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  Table of Contents    
CASE REPORT  
Year : 2016  |  Volume : 59  |  Issue : 3  |  Page : 355-358
Lipoleiomyoma of the left broad ligament with dermoid cyst in ipsilateral ovary and synchronous multiple benign lesions of female genital tract: An unusual association


1 Department of Obstetrics and Gynaecology, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India
2 Department of Pathology, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India

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Date of Web Publication10-Aug-2016
 

   Abstract 

Lipoleiomyoma of the uterus is a rare variant of leiomyoma, and lipoleiomyoma of the broad ligament is still rarer, with only a handful of cases being reported. The present case was a perimenopausal woman who presented with a huge lower abdominal mass. Ultrasonography and computed tomography showed a heterogeneous solid mass in the left adnexa. The histopathological findings confirmed the nature of the lesions as a benign lipoleiomyoma with dermoid cyst of the left ovary and its other associated benign lesions, were the interesting features seen in this case which were not suspected clinically and radiologically.

Keywords: Broad ligament, dermoid cyst, lipoleiomyoma

How to cite this article:
Mishra SS, Saha A, Mishra P, Jena SK. Lipoleiomyoma of the left broad ligament with dermoid cyst in ipsilateral ovary and synchronous multiple benign lesions of female genital tract: An unusual association. Indian J Pathol Microbiol 2016;59:355-8

How to cite this URL:
Mishra SS, Saha A, Mishra P, Jena SK. Lipoleiomyoma of the left broad ligament with dermoid cyst in ipsilateral ovary and synchronous multiple benign lesions of female genital tract: An unusual association. Indian J Pathol Microbiol [serial online] 2016 [cited 2019 Jun 19];59:355-8. Available from: http://www.ijpmonline.org/text.asp?2016/59/3/355/188117



   Introduction Top


Broad ligament lipoleiomyoma is a rare benign mesenchymal neoplasm, considered to be a variant of common benign neoplasm of the female genital tract, leiomyoma. It is composed of intimate mixture of varying proportion of bland smooth muscle cells and mature adipose tissue.[1],[2] Lipoleiomyoma is most commonly located in the uterine corpus although cervical, ovarian, broad ligament, and retroperitoneal locations have also been reported.[1],[2] We have found only eight cases of lipoleiomyomas of the broad ligament reported till date through an English literature review.[1],[3],[4],[5],[6],[7],[8],[9] To the best of our knowledge, this is probably the first case of broad ligament lipoleiomyomata in a perimenopausal woman associated with mesonephric cyst, dermoid cyst of ipsilateral ovary, endometriosis of the other ovary, benign endometrial polyp, multiple intramural and subserosal leiomyomata with adenomyosis. This case is reported here for its rarity.


   Case Report Top


A 45-year-old multiparous, perimenopausal woman presented with a history of abdominal pain, constipation, and difficulty in micturition with feeling of incomplete voiding for 1 month and was referred from outside to our tertiary care center with the provisional diagnosis of malignant ovarian tumor. Her menstrual cycles were regular. Her past history was significant for having undergone surgery twice for giant cell tumor of the left femur 12 years back. She was not known to be diabetic, hypertensive, hypothyroid, or hyperlipidemic.

Per-abdominal examination revealed a bilobed mobile mass corresponding to 24 weeks of gestation, firm in consistency with regular margins arising from the pelvis. On per-vaginum examination, uterus was irregularly enlarged, cervix moved with movement of the mass, and the pouch of Douglas (POD) was full with the mass.

Ultrasonography (USG) of the abdomen and pelvis showed bulky uterus and a huge solid hyperechoic mass, separate from the uterus and filling the POD, extending into the left adnexa [Figure 1]a. The presence of hydronephrosis on the left side was observed. No ascites or lymphadenopathy was present. On color Doppler, there was increased vascularity within the mass and bridging vascular sign was found to be present between the uterus and mass, giving rise to suspicion of the broad ligament fibroid [Figure 1]b. Contrast-enhanced computed tomography (CT) scan of the abdomen and pelvis showed a heterogeneously enhancing solid pelvic mass of size 15 cm × 13 cm with well-defined margins, occupying the lower abdomen, and extending into the pelvis and POD, with displacement of the iliac vessels and ureters outward [Figure 1]c. Radiological findings were suggestive of a broad ligament fibroid, and at this point of time, exploratory laparotomy along with total abdominal hysterectomy with bilateral salpingo-oophorectomy and excision of the mass was planned. Tumor markers CA-125, human chorionic gonadotropin, alpha-fetoprotein, carcinoembryonic antigen, and lactate dehydrogenase were normal. At laparotomy, abdomen was opened by midline incision. The uterus was bulky in size. There was a 15 cm × 13 cm soft and cystic fibroid in the region of the left broad ligament, with flimsy adhesions to the peritoneal fat [Figure 1]d. Total abdominal hysterectomy with bilateral salpingo-oophorectomy along with resection of the tumor was then performed.
Figure 1: (a) Pelvic ultrasonography showing bulky uterus and a huge solid hyperechoic mass, separate from the uterus, and filling the pouch of Douglas. (b) Color Doppler showing “bridging vascular sign.” (c) Contrast-enhanced computed tomography scan showed a heterogeneously enhancing solid mass, displacing the uterus, bilateral iliac vessels, and ureters. (d) 15 cm × 13 cm soft and cystic fibroid in the region of the left broad ligament, with flimsy adhesions to the peritoneal fat

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On gross examination of the resected specimen, the uterus with cervix and bilateral appendages measured 25 cm × 6 cm × 4.2 cm and a separately sent well-circumscribed partially encapsulated mass measured 17 cm × 16 cm × 13 cm [Figure 2]a and [Figure 2]b. On cut section, the endometrial cavity showed a polyp measuring 3 cm × 2 cm, [Figure 2]c myometrium showed two subserosal and two intramural leiomyomata measuring 0.3 cm -1 cm. The myometrium appeared trabeculated. There was a well-circumscribed, small globular mass measuring 1.5 cm × 1.5 cm in the left parametrium of the broad ligament, with adherent small cyst measuring 0.6 cm [Figure 2]d. Cut section of the mass revealed a fairly well-circumscribed lobulated mass, containing yellowish areas. Cut section of the separately sent huge mass showed a firm, coarsely whorled, grayish-white to yellow in appearance. Focal areas showed myxoid changes. Bilateral adnexa appeared grossly unremarkable except for a small cyst measuring 0.8 cm in the left ovary [Figure 2]e. The cervix was grossly unremarkable.
Figure 2: (a) Hysterectomy with bilateral salpingo-oophorectomy specimen along with separately sent globular mass. (b) Globular mass was well-circumscribed, tan gray-white with yellowish areas and whorled appearance. (c) Endometrial polyp (arrow). (d) Left parametrial fibroid with yellowish cut surface. (e) Left ovary with a cyst measuring 0.8 cm (arrow)

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Histological examination of both broad ligament masses showed smooth muscle cells in short and long interweaving fascicles interspersed regularly with lobules of mature adipocytes [Figure 3]a. The smooth muscles had cytologically bland, oval nuclei, and longitudinally oriented fibrillar eosinophilic cytoplasm. The adipose component was mature without any lipoblast. Areas of angiomatous hyperplasia with thick-walled tortuous hyalinized vascular channels and foci of myxoid degeneration were present [Figure 3]b. No necrosis or increased mitotic activity was seen. A plenty of mast cells and lymphocytes was scattered in the background. Immunohistochemically, the spindle cells were strongly positive for desmin and smooth muscle actin [Figure 4]a and [Figure 4]b and negative for CD 34, CD 117, and human melanoma black (HMB) 45 [Figure 4]c and [Figure 4]d. Based on the above findings, both the tumors were diagnosed as benign lipoleiomyoma. In addition, ipsilateral parametrium showed mesonephric cyst [Figure 3]c. The endomyometrium showed a benign endometrial polyp [Figure 3]d, with intramural leiomyomata and adenomyosis. Right ovary showed foci of endometriosis [Figure 3]e. To our utter “surprise,” the 0.8 cm cystic lesion of the left ovary showed derivatives of all the three germ cell layers [Figure 3]f. No immature tissue was identified. A diagnosis of benign mature cystic teratoma was made. The cervix and bilateral  Fallopian tube More Detailss were histologically unremarkable. The final histopathological diagnosis was lipoleiomyomata of the left broad ligament with mesonephric cyst, endometrial polyp, adenomyosis, intramural leiomyomata, associated with endometriosis of the right ovary and dermoid cyst of the left ovary.
Figure 3: (a) Lipoleiomyoma showing fascicles of bland smooth muscle cells admixed with mature adipocytes (H and E, ×40 and 200× [inset]). (b) Focal myxoid degeneration (H and E, ×100). (c) Mesonephric cyst (H and E, ×100). (d) Endometrial polyp (H and E, ×40). (e) Ovarian endometriosis (H and E, ×200). (f) Ovarian dermoid (H and E, ×100)

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Figure 4: (a and b) Tumor cells show strong positivity for desmin and smooth muscle actin (×200, ×100). (c) Tumor cells are negative for CD 34 (×40). (d) Tumor cells are negative for CD 117 and inset shows negative staining for HMB 45 (×100, ×100)

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   Discussion Top


Lipoleiomyoma of the broad ligament is extremely rare accounting for 0.03–2.9% of all leiomyomata.[9] It was first described in 1916 by Lobstein.[10] [Table 1] summarizes all the eight cases of lipoleiomyomas of the broad ligament reported so far in English literature. Association with various manifestations of hyperestrogenic status, such as adenomyosis, endometriosis, endometrial hyperplasia, polyps, and different gynecological malignancies, has been reported.[9] The association of lipoleiomyoma with benign lesions seen in our case such as endometrial polyp, adenomyosis, and endometriosis of the right ovary could be attributed to the hyperestrogenic state, which could be an important factor in the development of lipoleiomyoma.
Table 1: Summary of all the cases of broad ligament lipoleiomyoma reported in English literature till date

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In contrast to usual leiomyomata, lipoleiomyomas usually occur in perimenopausal women, with reported mean age of patients being 54.[1] Most patients are asymptomatic, but others may present with pelvic pain, palpable mass, or abnormal bleeding.[11] Lipoleiomyomas of the broad ligament mimics cystic teratoma by virtue of the hyperechogenic shadows on USG produced by fatty tissue. However, the presence of “beak sign” or “bridging vascular sign” on color Doppler differentiates them from solid ovarian tumors. CT and magnetic resonance imaging further aid in the diagnosis.

The histopathological finding of smooth muscle cells admixed with mature adipocytes is required to designate a neoplasm as lipoleiomyoma. The adipocytes may be evenly or focally distributed throughout the tumor. The proportion of adipocytes to smooth muscle is not clearly defined to classify a neoplasm as lipoleiomyoma, but it has been shown that the adipocytic component may vary widely ranging from 5% to 95% of the tumor mass.[12] These tumors grossly may contain microscopic foci of adipocytes resembling regular leiomyomas, or rich amount adipocytes may result in yellow and lobulated cut surface mimicking lipoma [1] Our case showed intersecting fascicles of smooth muscle cells with regular areas showing mature adipocytes. The vasculature in the stroma mimicked blood vessels seen in angiomyolipoma of the kidney, and hence, histologically, angiomyolipoma was a very close differential in our case but a negative HMB 45 staining helped us to differentiate the two. This entity of lipoleiomyoma is to be differentiated from lipomatous degeneration in a leiomyoma where the degenerative changes are always focal.

Other common differentials of this tumor are nonteratomatous lipomatous ovarian tumor, benign pelvic lipomas, liposarcomas, carcinosarcoma with heterologous liposarcomatous differentiation, benign or malignant degeneration of ordinary leiomyoma, pelvic fibromatosis, myxoid mesenchymal tumor, etc.[1],[2] Histogenesis of lipoleiomyoma is controversial.[12] Sieinski proposed various theories regarding the origin of lipomatous tumors in the uterus as follows: (1) misplaced embryonal mesodermal rests with a potential for lipoblastic differentiation, (2) lipoblast or pluripotent cells migrating along uterine arteries and nerves, (3) adipose metaplasia of smooth muscle cells.[13] However, without substantial proof, the pathogenesis remains inconclusive.

The case, thus, highlights the association of lipoleiomyoma with multiple benign lesions of the female genital tract. As the leiomyomas are benign, the patient can be spared from elaborate surgery.

Acknowledgement

We acknowledge Dr. Narbadyswari Deep Bag for her valuable radiological opinion in this case.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Wang X, Kumar D, Seidman JD. Uterine lipoleiomyomas: A clinicopathologic study of 50 cases. Int J Gynecol Pathol 2006;25:239-42.  Back to cited text no. 1
    
2.
Aung T, Goto M, Nomoto M, Kitajima S, Douchi T, Yoshinaga M, et al. Uterine lipoleiomyoma: A histopathological review of 17 cases. Pathol Int 2004;54:751-8.  Back to cited text no. 2
    
3.
Fernández FA, Val-Bernal F, Garijo-Ayensa F. Mixed lipomas of the uterus and the broad ligament. Appl Pathol 1989;7:70-1.  Back to cited text no. 3
    
4.
Bajaj P, Kumar G, Agarwal K. Lipoleiomyoma of broad ligament: A case report. Indian J Pathol Microbiol 2000;43:457-8.  Back to cited text no. 4
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5.
Cinel L, Düsmez D, Nabaei SH, Taner D, Pata O. Two intraligamentary lipomatous tumors with immunohistochemical features. Acta Obstet Gynecol Scand 2002;81:786-7.  Back to cited text no. 5
    
6.
Maryanski J, Gulak G, Pawlowski W. Lipoleiomyoma of the broad ligament of the uterus. Int J Gynaecol Obstet 2009;107:257.  Back to cited text no. 6
    
7.
Salman MC, Atak Z, Usubutun A, Yuce K. Lipoleiomyoma of broad ligament mimicking ovarian cancer in a postmenopausal patient: Case report and literature review. J Gynecol Oncol 2010;21:62-4.  Back to cited text no. 7
    
8.
Kim HK, Kim JH, Hong SY, Choi YS, Oh HK, Lee TS. A uterine lipoleiomyoma of the broad ligament mimicking an ovarian tumor. Korean J Obstet Gynecol 2012;55:787-90.  Back to cited text no. 8
    
9.
Akbulut M, Gündogan M, Yörükoglu A. Clinical and pathological features of lipoleiomyoma of the uterine corpus: A review of 76 cases. Balkan Med J 2014;31:224-9.  Back to cited text no. 9
    
10.
Willén R, Gad A, Willén H. Lipomatous lesions of the uterus. Virchows Arch A Pathol Anat Histol 1978;377:351-61.  Back to cited text no. 10
    
11.
Wahal SP, Mardi K. Lipoleiomyoma of uterus and lipoma of broad ligament – A rare entity. J Cancer Res Ther 2014;10:434-6.  Back to cited text no. 11
    
12.
Bolat F, Kayaselçuk F, Canpolat T, Erkanli S, Tuncer I. Histogenesis of lipomatous component in uterine lipoleiomyomas. Turk J Pathol 2007;23:82-6.  Back to cited text no. 12
    
13.
Sieinski W. Lipomatous neometaplasia of the uterus. Report of 11 cases with discussion of histogenesis and pathogenesis. Int J Gynecol Pathol 1989;8:357-63.  Back to cited text no. 13
    

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Correspondence Address:
Dr. Pritinanda Mishra
Department of Pathology, All India Institute of Medical Sciences, Bhubaneswar, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.188117

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