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感染性先天性腰骶部皮窦道伴脊髓圆锥表皮样脓肿:一种罕见的疾病。

Infected congenital lumbosacral dermal sinus tract with conus epidermoid abscess: a rare entity.

机构信息

Department of Neurosurgery, AIIMS, Raipur, 492099, India.

Department of Pathology & Laboratory Medicine, AIIMS, Raipur, India.

出版信息

Childs Nerv Syst. 2021 Mar;37(3):741-747. doi: 10.1007/s00381-020-04987-8. Epub 2020 Nov 27.

Abstract

PURPOSE

Dermal sinus is more commonly associated with intradural dermoid than an epidermoid cyst. Conus epidermoid cyst with dermal sinus is a rare entity. We are presenting a rare case of infected conus epidermoid cyst along with the dermal sinus in an 18-month-old girl presented with flaccid paraparesis with sphincter dysfunction and timely intervention leads to complete recovery. We had searched PubMed for previously reported similar cases and did a case-based review of the literature.

CASE REPORT

This 18-month-old girl with discharging lumbosacral sinus with fever since 3 days presented with flaccid paraparesis with sphincter dysfunction. Preoperative magnetic resonance imaging (MRI) showed a large enhancing lesion from L1-S1 along with the dermal sinus tract. Complete excision of the cyst along with the sinus tract, followed by long-term antibiotic therapy. The excision of the infected cyst was done through myelotomy under neuromonitoring, while some part of the capsule densely adherent to the neural tissue was left behind. The patient gradually improved following surgery and motor power of the lower limbs were [Formula: see text] while going home. Histopathology revealed epidermoid cyst with secondary inflammatory tissue. Follow-up MRI of the spine showed excision of the dermal sinus tract and cyst with postoperative changes. At 1-year follow-up, the patient was asymptomatic without any focal deficits.

CONCLUSION

Early surgical intervention followed by long-term antibiotic therapy is a must for good functional recovery in patients of an infected dermal sinus tract with associated cyst. While excising cyst through myelotomy, some part of the capsule densely adherent to neural tissue may be left behind. Regular follow-up in the first year of surgery is essential to look for the recurrence of the lesion.

摘要

目的

皮窦与硬脊膜内皮样瘤的关系比表皮样囊肿更为常见。伴有皮窦的脊髓圆锥表皮样囊肿是一种罕见的实体。我们报告了一例 18 个月大的女孩患有感染性脊髓圆锥表皮样囊肿伴皮窦的罕见病例,表现为弛缓性截瘫伴括约肌功能障碍,及时干预可导致完全康复。我们在 PubMed 上搜索了以前报道的类似病例,并对文献进行了基于病例的回顾。

病例报告

这名 18 个月大的女孩因腰骶部窦道有分泌物伴发热 3 天就诊,表现为弛缓性截瘫伴括约肌功能障碍。术前磁共振成像(MRI)显示 L1-S1 有一个大的增强病变,伴有皮窦道。我们对囊肿和窦道进行了完全切除,随后进行了长期的抗生素治疗。在神经监测下通过椎板切开术切除感染的囊肿,而部分囊壁与神经组织紧密粘连。术后,患者逐渐恢复,下肢运动功能为[Formula: see text],出院回家。组织病理学显示表皮样囊肿伴继发性炎症组织。脊柱 MRI 随访显示皮窦道和囊肿切除术后改变。在 1 年的随访中,患者无症状,无任何局灶性缺损。

结论

对于感染性皮窦道伴发囊肿的患者,早期手术干预加长期抗生素治疗是获得良好功能恢复的必要条件。在通过椎板切开术切除囊肿时,部分与神经组织紧密粘连的囊壁可能会被遗留。术后第一年定期随访对于寻找病变复发至关重要。

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