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Concurrent Craniosynostosis and Positional Plagiocephaly: A Scoping Review 并发颅畸形和位置性长颅畸形:范围审查
Pub Date : 2024-05-11 DOI: 10.1177/27325016241252953
Gwendolyn E. Daly, Peter Ferrin, J. Carboy, Lori K. Howell, Nathan R Selden, Erik M. Wolfswinkel
Craniosynostosis and positional plagiocephaly (PP) both lead to deformities of the infant skull. In some cases, patients may present with concurrent craniosynostosis and PP. The forces exerted on the skull by both conditions can complicate and delay correct diagnosis and require adjustments to surgical planning. The purpose of this scoping review was to identify and analyze the existing peer-reviewed literature covering the diagnosis and treatment of concurrent craniosynostosis and PP. In accordance with PRISMA guidelines, PubMed, MEDLINE, and EMBASE were searched for articles discussing concurrent diagnoses of craniosynostosis and PP. Out of 633 articles discovered, 7 met inclusion criteria and discussed a cumulative total of 10 patients. The average age of referral to a craniofacial team was 17 ± 22.2 months. The most common reason for referral was craniofacial asymmetry (90%, n = 9). 40% presented with unilateral coronal synostosis (n = 4), 40% with lambdoid synostosis (n = 4), 10% with sagittal synostosis (n = 1), and 10% with metopic synostosis (n = 1). The most commonly reported clinical features were occipital flattening (90%, n = 9), ear displacement (80%, n = 8), and frontal bossing (30%, n = 3). Delayed or incorrect initial diagnosis was noted in 70% (n = 7) of patients. CT scan was the most commonly employed diagnostic tool, utilized in 100% of patients. 70% of patients (n = 7) underwent surgery at an average age of 6.75 ± 3 months. Two studies, accounting for 2 patients reported, noted altering their surgical technique to account for PP in addition to the synostotic suture. Coincident craniosynostosis and PP can pose a complicated diagnostic and therapeutic challenge. An in-depth understanding of the most reliable physical exam findings associated with each type of craniosynostosis combined with PP will facilitate accurate diagnosis. Consideration of the concurrent forces on the skull created by craniosynostosis and PP aid in the design of a tailored surgical plan.
颅骨发育不全(Craniosynostosis)和位置性头盖骨畸形(PP)都会导致婴儿颅骨畸形。在某些情况下,患者可能会同时出现颅骨发育不良和位置性颅骨发育不良。这两种疾病对颅骨的作用力会使正确诊断变得复杂和延迟,并需要调整手术计划。本次范围界定综述旨在识别和分析现有的同行评议文献,这些文献涉及并发颅骨发育不良和 PP 的诊断和治疗。根据 PRISMA 指南,我们在 PubMed、MEDLINE 和 EMBASE 上检索了讨论并发颅骨发育不良和 PP 诊断的文章。在发现的 633 篇文章中,有 7 篇符合纳入标准,累计讨论了 10 名患者。转诊到颅颌面团队的平均年龄为(17 ± 22.2)个月。最常见的转诊原因是颅面不对称(90%,n = 9)。40%为单侧冠状突畸形(4例),40%为羊角突畸形(4例),10%为矢状突畸形(1例),10%为偏侧突畸形(1例)。最常见的临床特征是枕骨变平(90%,9 例)、耳朵移位(80%,8 例)和额部隆起(30%,3 例)。70%(7 例)患者的初步诊断延迟或不正确。CT 扫描是最常用的诊断工具,100% 的患者都进行了 CT 扫描。70%的患者(7 例)在平均年龄(6.75 ± 3 个月)时接受了手术。有两项研究(占报告患者总数的 2%)指出,除了合体缝合外,他们还改变了手术技术以考虑 PP。并发颅骨发育不良和 PP 可带来复杂的诊断和治疗难题。深入了解与每种颅合畸形合并 PP 相关的最可靠的体格检查结果将有助于准确诊断。考虑到颅骨失稳和 PP 同时对颅骨产生的作用力,有助于设计量身定制的手术方案。
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引用次数: 0
A Mandibular Brown Tumor: A Pediatric Case Report and Summary of Adolescence Cases 下颌骨棕色瘤:儿科病例报告和青少年病例摘要
Pub Date : 2024-05-11 DOI: 10.1177/27325016241253276
Gabriella E. Glassman, Joseph K. Williams, Colin M. Brady
Mandibular brown tumors are often radiographically and histologically indistinguishable from cherubism, aneurysmal bone cyst, giant cell granuloma, and odontogenic fibroma. Laboratory findings of hyperparathyroidism (HPT) aid in directing therapy. We present a case of a 14-year-old male with a brown tumor of the mandible associated with secondary HPT due to vitamin D deficiency. Core biopsy exhibited a proliferation of plump spindle cells and osteoclast-like giant cells within variably fibrous stroma. Parathyroid hormone (PTH) levels were elevated to 288 pg/mL (normal 8.5-77.1 pg/mL) and vitamin D was low at a level of 6.9 ng/mL (normal > 20.0 ng/mL). Six months after the introduction of weekly vitamin D supplementation, PTH and vitamin D levels improved but remained abnormal with PTH of 86.8 pg/mL and vitamin D of 9.3 ng/mL. Alongside an improvement in laboratory indices, the tumor character stabilized and became less clinically friable; however, the burden remained unchanged. At 9 months, the patient underwent surgical debulking of the tumor with local soft tissue coverage. A review of pediatric literature was performed, among which 16 patients were identified. Most children who developed brown tumors were found to have secondary HPT and ultimately 44% underwent some degree of surgical debridement of the mass.
下颌骨褐色瘤通常在放射学和组织学上与樱桃核、动脉瘤性骨囊肿、巨细胞肉芽肿和牙源性纤维瘤难以区分。甲状旁腺功能亢进(HPT)的实验室检查结果有助于指导治疗。我们报告了一例14岁男性患者的病例,他患有下颌骨棕色肿瘤,因缺乏维生素D而继发HPT。核心活检结果显示,在不同的纤维基质内有丰满的纺锤形细胞和破骨细胞样巨细胞增生。甲状旁腺激素(PTH)水平升高至288 pg/mL(正常值为8.5-77.1 pg/mL),维生素D水平低至6.9 ng/mL(正常值大于20.0 ng/mL)。每周补充维生素 D 六个月后,PTH 和维生素 D 水平有所改善,但仍不正常,PTH 为 86.8 pg/mL,维生素 D 为 9.3 ng/mL。在实验室指标改善的同时,肿瘤特征也趋于稳定,在临床上变得不那么易碎;然而,肿瘤的负担仍保持不变。9 个月后,患者接受了手术切除肿瘤,并进行了局部软组织覆盖。我们查阅了儿科文献,发现其中有 16 例患者。大多数患棕色瘤的儿童都被发现患有继发性HPT,最终44%的患儿接受了某种程度的肿块手术清创。
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引用次数: 0
Pericranial Flap Reconstruction in Pediatric Patients Undergoing Neuromodulatory Surgery: Impact on Wound Healing 接受神经调节手术的小儿患者的颅周皮瓣重建:对伤口愈合的影响
Pub Date : 2024-04-14 DOI: 10.1177/27325016241246784
Lawrence O. Lin, Annie Orr, Gregory D. Pearson, Jonathan Pindrik, Ammar Shaikhouni, Alyssa Fogolin, I. Khansa
Pediatric patients with medically-intractable epilepsy may require modulation of seizure foci to reduce frequency and severity of seizures. These neuromodulatory procedures, which include responsive neurostimulator (RNS) and deep brain stimulator (DBS), involve the implantation of high-profile and long-term cranial hardware, placing patients at risk of wound healing complications, hardware exposure and infection. A multidisciplinary neurosurgical and plastic surgical approach utilizing pericranial flap coverage of hardware may decrease wound healing complications in these cases. This study compares the wound-healing outcomes in patients undergoing RNS/DBS insertion with and without pericranial flap coverage. A retrospective chart review was conducted of all patients who underwent RNS and DBS insertion with or without pericranial flap coverage at a level 1 pediatric medical center between 2014 and 2022. Wound healing outcomes at 60 days were evaluated. Data were compared using Fisher’s exact test. Twenty-seven patients underwent 29 neuromodulatory epilepsy procedures (14 DBS, 15 RNS). Twenty of the procedures included a pericranial flap to cover hardware. Median length of stay was 2.0 days for both cohorts. Two subjects without pericranial flap reconstruction had a wound healing complication (22.2%), compared to none of the procedures that included a pericranial flap ( P = .09). In pediatric patients undergoing insertion of responsive neurostimulators and deep brain stimulators for medically-intractable epilepsy, a multidisciplinary neurosurgery and plastic surgery approach utilizing pericranial flap reconstruction may reduce the risk of postoperative wound complications.
患有药物难治性癫痫的小儿患者可能需要对癫痫发作灶进行调节,以减少癫痫发作的频率和严重程度。这些神经调节手术包括反应性神经刺激器(RNS)和脑深部刺激器(DBS),需要植入引人注目的长期头颅硬件,使患者面临伤口愈合并发症、硬件暴露和感染的风险。利用颅周皮瓣覆盖硬件的多学科神经外科和整形外科方法可减少此类病例的伤口愈合并发症。本研究比较了有无颅周皮瓣覆盖的 RNS/DBS 植入患者的伤口愈合效果。研究人员对一家一级儿科医疗中心在 2014 年至 2022 年期间接受 RNS 和 DBS 植入术(带或不带颅周皮瓣覆盖)的所有患者进行了回顾性病历审查。对 60 天的伤口愈合结果进行了评估。数据比较采用费雪精确检验。27 名患者接受了 29 次神经调节癫痫手术(14 次 DBS,15 次 RNS)。其中 20 例手术包括覆盖硬件的颅周皮瓣。两组患者的中位住院时间均为 2.0 天。两名未进行颅周皮瓣重建的受试者出现了伤口愈合并发症(22.2%),而包含颅周皮瓣的手术中没有出现并发症(P = 0.09)。对于接受植入反应性神经刺激器和脑深部刺激器治疗药物难治性癫痫的儿科患者,利用颅周皮瓣重建的多学科神经外科和整形外科方法可降低术后伤口并发症的风险。
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引用次数: 0
Unveiling a Misdiagnosed Midline Cervical Cleft: A Case Report and Review of the Literature 揭开被误诊的中线颈裂的面纱:病例报告与文献综述
Pub Date : 2024-04-08 DOI: 10.1177/27325016241246223
Nikki Rezania, Elizabeth S. O’Neill, Kelly A. Harmon, Sydney H. Arnold, Christina Tragos
Congenital midline cervical cleft (CMCC) is a rare congenital abnormality of the ventral neck that is often misdiagnosed. We present a case of a 12-year-old male with a history of incomplete CMCC excision and limited neck extension. We highlight the need for increased awareness of this anomaly and propose a clinical algorithm for clinicians when faced with this rare congenital condition. CMCC typically presents at birth and involves a defect along the midline of the neck, characterized by a nipple-like protrusion of the skin and a fibrous cord with an underlying sinus. Micrognathia and mandibular growth restriction are anticipated consequences of CMCC, requiring early intervention. Current treatment guidelines recommend surgical excision prior to age two using a Z-plasty technique. This report emphasizes the rarity and diagnostic challenges of CMCC. It underscores the significance of accurate identification and appropriate surgical planning in a timely manner. Based on the authors’ experience with this case and a review of the literature, we propose a clinical algorithm aimed to enhance CMCC diagnosis and management.
先天性颈中线裂(CMCC)是一种罕见的先天性颈部腹侧畸形,经常被误诊。我们介绍了一例 12 岁男性的病例,他曾有过 CMCC 不完全切除和颈部伸展受限的病史。我们强调需要提高对这种异常的认识,并为临床医生在面对这种罕见的先天性疾病时提出了一种临床算法。CMCC 通常在出生时出现,涉及沿颈部中线的缺损,其特点是皮肤呈乳头状突起,纤维索下有窦道。小颌畸形和下颌骨生长受限是 CMCC 的预期后果,需要及早干预。目前的治疗指南建议在两岁前使用 Z 形成形术进行手术切除。本报告强调了 CMCC 的罕见性和诊断难度。它强调了及时准确识别和制定适当手术计划的重要性。根据作者对该病例的经验和文献综述,我们提出了一种临床算法,旨在加强 CMCC 的诊断和管理。
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引用次数: 0
Humility in the Operating Room: The Essential Virtue of the Evidence-Based Surgeon 手术室中的谦逊:循证外科医生的基本美德
Pub Date : 2024-04-03 DOI: 10.1177/27325016241244530
Robin J. Evans, Kristen E. Hughes
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引用次数: 0
Maxillary and Midface Advancement With Rigid External Distraction: A Review and Technical Recommendations for Optimized Halo Placement 刚性外牵引下的上颌和中面部前移:优化光晕安置的综述和技术建议
Pub Date : 2024-04-03 DOI: 10.1177/27325016241241962
Okensama M. La-Anyane, Nikki Rezania, Kelly A. Harmon, Christina Tragos, John W. Polley, Alvaro A. Figueroa
Background: The use of a rigid external distraction (RED) is recognized as a viable treatment option for patients with severe maxillary and midface hypoplasia. However, there is limited research on the technical placement of the RED device and its impact on patient outcomes. This systematic review examined variations in RED placement as reported in published clinical photos from journal articles, discusses its potential connection to distraction outcomes, and provides suggestions for proper placement of the device. Material and Methods: Articles in which RED was utilized and included clinical photos of patients wearing the RED device were systematically reviewed using PRISMA guidelines. Data extraction and RED placement photo assessment were performed, including subjective analysis of the images and objective angular measurement of device placement. Results: Despite the reporting of satisfactory outcomes in 83% of the reviewed articles, 64% of the photos depicted variations to the recommended halo frame placement. Of these photos, the halo frame was angled inferiorly in 73.8% and 45.16% of those studies depicting inferiorly angled halos reported adverse events. The vertical bar placement was angled posteriorly in 45% of the articles, and 44.4% of these articles reported complications. Conclusion: Photos from the literature show a high variability in RED device placement, with varying outcomes and complication rates. When placed optimally, with consideration to technical placement details such as cranial pin placement and distraction vector angles, the RED system should allow predictable maxillary and midface advancements with minimal complications.
背景:对于严重上颌和中面部发育不良的患者来说,使用硬质外部牵引(RED)被认为是一种可行的治疗方案。然而,关于 RED 装置的技术位置及其对患者治疗效果的影响的研究却很有限。这篇系统性综述研究了期刊文章中发表的临床照片所报告的 RED 置放位置的变化,讨论了其与牵引效果的潜在联系,并为正确放置该装置提供了建议。材料与方法:采用 PRISMA 指南对使用了 RED 并包含佩戴 RED 装置的患者临床照片的文章进行了系统性审查。进行了数据提取和 RED 置放照片评估,包括图像的主观分析和装置置放的客观角度测量。结果:尽管 83% 的综述文章报告了令人满意的结果,但 64% 的照片描述了与推荐的光环框架放置位置不同的情况。在这些照片中,73.8%的照片中的光环框架向下倾斜,在描述光环向下倾斜的研究中,45.16%报告了不良事件。45%的文章将垂直条放置在后方,其中44.4%的文章报告了并发症。结论文献中的照片显示,RED 装置的放置存在很大差异,结果和并发症发生率也各不相同。如果考虑到技术安置细节(如颅针安置和牵引矢量角度),以最佳方式安置,RED 系统应能实现可预测的上颌和中面部前移,并将并发症降至最低。
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引用次数: 0
Modification of Noordhoff Flap for Tubercle Reconstruction in Unilateral Cleft Lip Repair 在单侧唇裂修复术中改良 Noordhoff 皮瓣以重建唇结节
Pub Date : 2024-04-01 DOI: 10.1177/27325016241245049
Joel M. Prince, Erin Poole, Christian El Amm
Background: Tubercle aesthetics are under-emphasized in unilateral cleft lip repair. Teenagers and adults with repaired cleft lip often display a central whistle deformity medial to the cleft lip scar. Lateral segment vermillion elements that are often discarded at the time of the initial repair can be used for tubercle reconstruction. Methods: We obtained anthropometric measurements of the dry vermilion around the central tubercle in neonates with unilateral cleft lip to determine the extent and morphology of deficiencies. A modification of the Noordhoff vermillion flap is proposed with a trapezoidal configuration to precisely match the measured, dry vermillion deficiency. Results: Photographic documentation illustrates the surgical technique and results. Conclusions: Tubercle reconstruction is described using a modification of the Noordhoff flap. Four-year results are encouraging. Observations through maturity are needed.
背景:在单侧唇裂修复中,唇管美学未得到足够重视。唇裂修复后的青少年和成人通常会在唇裂疤痕的内侧出现中央啸叫畸形。初次修复时常被丢弃的侧段朱砂可用于唇小结重建。方法:我们对单侧唇裂新生儿中央结节周围的干朱砂进行了人体测量,以确定缺损的程度和形态。我们提出了诺德霍夫(Noordhoff)朱砂瓣的一种改良方法,其梯形结构可精确匹配测量到的干性朱砂缺损。结果:照片记录说明了手术技巧和结果。结论:介绍了使用诺德霍夫皮瓣的改良方法进行朱唇重建。四年后的结果令人鼓舞。需要对成熟期进行观察。
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引用次数: 0
The CHIP Score: A Valuable Method for Determining Risk of Surgical Intervention for Hemangioma Patients CHIP 评分:确定血管瘤患者手术干预风险的有效方法
Pub Date : 2024-03-28 DOI: 10.1177/27325016241242372
Pearl Shah, Amanda Royek, Anand R. Kumar
Infantile hemangiomas (IHs) are the most common benign tumor of infancy. These tumors may have wide-ranging effects on functional and cosmetic outcomes for patients. With variations in recommendations for surgery among providers, it can be difficult to counsel patients and their families on potential surgical treatment. Through the development of the Case Western Hemangioma Intervention Predictor (CHIP) score, this study aims to improve the consistency of counseling regarding surgery and timing of surgical referral. This was a retrospective review of all patients (179) treated for IHs at a single tertiary care center over 17 years. Patients were divided equally into 2 cohorts. Descriptive statistics and correlation plots were performed on the first cohort to evaluate which disease factors (including size, location, and complications) and patient factors significantly correlated with the decision to pursue surgical treatment (surgical risk). These factors were used to form a CHIP score, which was then validated through logistic regression with length of medical management as a covariate against the second cohort of patients. After controlling for length of medical management to treat IH, lack of medical management, functional impairment, and ulceration were found to be significantly correlated with the risk of surgical intervention ( P < .05). When validated against the second half of our cohort, a CHIP score of 3 (of a maximum score of 3) was found to have a specificity of 92% and a sensitivity of 81% in predicting risk of surgical intervention. IHs can have clinical characteristics that may be predictors of complexity and surgical intervention. The CHIP score can assist in educating patients and families on surgery as a treatment option and guiding appropriate referrals.
婴儿血管瘤(IHs)是婴儿期最常见的良性肿瘤。这些肿瘤可能会对患者的功能和外观造成广泛的影响。由于各医疗机构对手术的建议不尽相同,因此很难就潜在的手术治疗为患者及其家属提供咨询。通过开发凯斯西血管瘤干预预测因子(CHIP)评分,本研究旨在提高手术咨询的一致性和手术转诊的时机。这是一项回顾性研究,研究对象是 17 年来在一家三级医疗中心接受治疗的所有 IH 患者(179 人)。患者被平均分为两组。对第一个队列进行了描述性统计和相关图分析,以评估哪些疾病因素(包括大小、位置和并发症)和患者因素与手术治疗的决定(手术风险)显著相关。这些因素被用来形成 CHIP 评分,然后通过逻辑回归对第二组患者进行验证,并将内科治疗时间作为协变量。在控制了治疗 IH 的医疗管理时间后,发现缺乏医疗管理、功能障碍和溃疡与手术干预风险显著相关(P < .05)。在对我们队列的后半部进行验证时发现,CHIP 评分 3 分(最高分 3 分)在预测手术干预风险方面的特异性为 92%,灵敏度为 81%。IHs 的临床特征可能是复杂性和手术干预的预测因素。CHIP 评分可以帮助患者和家属了解手术作为一种治疗选择,并指导适当的转诊。
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引用次数: 0
Schwannoma of Facial Region: A Pathology Often Missed in the Initial Diagnostic Hypothesis: A Case Report 面部区域的许旺瘤:一种在最初诊断假设中经常被忽略的病理学:病例报告
Pub Date : 2024-03-27 DOI: 10.1177/27325016241242006
Md Kalim Ansari, Akash Ganguly, Tabishur Rahman, Mohd Aswad Khan, G. N, Abhiparna Kundu
Schwannoma, also known as neurilemmoma, is a benign tumor originating from Schwann cells and may involve any nerve in the body but its most common occurrence is in the head and neck region. When it involves a nerve of face, it is often confused with dermoid cyst. We present the case of a 23-year-old male patient with Schwannoma arising from the area just below the lateral canthus of the left eye where the initial diagnostic hypothesis was dermoid cyst but later confirmed as Schwannoma on histopathological analysis.
许旺瘤又称神经瘤,是一种起源于许旺细胞的良性肿瘤,可累及身体的任何神经,但最常见于头颈部。当其累及面部神经时,常与皮样囊肿混淆。本病例是一名 23 岁男性患者,其左眼外侧眼眶下方区域出现许旺瘤,最初的诊断假设是皮样囊肿,但后来经组织病理分析证实为许旺瘤。
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引用次数: 0
Infantile Myofibroma of the Frontal Bone: A Case Report 婴儿额骨肌纤维瘤:病例报告
Pub Date : 2024-03-27 DOI: 10.1177/27325016241241782
K. A. Alawi, Ruqaya Al Abri, S. A. Shaqsi, Jehad Al Habsi, Ahmed Al Azri, Taimoor Al Balushi
Infantile myofibroma stands as the most prevalent fibrous neoplasm within the pediatric population, despite its rarity. The majority of these tumors manifest within the initial 2 years of life. Clinically, myofibroma lesions are classified into solitary and multicentric tumors. We present a case of infantile myofibroma located within the frontal bone of a 16-month-old infant. Initial evaluation of the swelling and its spread was conducted using CT and MRI scans. Subsequently, the diagnosis was confirmed through a core needle biopsy. The tumor underwent surgical excision with 1 cm safety margin and immediate reconstruction with strips of calvaria bone graft. Histopathology of the specimen showed complete tumor clearance with actual margin of 7 mm. The surgical resection of cranial benign bone tumors should be meticulously planned with a multidisciplinary approach. Wherever possible, immediate reconstruction should be conducted to avoid the morbidity of a subsequent procedure.
婴幼儿肌纤维瘤虽然罕见,但却是儿科最常见的纤维性肿瘤。这些肿瘤大多在婴儿出生后的最初两年内发病。临床上,肌纤维瘤可分为单发性和多中心性两种。我们介绍了一例位于16个月大婴儿额骨内的婴儿肌纤维瘤。通过 CT 和 MRI 扫描对肿物及其扩散情况进行了初步评估。随后,通过核心针刺活检确诊。手术切除了肿瘤,留出了 1 厘米的安全边缘,并立即进行了腓骨移植重建。标本的组织病理学检查显示肿瘤完全清除,实际边缘为 7 毫米。颅骨良性骨肿瘤的手术切除应通过多学科方法精心策划。在可能的情况下,应立即进行重建,以避免后续手术的发病率。
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引用次数: 0
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