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一名成年患者骶前脓肿并发库拉里诺综合征:病例报告

Currarino syndrome in an adult presenting with a presacral abscess: a case report.

作者信息

Shoji Masatoshi, Nojima Naomi, Yoshikawa Akemi, Fukushima Wataru, Kadoya Naotaka, Hirosawa Hisashi, Izumi Ryohei

机构信息

Department of Gastroenterological Surgery, Division of Cancer Medicine, Graduate School of Medical Science, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa 920-8641, Japan.

出版信息

J Med Case Rep. 2014 Feb 27;8:77. doi: 10.1186/1752-1947-8-77.

Abstract

INTRODUCTION

Currarino syndrome (Currarino triad) was described in 1981 as a triad syndrome with a common embryogenesis in infants and with three characteristics: anorectal stenosis, a defect in the sacral bone, and a presacral mass. We describe here an unusual case of Currarino syndrome in an adult presenting with a presacral abscess but no meningitis.

CASE PRESENTATION

A 32-year-old Japanese man presented with fever, arthralgia and buttock pain. A digital rectal examination showed mild rectal stenosis with local warmth and tenderness in the posterior wall of his rectum. Computed tomography showed a scimitar-shaped deformity of his sacrum and an 8cm presacral mass, which continued to a pedicle of his deformed sacrum. This was diagnosed as Currarino syndrome with a presacral abscess. The abscess was drained by a perianal approach with our patient treated with antibiotics. His symptoms soon disappeared. After three months, an excision was performed through a posterior sagittal approach. His postoperative course was uneventful and he was discharged 10 days after surgery. A histopathological examination revealed an infected epidermoid cyst. He has been free from recurrence as of four years and six months after surgery.

CONCLUSIONS

We report a case of Currarino syndrome in an adult who presented with a presacral abscess but no meningitis. Abscess drainage followed by radical surgery resulted in a successful outcome.

摘要

引言

库拉里诺综合征(库拉里诺三联征)于1981年被描述为一种在婴儿中具有共同胚胎发育过程的三联征综合征,具有三个特征:肛门直肠狭窄、骶骨缺损和骶前肿块。我们在此描述一例成人库拉里诺综合征的罕见病例,该患者表现为骶前脓肿但无脑膜炎。

病例报告

一名32岁的日本男性出现发热、关节痛和臀部疼痛。直肠指检显示直肠轻度狭窄,直肠后壁局部发热和压痛。计算机断层扫描显示其骶骨呈弯刀形畸形,并有一个8厘米的骶前肿块,该肿块延续至其畸形骶骨的一个椎弓根。这被诊断为伴有骶前脓肿的库拉里诺综合征。通过肛周途径引流脓肿,并给予患者抗生素治疗。他的症状很快消失。三个月后,通过后矢状入路进行了切除手术。他的术后病程顺利,术后10天出院。组织病理学检查显示为感染性表皮样囊肿。截至手术四年零六个月,他未出现复发。

结论

我们报告一例成人库拉里诺综合征病例,该患者表现为骶前脓肿但无脑膜炎。脓肿引流后行根治性手术取得了成功。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/581d/4234930/d1bbe0446eed/1752-1947-8-77-1.jpg

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