Leiomyomas are benign uterine smooth muscle neoplasms with varied morphology that are well known to undergo secondary changes. Cotyledonoid dissecting leiomyoma is a rare and distinct form of leiomyoma that poses a diagnostic challenge for clinicians, radiologists, and pathologists and can be confused with malignant uterine neoplasms. Only a few cases have been reported so far in the literature. Here we report a case of a cotyledonoid dissecting leiomyoma in a 60-year-old woman, emphasize its gross and histological features, and provide a review of the literature.
Leiomyomas are benign smooth muscle neoplasms that arise from the myometrium and account for almost 75% of hysterectomy cases [
A 60-year-old woman presented with the complaint of a 1-year history of lower abdominal pain and third-degree uterovaginal descent. Her previous menstrual history was unremarkable. A vaginal examination revealed a bulky uterus with an ulceration on the anterior cervical lip. Ultrasonography revealed that the uterus was 7.5×3.4 cm in size with an endometrial thickness of 4.9 mm. Multiple uterine fibroids 2–4 cm in diameter were also noted. Both adenexa were unremarkable. A hysterectomy was performed and the specimen was sent for histopathological examination. Grossly, the cervix appeared hypertrophied and epidermidized. The endomyometrial thickness was 1.6 cm, while the endometrial thickness was 0.4 cm. Multiple fibroids up to 4×3.5 cm were noted. In addition, 2 subserosal fibroids 2 cm and 3.5 cm in diameter were seen. A cut section of the largest intramural fibroid and 1 subserosal fibroid revealed a solid, grayish-white, homogenous, and whorled appearance. The cut section of another subserosal fibroid revealed the presence of multiple grayish-white nodules (
Leiomyomas are the most common benign smooth muscle neoplasms of the uterus. A number of patterns of leiomyomas have been described. CDL, a very rare variant that is also commonly known as Sternberg tumor, was first reported by Roth et al. [
A variety of other unusual patterns of uterine leiomyoma have been described, such as parasitic leiomyoma, cellular leiomyoma, symplastic or bizarre leiomyoma, epithelioid leiomyoma, intravenous leiomyomatosis, and leiomyoma with secondary changes. Some CDL appear as large fungating masses with widespread extension into the broad ligament and pelvic cavity. Due to its rarity and a clinician’s lack of familiarity, such tumors are sometimes misdiagnosed as malignancies [
Gurbuz et al. [
SN | Reference | Age | Clinical presentation | Tumor size (cm) maximum dimension | Tumor location |
---|---|---|---|---|---|
1 | David et al. [1] | 65 | Abnormal uterine bleed | 15 | Uterine fundus and cervix |
48 | Uterine prolapse | 12 | Uterine fundus | ||
2 | Roth et al. [2] | 39 | Pelvic mass | 10.3 | Uterine cornua |
41 | Abnormal uterine bleed | 10 | Uterine cornua | ||
23 | Pelvic mass | 25 | Uterine cornua | ||
Unknown | Pelvic mass | 24 | Uterine cornua | ||
3 | Brand et al. [3] | 24 | Abdominal mass | NA | Uterine fundus |
4 | Roth and Reed [4] | 46 | Pelvic mass | 34 | Uterine cornua |
5 | Kim et al. [5] | 26 | Incidental | 12 | Posterior uterine wall |
6 | Cheuk et al. [6] | 55 | Abnormal uterine bleed | 10 | Uterine cornua |
7 | Stewart et al. [7] | 58 | Abdominopelvic mass | 16.4 | Uterine fundus |
8 | Jordan et al. [8] | 46 | Right adnexal mass | 22 | Uterine with extrauterine extension (all cases) |
46 | Pelvic mass | 20 | |||
46 | Pelvic mass | 10 | |||
46 | Pelvic mass | 18 | |||
36 | Abnormal uterine bleed | 13 | |||
34 | Uterine mass, infertility | 18 | |||
9 | Saeed et al. [9] | 27 | Pelvic mass | 41 | Uterine fundus |
10 | Maimoon et al. [10] | 40 | Urinary retention | 10 | Uterine isthmus |
11 | Shelekhova et al. [11] | 73 | Uterine mass | 8 | Uterine fundus |
12 | Gurbuz et al. [12] | 67 | Incidental | 10 | Uterine cornua |
13 | Weissferdt et al. [13] | 52 | Abnormal uterine bleed | 6.2 | Uterine fundus |
14 | Raga et al. [14] | 33 | Abdominal pain | 6 | Lateral part of uterus |
15 | Driss et al. [15] | 47 | Pelvic mass | 25 | Uterine with extrauterine extension |
16 | Preda et al. [16] | 41 | Uterine mass | 9 | Left and posterior uterine wall |
17 | Fukunaga et al. [17] | 56 | Constipation | 30 | Posterior uterine wall |
47 | Abdominal pain | 26 | Posterior uterine wall | ||
36 | Abnormal uterine bleed | 4 | Posterior uterine wall | ||
35 | Abdominal pain | 18 | Lateral uterine wall | ||
18 | Gezginç et al. [18] | 57 | Pelvic pain | 2.5, 4.5 | Intrauterine, lateral uterine wall |
19 | Agarwal et al. [19] | 52 | Abnormal uterine bleeding | 10 | Uterine cornua |
20 | Ersöz et al. [20] | 51 | Abnormal uterine bleeding | 8.5 | Subserosal |
21 | Roth et al. [21] | 33 | Abnormal uterine bleeding | 6.5, 13.5 | Posterior uterine wall |
22 | Tanaka et al. [22] | 36 | Incidental | 10 | Posterior & lateral uterine wall |
23 | Onu et al. [23] | 50 | Incidental | 10 | Uterine fundus |
24 | Kim et al. [24] | 43 | Abdominal mass | 13 | Uterine with extrauterine extension |
25 | Blake et al. [25] | 56 | Abnormal uterine bleeding | 30 | Uterine with extrauterine extension |
26 | Shimizu et al. [26] | 40 | Abnormal uterine bleeding | 10 | Posterior uterine wall |
27 | Rocha et al. [27] | 38 | Abnormal uterine bleeding | 25 | Uterine isthmus |
28 | Xu et al. [28] | 55 | Pelvic mass | 6 | Posterior uterine wall |
43 | Pelvic mass | 3 | Body of uterus | ||
37 | Pelvic mass | 30 | Periuterine | ||
48 | Lower abdominal pain | 6.7 | Right wall of uterus |
In conclusion, CDL is a unique and rare variant of leiomyoma with a characteristic gross nodular appearance and microscopic features. Increasing awareness among clinicians and pathologists regarding this rare entity will prevent inappropriate diagnosis and treatment.
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