Styloid Process Syndrome and Ischemic Stroke: A Rare but Important Cause

doi:10.3969/j.issn.1673-5765.2025.07.014

Abstract

Abstract: Styloid process syndrome can lead to bone-derived ischemic stroke and represents a rare etiology of cerebrovascular disease. Carotid dissection caused by it is prone to being underdiagnosed and misdiagnosed. This article reviews the relationship between styloid process syndrome and ischemic stroke by analyzing existing literature and discussing the anatomical basis, epidemiological characteristics, pathological mechanisms of inducing cerebrovascular disease, and relevant diagnosis and treatment of styloid process syndrome. It aims to enhance clinicians’ understanding of ischemic stroke caused by styloid process syndrome and to provide a reference for clinical diagnosis and treatment.

Key words: Styloid process syndrome; Ischemic stroke; Carotid dissection; Diagnosis

摘要： 茎突综合征可导致骨源性缺血性卒中，是一种罕见的脑血管病病因，由其引起的颈动脉夹层易被漏诊和误诊。本文通过分析现有文献，综述了茎突综合征与缺血性卒中的关系，探讨了茎突综合征的解剖学基础、流行病学特征、引起脑血管病的病理机制及相关的诊断和治疗，旨在提高临床医师对茎突综合征引起缺血性卒中的认识，为临床诊治工作提供参考。

关键词: 茎突综合征; 缺血性卒中; 颈动脉夹层; 诊断

CLC Number:

SHI Yanping, LUO Yan, ZHU Longhai, SHI Dai, CAO Yongjun, SHI Jijun. Styloid Process Syndrome and Ischemic Stroke: A Rare but Important Cause[J]. Chinese Journal of Stroke, 2025, 20(7): 917-922.

石艳萍, 罗颜, 朱龙海, 时代, 曹勇军, 石际俊. 茎突综合征与缺血性卒中：罕见但必须重视的病因[J]. 中国卒中杂志, 2025, 20(7): 917-922.

References

[1] JADAV D，KANCHAN T，SHEKHAWAT R S，et al. Elongated styloid process：an incidental autopsy finding of clinical and medico-legal significance[J/OL]. J Forensic Leg Med，2022，87：102334[2025-01-15]. https://doi.org/10.1016/j. jflm.2022.102334.
[2] HAERTL J，RENZ M，WUNDERLICH S，et al. Bony stroke：ischemic stroke caused by mechanical stress on brain supplying arteries from anatomical bone or cartilage anomalies[J]. Stroke，2023，54（5）：1246-1256.
[3] SHI D，LIU C C，WANG L J，et al. Bilateral carotid artery dissection associated with Eagle syndrome in a drug-addicted young adult with stroke：a case report[J]. Neurol Sci，2022，43（10）：6105-6109.
[4] CRUDDAS L，JOFFE M，BAKER D. Can styloid process and internal carotid artery anatomy be used to predict carotid artery dissection？[J/OL]. Ann Vasc Surg，2021，74：105-110[2025-01-15]. https://doi.org/10.1016/j.avsg.2020.12.034.
[5] RADAK D，TANASKOVIC S，KECMANOVIC V，et al. Bilateral Eagle syndrome with associated internal carotid artery kinking and significant stenosis[J/OL]. Ann Vasc Surg，2016，34：271[2025-01-15]. https://doi.org/10.1016/j.avsg.2016.01.015.
[6] SONG J H，AHN S K，CHO C B. Elongated styloid process as a cause of transient ischemic attacks[J]. JAMA Neurol，2013，70（8）：1072-1073.
[7] LUO Y，CAO Y J，CAO Z S，et al. Ischemic stroke caused by bilateral internal carotid artery dissection due to elongated styloid processes：a case report[J]. Am J Med，2024，137（8）：719-721.
[8] EAGLE W W. Elongated styloid process；further observations and a new syndrome[J]. Arch Otolaryngol，1948，47（5）：630-640.
[9] KAPUR E，VOLJEVICA A，ŠAHINOVIĆ M，et al. Styloid process length variations：an osteological study[J]. Acta Med Acad，2022，51（1）：46-51.
[10] ELMAS F，SHRESTHA B L. Eagle’s syndrome[J/OL]. N Engl J Med，2017，377（13）：e18[2025-01-15]. https://doi.org/10.1056/NEJMicm1703542.
[11] RAZAK A，SHORT J L，HUSSAIN S I. Carotid artery dissection due to elongated styloid process：a self-stabbing phenomenon[J]. J Neuroimaging，2014，24（3）：298-301.
[12] OGURA T，MINEHARU Y，TODO K，et al. Carotid artery dissection caused by an elongated styloid process：three case reports and review of the literature[J]. NMC Case Rep J，2014，2（1）：21-25.
[13] GALLETTA K，SINISCALCHI E N，CICCIÙ M，et al. Eagle syndrome：a wide spectrum of clinical and neuroradiological findings from cervico-facial pain to cerebral ischemia[J/OL]. J Craniofac Surg，2019，30（5）：e424-e428[2025-01-15]. https://doi.org/10.1097/SCS.0000000000005453.
[14] TADJER J，BÉJOT Y. Vascular variant of Eagle syndrome：a review[J/OL]. Front Neurol，2024，15：1463275[2025-01-15]. https://doi.org/10.3389/fneur.2024.1463275.
[15] JARRAR E，NAIJA S，GRISSA O，et al. Eagle syndrome：a rare etiology of ischemic stroke ：a case report[J]. Clin Neuroradiol，2022，32（3）：869-871.
[16] HASSANI M，GRØNLUND E W，ALBRECHTSEN S S，et al. Neurological phenotypes and treatment outcomes in Eagle syndrome：systematic review and meta-analysis[J/OL]. PeerJ，2024，12：e17423[2025-01-15]. https://doi.org/10.7717/peerj.17423.
[17] ALDAKKAN A，DUNN M，WARSI N M，et al. Vascular Eagle’s syndrome：two cases illustrating distinct mechanisms of cerebral ischemia[J]. J Radiol Case Rep，2017，11（8）：1-7.
[18] JELODAR S，GHADIRIAN H，KETABCHI M，et al. Bilateral ischemic stroke due to carotid artery compression by abnormally elongated styloid process at both sides：a case report[J/OL]. J Stroke Cerebrovasc Dis，2018，27（6）：E89-E91[2025-01-15]. https://doi.org/10.1016/j.jstrokecerebrovasdis.2017.12.018.
[19] RASER J M，MULLEN M T，KASNER S E，et al. Cervical carotid artery dissection is associated with styloid process length[J]. Neurology，2011，77（23）：2061-2066.
[20] RENARD D，AZAKRI S，ARQUIZAN C，et al. Styloid and hyoid bone proximity is a risk factor for cervical carotid artery dissection[J]. Stroke，2013，44（9）：2475-2479.
[21] AMORIM J M，PEREIRA D，RODRIGUES M G，et al. Anatomical characteristics of the styloid process in internal carotid artery dissection：case-control study[J]. Int J Stroke，2018，13（4）：400-405.
[22] IKENOUCHI H，TAKAGI M，NISHIMURA A，et al. Bilateral carotid artery dissection due to Eagle syndrome in a patient with vascular Ehlers-Danlos syndrome：a case report[J/OL]. BMC Neurol，2020，20：285[2025-01-15]. https://doi.org/10.1186/s12883-020-01866-2.
[23] BARGIEL J，GONTARZ M，GĄSIOROWSKI K，et al. Stylohyoid chain syndrome（Eagle syndrome）in conjunction with carotid artery dissection：a case report of surgical treatment[J/OL]. Diseases，2024，12（1）：24[2025-01-15]. https://doi.org/10.3390/diseases12010024.
[24] ZUBER M，MEDER J F，MAS J L. Carotid artery dissection due to elongated styloid process[J/OL]. Neurology，1999，53（8）：1886[2025-01-15]. https://doi.org/10.1212/wnl.53.8.1886.
[25] QURESHI S，FAROOQ M U，GORELICK P B. Ischemic stroke secondary to stylocarotid variant of Eagle syndrome[J]. Neurohospitalist，2019，9（2）：105-108.
[26] GALLETTA K，GRANATA F，LONGO M，et al. An unusual internal carotid artery compression as a possible cause of Eagle syndrome—a novel hypothesis and an innovative surgical technique[J/OL]. Surg Neurol Int，2019，10：174[2025-01-15]. https://doi.org/10.25259/SNI_317_2019.
[27] SHINDO T，ITO M，MATSUMOTO J，et al. A case of juvenile stroke due to carotid artery dissection from an elongated styloid process-revisiting conservative management[J/OL]. J Stroke Cerebrovasc Dis，2019，28（10）：104307[2025-01-15]. https://doi.org/10.1016/j.jstrokecerebrovasdis.2019.104307.
[28] BERRADA K，EL OUALI I，ZAHI H，et al. Bilateral carotid dissection due to Eagle syndrome[J]. Radiol Case Rep，2023，19（3）：927-933.
[29] LIU B，LI Q J，ZHENG Y Y，et al. Ischemic stroke due to stylocarotid artery syndrome：a case report and review[J/OL]. J Int Med Res，2024，52（5）：3000605241253745[2025-01-15]. https://doi.org/10.1177/03000605241253745.
[30] KAVI T，LAHIRI S. Teaching neuroimages：Eagle syndrome：cerebrovascular complications[J/OL]. Neurology，2016，87（2）：e17[2025-01-15]. https://doi.org/10.1212/WNL.0000000000002848.
[31] YAGHI S，ENGELTER S，DEL BRUTTO V J，et al. Treatment and outcomes of cervical artery dissection in adults：a scientific statement from the American Heart Association[J/OL]. Stroke，2024，55（3）：e91-e106[2025-01-15]. https://doi.org/10.1161/STR.0000000000000457.
[32] ZINKSTOK S M，VERGOUWEN M D I，ENGELTER S T，et al. Safety and functional outcome of thrombolysis in dissection-related ischemic stroke：a meta-analysis of individual patient data[J]. Stroke，2011，42（9）：2515-2520.
[33] ENGELTER S T，RUTGERS M P，HATZ F，et al. Intravenous thrombolysis in stroke attributable to cervical artery dissection[J]. Stroke，2009，40（12）：3772-3776.
[34] HOOKER J D，JOYNER D A，FARLEY E P，et al. Carotid stent fracture from stylocarotid syndrome[J]. J Radiol Case Rep，2016，10（6）：1-8.
[35] SVEINSSON O，KOSTULAS N，HERRMAN L. Internal carotid dissection caused by an elongated styloid process（Eagle syndrome）[J/OL]. BMJ Case Rep, 2013：bcr2013009878[2025-01-15]. https://doi.org/10.1136/bcr-2013-009878.
[36] CHRCANOVIC B R，CUSTÓDIO A L N，DE OLIVEIRA D R F. An intraoral surgical approach to the styloid process in Eagle’s syndrome[J]. Oral Maxillofac Surg，2009，13（3）：145-151.
[37] ARAVINDAN V，MARIMUTHU M，KRISHNA V K，et al. Extraoral versus intraoral approach for removal of styloid process in treatment of Eagle’s syndrome：a report of two cases[J/OL]. Cureus，2023，15（5）：e38720[2025-01-15]. https://doi.org/10.7759/cureus.38720.