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Primary Ovarian Leiomyoma: A Rare Cause of Ovarian Tumor in Adolescence
J Pediatr Adolesc Gynecol (2008) 21:33e36
Case Reports Primary Ovarian Leiomyoma: A Rare Cause of Ovarian Tumor in Adolescence Christina Wei, MD1, Nenad Lilic, MD2, Nicholas Shorter, MD1, and Eugene Garrow, MD1 1
Division of Pediatric Surgery, Downstate Medical Center of the State University of New York; 2Department of Pathology, Kings County Hospital Center, Brooklyn, New York, USA
Abstract. A pediatric case of ovarian leiomyoma is presented. A review of the literature indicates that, in contrast to older patients, pediatric/young adult patients usually present with bilateral disease and do not have associated uterine leiomyomas.
Primary ovarian leiomyoma is a rare benign tumor. Less than 50 cases have been reported,1,2 and it is only occasionally seen in the pediatric age group. We present a case of unilateral ovarian leiomyoma in a 13-year-old.
tissue. The mass was easily separated from the ovary, leaving a defect in the capsule, which was repaired. The left ovary was normal by inspection and was left intact. The uterus was normal. The postoperative course was uneventful. Pathology evaluation identified the mass as an ovarian leiomyoma, consisting of numerous, cavernous vascular channels, composed of interlacing smooth muscle bundles, interspersed with solid areas of leiomyoma. No nuclear atypia or mitoses were present. Fresh blood and organized thrombi filled cavernous trabeculae (Figs. 2 and 3). Focal acute hemorrhagic infarction accounted for the acute clinical presentation, and focal myxoid degeneration was present. Immunohistochemical stains for alphasmooth muscle actin and muscle-specific actin (HHF-35) were positive (Fig 3, inset). This tumor has the distinct morphologic features of a vascular leiomyoma.
Case Report
Discussion
A 13-year-old female was admitted with a one-day history of intermittent right lower quadrant abdominal pain associated with nausea and vomiting. On physical examination, she was found to have right lower quadrant tenderness without guarding. Rectal examination showed tenderness in the right side of the cul de sac. The white blood cell count was 10900/mm3, and the urinary pregnancy test was negative. An ultrasound study of the pelvis and a confirming CT scan showed a heterogeneous, enhancing pelvic mass, 31mm 19mm 20mm in size (Fig. 1). At laparotomy, there was a solid, vascular-appearing mass which was attached to the right ovary by only a narrow bridge of Address correspondence to: Eugene Garrow, MD, SUNY Downstate Medical Center, Brooklyn, NY 11203; E-mail: egarrow@ downstate.edu
Ovarian leiomyoma, a rare benign tumor, is most often seen in premenopausal women (up to 85% of cases)3,4 and accounts for 0.5e1% of all benign ovarian neoplasms.1,5 There are a number of theories of the origin of these tumors. They most likely arise from smooth muscle cells in the ovarian hilar blood vessels, but other possible origins include cells in the ovarian ligament, smooth muscle cells or multipotential cells in the ovarian stroma, undifferentiated germ cells, or cortical smooth muscle metaplasia.2,6 The youngest patient reported to date with ovarian leiomyoma is a 12-year-old who presented with a left ovarian leiomyosarcoma associated with bilateral leiomyoma.7 However, the significance of this case is unclear because she had a history of medulloblastoma, and her ovaries were exposed to radiation as part of her therapy, so this patient has not been included in
Ó 2008 North American Society for Pediatric and Adolescent Gynecology Published by Elsevier Inc.
1083-3188/08/$34.00 doi:10.1016/j.jpag.2007.07.001
Key Words. Ovarian leiomyoma—Pediatric ovarian tumor
Introduction
34
Wei et al: Primary Ovarian Leiomyoma
Fig. 1. Pelvic CT scan revealing a heterogeneous enhancing soft tissue mass in the right adnexal region.
our analysis. The next youngest reported patients prior to our report are two 16-year-olds, both with bilateral disease. For a summary of the pediatric/young adult cases, see Table 1. These tumors may be found by chance during routine physical examination or incidentally at surgery or autopsy.8 A variety of clinical presentations has been described: (1) no symptoms; (2) abdominal pain; (3) a palpable mass; (4) hydrothorax and/or ascites; (5) hydronephrosis; (6) slightly elevated tumor marker, CA-125.3,9e13 Case reports show a unilateral predominance, with no predilection for left or right ovary.4 However, of the eight reported bilateral cases, seven occurred in patients between the ages of 16 and 25, and with the exception of our patient, all the patients in the pediatric/young adult group have had bilateral disease.2,8,9,12e16 No bilateral cases have been reported in patients over the age of 35. Unilateral ovarian leiomyomas are frequently associated with other ipsilateral or contralateral ovarian lesions.1,2 This association was found in 40% of the
Fig 2. Dilated vascular channels filled with fresh blood and recent organized thrombi (H & E 40).
Fig. 3. (A) Delicate smooth muscle bundles outline vascular channels (H & E 200). (B) Marked immunoreactivity for smooth muscle actin ( 200).
cases (mean age 45.8 years) reported by Doss et al.1 Ovarian leiomyomas also often co-exist with uterine leiomyomas3,4,17 and are indistinguishable macroscopically and microscopically from them; but uterine fibroids are common, so the association may be coincidental. Alternatively, this could reflect a common etiology or similar hormonal responsiveness. The uterus was normal in all reported cases of patients aged 16e25 2,8,9,12e14,16 and was normal in our patient. Interestingly, many of the reported patients with ovarian leiomyomas at all ages were nulligravidas (10 out of the 14 patients in which an obstetrical history was given).5,11,17 In the pediatric/young adult age group, all were nulligravid.8,12,13 This suggests that estrogen may play an important role in the development and/or maintenance of these tumors (further evidence of the hormonal responsiveness of these tumors comes from a case identified at the time of caesarian section, in which the residual tumors regressed
Table 1. Pediatric/Young Adult Cases of Ovarian Leiomyoma Author
Age
Presenting complaints
Laboratory findings 3
Laparotomy findings
13
One day history of intermittent right lower quadrant pain associated with nausea and vomiting
WBC: 10900/mm Beta HCG: neg
Danihel16
16
History of polymenorrhea. Presented with pain in the left hypogastrium and a palpable mass in the left adnexal region.
Not available.
San Marco9
16
History of pelvic pain and increased abdominal girth.
CA125: wnl AFP: wnl CEA: wnl Beta HCG: neg
Lim8
17
Lower abdominal palpable mass. Irregular menses three months prior to admission.
Various tumor markers all wnl.
Kandalaft12
21
Mild abdominal pain.
CA-125: 103U/ml
Macri13
21
Lower abdominal pain and increasing girth of several months duration.
CEA-125: 103.5 U/ml. AFP: wnl LD: wnl Beta HCG: neg
Emovon2
24
Central abdominal pain, nausea and vomiting.
Not available.
Some small (!1 cm) nodules on right ovary. A hard, lobulated 10 cm left ovary.
Seinera14
25
Asymptomatic. Bilateral ovarian cysts and a uterine fibroid seen on pelvic ultrasonography (Subsequently, these findings were found to be erroneous).
Not available.
Both ovaries had a number of smooth, solid tumors, two of which were pedunculated. Uterus was normal.
Enucleation of the tumor from the right ovary.
Uneventful.
Left salpingo-oophorectomy. Partial right oophorectomy with tumor enucleation.
Patient menstrual cycle returned to normal.
Right salpingo-oophorectomy. Partial left oophorectomy.
30 months later the left ovary had enlarged 3 times its previous size.
Bilateral salpingo-oophorectomy.
Uneventful.
Bilateral oophorectomy and left salpingectomy. Complete removal of abnormal appearing right adnexa via a retroperitoneal approach with resection of the gonadal vessels and target precaval lymph nodes. Left oophorectomy after intraoperative biopsies failed to identify any normal stroma. Left oophorectomy, omentectomy and biopsies from the right ovary.
Not available.
Laparoscopic inspection of the abdomen converted to laparotomy. Tumors in the ovaries were localized by manual palpation and subsequently enucleated. The ovaries were reconstructed.
Patient was placed on OrthoNovum 1/35 for HRT. She remained asymptomatic and disease free after 2 years.
At 7th months, complained of increasing abdominal pain and girth. Ultrasound showed a 5x3 multicystic ovarian mass. Right oophorectomy was performed. Uneventful.
35
WNL5 within normal limit.
A solid, vascular-appearing mass that was attached to the right ovary by only a narrow bridge of tissue. Uterus and left ovary were normal. Multiple tumors in the left ovary with attachment to the left fallopian tube. The right ovary had a tumor with a sharply demarcated border. The right fallopian tube and the uterus appeared normal. A large tumor growing from the right ovary and a small tumor from the left ovary. Uterus was normal. Bilateral solid ovarian masses distinctly separate from the uterus. No adhesions, no infiltration of the surrounding structures. Uterus was normal. Bilateral large ovarian masses. Uterus was normal. 200mL of bloody ascites. Bilateral, irregular, solid ovarian tumors with an extensive parasitic blood supply and adhesions. Uterus and fallopian tubes appeared normal.
Post-operative course
Wei et al: Primary Ovarian Leiomyoma
Wei
Surgical procedure
36
Wei et al: Primary Ovarian Leiomyoma
following the pregnancy15). Another possibility is that some of these tumors may arise in developmentally abnormal ovaries, a possibility supported by the finding in some cases that there is no recognizable normal ovarian tissue present histologically.8,13 The gross characteristics of these tumors are variable. Many are small in size, but on occasion they can be very large. While some are solid, others have cystic components or are predominantly cystic. Hemorrhage, calcification and/or hyalinization are sometimes present. The radiographic imaging features of these tumors have been correspondingly mixed, and the heterogeneous, complex nature of many of these tumors, particularly the large ones, often suggests malignancy, particularly when there is associated ascites.6 Hysterectomy and bilateral salpingo-oophorectomy has been the usual surgical treatment in middle-aged and older patients. In the younger patients presenting with bilateral ovarian leiomyomas, bilateral oophorectomy has often been required, with uterine and, sometimes, tubal preservation.2,3,6,8,9 We found four previously reported cases of ovarian sparing procedures.2,9,14,15 In a fifth case13 an attempt at ovarian preservation was abandoned when multiple ovarian biopsies failed to identify any normal stroma. In our patient, the pedunculated nature of the tumor made ovarian preservation simple. Once the smooth muscle nature of the tumor is known, leiomyoma must be differentiated from leiomyosarcoma. The histologic features of malignancy have not been well defined due to the rarity of these tumors. Pathologists have traditionally utilized criteria that emphasize the level of mitotic activity.1 However, Prayson and Hart4 have described two cases of mitotically active ovarian leiomyoma which had a subsequent clinically benign course. Clearly, the significance of other factors such as necrosis and cellular atypia, which are now used in the evaluation of uterine smooth muscle tumors,18 needs to be determined. Other smooth muscle processes that can involve the ovary include parasitic uterine leiomyomas, ovarian smooth muscle metaplasia, intravenous leiomyomatosis, and leiomyomatosis peritonealis disseminata.4 Leiomyomas can also arise adjacent to the ovary in the broad ligament. Given the age of our patient, long term follow-up will be important in detecting tumor recurrence or the development of asynchronous bilaterality. The information presented here suggests that a risk of subsequent contralateral disease exists. The prognosis in older patients appears to be excellent, but, due to its rarity, little is known about the long term prognosis of this tumor in young patients. In summary, there are two important differences in the presentation of ovarian leiomyoma in the pediatric/
young adult population when compared to older adults. Pediatric/young adult patients tend to present with bilateral ovarian leiomyoma, and uterine fibroids are not found to co-exist.
References 1. Doss BJ, Wanek SM, Jacques SM, et al: Ovarian leiomyomas: clinicopathologic features in fifteen cases. Int J Gynecol Pathol 1999; 18:63 2. Emovon EU, Cahill DJ, Joels LA, et al: Bilateral primary ovarian leiomyoma: a rare cause of ovarian tumour in a young woman. J Obstet Gynecol 1999; 19:676 3. Pandit MJ, Watson NR, Mackenzie IZ: Leiomyoma of the ovary. J Obstet Gynaecol 1997; 17:503 4. Prayson RA, Hart WR: Primary smooth-muscle tumors of the ovary: a clinicopathologic study of four leiomyomas and two mitotically active leiomyomas. Arch Pathol Lab Med 1992; 116:1068 5. Van Winter JT, Stanhope CR: Giant ovarian leiomyoma associated with ascites and polymyositis. J Obstet Gynecol 1992; 80:560 6. Kim JC, Nam SL, Suh KS: Leiomyoma of the ovary mimicking mucinous cystadenoma. J Clin Imaging 2000; 24:34 7. Monk BJ, Nieberg R, Berek JS: Primary leiomyosarcoma of the ovary in a perimenarchal female. Gynecol Oncol 1993; 48:389 8. Lim S, Jeon H: Bilateral primary ovarian leiomyoma in a young woman: case report and literature review. Gynecol Oncol 2004; 95:733 9. San Marco L, Londero F, Stefanutti V, et al: Ovarian leiomyoma. Case report. Clin Exp Obstet Gynecol 1991; 18:145 10. Nicoll JJ, Cox PJ: Leiomyoma of the ovary with ascites and hydrothorax. Am J Obstet Gynecol 1989; 161:177 11. Khaffaf N, Khaffat H, Wukwtich S: Giant ovarian leiomyoma as a rare cause of acute abdomen and hydronephrosis. Obstet Gynecol 1996; 87:872 12. Kandalaft PL, Esteban JM: Bilateral massive ovarian leiomyomata in a young woman: a case report with review of the literature. Mod Pathol 1992; 5:586 13. Macri CI, Vasilev SA: Bilateral giant primary ovarian leiomyoma. J Reprod Med 1993; 38:480 14. Seinera P, Raspollini M, Privitera S: Bilateral ovarian leiomyoma. Acta Obstet Gynecol Scand 1997; 76:488 15. Daniel Y, Lessing JB, Bar-Am A, et al: Treatment of bilateral multiple primary ovarian leiomyomas during pregnancy by way of conservative surgery: a case report. Eur J Obstet Gynecol Reprod Biol 1997; 74:125 16. Danihel L, Losch A, Kainz C, et al: Bilateral primary leiomyoma of the ovary. Wien Klin Wochenschr 1995; 107:436 17. Fallahzadeh H, Dockerty MB, Lee RA: Leiomyoma of the ovary: a report of five cases and review of the literature. Am J Obstet Gynecol 1972; 113:394 18. Bell SW, Kempson RL, Hendrickson MR: Problematic uterine smooth muscle neoplasms: a clinicopathologic study of 213 cases. Am J Surg Pathol 1994; 18:535
Case Reports Primary Ovarian Leiomyoma: A Rare Cause of Ovarian Tumor in Adolescence Christina Wei, MD1, Nenad Lilic, MD2, Nicholas Shorter, MD1, and Eugene Garrow, MD1 1
Division of Pediatric Surgery, Downstate Medical Center of the State University of New York; 2Department of Pathology, Kings County Hospital Center, Brooklyn, New York, USA
Abstract. A pediatric case of ovarian leiomyoma is presented. A review of the literature indicates that, in contrast to older patients, pediatric/young adult patients usually present with bilateral disease and do not have associated uterine leiomyomas.
Primary ovarian leiomyoma is a rare benign tumor. Less than 50 cases have been reported,1,2 and it is only occasionally seen in the pediatric age group. We present a case of unilateral ovarian leiomyoma in a 13-year-old.
tissue. The mass was easily separated from the ovary, leaving a defect in the capsule, which was repaired. The left ovary was normal by inspection and was left intact. The uterus was normal. The postoperative course was uneventful. Pathology evaluation identified the mass as an ovarian leiomyoma, consisting of numerous, cavernous vascular channels, composed of interlacing smooth muscle bundles, interspersed with solid areas of leiomyoma. No nuclear atypia or mitoses were present. Fresh blood and organized thrombi filled cavernous trabeculae (Figs. 2 and 3). Focal acute hemorrhagic infarction accounted for the acute clinical presentation, and focal myxoid degeneration was present. Immunohistochemical stains for alphasmooth muscle actin and muscle-specific actin (HHF-35) were positive (Fig 3, inset). This tumor has the distinct morphologic features of a vascular leiomyoma.
Case Report
Discussion
A 13-year-old female was admitted with a one-day history of intermittent right lower quadrant abdominal pain associated with nausea and vomiting. On physical examination, she was found to have right lower quadrant tenderness without guarding. Rectal examination showed tenderness in the right side of the cul de sac. The white blood cell count was 10900/mm3, and the urinary pregnancy test was negative. An ultrasound study of the pelvis and a confirming CT scan showed a heterogeneous, enhancing pelvic mass, 31mm 19mm 20mm in size (Fig. 1). At laparotomy, there was a solid, vascular-appearing mass which was attached to the right ovary by only a narrow bridge of Address correspondence to: Eugene Garrow, MD, SUNY Downstate Medical Center, Brooklyn, NY 11203; E-mail: egarrow@ downstate.edu
Ovarian leiomyoma, a rare benign tumor, is most often seen in premenopausal women (up to 85% of cases)3,4 and accounts for 0.5e1% of all benign ovarian neoplasms.1,5 There are a number of theories of the origin of these tumors. They most likely arise from smooth muscle cells in the ovarian hilar blood vessels, but other possible origins include cells in the ovarian ligament, smooth muscle cells or multipotential cells in the ovarian stroma, undifferentiated germ cells, or cortical smooth muscle metaplasia.2,6 The youngest patient reported to date with ovarian leiomyoma is a 12-year-old who presented with a left ovarian leiomyosarcoma associated with bilateral leiomyoma.7 However, the significance of this case is unclear because she had a history of medulloblastoma, and her ovaries were exposed to radiation as part of her therapy, so this patient has not been included in
Ó 2008 North American Society for Pediatric and Adolescent Gynecology Published by Elsevier Inc.
1083-3188/08/$34.00 doi:10.1016/j.jpag.2007.07.001
Key Words. Ovarian leiomyoma—Pediatric ovarian tumor
Introduction
34
Wei et al: Primary Ovarian Leiomyoma
Fig. 1. Pelvic CT scan revealing a heterogeneous enhancing soft tissue mass in the right adnexal region.
our analysis. The next youngest reported patients prior to our report are two 16-year-olds, both with bilateral disease. For a summary of the pediatric/young adult cases, see Table 1. These tumors may be found by chance during routine physical examination or incidentally at surgery or autopsy.8 A variety of clinical presentations has been described: (1) no symptoms; (2) abdominal pain; (3) a palpable mass; (4) hydrothorax and/or ascites; (5) hydronephrosis; (6) slightly elevated tumor marker, CA-125.3,9e13 Case reports show a unilateral predominance, with no predilection for left or right ovary.4 However, of the eight reported bilateral cases, seven occurred in patients between the ages of 16 and 25, and with the exception of our patient, all the patients in the pediatric/young adult group have had bilateral disease.2,8,9,12e16 No bilateral cases have been reported in patients over the age of 35. Unilateral ovarian leiomyomas are frequently associated with other ipsilateral or contralateral ovarian lesions.1,2 This association was found in 40% of the
Fig 2. Dilated vascular channels filled with fresh blood and recent organized thrombi (H & E 40).
Fig. 3. (A) Delicate smooth muscle bundles outline vascular channels (H & E 200). (B) Marked immunoreactivity for smooth muscle actin ( 200).
cases (mean age 45.8 years) reported by Doss et al.1 Ovarian leiomyomas also often co-exist with uterine leiomyomas3,4,17 and are indistinguishable macroscopically and microscopically from them; but uterine fibroids are common, so the association may be coincidental. Alternatively, this could reflect a common etiology or similar hormonal responsiveness. The uterus was normal in all reported cases of patients aged 16e25 2,8,9,12e14,16 and was normal in our patient. Interestingly, many of the reported patients with ovarian leiomyomas at all ages were nulligravidas (10 out of the 14 patients in which an obstetrical history was given).5,11,17 In the pediatric/young adult age group, all were nulligravid.8,12,13 This suggests that estrogen may play an important role in the development and/or maintenance of these tumors (further evidence of the hormonal responsiveness of these tumors comes from a case identified at the time of caesarian section, in which the residual tumors regressed
Table 1. Pediatric/Young Adult Cases of Ovarian Leiomyoma Author
Age
Presenting complaints
Laboratory findings 3
Laparotomy findings
13
One day history of intermittent right lower quadrant pain associated with nausea and vomiting
WBC: 10900/mm Beta HCG: neg
Danihel16
16
History of polymenorrhea. Presented with pain in the left hypogastrium and a palpable mass in the left adnexal region.
Not available.
San Marco9
16
History of pelvic pain and increased abdominal girth.
CA125: wnl AFP: wnl CEA: wnl Beta HCG: neg
Lim8
17
Lower abdominal palpable mass. Irregular menses three months prior to admission.
Various tumor markers all wnl.
Kandalaft12
21
Mild abdominal pain.
CA-125: 103U/ml
Macri13
21
Lower abdominal pain and increasing girth of several months duration.
CEA-125: 103.5 U/ml. AFP: wnl LD: wnl Beta HCG: neg
Emovon2
24
Central abdominal pain, nausea and vomiting.
Not available.
Some small (!1 cm) nodules on right ovary. A hard, lobulated 10 cm left ovary.
Seinera14
25
Asymptomatic. Bilateral ovarian cysts and a uterine fibroid seen on pelvic ultrasonography (Subsequently, these findings were found to be erroneous).
Not available.
Both ovaries had a number of smooth, solid tumors, two of which were pedunculated. Uterus was normal.
Enucleation of the tumor from the right ovary.
Uneventful.
Left salpingo-oophorectomy. Partial right oophorectomy with tumor enucleation.
Patient menstrual cycle returned to normal.
Right salpingo-oophorectomy. Partial left oophorectomy.
30 months later the left ovary had enlarged 3 times its previous size.
Bilateral salpingo-oophorectomy.
Uneventful.
Bilateral oophorectomy and left salpingectomy. Complete removal of abnormal appearing right adnexa via a retroperitoneal approach with resection of the gonadal vessels and target precaval lymph nodes. Left oophorectomy after intraoperative biopsies failed to identify any normal stroma. Left oophorectomy, omentectomy and biopsies from the right ovary.
Not available.
Laparoscopic inspection of the abdomen converted to laparotomy. Tumors in the ovaries were localized by manual palpation and subsequently enucleated. The ovaries were reconstructed.
Patient was placed on OrthoNovum 1/35 for HRT. She remained asymptomatic and disease free after 2 years.
At 7th months, complained of increasing abdominal pain and girth. Ultrasound showed a 5x3 multicystic ovarian mass. Right oophorectomy was performed. Uneventful.
35
WNL5 within normal limit.
A solid, vascular-appearing mass that was attached to the right ovary by only a narrow bridge of tissue. Uterus and left ovary were normal. Multiple tumors in the left ovary with attachment to the left fallopian tube. The right ovary had a tumor with a sharply demarcated border. The right fallopian tube and the uterus appeared normal. A large tumor growing from the right ovary and a small tumor from the left ovary. Uterus was normal. Bilateral solid ovarian masses distinctly separate from the uterus. No adhesions, no infiltration of the surrounding structures. Uterus was normal. Bilateral large ovarian masses. Uterus was normal. 200mL of bloody ascites. Bilateral, irregular, solid ovarian tumors with an extensive parasitic blood supply and adhesions. Uterus and fallopian tubes appeared normal.
Post-operative course
Wei et al: Primary Ovarian Leiomyoma
Wei
Surgical procedure
36
Wei et al: Primary Ovarian Leiomyoma
following the pregnancy15). Another possibility is that some of these tumors may arise in developmentally abnormal ovaries, a possibility supported by the finding in some cases that there is no recognizable normal ovarian tissue present histologically.8,13 The gross characteristics of these tumors are variable. Many are small in size, but on occasion they can be very large. While some are solid, others have cystic components or are predominantly cystic. Hemorrhage, calcification and/or hyalinization are sometimes present. The radiographic imaging features of these tumors have been correspondingly mixed, and the heterogeneous, complex nature of many of these tumors, particularly the large ones, often suggests malignancy, particularly when there is associated ascites.6 Hysterectomy and bilateral salpingo-oophorectomy has been the usual surgical treatment in middle-aged and older patients. In the younger patients presenting with bilateral ovarian leiomyomas, bilateral oophorectomy has often been required, with uterine and, sometimes, tubal preservation.2,3,6,8,9 We found four previously reported cases of ovarian sparing procedures.2,9,14,15 In a fifth case13 an attempt at ovarian preservation was abandoned when multiple ovarian biopsies failed to identify any normal stroma. In our patient, the pedunculated nature of the tumor made ovarian preservation simple. Once the smooth muscle nature of the tumor is known, leiomyoma must be differentiated from leiomyosarcoma. The histologic features of malignancy have not been well defined due to the rarity of these tumors. Pathologists have traditionally utilized criteria that emphasize the level of mitotic activity.1 However, Prayson and Hart4 have described two cases of mitotically active ovarian leiomyoma which had a subsequent clinically benign course. Clearly, the significance of other factors such as necrosis and cellular atypia, which are now used in the evaluation of uterine smooth muscle tumors,18 needs to be determined. Other smooth muscle processes that can involve the ovary include parasitic uterine leiomyomas, ovarian smooth muscle metaplasia, intravenous leiomyomatosis, and leiomyomatosis peritonealis disseminata.4 Leiomyomas can also arise adjacent to the ovary in the broad ligament. Given the age of our patient, long term follow-up will be important in detecting tumor recurrence or the development of asynchronous bilaterality. The information presented here suggests that a risk of subsequent contralateral disease exists. The prognosis in older patients appears to be excellent, but, due to its rarity, little is known about the long term prognosis of this tumor in young patients. In summary, there are two important differences in the presentation of ovarian leiomyoma in the pediatric/
young adult population when compared to older adults. Pediatric/young adult patients tend to present with bilateral ovarian leiomyoma, and uterine fibroids are not found to co-exist.
References 1. Doss BJ, Wanek SM, Jacques SM, et al: Ovarian leiomyomas: clinicopathologic features in fifteen cases. Int J Gynecol Pathol 1999; 18:63 2. Emovon EU, Cahill DJ, Joels LA, et al: Bilateral primary ovarian leiomyoma: a rare cause of ovarian tumour in a young woman. J Obstet Gynecol 1999; 19:676 3. Pandit MJ, Watson NR, Mackenzie IZ: Leiomyoma of the ovary. J Obstet Gynaecol 1997; 17:503 4. Prayson RA, Hart WR: Primary smooth-muscle tumors of the ovary: a clinicopathologic study of four leiomyomas and two mitotically active leiomyomas. Arch Pathol Lab Med 1992; 116:1068 5. Van Winter JT, Stanhope CR: Giant ovarian leiomyoma associated with ascites and polymyositis. J Obstet Gynecol 1992; 80:560 6. Kim JC, Nam SL, Suh KS: Leiomyoma of the ovary mimicking mucinous cystadenoma. J Clin Imaging 2000; 24:34 7. Monk BJ, Nieberg R, Berek JS: Primary leiomyosarcoma of the ovary in a perimenarchal female. Gynecol Oncol 1993; 48:389 8. Lim S, Jeon H: Bilateral primary ovarian leiomyoma in a young woman: case report and literature review. Gynecol Oncol 2004; 95:733 9. San Marco L, Londero F, Stefanutti V, et al: Ovarian leiomyoma. Case report. Clin Exp Obstet Gynecol 1991; 18:145 10. Nicoll JJ, Cox PJ: Leiomyoma of the ovary with ascites and hydrothorax. Am J Obstet Gynecol 1989; 161:177 11. Khaffaf N, Khaffat H, Wukwtich S: Giant ovarian leiomyoma as a rare cause of acute abdomen and hydronephrosis. Obstet Gynecol 1996; 87:872 12. Kandalaft PL, Esteban JM: Bilateral massive ovarian leiomyomata in a young woman: a case report with review of the literature. Mod Pathol 1992; 5:586 13. Macri CI, Vasilev SA: Bilateral giant primary ovarian leiomyoma. J Reprod Med 1993; 38:480 14. Seinera P, Raspollini M, Privitera S: Bilateral ovarian leiomyoma. Acta Obstet Gynecol Scand 1997; 76:488 15. Daniel Y, Lessing JB, Bar-Am A, et al: Treatment of bilateral multiple primary ovarian leiomyomas during pregnancy by way of conservative surgery: a case report. Eur J Obstet Gynecol Reprod Biol 1997; 74:125 16. Danihel L, Losch A, Kainz C, et al: Bilateral primary leiomyoma of the ovary. Wien Klin Wochenschr 1995; 107:436 17. Fallahzadeh H, Dockerty MB, Lee RA: Leiomyoma of the ovary: a report of five cases and review of the literature. Am J Obstet Gynecol 1972; 113:394 18. Bell SW, Kempson RL, Hendrickson MR: Problematic uterine smooth muscle neoplasms: a clinicopathologic study of 213 cases. Am J Surg Pathol 1994; 18:535