Primary intracranial β-human chorionic gonadotropin–producing leiomyosarcoma in a 2-year-old immunocompetent child

Kelley, Brian C.; Arnold, Paul M.; Newell, Kathy L.

doi:10.3171/2012.4.peds1216

Cited by 10 publications

(6 citation statements)

References 24 publications

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“…After treatment with hCG, CX3CL1 transcript levels have been shown to increase in mice and Bax mRNA levels have been shown to decrease in human endometrial stromal cells . The expression of ß‐hCG by liposarcoma, leiomyosarcoma and synovial sarcoma has been reported . Hormones/receptors expressed by STSs may regulate chemokines and affect the prognosis of STS patients in a gender‐specific manner, as summarized in the suggested model (Fig.…”

Section: Discussionmentioning

confidence: 94%

“…46 The expression of ß-hCG by liposarcoma, leiomyosarcoma and synovial sarcoma has been reported. [47][48][49] Hormones/receptors expressed by STSs may regulate chemokines and affect the prognosis of STS patients in a gender-specific manner, as summarized in the suggested model ( Fig. 3).…”

Section: Discussionmentioning

confidence: 99%

“…This hypothesis is supported by the expression of hormone receptors, including the estrogen and androgen receptors, in STSs and recent reports regarding the ectopic expression of the hormone human chorionic gonadotropin (hCG), which can regulate chemokines in different STS entities. [47][48][49] The correlation between elevated chemokine expression and a favorable prognosis may be related to chemokine-induced apoptotic pathways 35 and their influence on the cellular composition of the tumor microenvironment. 18…”

Section: Discussionmentioning

confidence: 99%

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High coexpression of CCL2 and CX3CL1 is gender‐specifically associated with good prognosis in soft tissue sarcoma patients

Kehlen

Greither

Wach

et al. 2014

Intl Journal of Cancer

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Chemokines are involved in both the negative and positive regulation of inflammatory processes, angiogenesis and cancer/cancer stem cell proliferation as well as the chemoattraction of tumor cells to metastatic sites. The aim of this study was to measure the mRNA expression levels of three chemokines, CCL2, CCL7 and CX3CL1, in soft tissue sarcomas (STSs) and to assess the correlations between these levels as well as their correlations with clinicopathological data and the disease-specific survival of STS patients. The mRNA levels of CCL2, CCL7 and CX3CL1 were analyzed in tumor tissues from 126 STS patients using qPCR. Low mRNA expression of CCL2 and CX3CL1 was significantly correlated with a worse prognosis (RR 5 1.98; p 5 0.019 and RR 5 2.10; p 5 0.014; multivariate Cox's regression analysis). A combined low expression of CCL2 and CX3CL1 was associated with a significantly increased risk of tumor-related death as compared to patients with high expression levels of both chemokines (RR 5 3.08; p 5 0.003). A gender-specific multivariate analysis revealed that female STS patients with low CX3CL1 mRNA expression had a 3.46-fold increased risk of death (p 5 0.004). Low expression of both CCL2 and CX3CL1 mRNAs resulted in an additive 5.37-fold increased risk of tumor-related death (p 5 0.003) as compared to those with high expression of both parameters in female patients. In conclusion, this is the first study to show a significant correlation between combined low expression of CCL2 and CX3CL1 and a poor prognosis for STS patients, particularly in female patients.Soft tissue sarcomas (STSs) are a heterogeneous group of malignant mesenchymal tumors. 1,2 Clinically relevant prognostic factors include tumor size, tumor grade, the localization of the primary tumor 3 and the type of tumor resection. In addition, several molecular factors with prognostic impact have been identified (reviewed in Refs. 4 and 5). However, there is still an urgent need to characterize further prognosisassociated molecular factors for an improved prognostication and treatment management. In several cancer types, chemokines have been discussed as biomarkers for detecting early metastasis, have been associated with tumor progression, and have been suggested as potential therapeutic targets. Chemokines represent a group of chemotactic cytokines responsible for orchestrating cell recruitment under both homeostatic and inflammatory conditions. The small (8-10 kDa) chemokine proteins are classified into four highly conserved groups-C, CC, CXC and CX3C-based on the position of the first two cysteines that are adjacent to the amino terminus. 6 The binding of the chemokine ligand to its seventransmembrane G-protein-coupled receptor activates downstream signaling that leads to cell movement in development, inflammation, wound healing and cancer. 7 Oncogenic

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Section: Discussionmentioning

confidence: 94%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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High coexpression of CCL2 and CX3CL1 is gender‐specifically associated with good prognosis in soft tissue sarcoma patients

Kehlen

Greither

Wach

et al. 2014

Intl Journal of Cancer

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“…Age of presentation in previous case reports ranged from 2 to 73 years. [4,5] There is usually no sex predilection and the median duration of symptoms before presentation is nearly 4 months. [6] Intraparenchymal location of the tumor is mostly observed in systemic metastases and primary examples are rare.…”

Section: Discussionmentioning

confidence: 99%

Primary extradural leiomyosarcoma involving cavernous sinus in an immunocompetent patient

Gulwani

Garg²

2017

IJNS

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C A S E R E P O R Tcranial nerve palsy. Routine laboratory studies were within normal limits. Serology was negative for HIV. Her magnetic resonance imaging brain [ Figures 1 and 2] revealed an isointense lesion on T2-weighted images with brilliant enhancement on contrast involving right middle cranial fossa base with cavernous sinus involvement and destruction of petrous bone. A possibility of skull base meningioma was considered. Operative detailsPatient underwent right temporal craniotomy, zygomatic osteotomy, interdural approach and tumor decompression under intra-operative neuronavigation. Intra-operatively, tumor was soft to firm, highly vascular, and extra-axial. The portion extending into the cavernous sinus was left behind due to significant bleeding. Resected tumor was sent for histopathology examination. Pathology findingsHistological examination revealed a tumor with pushing borders that was adhered to the nonneoplastic nerve bundle. Tumor was composed of spindle shaped cells that were arranged in interweaving fascicles. The tumor cells had cigar shaped nuclei with pointed ends and indistinct cytoplasmic borders [ Figure 3a and b]. There was brisk mitotic activity with several bizarre and giant nuclei. The stroma of tumor was richly vascular with areas of hyalinization. On immunohistochemistry (IHC), the tumor cells were positive for desmin, smooth muscle actin and vimentin [ Figure 4a]. The tumor cells were negative for S-100, epithelial membrane antigen (EMA), glial fibrillary acidic protein and CD117. Ki-67 staining revealed a high proliferation index of 25-30% [ Figure 4b].

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“…There are rare reports of pure leiomyosarcomas secreting β-hCG in the literature. [8][9][10] In the absence of the GCT components just mentioned, the neoplastic smooth muscle component of the tumor with expression of β-hCG was the likely source for the elevated serum β-hCG levels.…”

Section: Discussionmentioning

confidence: 99%

A Case of a Beta-Human Chorionic Gonadotropin Secreting Sinonasal Teratocarcinosarcoma

2014

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Background and Importance Sinonasal teratocarcinosarcoma (SNTCS) is an unusual and aggressive neoplasm characterized by the combination of teratoma and carcinosarcoma features that is often misdiagnosed based on a biopsy sample. Approximately 60 cases have been reported in the English literature, but none have been associated with serum tumor markers. We report a case of SNTCS with elevation of serum β-human chorionic gonadotropin (hCG) level. Clinical Presentation The patient was a 22-year-old pregnant woman at diagnosis, and her β-hCG level was appropriately elevated. Her initial treatment was a surgical resection followed by radiation therapy (RT). Two years later, the patient had a pregnancy work-up after a sexual assault. Her serum β-hCG was elevated, but a nongravid uterus was seen on ultrasound. Magnetic resonance imaging (MRI) of the head revealed an intracranial tumor recurrence in the right frontal lobe. During salvage RT, β-hCG decreased rapidly to normal levels, with a complete tumor response on MRI. Retrospective examination of the initial surgical specimen showed expression of β-hCG associated with the anaplastic mesenchymal component. Conclusion This is the first reported case of a β-hCG secreting SNTCS. The source of β-hCG production was traced to a neoplastic smooth muscle component of the tumor.

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Primary intracranial β-human chorionic gonadotropin–producing leiomyosarcoma in a 2-year-old immunocompetent child

Cited by 10 publications

References 24 publications

High coexpression of CCL2 and CX3CL1 is gender‐specifically associated with good prognosis in soft tissue sarcoma patients

High coexpression of CCL2 and CX3CL1 is gender‐specifically associated with good prognosis in soft tissue sarcoma patients

Primary extradural leiomyosarcoma involving cavernous sinus in an immunocompetent patient

A Case of a Beta-Human Chorionic Gonadotropin Secreting Sinonasal Teratocarcinosarcoma

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