscopic resection and many were destroyed with the neodymium:yttrium-aluminumgarnet laser.
anemia since menarche. She had been taking oral contraceptives for 10 years with slight improvement. Dilatation and curettage (D&C) 2 years earlier yielded four small submucous myomas 0.5 cm in diameter. The uterus was myomatous, the size of a 6-week pregnancy. Hemoglobin was 10 g. Office hysteroscopy was difficult because of bleeding, but a pedunculated submucous myoma could be seen. Because of the severity of bleeding, the woman was admitted to the hospital, Laminaria was inserted into the cervix, and the next day she underwent hysteroscopic removal of 18 myomata measuring 0.9 to 3.0 cm in diameter. Other myomas were felt within the uterus, but because of profuse bleeding it was decided to stop the procedure. A Foley catheter was inserted into the uterus for tamponade. The patient's hemoglobin dropped to 7 g. The catheter was removed the next day and the patient was discharged home taking oral iron therapy.
The rapidly expanding use of the hysteroscope for the evaluation and treatment of abnormal uterine bleeding has led to the diagnosis of many pedunculated submucous leiomyomata. 1 3 Usually these are solitary, but on occasion, two or three pedunculated myomas may be seen and treated hysteroscopically. In the past few years we have had the occasion to evaluate and successfully treat three women with innumerable submucous leiomyomata. We were unable to find any similar cases in the literature. This entity is of particular concern, as hysteroscopy is required for proper diagnosis and treatment.
Case Reports Patient No. 1 A 31-year-old woman experienced profuse menorrhagia of 8 months' duration. She had von Willebrand disease, which caused her to have menorrhagia with
From the Departments of Obstetrics and Gynecology (Dr. Goldrath) and Pathology (Dr. Husain), Wayne State University School of Medicine, and Section of Gynecology (Dr. Goldrath) and Department of Laboratory Medicine (Dr. Husain), Sinai Hospital, Detroit, Michigan. Address reprint requests to Milton H. Goldrath, M.D., Department of Obstetrics and Gynecology, Sinai Hospital, 6767 West Outer Drive, Detroit, MI 48235; fax 313 493 7503.
263
Hysteroscopic Management of Endometrial Leiomyomatosis Goldrath and Husain
Sixteen months later she returned stating that her menses were becoming heavier. Office hysteroscopy revealed one sizable myoma in the fundus as well as several tiny myomas. There were no synechiae. She was prescribed danazo1800 mg/day. The couple elected not to proceed with any assisted reproductive methods. Sixteen months later the woman returned still taking danazol and with a moderate amount of breakthrough bleeding. Office hysteroscopy revealed numerous tiny myomas and submucous myomas in the cornual areas. Three months later at operative hysteroscopy, more than 50 minute myomas, estimated to be 1 to 3 mm in diameter, were destroyed and the larger submucous myomas were resected with the Nd:YAG laser. Two months postoperatively office hysteroscopy revealed no myomas. Two broad synechiae were seen and easily broken up with the hysteroscope. A total of 103 myomas had been removed and more than 150 destroyed with the Nd:YAG laser. Two further contacts, almost a year apart, were made with the patient by telephone. Her menses were normal and she had still not conceived.
Her menorrhagia improved. Several office hysteroscopies revealed numerous small submucous myomas (Figure 1), and over the next 6 months 15 were removed in the office with polyp forceps; however, hysteroscopy revealed many remaining. Menses soon became heavy again and the patient was prescribed danazo1800 mg/day in an attempt to delay definitive hysteroscopic surgery until she decided to bear children. She informed us that she was married 7 months later and, following our advice, hoped to conceive as soon as the myomas were hysteroscopically removed. Two months after her marriage she was still taking danazol and experiencing very little bleeding. At outpatient operative hysteroscopy many minute myomas were seen and destroyed with the neodymium: yttrium-aluminum-garnet (Nd:YAG) laser. Forty-nine myomas were removed with polyp forceps. The patient was discharged the same day with instructions to stop danazol and start conjugated estrogen 2.5 mg/day for 60 days. She was told to take medroxyprogesterone acetate 5 mg/day from day 51 through day 60 to produce orderly withdrawal bleeding. After the withdrawal period, office hysteroscopy was normal and the woman was instructed to attempt conception. Three months later office hysteroscopy was again performed; no myomas were seen and the patient was having relatively normal menses. Five months later she had not become pregnant. Her menses remained relatively normal, and office hysteroscopy revealed no myomas. Infertility work-up revealed her husband to be oligospermic.
Patient No. 2 A 31-year-old gravida 1, para 0, abortus 1 (induced) woman had a 4-year history of profuse menorrhagia. A D&C performed in the past was not helpful. On examination her uterus was myomatous and approximately the size of an 8-week pregnancy. Office hysteroscopy revealed a large, mostly pedunculated submucous myoma on the posterior wall. Other small myomas were seen in the fundus. At vaginal myomectomy using Laminaria to dilate the cervix, seven myomas measuring 1.0 to 3.0 cm were removed. Others were believed still to be present. Six weeks later the woman's menses had improved. Office hysteroscopy revealed eight small, pedunculated myomas in the endometrial cavity. Since she did not wish to conceive at that time, danazo1800 mg/day was prescribed, but the patient decided not to take it. Approximately 1 year later she returned stating her menses were profuse. Office hysteroscopy revealed 10 to 15 submucous myomas, several quite large. She still did not wish to conceive. She refused hysteroscopic resection and oral contraception. One year later she was again seen, again with profuse menses. Her uterus was myomatous and the size of a 6-week pregnancy. Office hysteroscopy revealed more than 15 submucous myomas. She was given danazol 800 mg/day, and 1 month later underwent
FIGURE 1. Office hysteroscopic view of patient no. 1 while taking danazol. Note the various-size myomata, including seedlings.
264
February ] 997, Vol. 4, No. 2
The Journal of the American Association of Gynecologic Laparoscopists
resection o f 10 myomas. Postoperatively, conjugated estrogen and medroxyprogesterone acetate in a regimen similar to that of patient 1 were prescribed. One month later office hysteroscopy revealed a midline synechia. The tubal ostia were seen. There was a myoma in the right horn. The patient refused resection of the myoma at that time. Six months later she complained of profuse bleeding and was treated with a gonadotropin-releasing hormone analog. After 4 months office hysteroscopy disclosed 20 to 30 myomata. The tubal ostia were seen. At operative hysteroscopy performed 1 month later, 25 small myomas were destroyed, the midline synechia was lysed with the Nd:YAG laser, and 10 myomas were removed with polyp forceps. She was prescribed the same hormone regimen as patient no. 1. A total of 25 myomas had been destroyed with the Nd:YAG laser and 27 removed. The woman was next seen two months later after a normal withdrawal period. Office hysteroscopy revealed normal-appearing endometrium and no synechiae or myomata. She was advised to conceive and did so 6 months later. She spotted during the first trimester, and the fibroids grew quite large. She had many contractions during the pregnancy and was treated with bedrest and terbutaline. Spontaneous rupture of membranes occurred at 37 weeks and a cesarean section was performed because of myomas that prevented the application of the fetal head to the cervix. Cesarean section was described as being difficult because of the myomas. Postpartum the patient had considerable pain and bleeding. When menses recurred, they were heavy, and a total abdominal hysterectomy was performed 4 months postpartum. The uterus weighed 460 g. Submucous myomas were apparent and were quite large (Figure 2).
FIGURE 2. Hysterectomy specimen from patient 2. Hysterectomy was performed 4 months postpartum. Note large submucous myomata.
months later. The rationale for delay was to remove the myomas and have the patient conceive shortly thereafter. At operative hysteroscopy 20 minute myomas were destroyed and 39 were resected with the Nd:YAG laser. The patient was prescribed conjugated estrogen 5 mg for 60 days with medroxyprogesterone acetate 5 mg from days 51 to 60. Repeat treatment was planned, as all myomas were not destroyed. This was purposely staged to avoid extensive destruction of the endometrium with subsequent adhesion formation. Hysteroscopy 3 months later revealed a few small synechiae plus several submucous myomata. Operative hysteroscopy was done later that month, destroying 20 seedling myomas and resecting 7 with the Nd:YAG laser. The synechiae were lysed. The same regimen of conjugated estrogens and medroxyprogesterone acetate was prescribed. Three months later office hysteroscopy revealed a normal cavity with narrow horns. The endometrium appeared normal, and no synechiae or myomata were present. The tubal ostia were seen and were open. The shoulder sign was positive. The patient was advised to conceive as promptly as possible. A total of 46 myomas were removed and 40 were destroyed with the Nd:YAG laser. The woman conceived four months later and was delivered at 38 weeks by cesarean section for a complete placenta previa. Placenta accreta was present. No submucous myomas were noted in the cesarean section surgical record.
Patient No. 3 A 31-year-old gravida 2, para 0, abortus 2 (induced) woman was first seen because of 2 years of profuse menorrhagia. She had been taking oral contraceptives for 2 years in an attempt to control the bleeding. At D&C performed 1 month previously three small myomas were removed. It was noted that another remained. Office hysteroscopy revealed more than 20 small pedunculated myomata and many minute submucosal ones. The patient was prescribed danazol 800 mg/day to control bleeding until surgery was performed several
265
Hysteroscopic Management of Endometrial Leiomvomatosis Gddrath and Husain
At the 6 weeks postpartum the woman's uterus was said to be of normal size, and she was treated with oral contraceptives. Her menses were normal for the next 4 months. She is contemplating another pregnancy in the near future.
The hysterectomy specimen from patient 2 showed a few large intramural myometrial nodules and, in a few areas, multiple nodules, possibly representing diffuse leiomyomatosis. The endometrium was not reported to be significantly involved.
Specimen Pathology
Discussion
All endoscopically resected nodules had common morphologic findings. They ranged from 1.0 to 30.0 ram. Some of the smaller ones were totally surrounded by endometrium and others were partially covered by identifiable endometrial tissue. The nodules were not oriented around vascular structures. Unduly prominent mitotic activity or nuclear crowding was not observed. Features of infarction or necrosis were also lacking (Figures 3 and 4).
Large numbers of minute leiomyomata are reminiscent of the condition known as diffuse leiomyomatosis of the uterus. This term was first used by Lapan and Solomon in 1979, 4 who found three similar cases reported in the literature and reported two of their own. Since that time there have been seven additional cases reported. The largest series reported four cases and provided a detailed pathologic description of the lesions. 5 Endometrial leiomyomatosis and diffuse leiomyomatosis of the uterus are similar in that both conditions have innumerable numbers of tiny myomas. These myomas are benign. The similarity ends there. Diffuse leiomyomatosis of the uterus is characterized by a large uterus, up to 1200 g. In the eight cases that mentioned the endometrium, the endometrium was free of myomataJ -7 Two of three cases contained submucous myomata, but they were not described as being pedunculated. 8They may be similar to our patients. In endometrial leiomyomatosis, by our definition, the endometrium is extensively involved. The uteri in our patients were small on initial examination, none larger than 6 to 8 weeks' gestation. Obviously other myomas were present. The hysterectomy specimen from the second patient was quite large (460 g) and generally contained large myomata. In some areas the findings were compatible with diffuse leiomyomatosis. Since the other two women did not undergo hysterectomy, we do not know if diffuse leiomyomatosis is present within their myometrium, although this is doubtful because the uteri did not grow very large. Patients 1 and 3 who did not have a hysterectomy are doing quite well. Whether they have diffuse leiomyomatosis is not readily apparent. Ultrasonic examinations showed only small leiomyomata rather than a diffuse process. Since at the present time there is no indication for hysterectomy in these women, the histology of the myometrium remains unknown. Although all three women had voluntarily not borne children when first seen, the second and third had had previous voluntary terminations of pregnancy. Whether endometrial leiomyomatosis was present at
FIGURE 3. Low-power photomicrograph shows small leiomyoma covered by endometrium on the surface.
FIGURE 4. Medium-power photomicrograph shows minute leiomyoma in the endometrium deep to a gland.
266
February 1997, VoJ.4, No. 2
TheJournaJof the American Association of GynecoJogic Laparoscopists
the time they were pregnant is unknown; however, these terminations did antedate the onset of severe menorrhagia. Therapy was directed toward controlling menorrhagia with preservation of reproductive capability. This was achieved in all three patients for various periods of time. There was some recurrence of the myomas over the treatment period. The first patient, unfortunately, did not conceive, presumably due to oligospermia. The other two did conceive. The surgical approach to these myomas by operative hysteroscopy is worthy of discussion. In the first two women the submucous myomata seen hysteroscopically were of quite good size, and we elected to perform vaginal myomectomy by dilating the cervix with Laminaria and using polyp forceps. 2'3Follow-up of those patients and primary approach in the third involved using the Nd:YAG laser to remove the tiny myomas with as little destruction of the endometrium as possible. In attempting to expose as many minute submucous myomas as possible, the patients were treated preoperatively with danazol 800 mg/day to produce atrophy of the endometrium. Immediately after treatment with the Nd:YAG laser, they were given large dosages of estrogen for 60 days and withdrawn with l0 days of medroxyprogesterone acetate on days 51 through 60. This was an attempt to prevent synechiae from forming between larger areas of the damaged endometrium. There were far too many myomas present to avoid destroying them on the opposing surfaces as a method of preventing synechiae. We were quite successful in all these patients. Only a few synechiae formed in patients 2 and 3, and these were easily treated at the time of subsequent destruction of submucous myomata. We did not elect to use an intrauterine catheter or intrauterine device to prevent adhesions. It has been our approach to administer large doses of estrogen to encourage rapid endometrial regrowth, and the success of the regimen is attested to by these patients.
These cases are noteworthy in that they demonstrate that hysteroscopic surgery can be successful even if it takes great perseverance. Two of the women became pregnant and delivered living children, however, patient no. 2 required a hysterectomy. The third woman is looking forward to a second pregnancy; the first hopes to conceive. References
. Goldrath MH, Sherman A: Office hysteroscopy and suction curettage: Can we eliminate the hospital diagnostic dilatation and curettage? Am J Obstet Gyneco1220-229, 1985 . Goldrath MH: Vaginal removal of the pedunculated submucous myoma: The use ofLaminaria. Obstet Gynecol 70:670-672, 1987 . Goldrath MH: Vaginal removal of the pedunculated submucous myoma: Historical observations and development of a new procedure. J Reprod Med 35(10):921-924, 1990 . Lapan B, Solomon L: Diffuse leiomyomatosis of the uterus precluding myomectomy. Obstet Gynecol 53:82S-84S, 1979 . Clement PB, Young RH: Diffuse leiomymatosis of the uterus: A report of four cases. Int J Gynecol Pathol 6:322-330, 1987 . Grignon DJ, Carey MR, Kirk ME, et al: Diffuse uterine leiomyomatosis:A case study with pregnancy complicated by intrapartum hemorrhage. Obstet Gyneco169:477-480, 1987 7. Lai FM, Wong FW, Allen PW: Diffuse uterine leiomyomatosis with hemorrhage. Arch Pathol Lab Med 115:834-837, 1991 8. Fedele L, Zamberletti D, Carivelli S, et al: Diffuse Uterine leiomyomatosis. Acta Eur Fertil 13:125-131, 1982
267
TheJournal of the American Association of Gynecologic Laparoscopists
The HysteroscopicManagement of Endometrial Leiomyomatosis Milton H. Goldrath, M.D., and Mujtaba Husain, M.D.
Abstract Uterine myomata are frequently the cause of abnormal uterine bleeding. They may be removed successfully by hysteroscopy. As a rule, the myomata are single, although on occasion several pedunculated myomas may be present. Three women were diagnosed with extremely large numbers of submucous leiomyomata. Many of the lesions were removed by hysteroscopic resection and many were destroyed with the neodymium:yttrium-aluminumgarnet laser.
anemia since menarche. She had been taking oral contraceptives for 10 years with slight improvement. Dilatation and curettage (D&C) 2 years earlier yielded four small submucous myomas 0.5 cm in diameter. The uterus was myomatous, the size of a 6-week pregnancy. Hemoglobin was 10 g. Office hysteroscopy was difficult because of bleeding, but a pedunculated submucous myoma could be seen. Because of the severity of bleeding, the woman was admitted to the hospital, Laminaria was inserted into the cervix, and the next day she underwent hysteroscopic removal of 18 myomata measuring 0.9 to 3.0 cm in diameter. Other myomas were felt within the uterus, but because of profuse bleeding it was decided to stop the procedure. A Foley catheter was inserted into the uterus for tamponade. The patient's hemoglobin dropped to 7 g. The catheter was removed the next day and the patient was discharged home taking oral iron therapy.
The rapidly expanding use of the hysteroscope for the evaluation and treatment of abnormal uterine bleeding has led to the diagnosis of many pedunculated submucous leiomyomata. 1 3 Usually these are solitary, but on occasion, two or three pedunculated myomas may be seen and treated hysteroscopically. In the past few years we have had the occasion to evaluate and successfully treat three women with innumerable submucous leiomyomata. We were unable to find any similar cases in the literature. This entity is of particular concern, as hysteroscopy is required for proper diagnosis and treatment.
Case Reports Patient No. 1 A 31-year-old woman experienced profuse menorrhagia of 8 months' duration. She had von Willebrand disease, which caused her to have menorrhagia with
From the Departments of Obstetrics and Gynecology (Dr. Goldrath) and Pathology (Dr. Husain), Wayne State University School of Medicine, and Section of Gynecology (Dr. Goldrath) and Department of Laboratory Medicine (Dr. Husain), Sinai Hospital, Detroit, Michigan. Address reprint requests to Milton H. Goldrath, M.D., Department of Obstetrics and Gynecology, Sinai Hospital, 6767 West Outer Drive, Detroit, MI 48235; fax 313 493 7503.
263
Hysteroscopic Management of Endometrial Leiomyomatosis Goldrath and Husain
Sixteen months later she returned stating that her menses were becoming heavier. Office hysteroscopy revealed one sizable myoma in the fundus as well as several tiny myomas. There were no synechiae. She was prescribed danazo1800 mg/day. The couple elected not to proceed with any assisted reproductive methods. Sixteen months later the woman returned still taking danazol and with a moderate amount of breakthrough bleeding. Office hysteroscopy revealed numerous tiny myomas and submucous myomas in the cornual areas. Three months later at operative hysteroscopy, more than 50 minute myomas, estimated to be 1 to 3 mm in diameter, were destroyed and the larger submucous myomas were resected with the Nd:YAG laser. Two months postoperatively office hysteroscopy revealed no myomas. Two broad synechiae were seen and easily broken up with the hysteroscope. A total of 103 myomas had been removed and more than 150 destroyed with the Nd:YAG laser. Two further contacts, almost a year apart, were made with the patient by telephone. Her menses were normal and she had still not conceived.
Her menorrhagia improved. Several office hysteroscopies revealed numerous small submucous myomas (Figure 1), and over the next 6 months 15 were removed in the office with polyp forceps; however, hysteroscopy revealed many remaining. Menses soon became heavy again and the patient was prescribed danazo1800 mg/day in an attempt to delay definitive hysteroscopic surgery until she decided to bear children. She informed us that she was married 7 months later and, following our advice, hoped to conceive as soon as the myomas were hysteroscopically removed. Two months after her marriage she was still taking danazol and experiencing very little bleeding. At outpatient operative hysteroscopy many minute myomas were seen and destroyed with the neodymium: yttrium-aluminum-garnet (Nd:YAG) laser. Forty-nine myomas were removed with polyp forceps. The patient was discharged the same day with instructions to stop danazol and start conjugated estrogen 2.5 mg/day for 60 days. She was told to take medroxyprogesterone acetate 5 mg/day from day 51 through day 60 to produce orderly withdrawal bleeding. After the withdrawal period, office hysteroscopy was normal and the woman was instructed to attempt conception. Three months later office hysteroscopy was again performed; no myomas were seen and the patient was having relatively normal menses. Five months later she had not become pregnant. Her menses remained relatively normal, and office hysteroscopy revealed no myomas. Infertility work-up revealed her husband to be oligospermic.
Patient No. 2 A 31-year-old gravida 1, para 0, abortus 1 (induced) woman had a 4-year history of profuse menorrhagia. A D&C performed in the past was not helpful. On examination her uterus was myomatous and approximately the size of an 8-week pregnancy. Office hysteroscopy revealed a large, mostly pedunculated submucous myoma on the posterior wall. Other small myomas were seen in the fundus. At vaginal myomectomy using Laminaria to dilate the cervix, seven myomas measuring 1.0 to 3.0 cm were removed. Others were believed still to be present. Six weeks later the woman's menses had improved. Office hysteroscopy revealed eight small, pedunculated myomas in the endometrial cavity. Since she did not wish to conceive at that time, danazo1800 mg/day was prescribed, but the patient decided not to take it. Approximately 1 year later she returned stating her menses were profuse. Office hysteroscopy revealed 10 to 15 submucous myomas, several quite large. She still did not wish to conceive. She refused hysteroscopic resection and oral contraception. One year later she was again seen, again with profuse menses. Her uterus was myomatous and the size of a 6-week pregnancy. Office hysteroscopy revealed more than 15 submucous myomas. She was given danazol 800 mg/day, and 1 month later underwent
FIGURE 1. Office hysteroscopic view of patient no. 1 while taking danazol. Note the various-size myomata, including seedlings.
264
February ] 997, Vol. 4, No. 2
The Journal of the American Association of Gynecologic Laparoscopists
resection o f 10 myomas. Postoperatively, conjugated estrogen and medroxyprogesterone acetate in a regimen similar to that of patient 1 were prescribed. One month later office hysteroscopy revealed a midline synechia. The tubal ostia were seen. There was a myoma in the right horn. The patient refused resection of the myoma at that time. Six months later she complained of profuse bleeding and was treated with a gonadotropin-releasing hormone analog. After 4 months office hysteroscopy disclosed 20 to 30 myomata. The tubal ostia were seen. At operative hysteroscopy performed 1 month later, 25 small myomas were destroyed, the midline synechia was lysed with the Nd:YAG laser, and 10 myomas were removed with polyp forceps. She was prescribed the same hormone regimen as patient no. 1. A total of 25 myomas had been destroyed with the Nd:YAG laser and 27 removed. The woman was next seen two months later after a normal withdrawal period. Office hysteroscopy revealed normal-appearing endometrium and no synechiae or myomata. She was advised to conceive and did so 6 months later. She spotted during the first trimester, and the fibroids grew quite large. She had many contractions during the pregnancy and was treated with bedrest and terbutaline. Spontaneous rupture of membranes occurred at 37 weeks and a cesarean section was performed because of myomas that prevented the application of the fetal head to the cervix. Cesarean section was described as being difficult because of the myomas. Postpartum the patient had considerable pain and bleeding. When menses recurred, they were heavy, and a total abdominal hysterectomy was performed 4 months postpartum. The uterus weighed 460 g. Submucous myomas were apparent and were quite large (Figure 2).
FIGURE 2. Hysterectomy specimen from patient 2. Hysterectomy was performed 4 months postpartum. Note large submucous myomata.
months later. The rationale for delay was to remove the myomas and have the patient conceive shortly thereafter. At operative hysteroscopy 20 minute myomas were destroyed and 39 were resected with the Nd:YAG laser. The patient was prescribed conjugated estrogen 5 mg for 60 days with medroxyprogesterone acetate 5 mg from days 51 to 60. Repeat treatment was planned, as all myomas were not destroyed. This was purposely staged to avoid extensive destruction of the endometrium with subsequent adhesion formation. Hysteroscopy 3 months later revealed a few small synechiae plus several submucous myomata. Operative hysteroscopy was done later that month, destroying 20 seedling myomas and resecting 7 with the Nd:YAG laser. The synechiae were lysed. The same regimen of conjugated estrogens and medroxyprogesterone acetate was prescribed. Three months later office hysteroscopy revealed a normal cavity with narrow horns. The endometrium appeared normal, and no synechiae or myomata were present. The tubal ostia were seen and were open. The shoulder sign was positive. The patient was advised to conceive as promptly as possible. A total of 46 myomas were removed and 40 were destroyed with the Nd:YAG laser. The woman conceived four months later and was delivered at 38 weeks by cesarean section for a complete placenta previa. Placenta accreta was present. No submucous myomas were noted in the cesarean section surgical record.
Patient No. 3 A 31-year-old gravida 2, para 0, abortus 2 (induced) woman was first seen because of 2 years of profuse menorrhagia. She had been taking oral contraceptives for 2 years in an attempt to control the bleeding. At D&C performed 1 month previously three small myomas were removed. It was noted that another remained. Office hysteroscopy revealed more than 20 small pedunculated myomata and many minute submucosal ones. The patient was prescribed danazol 800 mg/day to control bleeding until surgery was performed several
265
Hysteroscopic Management of Endometrial Leiomvomatosis Gddrath and Husain
At the 6 weeks postpartum the woman's uterus was said to be of normal size, and she was treated with oral contraceptives. Her menses were normal for the next 4 months. She is contemplating another pregnancy in the near future.
The hysterectomy specimen from patient 2 showed a few large intramural myometrial nodules and, in a few areas, multiple nodules, possibly representing diffuse leiomyomatosis. The endometrium was not reported to be significantly involved.
Specimen Pathology
Discussion
All endoscopically resected nodules had common morphologic findings. They ranged from 1.0 to 30.0 ram. Some of the smaller ones were totally surrounded by endometrium and others were partially covered by identifiable endometrial tissue. The nodules were not oriented around vascular structures. Unduly prominent mitotic activity or nuclear crowding was not observed. Features of infarction or necrosis were also lacking (Figures 3 and 4).
Large numbers of minute leiomyomata are reminiscent of the condition known as diffuse leiomyomatosis of the uterus. This term was first used by Lapan and Solomon in 1979, 4 who found three similar cases reported in the literature and reported two of their own. Since that time there have been seven additional cases reported. The largest series reported four cases and provided a detailed pathologic description of the lesions. 5 Endometrial leiomyomatosis and diffuse leiomyomatosis of the uterus are similar in that both conditions have innumerable numbers of tiny myomas. These myomas are benign. The similarity ends there. Diffuse leiomyomatosis of the uterus is characterized by a large uterus, up to 1200 g. In the eight cases that mentioned the endometrium, the endometrium was free of myomataJ -7 Two of three cases contained submucous myomata, but they were not described as being pedunculated. 8They may be similar to our patients. In endometrial leiomyomatosis, by our definition, the endometrium is extensively involved. The uteri in our patients were small on initial examination, none larger than 6 to 8 weeks' gestation. Obviously other myomas were present. The hysterectomy specimen from the second patient was quite large (460 g) and generally contained large myomata. In some areas the findings were compatible with diffuse leiomyomatosis. Since the other two women did not undergo hysterectomy, we do not know if diffuse leiomyomatosis is present within their myometrium, although this is doubtful because the uteri did not grow very large. Patients 1 and 3 who did not have a hysterectomy are doing quite well. Whether they have diffuse leiomyomatosis is not readily apparent. Ultrasonic examinations showed only small leiomyomata rather than a diffuse process. Since at the present time there is no indication for hysterectomy in these women, the histology of the myometrium remains unknown. Although all three women had voluntarily not borne children when first seen, the second and third had had previous voluntary terminations of pregnancy. Whether endometrial leiomyomatosis was present at
FIGURE 3. Low-power photomicrograph shows small leiomyoma covered by endometrium on the surface.
FIGURE 4. Medium-power photomicrograph shows minute leiomyoma in the endometrium deep to a gland.
266
February 1997, VoJ.4, No. 2
TheJournaJof the American Association of GynecoJogic Laparoscopists
the time they were pregnant is unknown; however, these terminations did antedate the onset of severe menorrhagia. Therapy was directed toward controlling menorrhagia with preservation of reproductive capability. This was achieved in all three patients for various periods of time. There was some recurrence of the myomas over the treatment period. The first patient, unfortunately, did not conceive, presumably due to oligospermia. The other two did conceive. The surgical approach to these myomas by operative hysteroscopy is worthy of discussion. In the first two women the submucous myomata seen hysteroscopically were of quite good size, and we elected to perform vaginal myomectomy by dilating the cervix with Laminaria and using polyp forceps. 2'3Follow-up of those patients and primary approach in the third involved using the Nd:YAG laser to remove the tiny myomas with as little destruction of the endometrium as possible. In attempting to expose as many minute submucous myomas as possible, the patients were treated preoperatively with danazol 800 mg/day to produce atrophy of the endometrium. Immediately after treatment with the Nd:YAG laser, they were given large dosages of estrogen for 60 days and withdrawn with l0 days of medroxyprogesterone acetate on days 51 through 60. This was an attempt to prevent synechiae from forming between larger areas of the damaged endometrium. There were far too many myomas present to avoid destroying them on the opposing surfaces as a method of preventing synechiae. We were quite successful in all these patients. Only a few synechiae formed in patients 2 and 3, and these were easily treated at the time of subsequent destruction of submucous myomata. We did not elect to use an intrauterine catheter or intrauterine device to prevent adhesions. It has been our approach to administer large doses of estrogen to encourage rapid endometrial regrowth, and the success of the regimen is attested to by these patients.
These cases are noteworthy in that they demonstrate that hysteroscopic surgery can be successful even if it takes great perseverance. Two of the women became pregnant and delivered living children, however, patient no. 2 required a hysterectomy. The third woman is looking forward to a second pregnancy; the first hopes to conceive. References
. Goldrath MH, Sherman A: Office hysteroscopy and suction curettage: Can we eliminate the hospital diagnostic dilatation and curettage? Am J Obstet Gyneco1220-229, 1985 . Goldrath MH: Vaginal removal of the pedunculated submucous myoma: The use ofLaminaria. Obstet Gynecol 70:670-672, 1987 . Goldrath MH: Vaginal removal of the pedunculated submucous myoma: Historical observations and development of a new procedure. J Reprod Med 35(10):921-924, 1990 . Lapan B, Solomon L: Diffuse leiomyomatosis of the uterus precluding myomectomy. Obstet Gynecol 53:82S-84S, 1979 . Clement PB, Young RH: Diffuse leiomymatosis of the uterus: A report of four cases. Int J Gynecol Pathol 6:322-330, 1987 . Grignon DJ, Carey MR, Kirk ME, et al: Diffuse uterine leiomyomatosis:A case study with pregnancy complicated by intrapartum hemorrhage. Obstet Gyneco169:477-480, 1987 7. Lai FM, Wong FW, Allen PW: Diffuse uterine leiomyomatosis with hemorrhage. Arch Pathol Lab Med 115:834-837, 1991 8. Fedele L, Zamberletti D, Carivelli S, et al: Diffuse Uterine leiomyomatosis. Acta Eur Fertil 13:125-131, 1982
267