Undifferentiated high-grade pleomorphic sarcoma of ethmoid sinus: a case report and literature review

Zhu, Yupeng; Hao, Dapeng; Tang, Xiaoyan; Liang, Sun

doi:10.1016/j.bjorl.2017.05.004

Cited by 8 publications

(7 citation statements)

References 10 publications

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“…In our department, radical resection with negative margins has been performed for the UPS patients. Comparatively, the prognosis of UPS arising from oral-maxillary area is much poorer when compared with UPS in other regions [14,15]. In this study, we also observed that the cases with better responses to the neoadjuvant chemotherapy (anlotinib) had improved OS, and IHC staining also indicated the positive expression of CD31 and ERG in some cases.…”

Section: Discussionsupporting

confidence: 63%

Primary Undifferentiated Pleomorphic Sarcoma in Oral-maxillary Area: Retrospective Study and Molecular Analysis

Xiao

You

et al. 2020

Preprint

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BackgroundUndifferentiated pleomorphic sarcoma (UPS) in oral-maxillary area is rarely reported. Herein, we aimed to investigate the clinical characteristics, treatment strategies, prognosis, and molecular features of the oral-maxillary UPS. MethodsRetrospectively, we reviewed the UPS patients who were diagnosed and treated in our department. The medical histories, imaging features, histopathological characteristics, treatment strategies, clinical outcomes were summarized and analyzed. Besides, the molecular features were demonstrated by whole exonic sequencing. ResultsTotally, 10 cases with primary oral-maxillary UPS were included. The rapidly progressive UPS can easily develop at advanced and life-threatening stage, especially concerning the complex anatomical structures and spaces in the oral-maxillary area. The finial diagnosis for UPS greatly depends on histological findings and immunohistochemistry under the exclusion of all the possible differential diagnosis. Retrospectively, the treatment strategies for the involved cases still referred to those of oral squamous cell carcinoma. Statistically, the median overall survival (OS) for all the included cases was 7.75 months (range: 5-17 months). Comparatively, 3 cases had improved OS (median survival: 17 months, range: 17-18 months) experienced PR/SD with neoadjuvant chemotherapy (anlotinib). Cancer driver genes detection revealed GBP4 as a candidate driver gene for the oral-maxillary UPS. Additionally, a missense mutation in gene PIK3CA (p.E545K) was also identified. ConclusionOur findings can greatly expand the knowledge about the oral-maxillary UPS, and provide molecular evidences to improve the therapeutic options for the oral-maxillary UPS. Further studies are warranted to validate our discoveries on the oral-maxillary UPS.

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Section: Discussionsupporting

confidence: 63%

Primary Undifferentiated Pleomorphic Sarcoma in Oral-maxillary Area: Retrospective Study and Molecular Analysis

Xiao

You

et al. 2020

Preprint

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“…UPS, formerly called malignant fibrous histiocytoma and declassified by the World Health Organization in 2002, is a rare and malignant subtype characterized by a lack of specific immunohistochemical markers for any specific lineage of differentiation. 5,6 It represents the fourth most common soft tissue sarcoma and has an incidence of about 0.08-1 per 100 000. 7 In the present case, both preoperative imaging modalities and hormonal studies indicated a possible functional adrenal tumor secreting cortisol and catecholamine.…”

Section: Discussionmentioning

confidence: 99%

Undifferentiated pleomorphic sarcoma of the retroperitoneum mimicking a cortisol‐ and catecholamine‐secreting adrenal tumor

2022

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Introduction Retroperitoneal tumors with endocrine abnormalities are suspected to be functional adrenal tumors. Retroperitoneal soft tissue sarcomas are rare tumors, without endocrine potential. Case presentation A 60‐year‐old male was referred for a 15 cm mass in the left suprarenal space. His plasma cortisol and catecholamine levels were elevated. He underwent open left adrenalectomy with radical nephrectomy and his endocrinological abnormalities were improved. Pathological findings suggested that it had originated from the retroperitoneal fat tissue, and a diagnosis of undifferentiated pleomorphic sarcoma was made based on the results of immunohistochemical analysis and fluorescence in situ hybridization. Interestingly, neither cortisol nor catecholamine was elevated when, 6 months after surgery, local recurrence developed. Conclusion This is the first reported case of undifferentiated pleomorphic sarcoma accompanied by high levels of cortisol and catecholamine. We should keep in mind the possibility of tumors like retroperitoneal soft tissue sarcomas inducing endocrine abnormalities.

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“…UPS is derived from primitive undifferentiated mesenchymal cells and lacks specific immunohistochemical markers[ 3 ]. Immunohistochemically, Vim, CD68, AAT, and AACT are usually positive, while epithelial markers such as CK5/6 and SMA are generally negative[ 4 ].…”

Section: Discussionmentioning

confidence: 99%

Local hyperthermia combined with chemotherapy for the treatment of multiple recurrences of undifferentiated pleomorphic sarcoma: A case report

Zhou¹,

Wang²,

Zhang³

et al. 2022

WJCC

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BACKGROUND Undifferentiated pleomorphic sarcoma (UPS) is a type of soft tissue sarcoma, the histologic origin and differentiation direction of which are still unclear. There are few treatment options for UPS other than surgery. Herein we describe a patient who had multiple recurrences of UPS postoperatively, but R0 resection was achieved by local hyperthermia combined with chemotherapy, thus providing a new treatment approach for similar situations. CASE SUMMARY A 65-year-old man sought evaluation from a physician for a mass on his right back. After surgery, the pathologic diagnosis was fibrosarcoma. During the follow-up evaluations until 2021, the patient had four relapses of varying degrees. Postoperative pathology confirmed the recurrence of UPS on the right back. In March 2021, he underwent local hyperthermia combined with two cycles of chemotherapy for recurring lesions. After magnetic resonance imaging re-examination and preoperative examination, the patient chose surgery again. During the operation, the tumors were easy to excise, the amount of bleeding decreased significantly, and the pathologic evaluation confirmed that one of the specimens was an R0 excision. CONCLUSION Local hyperthermia combined with chemotherapy enables R0 resection to be achieved in patients with advanced UPS recurrence.

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Undifferentiated high-grade pleomorphic sarcoma of ethmoid sinus: a case report and literature review

Cited by 8 publications

References 10 publications

Primary Undifferentiated Pleomorphic Sarcoma in Oral-maxillary Area: Retrospective Study and Molecular Analysis

Primary Undifferentiated Pleomorphic Sarcoma in Oral-maxillary Area: Retrospective Study and Molecular Analysis

Undifferentiated pleomorphic sarcoma of the retroperitoneum mimicking a cortisol‐ and catecholamine‐secreting adrenal tumor

Local hyperthermia combined with chemotherapy for the treatment of multiple recurrences of undifferentiated pleomorphic sarcoma: A case report

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