Shoji Masatoshi, Nojima Naomi, Yoshikawa Akemi, Fukushima Wataru, Kadoya Naotaka, Hirosawa Hisashi, Izumi Ryohei
Department of Gastroenterological Surgery, Division of Cancer Medicine, Graduate School of Medical Science, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa 920-8641, Japan.
J Med Case Rep. 2014 Feb 27;8:77. doi: 10.1186/1752-1947-8-77.
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.
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.
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.
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。
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。
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。