Metastatic leiomyosarcoma in the oral cavity: Case report with protein expression profiles

https://doi.org/10.1016/j.jcms.2009.06.010Get rights and content

Summary

Leiomyosarcoma (LMS) is a relatively uncommon malignant tumour derived from smooth muscle cells that rapidly metastasizes to distant regions. It rarely reaches oral tissues in which smooth muscle tissues are absent.

We report the case of a 56-year-old woman who presented with LMS in the maxilla that had metastasized from a primary tumour in her uterus, received a total hysterectomy with bilateral salpingo–oophorectomy 9 months earlier. To reveal the poor prognosis of metastatic LMS, a total of 26 antibodies against different factors related to the proliferation, apoptosis, necrosis, and angiogenesis were simultaneously applied on the immunohistochemistry and immuno-blot detection in order to screen for expression n of different proteins in the metastatic LMS.

Compared with the immunoreactions of primary uterine LMS, the different antibodies for cellular proliferation, i.e., proliferating cell nuclear antigen (PCNA), multiple primary neoplasm-2 (MPN-2), Max, p21, CDK4, p53, Rb-1, Bad, Bcl-2, epidermal growth factor receptor (EGF-R), hepatocyte growth factor (HGF), C-erbb2, Maspin, and DMBT-1, and those for angiogenesis, i.e., vWF, CD31, and Angiogenin, were more intensely expressed, while Bax, p16, Wnt-1, E-cadherin, and APC were relatively weakly expressed. In particular, beta-catenin was densely localized to the nuclei of tumour cells.

These data suggest that rapid proliferation of the tumour cells is related to over-expression of different oncogenes, and that the infiltrative growth and early distant metastasis of these tumour cells are related to over-expression of angiogenesis factors. A total of seven cases of metastatic LMS to the oral cavity that had been published in the English literature were reviewed, and the reason for the poor prognosis in the metastatic LMS is suggested in this case report.

Introduction

Leiomyosarcoma (LMS) is found rarely in the oral cavity and is associated with aggressive clinical behaviour and low survival. This report describes a LMS in the right upper buccal gingiva metastasized from a primary lesion in the uterus. Total seven cases of metastatic LMS to the oral cavity, including our own, have been published in the English literature to date. Clinical, histological, immunohistochemical, and immuno-blot detections using 26 antibodies against different factors related to the proliferation, apoptosis, necrosis, and angiogenesis were performed in order to screen for expression of different proteins in the metastatic LMS.

Section snippets

Case report

A 56-year-old Korean woman was referred to the Department of Oral and Maxillofacial Surgery, Kangnung National University Dental Hospital, complaining of painful swelling with a necrotic ulcer measuring roughly 4.0 × 3.0 cm on the right upper buccal gingiva (Fig. 1A). Cervical or submandibular lymphadenopathy was not detected. She had no previous dental history relevant to the lesion but had undergone a total hysterectomy with bilateral salpingo–oophorectomy for a diagnosis of LMS, nine months

Discussion

In general, soft tissue sarcomas, including LMSs of the head and neck, aggressively invade surrounding tissues and disseminate haematogenously. Dissemination is frequently to the lungs, and regional lymph node involvement tends to occur later. LMS has a higher propensity to metastasize than do fibrosarcoma and liposarcoma (Hajdu, 2007, Ethunandan et al., 2007). Metastatic LMSs to the oral cavity are extremely rare, and we were able to review only six cases of metastatic LMS to the oral cavity

Conclusions

The prognosis of metastatic LMS is generally poor, and five patients did not survive more than two years after the initial diagnosis of intraoral metastatic lesion (Kaziro, 1981, Bogart et al., 1990, Allen et al., 1993, Vora and Levin, 2003). When compared with the immunoreactions of normal gingiva and primary uterine LMS, most oncogenes for cellular proliferation and angiogenesis in the metastatic oral LMS were more intensely expressed. These data suggest that rapid proliferation of the

Conflict of interest

There are no conflicts of interest in this article.

Acknowledgement

This work was supported by the Korea Research Foundation Grant funded by the Korean government (MOEHRD, Basic Research Promotion Fund) (KRF-2009-R1A4A002-0075286).

References (9)

There are more references available in the full text version of this article.

Cited by (31)

  • Metastasis of leiomyosarcoma to the sublingual region

    2024, Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology
  • A rare pediatric variant of organized hematoma in the maxillary sinus

    2015, Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology
  • Melanotic and non-melanotic malignancies of the face and external ear - A review of current treatment concepts and future options

    2014, Cancer Treatment Reviews
    Citation Excerpt :

    LMS of the oral and perioral region can be differentiated based upon the histological features in LMS of the oral soft tissue, LMS of the facial skin, and LMS of the jawbone. Primary LMS of the head and neck and metastases originating from the female genitalia, especially from the uterus, are known [182]. Superficial LMS are classified into two subtypes according to their primary site of origin: the cutaneous forms arise from the arrector pili muscles of hair follicles or dartos muscles of genital skin, are associated with epidermal change, such as ulceration, umbilication, and color changes, and are usually small and grow slowly; the subcutaneous forms arise from the smooth muscle lining of arterioles and veins in the subcutaneous tissue, are not associated with surface changes, and usually grow faster.

  • Osteogenetic changes in elongated styloid processes of Eagle syndrome patients

    2014, Journal of Cranio-Maxillofacial Surgery
    Citation Excerpt :

    Microsections were stained with hematoxylin and eosin, followed by immunohistochemical staining using antibodies against osteonectin, osteocalcin, BMP-2, BMP-4, RANKL, HO-1, HSP-70, or HSP-90. All immunostaining was performed with an indirect triple sandwich method as previously described (Lee et al., 2005; Kim et al., 2009). Background cross-reaction was minimized by negative control staining without primary antibodies using the same procedures.

View all citing articles on Scopus
View full text