Atypical uterine leiomyoma: a case report and review of the literature

Journal of Medical Case Reports

Labinot Shahini²  Lloreta Kerliu³  Vjollca Sahatciu-Meka¹  Irma Kerliu-Saliu³  Suzana Manxhuka-Kerliu² 

[1] Faculty of Medicine, University of Prishtina, Mother Theresa Street NN, Prishtina, 10 000, Kosovo;Faculty of Medicine, Institute of Pathology, University of Prishtina, Mother Theresa Street NN, Prishtina, 10 000, Kosovo;Massachusetts College of Pharmacy and Health Sciences (MCPHS), 179 Longwood Avenue, Boston 02115, MA, USA

关键词: Prognostic markers;    Immunohistochemistry;    Histology;    Atypical uterine leiomyoma;   

Others  :  1235817
DOI  :  10.1186/s13256-016-0800-3

 received in 2015-03-22, accepted in 2016-01-04,  发布年份 2016

【 摘 要 】

Background

Atypical uterine leiomyomas show benign behavior. However, the distinction between leiomyomas and leiomyosarcomas may at times be problematic. We report a rare case of atypical uterine leiomyoma. We try to investigate potential immunohistochemical parameters that could be essential to distinguish cases of malignant smooth muscle tumors and those of uncertain or borderline histology.

Case presentation

A 56-year-old white ethnic Albanian woman from Kosovo presented with uterine bleeding because of uterine multiple leiomyomas. A hysterectomy with unilateral adnexectomy was performed. Her hysterectomy specimen contained multiple leiomyomas in submucosal, intramural and subserosal locations. The leiomyomas were well demarcated, firm and white with a whorled cut surface and one had foci of hemorrhage. Histology of most of the leiomyomas showed a whorled (fascicular) pattern of smooth muscle bundles separated by well-vascularized connective tissue. Smooth muscle cells were elongated with eosinophilic or occasional fibrillar cytoplasm and distinct cell membranes. Some of them developed areas of degeneration including hyaline change, with less than five mitotic figures per ten high power fields in most mitotically active areas, and no significant atypia. One leiomyoma was characterized by moderately to severely pleomorphic atypical tumor cells with low mitotic counts and no coagulative tumor cell necrosis. Immunohistochemistry showed strong immunoreactivity for vimentin, smooth muscle actin and desmin, while cyclin-dependent kinase inhibitor 2A (p16), and B-cell lymphoma 2 (bcl-2) showed focal immunoreactivity, estrogen and progesterone were positive, Ki-67 expressed a low proliferation index, whereas p21 and tumor suppressor gene p53 were negative.

Conclusions

The combination of evaluation of conventional morphologic criteria with cyclin-dependent kinase inhibitor 2A (p16), p21, progesterone, B-cell lymphoma 2, tumor suppressor gene p53 and Ki-67 expression may be of great value in the assessment of uterine smooth muscle tumors of uncertain or borderline histology.

【 授权许可】

   
2016 Manxhuka-Kerliu et al.

【 图 表 】

Fig. 1.

Fig. 2.

Fig. 3.

Fig. 4.

Fig. 5.

Fig. 6.

Fig. 7.

Figure 3.

Fig. 9.

Fig. 10.

【 参考文献 】

• [1]Sung CO, Ahn G, Song SY: Atypical leiomyomas of the uterus with long-term follow-up after myomectomy with immunohistochemical analysis for p16INK4A, p53, Ki-67, estrogen receptors, and progesterone receptors. Int J Gynecol Pathol 2009, 28(6):529-34.
• [2]Prayson RA, Hart WR: Pathologic considerations of uterine smooth muscle tumors. Obstet Gynecol Clin North Am 1995, 22(4):637-57.
• [3]Fattaneh A: Tavassoli Peter Devilee: Pathology and genetics of tumours of the breast and female genital organs. IARC Press, Lyon; 2003.
• [4]Ip PP, Cheung AN, Clement PB: Uterine smooth muscle tumors of uncertain malignant potential (STUMP): a clinicopathologic analysis of 16 cases. Am J Surg Pathol 2009, 33(7):992-1005.
• [5]Bodner-Adler B, Bodner K, Czerwenka K: Expression of p16 protein in patients with uterine smooth muscle tumors: an immunohistochemical analysis. Gynecol Oncol 2005, 96(1):62-6.
• [6]O’Neill CJ, McBride HA, Connolly LE: Uterine leiomyosarcomas are characterized by high p16, p53 and MIB1 expression in comparison with usual leiomyomas, leiomyoma variants and smooth muscle tumours of uncertain malignant potential. Histopathology 2007, 50(7):851-8.
• [7]Atkins KA, Arronte N, Darus CJ: The use of p16 in enhancing the histologic classification of uterine smooth muscle tumors. Am J Surg Pathol 2008, 32(1):98-102.
• [8]Ly A, Mills AM, McKenney JK: Atypical leiomyomas of the uterus: a clinicopathologic study of 51 cases. Am J Surg Pathol 2013, 37(5):643-9.
• [9]Mills AM, Ly A, Balzer BL: Cell cycle regulatory markers in uterine atypical leiomyoma and leiomyosarcoma: immunohistochemical study of 68 cases with clinical follow-up. Am J Surg Pathol 2013, 37(5):634-42.
• [10]Ünver NU, Acikalin MF, Öner Ü: Differential expression of P16 and P21 in benign and malignant uterine smooth muscle tumors. Arch Gynecol Obstet 2011, 284(2):483-90.
• [11]Bodner K, Bodner-Adler B, Kimberger O: Bcl-2 receptor expression in patients with uterine smooth muscle tumors: an immunohistochemical analysis comparing leiomyoma, uterine smooth muscle tumor of uncertain malignant potential, and leiomyosarcoma. J Soc Gynecol Investig 2004, 11(3):187-91.
• [12]Bodner K, Bodner-Adler B, Kimberger O: Estrogen and progesterone receptor expression in patients with uterine smooth muscle tumors. Fertil Steril 2004, 81(4):1062-6.
• [13]Mayerhofer K, Lozanov P, Bodner K: Ki-67 expression in patients with uterine leiomyomas, uterine smooth muscle tumors of uncertain malignant potential (STUMP) and uterine leiomyosarcomas (LMS). Acta Obstet Gynecol Scand 2004, 83(11):1085-8.
• [14]Petrović D, Babić D, Forko JI: Expression of Ki-67, P53 and progesterone receptors in uterine smooth muscle tumors. Diagnostic value. Coll Antropol. 2010, 34(1):93-7.
• [15]Hewedi IH, Radwan NA, Shash LS: Diagnostic value of progesterone receptor and p53 expression in uterine smooth muscle tumors. Diagn Pathol. 2012, 7:1. BioMed Central Full Text
• [16]Niemann TH, Raab SS, Lenel JC: p53 protein overexpression in smooth muscle tumors of the uterus. Hum Pathol 1995, 26(4):375-9.