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新生儿口咽先天性毛发状息肉表现为食管肿物:一例报告及文献复习

Congenital hairy polyp of the oropharynx presenting as an esophageal mass in a neonate, a case report and literature review.

作者信息

Richter Amy, Mysore Krupa, Schady Deb, Chandy Binoy

机构信息

Bobby R. Alford Department of Otolaryngology, Head and Neck Surgery, Texas Children's Hospital, Baylor College of Medicine, Houston, TX, United States.

Department of Pediatric Gastroenterology, Texas Children's Hospital, Baylor College of Medicine, Houston, TX, United States.

出版信息

Int J Pediatr Otorhinolaryngol. 2016 Jan;80:26-9. doi: 10.1016/j.ijporl.2015.11.015. Epub 2015 Nov 24.

Abstract

PURPOSE

To review the literature of congenital hairy polyps and describe the clinical presentation, operative management, and histologic findings of a congenital hairy polyp arising from the palatopharyngeus muscle in a neonate with recurrent choking episodes.

METHODS

Chart review of a 2-month-old male referred to a tertiary care pediatric hospital.

RESULTS

We present a case of a 2-month-old male who presented to the emergency room with recurrent episodes of choking and vomiting. The patient was previously healthy with no prior medical or neonatal history. The parents noted a small fleshy mass in the patient's oropharynx that he would chew on and swallow after several minutes. However, on physical exam, there was no evidence of oropharyngeal mass. The patient did not have respiratory distress. Imaging revealed a 22×7×11mm oblong, fatty mass in the lower cervical and upper thoracic esophagus with a thin stalk extending proximally to the upper collapsed esophagus. Intraoperative recorded laryngoscopy revealed a pedunculated soft palate mass attached to the right superior palatopharyngeus muscle. Histopathology revealed ectodermal and mesodermal elements in a polypoid structure lined by keratinizing squamous epithelium with adnexal structures and central mature adipose tissue, consistent with congenital hairy polyp resembling an accessory tragus of the ear and branchial anomaly. At 6-week follow up, the patient was doing well and gaining weight appropriately with no further choking episodes. There was no evidence of velopharyngeal dysfunction on follow up exam. The surgical site was completely healed and there was no evidence of recurrence.

DISCUSSION

Congenital hairy polyps of the naso- and oropharynx are rare but may present as airway or esophageal masses, causing respiratory distress or choking episodes in a pediatric patient. The pathologic findings of keratinizing squamous epithelium, adnexal structures, adipose and cartilage tissues resemble congenital accessory tragus and may be considered a branchial arch anomaly.

摘要

目的

回顾先天性毛发息肉的文献,并描述一名患有反复窒息发作的新生儿腭咽肌先天性毛发息肉的临床表现、手术治疗及组织学发现。

方法

对转诊至三级儿科医院的一名2个月大男性患儿的病历进行回顾。

结果

我们报告一例2个月大男性患儿,因反复出现窒息和呕吐发作就诊于急诊室。该患儿此前健康,无既往病史或新生儿病史。家长注意到患儿口咽有一个小肉质肿物,他会咀嚼几分钟后吞咽。然而,体格检查未发现口咽肿物。患儿无呼吸窘迫。影像学检查显示在下颈部和上胸部食管有一个22×7×11mm的椭圆形脂肪性肿物,有一细蒂向近端延伸至塌陷的上段食管。术中喉镜检查显示一个带蒂的软腭肿物附着于右侧腭咽肌上部。组织病理学显示息肉样结构中有外胚层和中胚层成分,内衬角化鳞状上皮,有附属结构和中央成熟脂肪组织,符合先天性毛发息肉,类似于耳副耳屏和鳃裂畸形。在6周随访时,患儿情况良好,体重增长正常且无进一步窒息发作。随访检查未发现腭咽功能不全的证据。手术部位完全愈合,无复发迹象。

讨论

鼻咽和口咽的先天性毛发息肉罕见,但可能表现为气道或食管肿物,导致儿科患者呼吸窘迫或窒息发作。角化鳞状上皮、附属结构、脂肪和软骨组织的病理表现类似于先天性副耳屏,可被认为是鳃弓畸形。

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