Tan Emily T C, Rostamzad Parinaz, Esser Yasmin S, Pleumeekers Mieke M, Loudon Sjoukje E
Department of Ophthalmology, Erasmus MC, University Medical Center, 3000 CA Rotterdam, The Netherlands.
Clinical Sciences for Health Professionals, Program in Clinical Health Sciences, University Medical Center Utrecht, Utrecht University, 3584 CS Utrecht, The Netherlands.
J Clin Med. 2023 Sep 19;12(18):6059. doi: 10.3390/jcm12186059.
(1) Background: Patients with unicoronal craniosynostosis (UCS) often show torticollis which can result from either an ocular cause or contraction of the sternocleidomastoid muscle. For clinicians, it is crucial to know the prevalence of ocular torticollis (OT) to ensure appropriate referral for treatment. Furthermore, associated ophthalmic features with OT in these patients are scarcely described. The aim of this study was to determine the prevalence of OT in non-syndromic UCS patients and investigate its associated ophthalmic features. (2) Methods: In this descriptive cross-sectional study medical records of non-syndromic UCS patients treated between 1994-2022 in one tertiary care hospital in The Netherlands were retrospectively reviewed. Collected data included: diagnosis and type of torticollis, binocular single vision (BSV), strabismus, ocular motility, alphabetical patterns, refractive error, and amblyopia. Patients were classified as OT, based on their ophthalmic and/or orthoptic diagnosis. Prevalence was determined with the 95% CI using the Clopper-Pearson exact test. Associations between OT and the ophthalmic features were determined using Chi-square or Fishers' exact test and its effect size was calculated using Cramer's V. (3) Results: In total, 146 patients were included, of whom 57 had torticollis. An ocular cause for the torticollis was found in 54 patients. The prevalence of OT was 37% (n = 146; 95% CI [0.292-0.454]). Significant associations were found between OT and strabismus ( < 0.001), ocular motility abnormalities ( < 0.001), alphabetical patterns ( < 0.001), and amblyopia ( = 0.002). BSV ( = 0.277) and refractive error ( = 1.0) were not significantly associated with OT. However, in OT the BSV was relatively poor (42.1%) and more frequently absent (26.3%) compared to the non-torticollis group (7% poor and 16.3% absent). In both groups, excyclotorsion was predominantly present (62.3%). (4) Conclusions: In 95% of cases, torticollis in UCS patients is ocular-related. Overall, one in three patients with UCS have OT. This study emphasizes the importance of a timely referral of all patients with UCS with torticollis to an orthoptist and/or ophthalmologist, specialized in diagnosing and treatment of OT, before considering physiotherapy.
（1）背景：单冠状缝早闭（UCS）患者常出现斜颈，其原因可能是眼部问题或胸锁乳突肌挛缩。对于临床医生而言，了解眼性斜颈（OT）的患病率对于确保适当转诊治疗至关重要。此外，这些患者中OT相关的眼科特征鲜有描述。本研究的目的是确定非综合征性UCS患者中OT的患病率，并调查其相关的眼科特征。（2）方法：在这项描述性横断面研究中，回顾性分析了1994年至2022年在荷兰一家三级护理医院接受治疗的非综合征性UCS患者的病历。收集的数据包括：斜颈的诊断和类型、双眼单视（BSV）、斜视、眼球运动、字母模式、屈光不正和弱视。根据眼科和/或视光学诊断，将患者分类为OT。使用Clopper-Pearson精确检验确定患病率及其95%置信区间（CI）。使用卡方检验或Fisher精确检验确定OT与眼科特征之间的关联，并使用Cramer's V计算其效应大小。（3）结果：总共纳入了146例患者，其中57例有斜颈。在54例患者中发现了斜颈的眼部原因。OT的患病率为37%（n = 146；95% CI [0.292 - 0.454]）。发现OT与斜视（< 0.001）、眼球运动异常（< 0.001）、字母模式（< 0.001）和弱视（= 0.002）之间存在显著关联。BSV（= 0.277）和屈光不正（= 1.0）与OT无显著关联。然而，与非斜颈组相比，OT组的BSV相对较差（42.1%）且更频繁缺失（26.3%）（非斜颈组分别为7%差和16.3%缺失）。在两组中，外旋转斜视占主导（62.3%）。（4）结论：在95%的病例中UCS患者的斜颈与眼部相关。总体而言，三分之一的UCS患者有OT。本研究强调，在考虑物理治疗之前，应及时将所有患有斜颈的UCS患者转诊至擅长诊断和治疗OT的视光师和/或眼科医生处。
