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MACROSCOPIC AND ULTRASOUND EVALUATION OF GIANT SUBMUCOSAL LEIOMYOMA WITH CYSTIC DEGENERATION: DIFFERENTIAL DIAGNOSIS



Abstract
The development of leiomyomas is a frequent event with high prevalence. In most cases of benign nature, we can reach the diagnosis just with typical ultrasound images and the clinical symptoms. We report the case of giant leiomyoma with cystic degeneration, which proposes an initial differential diagnosis with neoplasic disease.
Keywords
Leiomyomas
Degeneratives changes
Uterine Sarcoma
Ultrasound






















Introduction

Leiomyomas�are a high prevalence benign pathology, being the most common pelvic tumor in women. There are typical ultrasound images that allow their classification, according to their location in submucosal, intramural and subserosal. We can make the diagnosis in a simple way due to this characteristics as well the clinical manifestations. Even with low incidence, degenerative changes can occur and must show special interest in ultrasound exploration, as a challenge in the differential diagnosis with neoplastic disease based on a proper ultrasound assessment and follow-up .


Case presentation

Multiparous 39 years old patient, who consults for intermittent hypogastrium pain, with an eight-months evolution of increasing intensity, hypermenorrhoeas associated with increased waist circumference and intense anemic syndrome.

Physical examination evidenced a severe metrorrhagia, multiparous cervix without lesions, well epithelialized with a hard consistency, fixed and enlarged uterus, located one finger above the navel with a painful exploration. The analytical control shows hemoglobin in levels of 9.2 g / dL with a hematocrit of 29.3%.

The transvaginal ultrasound shows large uterus, with a dimension of 176 x 132 x146 mm, presenting therein a round cystic mass of 142 x 138 x 126 mm of well-defined boundary homogeneous content without excrescences, partitions or solid growth. It features poor vascularization, only in the lower pole with a RI of 0.54, all of this compatible with submucosal giant fibroid cystic degeneration. The endometrium is not clearly displayed because of  possible mass shift toward anterior face. Both ovaries displayed normal morphology and size (Figure 1).

During the study, a Magnetic Resonance (MR) was requested, it reported the existence of the mass described, it limited to uterine cavity without myometrial extension or cervical channel. The image could be diagnosed as a cystic degeneration versus sarcomatous endometrioid degeneration. The other pelvic organs in study were normal (Figure 2).

With all this findings, we decided to make a surgical resection, performing abdominal hysterectomy without oophorectomy, due to the age of the patient. During the surgery, after openning abdominal cavity, there was a large uterus according to a five months of gestation with a smooth surface, no adherences with good plane cleavage without infiltration, ascites or adenopathies (Figure 3).

After extraction of the 630 grams of weigh surgical piece, we proceeded to the incision, with high quantity jet of clear fluid, wich cytology and microbiology was negative. The Pathology study confirmed the diagnosis, being described as a submucosal leiomyoma with cystic degeneration and a thin capsule, displacing and compressing the underlying endometrium (Figure 4).

The patient had a normal postoperative period, requiring blood transfusion for low levels in postoperative control (Hemoglobin 7.3 g / dL) with a value of 10.1 mg / dL and Hematocrit of 32.2% before hospital discharge. The revision a month after surgery was normal.


Discussion

Uterine leiomyomas are defined as monoclonal smooth muscle growth, being the most common pelvic bening tumor in women [1].

The prevalence is very high, they can be diagnosed by ultrasound in 12-25% of women in reproductive age [2], but their presence is higher if we consider the asymptomatics ones, as shown by the study by Cramer et al, in which they found a rate of 77% in the presence of fibroids in hysterectomy surgical pieces [3]. Therefore, it is estimated that there is a risk of developing leiomyomas in 70% of white women and 80% of African-American women      throughout their lives [1]. 

Regarding clinical manifestations, most myomas are usually small and asymptomatic, but depending on the number, location and size, they may lead to abnormal uterine bleeding, pelvic discomfort and alterations in reproductive function. The  most common symptom in most cases is an unusual bleeding, with abundant and prolonged menstruation, but dealing with the fibroids, the occurrence of intermenstrual bleeding or menopausal bleeding is not common, so endometrial pathology should be studied [4].

The diagnosis is based on clinical examination and ultrasound study, preferably transvaginal ultrasound because of its  high sensitivity, subtracting many false positives in relation to adnexal pathology.

We can find degenerative changes in the myoma ultrasound study, especially in those of large size and years of evolution: the hyaline degeneration can be present (65% cases, the most frequent) especially  in submucosal fibroids, with appearance of acellular hyaline material, much tendency to have liquefaction transforming it into a cystic cavity (cystic degeneration) as occurred in the case presented. The calcification is typical in menopause with  areas of high refractive myoma inside or on the periphery, and finally the red degeneration (necrosis by rapid growth of the fibroid, typical during pregnancy around 10 weeks of gestation).

The differential diagnosis is considered in terms of two important diseases, being one of them a bening one, adenomyosis, and the oher a malignant one, sarcomatous degeneration.

Adenomyosis, defined as a diffusely implantation of ectopic endometrial tissue in myometrial thickness in women of reproductive age, sometimes gives isolated nodular growths, known as adenomyoma. They usually generate ultrasound images similar to myomas, but their differentiation is very important to focus on the evaluation of uterine morphology and endometrial myometrial junction, as well as to point out different studies, such as that published by Atri et al, focusing on comparing the ultrasound differences between with and without adenomyosis uteri [5].

In adenomyosis uterus, is typical the appearance of a myometrium with an ill-defined heterogeneous echostructure, with an alteration in the endometrial-myometrial junction, to distort this union, is not possible to distinguish  the underlying myometrium endometrium correctly, and this region appears occasionally erased and with even linear striations.

Regarding the clinical symptoms, adenomyosis usually associated abnormal uterine bleeding,  but dysmenorrhea will be the main complaint of the patient, being both symptons frecuently shown together. In the surgery the excision  is usually difficult, because there is not a good plane of separation from the underlying myometrium (in the absence of the pseudocapsule present in myomas).

The differential diagnosis is made with sarcomatous development, a malignant pathology. It is a rare entity, its incidencie in the U.S. is 3-7 per 100,000 inhabitants, with an average  of 60 years old at the diagnosis [6]. The clinical manifestations are similar to myomas, so the differential diagnosis is complicated. They may appear in advanced stages clinic metastases (usually lung and peritoneal infiltration), although there is some published case, as Ito et al, metastasis from benign myomas, being almost anecdotal [7].

Ultrasonography are similar, uterine myomas usually appear as spherical structures with regular edges and precise limits, a variable echogenicity with peritumoral halo (the presence of a  pseudocapsule is an unequivocal sign of benignity) and poor acoustic transmission with posterior shadowing. The greater the size is, the more heterogeneous they are, because for the appearance of necrosis and degeneration areas.

The sarcomas are described as focal mixed echogenic masses with central necrosis, with irregular vessels with low resistance rates and elevated systolic speed peak shown in color Doppler application.  This findings are very suggestive, although benign myomas sometimes can also generate this as well [8].
 
Many studies have attempted to use MR trying to differentiate the most of these two pathologies. Nowadays, the MR provides great information, but does not give a definitive diagnosis [9,10]. The hyperintensity marker  is typical but not safe, the sarcomatous lesions often have irregular borders, presence of calcifications, but even with it in different studies MR has a specificity of 93% with a positive predictive value around 53% [9].

Routinely MR should not be requested in suspected myomatous uterus, this imaging technique is better for the description of size, location of fibroids and other features. It will be applied only if there is doubt or pre approach surgery in selected cases.

There is a high interest in looking for  elevated serum markers for correct diagnosis, such as CA125 or different subtypes of lactate dehydrogenase. These are being studied in some patients diagnosed with uterine sarcoma, although they are still under investigation [11].

Even with the high frequency of pathology of myomas that exists, being in the majority of cases of benign character, knowledge is required in the differential diagnosis. 

The possibility of occurrence of degenerative changes, specially sarcomatous degeneration, obliges a correct assessment by ultrasound and clinical exploration, knowing the role of the use of other imaging techniques such as MR.



References


[1]	Day Baird D, Dunson DB, Hill MC. High cumulative incidence of uterine leiomyoma in black and white women: ultrasound evidence. Am J Obstet Gynecol 2003; 188:100

[2]	Downes E, Sikirica V, Gilabert-Estell�s J. The burden of uterine fibroids in five European countries. Eur J Obstet Gynecol Reprod Biol 2010; 152:96

[3]	Cramer SF, Patel A. The frecuency of uterine leiomyomas. Am J Clin Pathol 1990; 94-435.

[4]	Fraser IS, Critchley HO, Munro MG, et al. A process designed to lead to international agreement on terminologies and definitions used to describe abnormalities of menstrual bleeding. Fertil Steril 2007;87:466

[5]	Atri M, Reinhold C, Mehio AR, Chapman WB, Bret PM.  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed/10831700" Adenomyosis: US features with histologic correlation in an in-vitro study. Radiology. 2000;215(3):783-90.

[6]	Brooks SE, Zhan M, Cote T, Braquet CR. Surveillance, epidemiology, and end results analysis of 2677 cases of uterine sarcoma 1989-1999. Gynecol Oncol 2004;93:204

[7]	 HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Ito%20T%5BAuthor%5D&cauthor=true&cauthor_uid=22647339" Ito T,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Kawasaki%20N%5BAuthor%5D&cauthor=true&cauthor_uid=22647339" Kawasaki N,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Kinoshita%20M%5BAuthor%5D&cauthor=true&cauthor_uid=22647339" Kinoshita M,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Inada%20H%5BAuthor%5D&cauthor=true&cauthor_uid=22647339" Inada H,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Ikeda%20N%5BAuthor%5D&cauthor=true&cauthor_uid=22647339" Ikeda N. Benign metastasizing leiomyoma; report of a case. Kyobu Geka. 2012;65(6):513-5.

[8]	 Amant F, Coosemans A, Debiec-Rychter M, et al. Clinical management of uterine sarcomas. Lancet Oncol 2009;10:1188

[9]	  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Tirumani%20SH%5BAuthor%5D&cauthor=true&cauthor_uid=22699695" Tirumani SH,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Ojili%20V%5BAuthor%5D&cauthor=true&cauthor_uid=22699695" Ojili V,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Shanbhogue%20AK%5BAuthor%5D&cauthor=true&cauthor_uid=22699695" Shanbhogue AK,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Fasih%20N%5BAuthor%5D&cauthor=true&cauthor_uid=22699695" Fasih N,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Ryan%20JG%5BAuthor%5D&cauthor=true&cauthor_uid=22699695" Ryan JG,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Reinhold%20C%5BAuthor%5D&cauthor=true&cauthor_uid=22699695" Reinhold C. Current concepts in the imaging of uterine sarcoma. Abdom Imaging. 2012 Jun 15.

[10]	Tanaka YO, Nishida M, Tsunoda H, et al. Smooth muscle tumors of uncertain malignant potential and leiomyosarcomas of the uterus: MR findings. J Magn Reson Imaging 2004;20:998

[11]	Hoskins PJ, Le N. Preoperative tumor markers at diagnosis in woman with malignant mixed m�llerian tumors/carcinosarcoma of the uterus. Int Gynecol Cancer 2008;18:1200.

Figure Legendes

Figure 1
Transvaginal ultrasound cross section, which shows the large, homogenous mass

Figure 2
Sagittal MR, visualizing regular surface cystic mass confined to uterine cavity

Figure 3
Macroscopic visualization of surgical piece

Figure 4
Internal detail after incision and drainage, cyst capsule pulled by f�rceps
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