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一例罕见的先天性胸段静脉异常及心外静脉窦缺损病例,该无症状成年患者首次表现为脑脓肿:病例报告

An uncommon case of congenital thoracic venous anomaly and extracardiac sinus venosus defect in an asymptomatic adult first presenting with brain abscesses: a case report.

作者信息

Pettersson Staffan, Trzebiatowska-Krzynska Aleksandra, Engvall Jan

机构信息

Department of Cardiology, Linkoping University Hospital, 58185 Linkoping, Sweden.

Department of Clinical Physiology, Prof. Linkoping University Hospital, 58185 Linkoping, Sweden.

出版信息

Eur Heart J Case Rep. 2022 Feb 7;6(2):ytac052. doi: 10.1093/ehjcr/ytac052. eCollection 2022 Feb.

DOI:10.1093/ehjcr/ytac052
PMID:35299706
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8922713/
Abstract

BACKGROUND

Congenital thoracic venous anomalies (CTVAs) with right-to-left shunt constitute an uncommon source of paradoxical embolization in adults. We present a case of a healthy and physically fit individual with a rare asymptomatic anomaly first presenting with brain abscesses after a visit to the dental office; persistent left superior vena cavae (PLSVC) without bridging vein, over-riding right-sided superior vena cavae (RSVC) connected to the left atrium (LA), and an extracardiac sinus venosus defect.

CASE SUMMARY

A 29-year-old male presented to the neurosurgical unit due to intracranial abscesses requiring intervention following a visit to his dentist. The abscess cultures isolated bacteria commonly found in the normal oral flora. Transthoracic echocardiography revealed an enlarged coronary sinus consistent with PLSVC. An agitated saline study was performed and raised suspicion of simultaneous extra- and intracardiac shunting. Magnetic resonance angiography confirmed the presence of a PLSVC and revealed an RSVC connected to the LA; however, no intracardiac shunt was evident. Electrocardiogram-gated computed tomography was therefore conducted and discovered the rudimentary remains of the physiologic RSVC forming a connection to the right atrium, explaining the bilateral contrast loading seen on the agitated saline study and diagnosing an extracardiac sinus venosus defect (SVD). The patient recovered and has been referred for surgery.

DISCUSSION

This case illustrates a CTVA and a forme fruste type SVD resulting in a severe complication in a healthy adult. We highlight the diagnostic challenges posed, suggest early usage of agitated saline studies, and discuss the rationale for surgical correction of this patient.

摘要

背景

伴有右向左分流的先天性胸段静脉异常(CTVAs)是成人反常栓塞的罕见原因。我们报告一例健康且体格健壮的个体,其患有罕见的无症状异常,在就诊牙科诊所后首次出现脑脓肿;存在无桥静脉的持续左上腔静脉(PLSVC)、横跨至连接左心房(LA)的右侧上腔静脉(RSVC)以及心外窦静脉缺损。

病例摘要

一名29岁男性因颅内脓肿就诊神经外科,该脓肿是在他看过牙医后需要进行干预治疗的。脓肿培养分离出正常口腔菌群中常见的细菌。经胸超声心动图显示冠状窦扩大,符合PLSVC。进行了生理盐水激发试验,怀疑存在心外和心内同时分流。磁共振血管造影证实存在PLSVC,并显示有一条RSVC连接至LA;然而,未发现明显的心内分流。因此进行了心电图门控计算机断层扫描,发现生理性RSVC的残留部分与右心房形成连接,解释了生理盐水激发试验中出现的双侧造影剂充盈情况,并诊断为心外窦静脉缺损(SVD)。患者康复后已被转诊接受手术。

讨论

本病例说明了一种CTVAs和一种不完全型SVD在一名健康成人中导致了严重并发症。我们强调了所面临的诊断挑战,建议早期使用生理盐水激发试验,并讨论了对该患者进行手术矫正的理论依据。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7581/8922713/7b83814dbf45/ytac052f8.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7581/8922713/8e072385999c/ytac052f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7581/8922713/a540e8d929d8/ytac052f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7581/8922713/8983a28e4290/ytac052f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7581/8922713/932fed98d7e5/ytac052f4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7581/8922713/7b83814dbf45/ytac052f8.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7581/8922713/8e072385999c/ytac052f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7581/8922713/a540e8d929d8/ytac052f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7581/8922713/8983a28e4290/ytac052f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7581/8922713/932fed98d7e5/ytac052f4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7581/8922713/7b83814dbf45/ytac052f8.jpg

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