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通过荧光原位杂交分析颅部筋膜炎中的USP6基因重排:三例报告

USP6 Gene Rearrangement by FISH Analysis in Cranial Fasciitis: A Report of Three Cases.

作者信息

Salib Christian, Edelman Morris, Lilly Joshua, Fantasia John E, Yancoskie Aaron E

机构信息

Department of Pathology, Westchester Medical Center at New York Medical College, 100 Woods Rd., Valhalla, NY, 10543, USA.

Division of Anatomical Pathology, Cytopathology, Pediatric Pathology, Cohen Children's Medical Center, Hofstra-Northwell School of Medicine, 270-05 76th Avenue, New Hyde Park, NY, 11040, USA.

出版信息

Head Neck Pathol. 2020 Mar;14(1):257-261. doi: 10.1007/s12105-019-01018-0. Epub 2019 Feb 13.

Abstract

Cranial fasciitis (CF) is an uncommon benign myofibroblastic proliferation involving the soft and hard tissues of the cranium. It typically occurs in the pediatric population with a male predilection (male-to-female ratio 1.5:1). The clinical presentation is usually a rapidly expanding, painless nodule. Bone erosion may be appreciated radiographically. Histopathologic sections of CF show plump, fibroblast-like cells with pale, oval shaped nuclei and prominent nucleoli in a fibrous or myxoid background. Growth is self-limited and surgical excision is considered curative. Due to these features, CF is thought to be a variant of nodular fasciitis (NF). As with NF, CF may mimic a sarcomatous process and pose a diagnostic challenge to clinicians and pathologists alike. Erickson-Johnson et al. identified rearrangements of the ubiquitin-specific protease 6 (USP6) gene in 44 of 48 cases of NF. MYH9 was the fusion partner in 12 of these cases. To date, the molecular profile of CF has not been studied. Here we present the molecular findings in three cases of CF identified at our institution. Each case was subjected to fluorescence in-situ hybridization with appropriate negative controls. Two of three cases were positive for the USP6 gene rearrangement. The third case failed to hybridize, likely related to nucleic acid damage secondary to decalcification. Negative controls did not demonstrate the genetic rearrangement. These findings warrant further investigation of the USP6 gene rearrangement in CF, as it may prove helpful as a diagnostic adjunct in challenging cases.

摘要

颅骨筋膜炎(CF)是一种罕见的良性肌成纤维细胞增生性疾病,累及颅骨的软组织和硬组织。它通常发生于儿童群体,男性更易患病(男女比例为1.5:1)。临床表现通常为一个迅速增大的无痛性结节。影像学检查可能发现骨质侵蚀。CF的组织病理学切片显示,在纤维性或黏液样背景中,有丰满的、成纤维细胞样细胞,其细胞核呈淡色、椭圆形,核仁突出。生长具有自限性,手术切除被认为可治愈。由于这些特征,CF被认为是结节性筋膜炎(NF)的一种变体。与NF一样,CF可能类似肉瘤样病变,给临床医生和病理学家都带来诊断挑战。埃里克森 - 约翰逊等人在48例NF病例中的44例中发现了泛素特异性蛋白酶6(USP6)基因重排。其中12例的融合伴侣是MYH9。迄今为止,尚未对CF的分子特征进行研究。在此,我们展示了在我们机构确诊的3例CF的分子学研究结果。每例均进行了荧光原位杂交,并设置了适当的阴性对照。3例中的2例USP6基因重排呈阳性。第3例未能杂交,可能与脱钙导致的核酸损伤有关。阴性对照未显示基因重排。这些发现值得对CF中的USP6基因重排进行进一步研究,因为它可能在疑难病例的诊断辅助中被证明是有用的。

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引用本文的文献

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Mod Pathol. 2017 Nov;30(11):1577-1588. doi: 10.1038/modpathol.2017.78. Epub 2017 Jul 28.
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