Leiomyosarcoma of bone is just one of the variants of spindle cell sarcoma of bone characterised by the expression of desmin and other markers indicating a significant element of smooth muscle in the tumour, without osteoid production we have investigated the management and outcome of this rare type of primary malignant bone tumour. Method. Retrospective review of data stored on a prospective database. Results. In a database of 3364 patients with primary malignant bone sarcomas, 31 patients were identified with a primary leiomyosarcoma of bone. There were 12 males and 19 females with a mean age of 46 and tumour size of 8 cm. The most common site was the distal femur followed by the proximal tibia. Treatment was with chemotherapy and surgical resection. Seven of the patients had metastases at diagnosis. Surgery was carried out in 28 patients, 8 having amputation and 20 limb salvage. Three patients developed local recurrence, but half developed metastases. All patient disease-specific survival was 57% at five years and 44% at 10 yrs but for those without metastases was 82% and 60%, respectively. The only prognostic factors were metastases at diagnosis. Conclusion. Leiomyosarcoma of bone is a very rare primary malignant bone tumour affecting a predominantly older population. Despite the high incidence of metastases, survival is better than for other bone sarcomas for those without metastases at diagnosis.

https://downloads.hindawi.com/journals/sarcoma/2012/636849.pdf

Primary leiomyosarcoma of bone: analysis of prognosis.

Primary leiomyosarcoma of bone is a very rare tumor. Four cases are reported: 3 tumors were located in the femur and 1 in the talus. Clinical and imaging findings including radiographs, CT, and MRI are described. The final diagnosis was made by histopathological, immunohistochemical, and/or ultrastructural study after biopsy.

Primary leiomyosarcoma of bone: report of 4 cases.

Leiomyosarcoma is a rare type of malignant soft tissue tumor and also one of the most aggressive soft tissue sarcomas. It commonly occurs in uterus, abdominal viscera, retroperitoneal space and soft tissue of the extremities. Primary osseous leiomyosarcoma is a rare condition. Furthermore, primary leiomyosarcoma of the spine is extremely rare. Only few cases have been reported. However, the treatment and outcomes remains controversial. Therefore, the objective of this case report is to illustrate the management of this extremely rare disease by using total en bloc spondylectomy (TES) procedure, which is one of a suitable option for surgical resection of the spinal tumors. In this study, we presented an unusual case of a 61-year-old female, who was diagnosed as primary leiomyosarcoma of the twelfth thoracic spine with liver metastasis, treated with total en bloc spondylectomy of the twelfth thoracic vertebra followed by chemotherapy. On last follow-up, 6 months after the surgery, the clinical outcome remained in good condition and no signs of local recurrence. In conclusion, primary leiomyosarcoma of the spine is an extremely rare and difficult to diagnosis. Immunohistochemistry studies are very important for confirmation of the diagnosis. Standard treatment remains controversial. However, surgical resection is still treatment of choice. One of the most effective surgical options is TES, which give the better outcome and minimize local recurrence, if indicate.

Primary leiomyosarcoma of the spine treated with total en bloc spondylectomy: the first case in Thailand—a case report and literature review

Primary leiomyosarcoma (LMS) of bone is a rare and aggressive mesenchymal malignancy that differentiates toward smooth muscle. Complete resection is the only curable treatment, and novel therapeutic approaches for primary LMS of bone have long been desired. Patient-derived xenografts (PDXs) and cell lines are invaluable tools for preclinical studies. Here, we established PDXs from a patient with primary LMS of bone and a cell line from an established PDX. Bone primary LMS tissue was subcutaneously implanted into highly immune-deficient mice. After two passages, a piece of the tumor was subjected to tissue culturing, and a morphological evaluation and proteomic analysis were performed on the PDX and the established cell line. Moreover, the responses of the established cell line to anti-cancer drugs were examined. Microscopic observations revealed that the PDX tumors retained their original histology. The cell line was established from the third-generation PDX and named NCC-LMS1-X3-C1. The cells were maintained for over 18 mo and 40 passages. The cells exhibited a spindle shape and aggressive growth. Mass spectrometric protein identification revealed that the original tumor tissue, PDX tumor tissue, and NCC-LMS1-X3-C1 cells had similar but distinct protein expression profiles. We previously established the cell line, NCC-LMS1-C1, from the tumor tissue of same patient. We found that the response to drug treatments was different between NCC-LMS1-X3-C1 and NCC-LMS1-C1, suggesting the heterogeneous traits of tumor cells in the identical tumor tissue. This set of PDXs and stable cell line will be a useful resource for bone LMS research.

Establishment and characterization of patient-derived xenograft and its cell line of primary leiomyosarcoma of bone

Resume Les lymphomes malins primitifs du sein sont rares : 0,04 a 0,53 % de tous les cancers du sein. Leur symptomatologie clinique est polymorphe. L’imagerie medicale est non-specifique. Le diagnostic peut etre evoque a l’examen cytologique, sa confirmation est toujours histologique. La survenue d’un lymphome malin non hodgkinien impose un bilan d’extension soigneux. Le pronostic et le traitement rejoignent ceux des autres localisations lymphomatoses. Les auteurs rapportent une observation de lymphome malin non hodgkinien primitif du sein avec une revue de la litterature des aspects epidemiologiques, cliniques, histologiques et therapeutiques de ce neoplasme.

Lymphome malin non hodgkinien primitif du sein

L'os est compose d'une variete de tissus qui peuvent etre l'origine de lesions neoplasiques primitives. En consequence, il peut etre le site de divers sarcomes des tissus mous, en particulier, le leiomyosarcome. Le leiomyosarcome de l'os est considere comme primitif apres exclusion d'une extension osseuse d'une tumeur des parties molles de voisinage ou de la metastase d'un leiomyosarcome situe ailleurs. Nous rapportons un cas de leiomyosarcome primitif osseux, de localisation femorale gauche, chez une femme de 40 ans, confirme par une etude immuno-histochimique et ultrastructurale. Une resection du tiers inferieur du femur et une prothese massive gauche ont ete pratiquees. Un traitement complementaire par radiotherapie fractionnee a ete administre a la dose totale de 75Gy. L'evolution post-operatoire, un an apres, a ete marquee par une recidive locale et des metastases pulmonaires. Apres la metastasectomie pulmonaire et malgre l'amputation de la cuisse gauche, la malade a developpe une recidive locale au niveau du moignon de l'amputation et a ete jugee au dessus de toute ressource therapeutique. Nous discutons, a la lumiere des donnees de la litterature, les aspects anatomo-cliniques, immunohistochimiques et ultrastructuraux de cette tumeur rare.

[Primary bone leiomyosarcoma. Anatomo-clinical, immunohistochemical, and ultrastructural study].

Le lymphome malin non hodgkinien primitif du sein: À propos d'une observation

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[Primary bone leiomyosarcoma. Anatomo-clinical, immunohistochemical, and ultrastructural study].

Le lymphome malin non hodgkinien primitif du sein: À propos d'une observation