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坐骨直肠窝罕见孤立性纤维瘤:病例报告

A rare solitary fibrous tumor in the ischiorectal fossa: a case report.

作者信息

Morikawa Kazuhiko, Takenaga Shinsuke, Masuda Koichi, Kano Asami, Igarashi Takao, Ojiri Hiroya, Ueda Kaoru, Ishiyama Mamoru, Fukasawa Nei

机构信息

Department of Radiology, The Jikei University Katsushika Medical Center, 6-41-2, Aoto, Katsushika-ku, Tokyo, 125-8506, Japan.

Department of Radiology, The Jikei University School of Medicine, 3-25-8, Nishi- Shimbashi, Minato-ku, Tokyo, 105-8461, Japan.

出版信息

Surg Case Rep. 2018 Oct 3;4(1):126. doi: 10.1186/s40792-018-0533-1.

Abstract

BACKGROUND

A solitary fibrous tumor (SFT) is a rare mesenchymal tumor that occurs mostly in pleural sites, and an SFT occurring in the ischiorectal fossa is extremely rare. Because of the rarity, there are few reports detailing an SFT in the ischiorectal fossa.

CASE PRESENTATION

A pararectal tumor was incidentally found in a 42-year-old man during a routine medical examination. The patient had no symptoms and no previous medical history. In the physical examination, a smooth-margined and hard elastic mass was felt, and in a digital rectal examination, the rectal mucosa appeared normal. A computed tomography (CT) scan showed a 5-cm, well-defined, solid mass in the left ischiorectal fossa. Contrast-enhanced CT in the early phase showed intense heterogeneous enhancement that persisted during the delayed phase. T2-weighted images of magnetic resonance imaging yielded heterogeneous intermediate and low signal intensity. Intense arterial enhancement suggested a hypervascular nature, and persistent delayed enhancement and low signal bands on T2-weighted images suggested a fibrous component of the mass. An SFT was suspected. Most SFTs are benign but have malignant potential. Our patient did not hope for surgery if the tumor was benign; therefore, an ultrasound-guided transperineal core needle biopsy was performed to decide on a treatment strategy. Microscopic examination showed tumor cells appearing as spindle and fibroblast-like cells within a collagenous stroma. Immunohistochemistry identified CD34 and vimentin, supporting the diagnosis of an SFT. The patient consented to excision of the mass. He was placed in a prone jackknife position, and the tumor was removed transperineally using a posterior approach (modified Kraske procedure). The levator ani muscle, external sphincter muscles, and rectum were not involved and separated from the tumor. The tumor was successfully resected en bloc with no complications. Five uneventful days post surgery, the patient was discharged. There was no local recurrence during the year following surgery.

CONCLUSION

Imaging findings reflect the tissue characterization such as hypervascularity and fibrous nature of SFTs. We have presented a rare case of an SFT in the ischiorectal fossa with useful imaging findings for diagnosis, treatment strategy, and successful surgical removal using a posterior approach.

摘要

背景

孤立性纤维瘤(SFT)是一种罕见的间叶组织肿瘤,主要发生于胸膜部位,发生于坐骨直肠窝的SFT极为罕见。由于其罕见性,关于坐骨直肠窝SFT的详细报道较少。

病例介绍

一名42岁男性在常规体检时偶然发现直肠旁肿瘤。患者无症状,既往无病史。体格检查时可触及边缘光滑、质地硬且有弹性的肿块,直肠指检时直肠黏膜外观正常。计算机断层扫描(CT)显示左侧坐骨直肠窝有一个5厘米、边界清晰的实性肿块。增强CT早期显示强烈的不均匀强化,延迟期持续存在。磁共振成像的T2加权图像显示不均匀的中等和低信号强度。动脉期强烈强化提示肿瘤血供丰富,延迟期持续强化及T2加权图像上的低信号带提示肿块的纤维成分。怀疑为SFT。大多数SFT是良性的,但有恶变潜能。如果肿瘤是良性的,我们的患者不希望接受手术;因此,进行了超声引导下经会阴粗针穿刺活检以确定治疗策略。显微镜检查显示肿瘤细胞呈梭形和成纤维细胞样,位于胶原性间质内。免疫组化检测到CD34和波形蛋白,支持SFT的诊断。患者同意切除肿块。患者取俯卧折刀位,经会阴采用后入路(改良Kraske手术)切除肿瘤。肛提肌、外括约肌和直肠未受累,与肿瘤分离。肿瘤成功完整切除,无并发症。术后5天恢复顺利,患者出院。术后一年无局部复发。

结论

影像学表现反映了SFT的组织特征,如血供丰富和纤维性质。我们报告了一例罕见的坐骨直肠窝SFT病例,其影像学表现对诊断、治疗策略及采用后入路成功手术切除具有指导意义。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ed62/6170515/7904c3070ec0/40792_2018_533_Fig1_HTML.jpg

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