Correspondence
Lipoleiomyoma of uterus and lipoma of broad ligament‑a rare entity ABSTRACT Lipoleiomyoma of uterus are a rare variant of uterine leiomyoma. Clinically the symptoms are indistinguishable from an ordinary leiomyoma. It is diagnosed pre‑operatively as leiomyoma or mature ovarian teratoma. Majority of them are post operative chance finding. Solid tumors of broad ligament are also rare. Most of them are lateral extensions from the uterine tumors. Primary lipomas of broad ligament are rare. They are asymptomatic in majority of cases and are incidental post‑operative finding. We report this case because of the rarity of individual lesions and rarity of the combination and also speculate their histogenesis as adipose tissue is rare absent at both locations. KEY WORDS: Lipoleiomyoma, uterus, broad ligament, lipoma
INTRODUCTION Lipoleiomyoma of uterus ts a rare variant of uterine leiomyoma. Lipoma of broad ligament is still rarer. Less than 30 cases have been reported in literature. The exact pathogenesis of lipoleiomyoma is not known. Lipoleiomyoma consists of variable proportion of mature adipocytes and smooth muscle cells. These tumors generally occur in asymptomatic obese menopausal women. Solid tumors in broad ligament are rare. Those encountered are lateral extension of uterine tumors. A primary lipoma of broad ligament is uncommon since the amount of fat at this location is scanty. Some workers have suggested “a teratogenous element” and “a developmental anomaly” but without substantial proof, the pathogenesis remains inconclusive. CASE REPORT A 61-year-old female (para 4 + 0) presented with abdominal pain for 1 month. The patients menstrual history revealed menarche at the age of 14, regular menstrual cycles, 3-4 days duration at regular intervals of 30 days. She had undergone menopause 15 years ago. Gynaecological examination showed no abnormality of vulva, vagina and vaginal portion of the cervix. The uterine cavity was enlarged corresponding to 6 weeks of gestation. The adnexae were not palpable. Findings of vaginal ultrasonography suggested a well circumscribed 4 cm mass having semi‑solid character located in the fundus of the uterus. An echogenic adnexal mass was appreciated 434
on the left side of the uterus. The endometrium thickness was 1 mm. Both ovaries and tubes were normal. No ascitis was seen in the abdomen. The patient underwent abdominal hysterectomy for mass in fundus. Grossly, a hysterectomy specimen with bilateral salpigo‑oophectomy was received. It measured 10.5 × 6.5 × 3 cm and showed a well‑circumscribed sub‑mucosal mass in the fundus. Endometrium measured 1 mm and myometrium measured 2 cm in thickness. The sub‑mucosal mass measured 4 cm in greatest diameter. Cut surface was soft and yellow. No areas of hemorrhage or necrosis were identified [Figure 1]. A separate nodule measuring 1 cm in diameter was present in the left parametrium of broad ligament. Microscopic examination showed cystic atrophy of the endometrium, senile changes in the myometrium, bilateral fallopian tubes and ovaries within normal limits. The sub‑mucosal mass showed a mixture of spindle‑shaped smooth muscle cells without atypical nuclei in a whorled pattern and mature fat cells in the sub‑mucosal mass [Figure 2]. Nuclei of smooth muscle cells were elongated, had blunt ends, finely dispersed chromatin and small nucleoli [Figure 3]. Bizarre pleomorphic cells, mitotic figures or necrosis were not present. Between the muscle cells significant amount of fat was present. The adipose tissue was completely mature without any lipoblasts. Based on these findings, the tumor was diagnosed as Lipoleiomyoma. Sections from the nodule in the parametrium of the broad ligament revealed a circumscribed lesion composed of uniform sized
Shailja Puri Wahal, Kavita Mardi Department of Pathology, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India For correspondence: Dr. Shailja Puri Wahal, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India. E‑mail: drshailjadoe_11@ ymail.com
Access this article online Website: www.cancerjournal.net DOI: 10.4103/0973-1482.136682 PMID: *** Quick Response Code:
Journal of Cancer Research and Therapeutics - April-June 2014 - Volume 10 - Issue 2
Wahal and Mardi: Lipoleiomyoma of uterus and lipoma of broad ligament-A rare entity
adipocytes [Figure 4]. There was no connection between this lipomatous nodule and the lipoleiomyoma of the uterus. A diagnosis of Lipoma‑Broad ligament was given. DISCUSSION Lipomatous tumors of the uterus include a spectrum of lesions consisting of pure lipomas, lipoleiomyomas and fibromyolipoma.[1] Solid tumors in broad ligament are rare. Those encountered are lateral extension of uterine tumors. A primary lipoma of broad ligament is uncommon.[2] Lipoleiomyoma is a rare uterine fatty tumor. Myolipoma of soft tissue was first described by Meis and Enzinger in 1991. These tumors show characteristic histological findings composed of benign smooth muscle cells and mature adipose tissue. In uterus similar tumors are called lipoleiomyomas.[3] Lipoleiomyomas occur in different locations including cervix. The commonest location in uterus is the fundus.[4] Pathogenesis of lipoleiomyoma remains unclear. There are two main theories, namely adipose metaplasia of smooth muscle cells
and a multipotential mullerian cell origin.[5] These tumors are asymptomatic and are common in obese menopausal women.[1] Various lipid metabolic disorders associated with estrogen deficiency occurring in pre‑ and post‑ menopausal women, possibly promotes intracellular deposition of lipid.[6] An incidence of 0.28% of all leiomyomas and 0.39% of all hysterectomies was reported from National Taiwan University Hospital between January 1994 and December 1998 by K.C. Lin et al. who analysed 2878 leiomyoma cases and 2071 hysterectomies specimens.[7] Although most patients are asymptomatic, they can present with symptoms similar to leiomyomas of the same size and location such as abdominal pain, pelvic pain, palpable mass and menstrual abnormalities. Differential diagnosis includes mature ovarian teratoma, malignant degeneration of benign immature teratoma, non‑teratomatous lipomatous ovarian tumor, pelvic lipoma, pelvic liposarcoma, very rare lipomatous tumors of the uterus, angiomyolipoma, fibromyolipoma and myelolipoma. Uterine lipoleiomyomas are often diagnosed pre‑operatively as uterine leiomyomas or mature ovarian teratoma.
Figure 1: Gross hysterectomy specimen showing a sub-mucosal well circumscribed mass with soft yellow-white cut surface
Figure 2: Lipoleiomyoma characterized by bundles of fusiform smooth muscle cells admixed with mature fat cells (H and E, ×10)
Figure 3: Smooth muscle cells with elongated nuclei and mature adipocytes (H and E, ×40)
Figure 4: Broad ligament lipoma showing mature adipose tissue with capillaries (H and E, ×40)
Journal of Cancer Research and Therapeutics - April-June 2014 - Volume 10 - Issue 2
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Wahal and Mardi: Lipoleiomyoma of uterus and lipoma of broad ligament‑A rare entity
The key to distinguishing the tumors from lipoleiomyomas is to ascertain the primary site of development‑uterus or adnexa
Gynecologists should be aware of the possibility of retroperitoneal broad ligament lipomatous tumors presenting as adnexal masses.[9]
Imaging of these lesions allow for differentiation from cystic ovarian neoplasms, which may require surgical therapy.[8] Lipoleiomyomas are benign tumors of the uterus that do not directly affect mortality.
To our knowledge this is the first case report of a combination of Lipoleiomyoma of uterus with a broad ligament lipoma.
Retroperitoneal benign lipomas are extremely rare and represent about 2.9% of all primary retroperitoneal tumors.[9] A retroperitoneal lipoma arising from broad ligament is exceedingly rare.[10] No such case has been reported in last 5 years. John Bland Sutton reported a case of broad ligament lipoma co‑existent with ovarian mature teratoma. On careful examination it was found that the lipomatous tumor in the broad ligament was the tissue from ruptured ovarian teratoma burrowing along the line of least resistance and made its way between the layers of the broad ligament.[10]
1. Houser LM, Carrasco CH, Sheehan CR Jr. Lipomatous tumour of ultrasonic appearance of uterus: Radiographic and ultrasonic appearance Br J Radiol 1979:52:992‑3. 2. Cantin PF. Lipoma of broad ligament. Br Med J. 1959:1242. 3. Oh MH, Cho IC, Kang YI, Kim CY, Kim DS, Cho HD, et al. A case of retroperitoneal lipoleiomyoma. J Korean Med Sci 2001:16:250-2. 4. Kurman RJ. Mesenchymal tumors of the uterus. Blaustein’s Female Pathology the Female Genital Tract. 6th ed: New Springer, 2002:467 5. Dellacha A, Di Marco A, Foglia G, Fulcheri E. Lipoleiomyomyoma of the uterus. Pathologica 1997:89:737‑41. 6. Lin KC, Sheu BC, Huang SC. Lipoleiomyoma of the uterus. Int J Gynaecol Obstet 1999:67:47‑9. 7. Avritscher R, Iyer RB, Ro J, Whitman J. Lipoleiomyoma of the uterus. AJR Am J Roentgenol 2001:177:856. 8. Armstrong JR, Cohn I Pr. Primary malignant retroperitoneal tumors. Am J Surg. 1965:110:937-43. 9. Eltabbakh GH. Broad ligament lipoma presenting as a pelvic mass: A case report. J Reprod Med 2007:52:543‑4. 10. Lockyer C. Lipoma of broad ligament. Proc R Soc Med. 1919:12:195-9.
A true lipoma of broad ligament was reported by Lockyer again in association with an ovarian mature teratoma. However, in this case the lipomatous tumor was well encapsulated and was separate from the cyst wall. A primary lipoma of broad ligament is rare. Scant amount of fat in this retroperitoneal place supports the fact that adipose tissue in broad ligament is pathological. Doran and Borrman have suggested “a teratogenic element” and “a developmental anomaly” respectively for this process.[10] However, these two hypothesis lack substantial proof.
436
REFERENCES
Cite this article as: Wahal SP, Mardi K. Lipoleiomyoma of uterus and lipoma of broad ligament-a rare entity. J Can Res Ther 2014;10:434-6.
Source of Support: Nil, Conflict of Interest: None declared.
Journal of Cancer Research and Therapeutics - April-June 2014 - Volume 10 - Issue 2
Copyright of Journal of Cancer Research & Therapeutics is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
Lipoleiomyoma of uterus and lipoma of broad ligament‑a rare entity ABSTRACT Lipoleiomyoma of uterus are a rare variant of uterine leiomyoma. Clinically the symptoms are indistinguishable from an ordinary leiomyoma. It is diagnosed pre‑operatively as leiomyoma or mature ovarian teratoma. Majority of them are post operative chance finding. Solid tumors of broad ligament are also rare. Most of them are lateral extensions from the uterine tumors. Primary lipomas of broad ligament are rare. They are asymptomatic in majority of cases and are incidental post‑operative finding. We report this case because of the rarity of individual lesions and rarity of the combination and also speculate their histogenesis as adipose tissue is rare absent at both locations. KEY WORDS: Lipoleiomyoma, uterus, broad ligament, lipoma
INTRODUCTION Lipoleiomyoma of uterus ts a rare variant of uterine leiomyoma. Lipoma of broad ligament is still rarer. Less than 30 cases have been reported in literature. The exact pathogenesis of lipoleiomyoma is not known. Lipoleiomyoma consists of variable proportion of mature adipocytes and smooth muscle cells. These tumors generally occur in asymptomatic obese menopausal women. Solid tumors in broad ligament are rare. Those encountered are lateral extension of uterine tumors. A primary lipoma of broad ligament is uncommon since the amount of fat at this location is scanty. Some workers have suggested “a teratogenous element” and “a developmental anomaly” but without substantial proof, the pathogenesis remains inconclusive. CASE REPORT A 61-year-old female (para 4 + 0) presented with abdominal pain for 1 month. The patients menstrual history revealed menarche at the age of 14, regular menstrual cycles, 3-4 days duration at regular intervals of 30 days. She had undergone menopause 15 years ago. Gynaecological examination showed no abnormality of vulva, vagina and vaginal portion of the cervix. The uterine cavity was enlarged corresponding to 6 weeks of gestation. The adnexae were not palpable. Findings of vaginal ultrasonography suggested a well circumscribed 4 cm mass having semi‑solid character located in the fundus of the uterus. An echogenic adnexal mass was appreciated 434
on the left side of the uterus. The endometrium thickness was 1 mm. Both ovaries and tubes were normal. No ascitis was seen in the abdomen. The patient underwent abdominal hysterectomy for mass in fundus. Grossly, a hysterectomy specimen with bilateral salpigo‑oophectomy was received. It measured 10.5 × 6.5 × 3 cm and showed a well‑circumscribed sub‑mucosal mass in the fundus. Endometrium measured 1 mm and myometrium measured 2 cm in thickness. The sub‑mucosal mass measured 4 cm in greatest diameter. Cut surface was soft and yellow. No areas of hemorrhage or necrosis were identified [Figure 1]. A separate nodule measuring 1 cm in diameter was present in the left parametrium of broad ligament. Microscopic examination showed cystic atrophy of the endometrium, senile changes in the myometrium, bilateral fallopian tubes and ovaries within normal limits. The sub‑mucosal mass showed a mixture of spindle‑shaped smooth muscle cells without atypical nuclei in a whorled pattern and mature fat cells in the sub‑mucosal mass [Figure 2]. Nuclei of smooth muscle cells were elongated, had blunt ends, finely dispersed chromatin and small nucleoli [Figure 3]. Bizarre pleomorphic cells, mitotic figures or necrosis were not present. Between the muscle cells significant amount of fat was present. The adipose tissue was completely mature without any lipoblasts. Based on these findings, the tumor was diagnosed as Lipoleiomyoma. Sections from the nodule in the parametrium of the broad ligament revealed a circumscribed lesion composed of uniform sized
Shailja Puri Wahal, Kavita Mardi Department of Pathology, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India For correspondence: Dr. Shailja Puri Wahal, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India. E‑mail: drshailjadoe_11@ ymail.com
Access this article online Website: www.cancerjournal.net DOI: 10.4103/0973-1482.136682 PMID: *** Quick Response Code:
Journal of Cancer Research and Therapeutics - April-June 2014 - Volume 10 - Issue 2
Wahal and Mardi: Lipoleiomyoma of uterus and lipoma of broad ligament-A rare entity
adipocytes [Figure 4]. There was no connection between this lipomatous nodule and the lipoleiomyoma of the uterus. A diagnosis of Lipoma‑Broad ligament was given. DISCUSSION Lipomatous tumors of the uterus include a spectrum of lesions consisting of pure lipomas, lipoleiomyomas and fibromyolipoma.[1] Solid tumors in broad ligament are rare. Those encountered are lateral extension of uterine tumors. A primary lipoma of broad ligament is uncommon.[2] Lipoleiomyoma is a rare uterine fatty tumor. Myolipoma of soft tissue was first described by Meis and Enzinger in 1991. These tumors show characteristic histological findings composed of benign smooth muscle cells and mature adipose tissue. In uterus similar tumors are called lipoleiomyomas.[3] Lipoleiomyomas occur in different locations including cervix. The commonest location in uterus is the fundus.[4] Pathogenesis of lipoleiomyoma remains unclear. There are two main theories, namely adipose metaplasia of smooth muscle cells
and a multipotential mullerian cell origin.[5] These tumors are asymptomatic and are common in obese menopausal women.[1] Various lipid metabolic disorders associated with estrogen deficiency occurring in pre‑ and post‑ menopausal women, possibly promotes intracellular deposition of lipid.[6] An incidence of 0.28% of all leiomyomas and 0.39% of all hysterectomies was reported from National Taiwan University Hospital between January 1994 and December 1998 by K.C. Lin et al. who analysed 2878 leiomyoma cases and 2071 hysterectomies specimens.[7] Although most patients are asymptomatic, they can present with symptoms similar to leiomyomas of the same size and location such as abdominal pain, pelvic pain, palpable mass and menstrual abnormalities. Differential diagnosis includes mature ovarian teratoma, malignant degeneration of benign immature teratoma, non‑teratomatous lipomatous ovarian tumor, pelvic lipoma, pelvic liposarcoma, very rare lipomatous tumors of the uterus, angiomyolipoma, fibromyolipoma and myelolipoma. Uterine lipoleiomyomas are often diagnosed pre‑operatively as uterine leiomyomas or mature ovarian teratoma.
Figure 1: Gross hysterectomy specimen showing a sub-mucosal well circumscribed mass with soft yellow-white cut surface
Figure 2: Lipoleiomyoma characterized by bundles of fusiform smooth muscle cells admixed with mature fat cells (H and E, ×10)
Figure 3: Smooth muscle cells with elongated nuclei and mature adipocytes (H and E, ×40)
Figure 4: Broad ligament lipoma showing mature adipose tissue with capillaries (H and E, ×40)
Journal of Cancer Research and Therapeutics - April-June 2014 - Volume 10 - Issue 2
435
Wahal and Mardi: Lipoleiomyoma of uterus and lipoma of broad ligament‑A rare entity
The key to distinguishing the tumors from lipoleiomyomas is to ascertain the primary site of development‑uterus or adnexa
Gynecologists should be aware of the possibility of retroperitoneal broad ligament lipomatous tumors presenting as adnexal masses.[9]
Imaging of these lesions allow for differentiation from cystic ovarian neoplasms, which may require surgical therapy.[8] Lipoleiomyomas are benign tumors of the uterus that do not directly affect mortality.
To our knowledge this is the first case report of a combination of Lipoleiomyoma of uterus with a broad ligament lipoma.
Retroperitoneal benign lipomas are extremely rare and represent about 2.9% of all primary retroperitoneal tumors.[9] A retroperitoneal lipoma arising from broad ligament is exceedingly rare.[10] No such case has been reported in last 5 years. John Bland Sutton reported a case of broad ligament lipoma co‑existent with ovarian mature teratoma. On careful examination it was found that the lipomatous tumor in the broad ligament was the tissue from ruptured ovarian teratoma burrowing along the line of least resistance and made its way between the layers of the broad ligament.[10]
1. Houser LM, Carrasco CH, Sheehan CR Jr. Lipomatous tumour of ultrasonic appearance of uterus: Radiographic and ultrasonic appearance Br J Radiol 1979:52:992‑3. 2. Cantin PF. Lipoma of broad ligament. Br Med J. 1959:1242. 3. Oh MH, Cho IC, Kang YI, Kim CY, Kim DS, Cho HD, et al. A case of retroperitoneal lipoleiomyoma. J Korean Med Sci 2001:16:250-2. 4. Kurman RJ. Mesenchymal tumors of the uterus. Blaustein’s Female Pathology the Female Genital Tract. 6th ed: New Springer, 2002:467 5. Dellacha A, Di Marco A, Foglia G, Fulcheri E. Lipoleiomyomyoma of the uterus. Pathologica 1997:89:737‑41. 6. Lin KC, Sheu BC, Huang SC. Lipoleiomyoma of the uterus. Int J Gynaecol Obstet 1999:67:47‑9. 7. Avritscher R, Iyer RB, Ro J, Whitman J. Lipoleiomyoma of the uterus. AJR Am J Roentgenol 2001:177:856. 8. Armstrong JR, Cohn I Pr. Primary malignant retroperitoneal tumors. Am J Surg. 1965:110:937-43. 9. Eltabbakh GH. Broad ligament lipoma presenting as a pelvic mass: A case report. J Reprod Med 2007:52:543‑4. 10. Lockyer C. Lipoma of broad ligament. Proc R Soc Med. 1919:12:195-9.
A true lipoma of broad ligament was reported by Lockyer again in association with an ovarian mature teratoma. However, in this case the lipomatous tumor was well encapsulated and was separate from the cyst wall. A primary lipoma of broad ligament is rare. Scant amount of fat in this retroperitoneal place supports the fact that adipose tissue in broad ligament is pathological. Doran and Borrman have suggested “a teratogenic element” and “a developmental anomaly” respectively for this process.[10] However, these two hypothesis lack substantial proof.
436
REFERENCES
Cite this article as: Wahal SP, Mardi K. Lipoleiomyoma of uterus and lipoma of broad ligament-a rare entity. J Can Res Ther 2014;10:434-6.
Source of Support: Nil, Conflict of Interest: None declared.
Journal of Cancer Research and Therapeutics - April-June 2014 - Volume 10 - Issue 2
Copyright of Journal of Cancer Research & Therapeutics is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
