1 UCSF Pediatric Headache, University of California, San Francisco, CA, USA.
2 UCSF Child Neurology, University of California, San Francisco, CA, USA.
Cephalalgia. 2018 Feb;38(2):374-382. doi: 10.1177/0333102416681570. Epub 2017 Jan 18.
Objective Neck-Tongue syndrome (NTS) is characterized by brief attacks of neck or occipital pain, or both, brought out by abrupt head turning and accompanied by ipsilateral tongue symptoms. As the disorder is rare, we undertook a systematic review of the literature to identify all reported cases in order to phenotype clinically the disorder and subsequently inform clinical diagnostic criteria. Methods Two electronic databases were searched using the search term "neck tongue syndrome". All English language references were reviewed in full. Cases were abstracted using a standardized abstraction form and the references of the retrieved articles were reviewed by hand to identify additional references and cases. Conference proceedings from recent headache meetings were searched. We also report six new cases from our centers. Results There were 39 primary cases, 56% of which were female. Mean age (SD) at onset was 16 (12) years. Twenty (53%) experienced neck pain, seven (18%) occipital pain, and 11 (29%) both. Pain was most often sharp or stabbing and severe, lasting several seconds to several minutes. Eleven experienced numbness and/or tingling in the neck/occiput following the pain. Thirty-six had an accompanying tongue sensory disturbance and three a motor/posture disturbance; five had both. Thirteen had other headaches, and four a family history of Neck-Tongue syndrome. Conclusions Neck-Tongue syndrome typically has pediatric or adolescent onset, suggesting that ligamentous laxity during growth and development may facilitate transient subluxation of the lateral atlantoaxial joint with sudden head turning. Familial cases suggest a genetic predisposition in some individuals. Neck-Tongue syndrome should be re-instated in the International Classification of Headache Disorders.
Objective Neck-Tongue syndrome (NTS) 以突发转头诱发的短暂颈部或枕部疼痛为特征，或两者兼有，同时伴有同侧舌症状。由于该疾病较为罕见，我们进行了系统的文献回顾，以识别所有报告的病例，从而对该疾病进行临床表型分析，并随后为临床诊断标准提供信息。
方法 我们使用搜索词“neck tongue syndrome”在两个电子数据库中进行搜索。全面审查了所有英语文献。使用标准化的摘要表格提取病例，并通过手动审查检索文章的参考文献来识别其他参考文献和病例。搜索了最近头痛会议的会议论文集。我们还报告了来自我们中心的 6 个新病例。
结果 共有 39 例原发性病例，其中 56%为女性。发病时的平均年龄（标准差）为 16（12）岁。20 例（53%）经历颈部疼痛，7 例（18%）经历枕部疼痛，11 例（29%）同时经历颈部和枕部疼痛。疼痛通常是尖锐或刺痛的，严重程度为数秒到数分钟。11 例在疼痛后出现颈部/枕部麻木和/或刺痛。36 例伴有舌感觉障碍，3 例伴有运动/姿势障碍；5 例同时伴有两者。13 例伴有其他头痛，4 例伴有 Neck-Tongue syndrome 家族史。
结论 Neck-Tongue syndrome 通常在儿童或青少年期发病，这表明生长发育期间韧带松弛可能会促进 lateral atlantoaxial 关节的瞬时半脱位，导致突然转头。家族病例表明一些个体存在遗传易感性。Neck-Tongue syndrome 应重新列入国际头痛疾病分类。
