Journal of Obstetrics and Gynaecology

ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20

Preoperative diagnosis of a uterine lipoleiomyoma using transvaginal ultrasound: A case report of an unusual entity J. F. Carvalho, A. Cubal, S. Torres, A. Morgado, C. Oliveira & O. do Carmo To cite this article: J. F. Carvalho, A. Cubal, S. Torres, A. Morgado, C. Oliveira & O. do Carmo (2014) Preoperative diagnosis of a uterine lipoleiomyoma using transvaginal ultrasound: A case report of an unusual entity, Journal of Obstetrics and Gynaecology, 34:3, 281-282 To link to this article: http://dx.doi.org/10.3109/01443615.2013.863271

Published online: 29 Jan 2014.

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Date: 04 November 2015, At: 07:24

Gynaecology Case Reports 281

Preoperative diagnosis of a uterine lipoleiomyoma using transvaginal ultrasound: A case report of an unusual entity J. F. Carvalho, A. Cubal, S. Torres, A. Morgado, C. Oliveira & O. do Carmo Department of Obstetrics and Gynaecology, Tâmega and Sousa Hospital Centre, Penafiel, Portugal DOI: 10.3109/01443615.2013.863271

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Figure 5. Photomicrograph of the cyst wall with focal cautery effect showing the fibrous cyst wall lined by columnar serous-type epithelium (original magnification,  150).

examination. Reported sites of ectopic tubal epithelium include the vagina, cervix, uterus, peritoneum, appendix, colon, and umbilicus (Liang et al. 2007; Papavramidis et al. 2010). Occasionally, vague and nonspecific symptoms, including chronic pelvic pain or menorrhagia prompt radiologic investigation (Taneja et al. 2010). In a rare subset of cases, termed florid cystic endosalpingiosis, the presentation involves the presence of multiple macroscopic tumourlike pelvic masses (Heatley and Russell 2001). On histologic evaluation, endosalpingiosis appears as a cystic mass with a serous epithelial lining. The absence of cellular stratification or mitotic activity differentiates this entity from malignant lesions. The lack of typical endometrial-like stroma and no indication of cyclical hemorrhagic response help distinguish this lesion from endometriosis (Rosenberg et al. 2011). At sonography, these lesions can present as anechoic adnexal masses with negative Doppler and consequently mimic cystic ovarian neoplasms or degenerated leiomyomas (Rosenberg et al. 2011). Characteristics on MRI have been sparsely reported. The reports include a single cystic lesion in the uterine fundus, a simple ovarian cyst, and a multiseptated cystic mass in the pouch of Douglas (Taneja et al. 2010). In this case of florid cystic endosalpingiosis, the presentation was that of multiple cystic masses along the uterine surface and the cervix, mimicking recurrence of tumour in a patient with history of ovarian serous cystadenoma. Florid cystic endosalpingiosis is an important benign entity to be aware of in cases of suspected ovarian and pelvic neoplasms. The ability to make this diagnosis preoperatively may preclude surgery, or potentially alter the surgical plan to a less invasive option such as laparoscopy. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.

References Heatley MK, Russell P. 2001. Florid cystic endosalpingiosis of the uterus. Journal of Clinical Pathology 54:399–400. Hesseling MH, De Wilde RL. 2000. Endosalpingiosis in laparoscopy. Journal of the American Association of Gynecologic Laparoscopy 7:215–219. Kurman RJ, Vang R, Junge J, et al. 2011. Papillary tubal hyperplasia: the putative precursors of ovarian atypical proliferative (borderline) serous tumors, noninvasive implants and endosalpingiosis. American Journal of Surgical Pathology 35:1605–1614. Liang JJ, Malpica A, Broaddus RR. 2007. Florid cystic endosalpingiosis presenting as an obstructive colon mass mimicking malignancy: case report and literature review. Journal of Gastrointestinal Cancer 38:83–86. Papavramidis TS, Sapalidis K, Michalopoulos N et al. 2010. Umbilical endosalpingiosis: a case report. Journal of Medical Case Reports 24:287. Rosenberg P, Nappi L, Santoro A et al. 2011. Pelvic mass-like florid cystic endosalpingiosis of the uterus: a case report and a review of literature. Archives of Gynecology and Obstetrics 283:519–523. Taneja S, Sidhu R, Khurana A et al. 2010. MRI appearance of florid cystic endosalpingiosis of the uterus: a case report. Korean Journal of Radiology 11:476–479.

Correspondence: J. F. Carvalho, Department of Gynaecology and Obstetrics, Tâmega and Sousa Hospital Centre, Rua da Vinha, no. 124, Valbom, 4420–578, Gondomar. Portugal. E-mail: [email protected]

Introduction Lipoleiomyomas are uncommon benign neoplasms of the uterus and are considered to be a variant of uterine myomas. The reported incidence in the literature varies from 0.03–0.2% of all uterine myomas and 50 cases have been reported in the last 15 years (Prieto et al. 2000). These tumours are usually found in postmenopausal, obese women with typical uterine leiomyomas. Although most patients are asymptomatic, they can present with symptoms similar to leiomyomas of the same size and location, such as palpable mass, menstrual abnormalities and abdominal/ pelvic pain (Oppenheimer et al. 1982). They are most frequently found in the uterine corpus and are usually intramural fibroids, but they can also be found anywhere in the uterus or cervix and may be subserosal (Aizenstein et al. 1991). Although its definite diagnosis is histological, today, with the advancement of imaging techniques such as ultrasound (US) and magnetic resonance imaging (MRI), we can have strong diagnostic accuracy of these lesions preoperatively (Dodd and Budzik 1990; Prieto et al. 2000). We report a case of a uterine lipoleiomyoma whose diagnosis was suspected preoperatively in the US scan and was confirmed postoperatively by histological examination.

Case report A 54-year-old postmenopausal woman presented with pelvic pain over the previous 6 months. The patient was obese (BMI: 35.26 kg/m2), with high blood pressure, dyslipidemia and type II diabetes mellitus. The patient’s history revealed that she had attained menarche at the age of 12 years; had regular menstrual cycles of 3–5 days’ duration of moderate intensity and at 28-day intervals. She was a gravida 2 para 2, had attained menopause at the age of 50 years and had not received any hormone therapy. Gynaecological examination revealed vulvovaginal atrophy; at the bimanual palpation, the posterior vaginal fornix was occupied by a nodular formation, of elastic consistency, painless and apparently of uterine origin. The uterus was enlarged and it could be felt abdominally approximately 10 cm above the symphysis pubis. Transvaginal US revealed

Figure 1. Uterine lipoleiomyoma suspected in transvaginal US.

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a large, well-defined, homogeneously hyperechoic mass with regular margins, measuring 8.2  5.7  8.8 cm, surrounded by a hypoechoic rim, in the posterior wall of the uterus, and it raised suspicion of a uterine lipoleiomyoma (Figure 1). An MRI was performed and it showed a large lobulated mass arising from the posterior wall of the uterus, which had hyperintense signals on T1-weighted images, which suppressed on a fat-saturation sequence. A total abdominal hysterectomy with bilateral salpingo-oophorectomy was performed. A large, well-circumscribed solid mass with yellow cut-surface was found at the uterine body. Microscopically, the tumour was shown to be a lipoleiomyoma consisting of smoothmuscle cells and mature adipose tissue. The postoperative period was uneventful and the patient was discharged home 4 days after surgery, clinically well.

Female paraphimosis? Management of a large female urethral caruncle, trialling manual reduction S. Salim1, A. Taylor1 & C. Carter2 Departments of 1Obstetrics and Gynaecology and 2Urology, Royal Bournemouth and Christchurch Hospitals, Bournemouth, UK DOI: 10.3109/01443615.2013.868420 Correspondence: S. Salim, Department of Obstetrics and Gynaecology, Royal Bournemouth and Christchurch Hospital, Castle Lane East, Bournemouth BH7 7DW, UK. E-mail: [email protected]

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Discussion Lipoleiomyomas are benign uterine tumours. The exact aetiology is not well known, but they are mostly thought to represent fatty metamorphosis of the smooth muscle cells of leiomyomas. Histologically, they consist of smooth-muscle tissue, most commonly of the spindle-cell type, admixed with varying amounts of mature adipose tissue, without cytological atypia (Tsushima et al. 1997). Typical features in US and MRI make the diagnosis easier. The sonographic appearance is that of a hyperechoic mass, partially encased by a hypoechoic rind that is thought to represent a layer of myometrium surrounding the fatty component (Dodd and Budzik 1990; Prieto et al. 2000). On MRI, the lipomatous nature of the lesion is suggested by high signal intensity on T1-weighted images and chemical shift artifacts in the lesion (Dodd and Budzik 1990; Aizenstein et al. 1991; Ishigami et al. 1998). The differential diagnosis of the lipomatous mass in the pelvis includes benign cystic ovarian teratoma, non-teratomatous lipomatous ovarian tumour, benign pelvic lipoma, liposarcoma, lipoblastic lymphadenopathy and retroperitoneal cystic hamartoma (Friedman et al. 1981; Ekici and Vicdan 1993). Identification of the uterus as the organ of origin, and knowledge of the characteristic features of the lesions in imaging studies, as stated above, is the key to diagnose lipomatous lesions in the pelvis. Such a differentiation is crucial because lipomatous uterine tumours are generally asymptomatic and do not require surgery, unless they start causing symptoms (Ekici and Vicdan 1993). In our case, although we suspected the diagnosis, we decided to perform a total abdominal hysterectomy because the patient was symptomatic, with bilateral salpingo-oophorectomy, as she was a postmenopausal woman. This case serves as a reminder of this rare gynaecological entity and highlights the importance of a correct preoperative diagnosis to avoid unnecessary surgical morbidity. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.

References Aizenstein R, Wilbur AC, Aizenstein S. 1991. CT and MRI of uterine lipoleiomyoma. Gynecologic Oncology 40:274–276. Dodd GD III, Budzik RF. 1990. Lipomatous uterine tumours: diagnosis by ultrasound, CT and MRI. Journal of Computer Assisted Tomography 14: 629–632. Ekici E, Vicdan K. 1993. Uterine myolipoma: diagnosis by ultrasound. International Journal of Gynecology and Obstetrics 42:167–171. Friedman AC, Hartman DS, Sherman J. 1981. Computed tomography of abdominal fatty masses. Radiology 139:415–429. Ishigami K, Yoshimitsu K, Honda H. 1998. Uterine lipoleiomyoma: MRI appearances. Abdominal Imaging 23:214–216. Oppenheimer DA, Carroll BA, Young SW. 1982. Lipoleiomyoma of the uterus. Journal of Computer Assisted Tomography 6:640–642. Prieto A, Crespo C, Pardo A, Docal I, Calzada J, Alonso P. 2000. Uterine lipoleiomyomas: US and CT findings. Abdominal Imaging 25:655–657. Tsushima Y, Kita T, Yamamoto K. 1997. Uterine lipoleiomyoma: MRI, CT and ultrasonographic findings. British Journal of Radiology 70:1068–1070.

Introduction The history of the urethral caruncle can be traced back to 1750, when the lesion was first described as a clinical entity by Samuel Sharp (Becker 1975). Subsequent authors have described it as a polypoidlooking fleshy growth arising from the posterior lip of urethra (Becker 1975). The condition occurs primarily in postmenopausal women with several factors attributing to the aetiology. Rarely, it can be seen in perimenopausal and extremely rarely in pre-pubertal girls (Venyo 2012; Kim et al. 1993). Although rare, it is the most common benign tumour of the female urethra. Malignancy has been reported in a few rare cases but these account for 1.6–2% of all caruncles (Venyo 2012; Conces et al. 2012; Srinivas and Khan 1987; Kaneko et al. 2011). Overall, it is estimated that urethral cancer accounts for  0.02% of female cancers (Srinivas and Khan 1987; Kaneko et al. 2011; Chaudhari et al. 2010). Conservative management is the mainstay of treatment. Surgery is reserved for refractory cases or where biopsy is required to rule out malignancy (Venyo 2012; Srinivas and Khan 1987; Kaneko et al. 2011).

Case report The authors present a rare case of an unusually large urethral caruncle in a 67-year-old postmenopausal woman. She presented with the symptom of a lump in her vagina. There was no associated dysuria, pain or bleeding. During clinical assessment, she was found to have a 3 cm polypoid-looking mass, which was diagnosed as a benign urethral caruncle (Figure 1). A joint examination under anaesthesia was carried out with a urologist. Initial surgical management with excision was considered but discarded due to the large size of the lesion and also to avoid iatrogenic fibrosis and potential risk of distal urethral stenosis, secondary to excision. We therefore took the previously unreported approach of manual reduction. This approach is commonly used by urologists to reduce paraphimosis in males. A constant manual pressure was applied for 10 min. This reduced the oedema around the prolapsed tissue and achieved a complete reduction. In order to further reduce the pressure, an indwelling catheter was inserted for 10 days. Unfortunately, at a 1-month follow-up, it had recurred. The patient was counselled and referred to urology for surgical excision.

Discussion The exact aetiology of a urethral caruncle is yet to be established. However, a chain of events has been suggested in the development of the caruncle, especially in postmenopausal women (Venyo 2012; Kaneko et al. 2011). The normal female urethra is 3–4 cm long (Chaudhari et al. 2010; Ryu and Kim 2001; Kawashima et al. 2004). It has been suggested that after menopause, the hypoestrogenic state leads to the atrophy of urethral epithelium and subsequent mucosal prolapse (Venyo 2012; Chaudhari et al. 2010; Ryu and Kim 2001). The presentation is usually an asymptomatic small mass in the vagina, often incidentally found during clinical examination. It can also present with symptoms of pain, dysuria and bleeding (Becker 1975; Venyo 2012; Conces et al. 2012; Kaneko et al. 2011). Before initiating treatment, it is important to rule out urinary tract infection, especially with associated symptoms of dysuria. Cystoscopy is reserved for

Preoperative diagnosis of a uterine lipoleiomyoma using transvaginal ultrasound: a case report of an unusual entity.

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