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累及颅底和颞骨的非典型颌面放线菌病

Atypical Form of Cervicofacial Actinomycosis Involving the Skull Base and Temporal Bone.

作者信息

McCann Adam, Alvi Sameer A, Newman Jessica, Kakarala Kiran, Staecker Hinrich, Chiu Alexander, Villwock Jennifer A

机构信息

1 Department of Otolaryngology-Head and Neck Surgery, University of Kansas Health System, Kansas City, KS, USA.

2 University of Kansas School of Medicine, Kansas City, KS, USA.

出版信息

Ann Otol Rhinol Laryngol. 2019 Feb;128(2):152-156. doi: 10.1177/0003489418808541. Epub 2018 Oct 29.

Abstract

BACKGROUND

: Cervicofacial actinomycosis is an uncommon indolent infection caused by Actinomyces spp that typically affects individuals with innate or adaptive immunodeficiencies. Soft tissues of the face and neck are most commonly involved. Actinomyces osteomyelitis is uncommon; involvement of the skull base and temporal bone is exceedingly rare. The authors present a unique case of refractory cervicofacial actinomycosis with development of skull base and temporal bone osteomyelitis in an otherwise healthy individual.

METHODS

: Case report with literature review.

RESULTS

: A 69-year-old man presented with a soft tissue infection, culture positive for Actinomyces, over the right maxilla. Previous unsuccessful treatment included local debridement and 6 weeks of intravenous ceftriaxone. He was subsequently treated with conservative debridement and a prolonged course of intravenous followed by oral antibiotic. However, he eventually required multiple procedures, including maxillectomy, pterygopalatine fossa debridement, and a radical mastoidectomy to clear his disease. Postoperatively he was gradually transitioned off intravenous antibiotics.

CONCLUSIONS

: Cervicofacial actinomycosis involves soft tissue surrounding the facial skeleton and oral cavity and is typically associated with a history of mucosal trauma, surgery, or immunodeficiency. The patient was appropriately treated but experienced disease progression and escalation of therapy. Although actinomycosis is typically not an aggressive bacterial infection, this case illustrates the need for prompt recognition of persistent disease and earlier surgical intervention in cases of recalcitrant cervicofacial actinomycosis. Chronic actinomycosis has the potential for significant morbidity.

摘要

背景

颈面部放线菌病是由放线菌属引起的一种罕见的慢性感染,通常影响先天性或适应性免疫缺陷的个体。面部和颈部的软组织最常受累。放线菌性骨髓炎并不常见;累及颅底和颞骨极为罕见。作者报告了一例独特的难治性颈面部放线菌病病例,该病例在一名原本健康的个体中发展为颅底和颞骨骨髓炎。

方法

病例报告并进行文献综述。

结果

一名69岁男性患者右侧上颌骨出现软组织感染,放线菌培养阳性。先前的治疗未成功,包括局部清创和6周的静脉注射头孢曲松。随后,他接受了保守清创以及长时间的静脉注射,之后改为口服抗生素治疗。然而,他最终需要进行多次手术,包括上颌骨切除术、翼腭窝清创术和根治性乳突切除术以清除病灶。术后,他逐渐停用静脉抗生素。

结论

颈面部放线菌病累及面部骨骼和口腔周围的软组织,通常与黏膜创伤、手术或免疫缺陷病史相关。该患者接受了适当的治疗,但疾病仍进展且治疗升级。虽然放线菌病通常不是侵袭性细菌感染,但该病例表明,对于难治性颈面部放线菌病,需要及时识别持续性疾病并尽早进行手术干预。慢性放线菌病有可能导致严重的发病情况。

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