Center for Rheumatic Diseases, Matsuyama Red Cross Hospital, 1 Bunkyomachi, Ehime Prefecture, 790-8524, Matsuyama City, Japan.
Department of Respiratory Medicine, Japanese Red Cross Ishinomaki Hospital, Miyagi Prefecture, Ishinomaki City, Japan.
BMC Infect Dis. 2023 Mar 8;23(1):145. doi: 10.1186/s12879-023-08089-z.
Typical Lemierre's syndrome is usually secondary to an oropharyngeal infection. Recently, several cases following a primary infection site other than the oropharynx have been reported as atypical Lemierre's syndrome; although, these primary lesions are limited to the head and neck. This is the first case potentially sequential to infectious foci outside the head and neck.
We describe an atypical Lemierre's syndrome in a 72-year-old woman with rheumatoid arthritis, which occurred during the treatment of Streptococcus anginosus bacteremia acquired from a sacral ulcer infection related to rheumatoid vasculitis. At first, the symptoms resolved after the initial administration of vancomycin for the bacteremia caused by methicillin-resistant Staphylococcus aureus and Streptococcus anginosus that entered via a sacral ulcer. On the 8th day, the patient developed a fever of 40 °C and unexpectedly required 10 L of oxygen due to rapid deterioration of oxygenation temporarily. Immediately contrast-enhanced computed tomography was performed to investigate systemic thrombosis including pulmonary embolism. Afterward, the newly formed thrombi at the right external jugular vein, bilateral internal jugular veins, and the right small saphenous vein were detected, and apixaban was started. On the 9th day, the patient again had an intermittent fever of 39.7 °C, and continuous Streptococcus anginosus bacteremia was revealed; subsequently, clindamycin was administered. On the 10th day, she developed a left hemothorax; consequently, apixaban was discontinued, and a thoracic drain was inserted. She repeatedly had an intermittent fever of 40.3 °C, and contrast-enhanced computed tomography detected an abscess formation at the left parotid gland, pterygoid muscle group, and masseter muscle. After Lemierre's syndrome was diagnosed in combination with the abovementioned jugular vein thrombus, clindamycin was replaced with meropenem, and vancomycin was increased. Swelling of the lower part of the left ear became prominent with delay and peaked at approximately the 16th day. The subsequent treatment course was favorable, and she was discharged on the 41st day.
Clinicians should consider Lemierre's syndrome as the differential diagnosis of internal jugular vein thrombosis occurring during sepsis, even though an antibiotic is administered or a primary infection site is anything besides the oropharynx.
典型的勒米埃雷氏综合征通常继发于口咽感染。最近，一些病例报告了口咽以外的原发性感染部位为非典型勒米埃雷氏综合征; 尽管这些原发性病变仅限于头颈部。这是首例潜在继发于头颈部以外感染灶的病例。
我们描述了一例 72 岁类风湿关节炎女性的非典型勒米埃雷氏综合征，该患者在治疗由类风湿性脉管炎引起的骶部溃疡感染所致耐甲氧西林金黄色葡萄球菌和咽峡炎链球菌菌血症时发生。起初，在最初给予万古霉素治疗耐甲氧西林金黄色葡萄球菌和咽峡炎链球菌菌血症后，症状得到缓解，这些菌血症是通过骶部溃疡进入体内的。第 8 天，患者出现 40°C 的发热，由于氧合迅速恶化，意外需要 10 升氧气暂时支持。立即进行增强 CT 以检查包括肺栓塞在内的全身血栓形成。随后，在右侧颈外静脉、双侧颈内静脉和右侧小隐静脉发现新形成的血栓，并开始使用阿哌沙班。第 9 天，患者再次出现 39.7°C 的间歇性发热，连续出现咽峡炎链球菌菌血症; 随后给予克林霉素治疗。第 10 天，患者出现左侧血胸; 因此，停用阿哌沙班，并插入胸腔引流管。她反复出现 40.3°C 的间歇性发热，增强 CT 显示左侧腮腺、翼状肌群和咬肌形成脓肿。在结合上述颈内静脉血栓形成诊断为勒米埃雷氏综合征后，用美罗培南替换克林霉素，并增加万古霉素剂量。左耳下部肿胀延迟并在第 16 天左右达到高峰。随后的治疗过程顺利，第 41 天出院。
临床医生应将勒米埃雷氏综合征视为脓毒症期间颈内静脉血栓形成的鉴别诊断，即使给予抗生素或原发性感染部位不在口咽。
