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Expect the unexpected: A rare case of bilateral lymphoepithelial carcinoma of the parotid gland 意料之外:一个罕见的双侧腮腺淋巴上皮癌病例
IF 0.3 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-12-11 DOI: 10.1016/j.xocr.2025.100718
Umair Hafeez , Niall James McInerney , Sri Ganesh Kalimuthu , Olena Tkachuk
Lymphoepithelial carcinoma (LEC) of the salivary glands is a rare and aggressive malignancy strongly associated with Epstein–Barr virus (EBV) infection and more frequently reported in endemic regions such as Greenland and Southeast Asia. Bilateral involvement is exceptionally uncommon, particularly in non-endemic populations. We report the case of a 62-year-old male with bilateral, asymptomatic parotid swellings persisting for five years. Initial clinical assessment suggested a benign etiology, with no pain, facial nerve involvement, or other red flag symptoms. However, ultrasound, fine needle aspiration, and contrast-enhanced computed tomography of the left parotid revealed atypical features suspicious for malignancy, and histopathology confirmed LEC. The patient underwent left superficial parotidectomy with clear margins, followed by right superficial parotidectomy, which also confirmed LEC. This case represents a rare presentation of bilateral LEC in a non-endemic region. The indolent course and absence of classic malignant features highlight the diagnostic challenges of this entity. Early multidisciplinary evaluation is essential for persistent or atypical parotid swellings, and further research is warranted into the role of EBV in tumorigenesis outside endemic areas.
唾液腺淋巴上皮癌(LEC)是一种罕见的侵袭性恶性肿瘤,与eb病毒(EBV)感染密切相关,在格陵兰岛和东南亚等流行地区更为常见。双边参与极为罕见,特别是在非流行人群中。我们报告的情况下,62岁的男性与双侧,无症状腮腺肿胀持续五年。初步临床评估提示为良性病因,无疼痛、面神经受累或其他危险症状。然而,超声、细针穿刺和增强ct显示左腮腺不典型特征,怀疑为恶性肿瘤,组织病理学证实LEC。患者行左侧腮腺浅表切除术,边缘清晰,随后行右侧腮腺浅表切除术,也证实LEC。本病例是非流行地区罕见的双侧LEC。惰性过程和缺乏典型的恶性特征突出了这个实体的诊断挑战。早期多学科评估对于持续或非典型腮腺肿胀至关重要,进一步研究eb病毒在流行地区以外的肿瘤发生中的作用是必要的。
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引用次数: 0
Spontaneous catastrophic cervical hemorrhage in the setting of neurofibromatosis 神经纤维瘤病并发自发性灾难性宫颈出血
IF 0.3 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-12-04 DOI: 10.1016/j.xocr.2025.100720
Lavanya Nagappan , Yekaterina Shapiro , Luke Stanisce , Donald Solomon
Neurofibromatosis type 1 (NF1) can manifest in the head and neck in a variety of ways, most commonly presenting as solitary neurofibromas. The management of these lesions require multifaceted consideration of neurovascular involvement and airway compromise. Herein, we describe a 58-year-old male with a history of NF1 and extensive cervical neurofibromas who developed a spontaneous, expanding neck hematoma secondary to his tumors. He was initially stabilized with surgical drainage and endovascular embolization, but then subsequently represented with a second rapidly expanding neck hematoma resulting in acute respiratory distress. Failed intubation due to airway edema necessitated emergent cricothyrotomy and cardiopulmonary resuscitation. Additional embolization was performed but the patient eventually succumbed to global anoxic brain injury.
This case highlights the need for proactive multi-disciplinary management with specific attention to respiratory and hemodynamic stabilization in NF1 patients with large tumor burden. As with all neck hematomas, rapid respiratory decompensation may occur, requiring prompt recognition and early intervention. This case underscores the significance of a multidisciplinary approach involving emergency medicine, critical care, anesthesia, interventional radiology, and otolaryngology to recognize and address the potential for airway compromise in the face of a neck hematoma in patients with NF1.
1型神经纤维瘤病(NF1)可以在头颈部以多种方式表现,最常见的表现为孤立的神经纤维瘤。这些病变的处理需要多方面考虑神经血管受累和气道损害。在此,我们描述了一位58岁的男性,有NF1和广泛的颈部神经纤维瘤的病史,他的肿瘤发展为自发性的,扩大的颈部血肿。他最初通过手术引流和血管内栓塞稳定,但随后出现第二次迅速扩大的颈部血肿,导致急性呼吸窘迫。由于气道水肿插管失败,需要紧急环甲环切开术和心肺复苏。虽然进行了额外的栓塞治疗,但患者最终死于全身性缺氧脑损伤。本病例强调需要积极主动的多学科管理,特别关注具有大肿瘤负担的NF1患者的呼吸和血流动力学稳定。与所有颈部血肿一样,可能发生快速呼吸失代偿,需要及时识别和早期干预。该病例强调了多学科方法的重要性,包括急诊医学、重症监护、麻醉、介入放射学和耳鼻喉科,以识别和解决NF1患者面对颈部血肿时气道损害的可能性。
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引用次数: 0
Neglected middle ear foreign body with unusual eustachian tube migration; case report 中耳异物伴咽鼓管异常移位;病例报告
IF 0.3 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-11-29 DOI: 10.1016/j.xocr.2025.100717
Abdullah Almutairi , Ghadah Alotaibi , Alwaleed Alqahtani , Mohammed Almutairi , Essa Bakry
Foreign bodies in the ear, nose, and throat are common presentations that may carry a significant risk of complications. Middle ear foreign bodies are less frequently encountered and are most often associated with direct tympanic membrane perforation or iatrogenic causes, such as displaced tympanostomy tubes. In this report, we describe a case of a long-standing graphite pencil tip found within the middle ear, with a possible migration route through the Eustachian tube. This case highlights an unusual mechanism of entry and underscores the importance of careful evaluation in patients with chronic otologic symptoms.
耳、鼻、喉异物是常见的表现,可能会带来严重的并发症风险。中耳异物较少见,通常与直接的鼓膜穿孔或医源性原因有关,如鼓膜造瘘管移位。在本报告中,我们描述了一个在中耳内发现的长期存在的石墨铅笔尖端,其可能通过耳咽管迁移。本病例强调了一种不寻常的进入机制,并强调了对慢性耳科症状患者进行仔细评估的重要性。
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引用次数: 0
Incidental parapharyngeal ganglioneuroma: A case report on surgical management 意外性咽旁神经节神经瘤:手术治疗1例报告
IF 0.3 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-11-19 DOI: 10.1016/j.xocr.2025.100711
Michael G. Hahn , Courtney J. Hunter , André M. Wineland

Introduction

Parapharyngeal ganglioneuromas are exceedingly rare, benign tumors of the sympathetic nervous system, with fewer than 40 reported cases in this specific anatomical space over four decades. They frequently remain asymptomatic until they reach a significant size, an their rarity contributes to a limited understanding of optimal management strategies.

Case presentation

In this case report, a 16-year-old female presented with an incidental finding of a 5.2 × 3.9 × 2.0 cm right parapharyngeal space mass on a CT head. Further diagnostic imaging included a contrast-enhanced CT neck and an MRI neck. Flexible fiberoptic laryngoscopy was performed to confirm mass effect of the right lateral pharyngeal wall. Surgical excisional biopsy via an endoscope-assisted transoral approach resulted in subtotal resection due to proximity to the internal carotid artery. Pathology confirmed a benign mature ganglioneuroma (5.0 cm), positive for S100. She has been monitored with serial MRI imaging with evidence of known residual lesion and surrounding fibrosis.

Conclusion

This case highlights the rare presentation and diagnostic challenges of parapharyngeal ganglioneuromas. Achieving complete surgical resection can be complex due to tumor adherence and proximity to vital structures. Comprehensive diagnostic workup, surgical excision, and regular surveillance remain crucial for optimizing outcomes in these benign yet challenging tumors.
咽旁神经节神经瘤是一种极为罕见的交感神经系统良性肿瘤,在过去的四十年中,在这一特殊解剖空间中报道的病例不到40例。它们通常没有症状,直到它们达到相当大的尺寸,它们的罕见性有助于对最佳管理策略的有限理解。本病例报告中,一名16岁的女性在CT头部上偶然发现一个5.2 × 3.9 × 2.0 cm的右侧咽旁间隙肿块。进一步的诊断成像包括对比增强CT颈部和MRI颈部。软性纤维喉镜检查证实右侧咽外壁肿块效应。手术切除活检通过内窥镜辅助经口入路导致近全切除由于接近颈内动脉。病理证实为良性成熟神经节神经瘤(5.0 cm), S100阳性。对患者进行了连续MRI检查,发现有已知的残余病变和周围纤维化。结论本病例强调咽旁神经节瘤的罕见表现和诊断挑战。由于肿瘤粘附性和靠近重要结构,实现完全手术切除可能是复杂的。全面的诊断检查、手术切除和定期监测对于优化这些良性但具有挑战性的肿瘤的预后仍然至关重要。
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引用次数: 0
Speech outcomes following buccal fat pad interposition in palatal repair: A clinical outcome analysis 口腔脂肪垫介入腭修复术后的语言预后:一项临床结果分析
IF 0.3 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-11-19 DOI: 10.1016/j.xocr.2025.100716
Manu Gupta , Sanjeev Kumar , S. Manu Prasad , Partishtha Sharma , Harish Kumar Agarwala , Vivek Kumar Jha

Background

Cleft palate repair aims not only to achieve anatomical closure but also to restore normal velopharyngeal (VP) function essential for intelligible speech. Despite technical refinements, complications such as fistula formation, wound dehiscence, and persistent velopharyngeal insufficiency (VPI) continue to affect surgical success and speech outcomes. The buccal fat pad (BFP) has emerged as a reliable, well-vascularized graft for intraoral reconstruction, offering potential benefits in enhancing healing and speech function.

Aim

To evaluate the effect of BFP interposition during Bardach two-flap palatoplasty on surgical healing, speech outcomes, and velopharyngeal competence in paediatric cleft palate patients.

Materials and methods

A prospective case series of 26 patients (17 males, 9 females; mean age 14 ± 2 months) with complete cleft palate underwent Bardach two-flap palatoplasty with BFP interposition between April 2022 and January 2024. Speech evaluations were performed preoperatively and at 3, 6, and 12 months postoperatively using age-appropriate perceptual assessments focusing on resonance, articulation, intelligibility, and VP competence. Postoperative complications, including fistula and donor site morbidity, were recorded. Data were analysed using SPSS v22, with significance set at p < 0.05.

Results

BFP provided a consistent, well-vascularized interpositional layer, resulting in satisfactory wound healing. Oronasal fistula occurred in 8 % and wound dehiscence in 4 % of cases. Donor site morbidity was minimal (4 %) and resolved spontaneously. Hypernasality decreased from 76 % preoperatively to 12 % at 12 months, nasal air emission from 64 % to 10 %, and articulation errors from 70 % to 14 %, while speech intelligibility improved from 42 % to 88 % (p < 0.05). Adequate VP closure increased from 23 % preoperatively to 88 % postoperatively.

Conclusion

BFP interposition during Bardach two-flap palatoplasty is a safe and effective adjunct for cleft palate repair. It enhances tissue healing, minimizes fistula formation, and significantly improves postoperative speech outcomes and velopharyngeal competence. Given its simplicity, minimal morbidity, and functional advantages, BFP-assisted palatoplasty represents a valuable technique in optimizing both structural and speech outcomes in cleft palate surgery.
背景腭裂修复的目的不仅是实现解剖上的闭合,而且是恢复正常的腭咽(VP)功能,这对清晰的言语至关重要。尽管技术上有所改进,但瘘形成、伤口裂开和持续性腭咽功能不全(VPI)等并发症继续影响手术成功和言语预后。口腔脂肪垫(BFP)已成为一种可靠的、血管化良好的口腔内重建移植物,在增强愈合和语言功能方面具有潜在的好处。目的探讨Bardach双瓣腭成形术中BFP介入对小儿腭裂患者手术愈合、语言预后和腭咽功能的影响。材料与方法对2022年4月至2024年1月26例完全性腭裂患者(男17例,女9例,平均年龄14±2个月)行BFP介入Bardach双瓣腭裂成形术进行前瞻性研究。术前、术后3个月、6个月和12个月进行语言评估,使用与年龄相适应的感知评估,重点是共振、发音、可理解性和VP能力。记录术后并发症,包括瘘和供区发病率。数据采用SPSS v22进行分析,p <; 0.05为显著性。结果bfp提供了一致的、血管化良好的间置层,创面愈合满意。8%的病例发生口鼻瘘,4%的病例发生伤口裂开。供体部位的发病率极低(4%)并自行消退。鼻音过高从术前的76%下降到12个月时的12%,鼻气排出率从64%下降到10%,发音错误从70%下降到14%,而语音清晰度从42%提高到88% (p < 0.05)。足够的VP闭合从术前的23%增加到术后的88%。结论Bardach双瓣腭成形术中bfp介入是一种安全有效的腭裂修复辅助手段。它促进组织愈合,最大限度地减少瘘形成,并显著改善术后语言预后和腭咽能力。由于其简单,低发病率和功能优势,bfp辅助腭成形术在优化腭裂手术的结构和语言结果方面是一种有价值的技术。
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引用次数: 0
Fibromatosis colli: A rare neck mass of the sternocleidomastoid muscle in an infant: A case report 结缔组织瘤病:婴儿胸锁乳突肌罕见颈部肿块1例报告
IF 0.3 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-11-17 DOI: 10.1016/j.xocr.2025.100714
Tesfa G/Meskel , Mesfin Wubishet , Nuri Mama , Ayalneh Demissie

Background

Fibromatosis colli (FC), also known as sternocleidomastoid pseudotumor of infancy, is a rare benign proliferation of fibrous tissue within the sternocleidomastoid muscle. It typically presents as a unilateral neck mass during the first weeks of life. Although self-limiting, early recognition is essential to prevent unnecessary investigations and to reduce the risk of complications such as congenital torticollis.

Case presentation

We report the case of a 3-week-old female neonate who presented with a painless, left-sided neck swelling of one week’s duration. Clinical examination revealed a firm, non-tender mass along the sternocleidomastoid muscle. Ultrasonography confirmed the diagnosis of Fibromatosis colli. The patient was managed conservatively with physiotherapy, resulting in near-complete resolution of the mass and restoration of normal neck mobility within three months.

Conclusion

Fibromatosis colli is an uncommon but clinically significant cause of neck swelling in neonates and may closely mimic neoplastic lesions. It should be considered in the differential diagnosis of unilateral neck masses in early infancy. Accurate diagnosis is typically achieved through targeted clinical examination complemented by ultrasonography. Early initiation of physiotherapy is highly effective, typically resulting in complete recovery without the need for surgical intervention. Timely recognition and differentiation from other neonatal neck masses are crucial to avoid unnecessary invasive procedures, alleviate parental anxiety, and prevent complications such as persistent torticollis and craniofacial asymmetry.
背景:乳突纤维瘤病(FC),也称为婴儿期胸锁乳突假瘤,是一种罕见的胸锁乳突肌内纤维组织的良性增生。在生命的最初几周,它通常表现为单侧颈部肿块。虽然有局限性,但早期识别对于防止不必要的调查和减少并发症(如先天性斜颈)的风险至关重要。病例介绍我们报告的情况下,3周大的女性新生儿谁提出了无痛,左侧颈部肿胀的一个星期的时间。临床检查发现沿胸锁乳突肌有一坚固无压痛肿块。超声检查证实为大肠纤维瘤病。患者接受了保守的物理治疗,三个月内肿块几乎完全消退,颈部活动恢复正常。结论大肠纤维瘤病是一种罕见但临床意义重大的新生儿颈部肿胀病因,与肿瘤病变相似。在婴儿期早期单侧颈部肿块的鉴别诊断中应予以考虑。准确的诊断通常通过有针对性的临床检查和超声检查来实现。早期开始物理治疗是非常有效的,通常导致完全恢复而不需要手术干预。及时识别和区分其他新生儿颈部肿块对于避免不必要的侵入性手术,减轻父母的焦虑,防止并发症如持续性斜颈和颅面不对称是至关重要的。
{"title":"Fibromatosis colli: A rare neck mass of the sternocleidomastoid muscle in an infant: A case report","authors":"Tesfa G/Meskel ,&nbsp;Mesfin Wubishet ,&nbsp;Nuri Mama ,&nbsp;Ayalneh Demissie","doi":"10.1016/j.xocr.2025.100714","DOIUrl":"10.1016/j.xocr.2025.100714","url":null,"abstract":"<div><h3>Background</h3><div>Fibromatosis colli (FC), also known as sternocleidomastoid pseudotumor of infancy, is a rare benign proliferation of fibrous tissue within the sternocleidomastoid muscle. It typically presents as a unilateral neck mass during the first weeks of life. Although self-limiting, early recognition is essential to prevent unnecessary investigations and to reduce the risk of complications such as congenital torticollis.</div></div><div><h3>Case presentation</h3><div>We report the case of a 3-week-old female neonate who presented with a painless, left-sided neck swelling of one week’s duration. Clinical examination revealed a firm, non-tender mass along the sternocleidomastoid muscle. Ultrasonography confirmed the diagnosis of Fibromatosis colli. The patient was managed conservatively with physiotherapy, resulting in near-complete resolution of the mass and restoration of normal neck mobility within three months.</div></div><div><h3>Conclusion</h3><div>Fibromatosis colli is an uncommon but clinically significant cause of neck swelling in neonates and may closely mimic neoplastic lesions. It should be considered in the differential diagnosis of unilateral neck masses in early infancy. Accurate diagnosis is typically achieved through targeted clinical examination complemented by ultrasonography. Early initiation of physiotherapy is highly effective, typically resulting in complete recovery without the need for surgical intervention. Timely recognition and differentiation from other neonatal neck masses are crucial to avoid unnecessary invasive procedures, alleviate parental anxiety, and prevent complications such as persistent torticollis and craniofacial asymmetry.</div></div>","PeriodicalId":37154,"journal":{"name":"Otolaryngology Case Reports","volume":"38 ","pages":"Article 100714"},"PeriodicalIF":0.3,"publicationDate":"2025-11-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145570586","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
A rare case of isolated frontal sinus fungus ball 罕见孤立额窦真菌球1例
IF 0.3 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-11-14 DOI: 10.1016/j.xocr.2025.100715
Shin Ito , Satoko Kubo , Ryuzaburo Higo , Akane Hashizume , Fumihiko Matsumoto
Fungal sinusitis is a common otolaryngological disease; however, most cases involve the maxillary sinus, and cases of isolated fungus balls (FBs) in the frontal sinus are extremely rare. Herein, we describe the management of an isolated frontal sinus FB in a patient without underlying immunodeficiency. A 58-year-old man presented with a 1-month history of pain in his left forehead. Sinus computed tomography (CT) showed soft-tissue shadows with calcification in the left frontal sinus, and magnetic resonance imaging showed low signal intensity on T2-weighted images at the site of calcification on CT, suggesting the presence of an FB. The patient had a relatively severe and unique anatomical condition that appeared challenging to manage. The endoscopic modified Lothrop procedure (EMLP) was selected, and surgery was performed 3 months after the initial visit. An inside-out approach was used to reach the frontal sinus and unify the bilateral frontal sinuses into a single cavity. A powered irrigation system was used to irrigate and completely remove the entire fungal mass. The pathological findings showed clusters of Aspergillus. In summary, the EMLP was successfully performed, and the FB was completely removed without any complications in this case. The patient remains recurrence-free to date, and the wound site is easily observable via a fiberscope.
真菌性鼻窦炎是一种常见的耳鼻喉疾病;然而,大多数病例涉及上颌窦,额窦孤立真菌球(FBs)的病例极为罕见。在此,我们描述了一个孤立的额窦FB患者的管理没有潜在的免疫缺陷。男,58岁,左前额疼痛1个月。鼻窦CT示左侧额窦软组织影伴钙化,磁共振CT示钙化部位t2加权低信号,提示FB存在。患者有一个相对严重和独特的解剖条件,似乎具有挑战性的管理。选择内镜下改良Lothrop手术(EMLP),并在初次就诊3个月后进行手术。采用由内向外入路到达额窦,并将双侧额窦统一为一个腔。使用动力灌溉系统冲洗并完全去除整个真菌团块。病理表现为曲霉聚集。总之,EMLP手术成功,FB完全切除,无任何并发症。患者至今无复发,伤口部位通过纤维镜很容易观察到。
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引用次数: 0
A 4-year-old girl with an unrepaired cleft palate: Outcomes of late intervention 1例4岁女童腭裂未修复:晚期干预的结果
IF 0.3 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-11-14 DOI: 10.1016/j.xocr.2025.100713
Mahdiye Tavakoli , Fariba Jahangiri , Gholamreza Bayazian , Nahid Jalilevand , Reyhane Mohamadi
Cleft lip and palate(CL/P) are among the most common orofacial birth defects, often associated with feeding, hearing,communication, and speech problems. A multidisciplinary approach is required for optimal management.
This report describes a 4-year-old girl with an unrepaired cleft palate who presented with multiple articulation errors, reduced speech intelligibility, and low intraoral pressure. Following surgical repair of the palate, she received intensive online speech therapy. Speech assessment demonstrated a significant improvement. Compensatory articulation errors decreased progressively and reached zero at follow-up. Based on perceptual assessment, hypernasality was significantly reduced after the cleft palate surgery.
唇腭裂(CL/P)是最常见的口腔面部出生缺陷之一,通常与喂养,听力,沟通和语言问题有关。最佳管理需要多学科方法。本报告描述了一名四岁女孩腭裂未修复,其表现为多重发音错误,言语清晰度降低和低口内压。在腭裂修复手术后,她接受了密集的在线语言治疗。语音评估显示有显著改善。补偿性发音错误逐渐减少,随访时达到零。根据知觉评估,腭裂手术后鼻音异常明显减少。
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引用次数: 0
A rare case report of sinonasal mucosal melanoma in the maxillary sinus 上颌窦鼻窦黏膜黑色素瘤1例
IF 0.3 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-11-13 DOI: 10.1016/j.xocr.2025.100712
Molham Alahmad , Venus Hussain Ameen , Mohammad Jomaa , Abdulmajeed Yousfan

Introduction

Sinonasal mucosal melanoma (SNMM) is an aggressive subtype of melanoma that represents <5 % of head and neck melanomas and constitutes less than 1 % of systemic melanoma cases.

Case presentation

A 54-year-old female presented with a 5-month history of unilateral recurrent epistaxis, anosmia, and nasal obstruction. Nasal endoscopy showed a bleeding fleshy lesion occupying the right nasal cavity, extending to the nasal floor and nasal septum. Pathological examination and immunohistochemistry (IHC) findings confirmed the diagnosis of SNMM.

Conclusion

SNMM in the head and neck region can manifest with nonspecific symptoms. The diagnosis often requires a combination of clinical, pathological, and IHC studies.
鼻窦粘膜黑色素瘤(SNMM)是一种侵袭性黑色素瘤亚型,占头颈部黑色素瘤的5%,占全身性黑色素瘤病例的不到1%。病例介绍:一名54岁女性,有5个月的单侧复发性鼻出血、嗅觉丧失和鼻塞病史。鼻内窥镜检查显示右鼻腔有出血肉质病变,并延伸至鼻底及鼻中隔。病理检查和免疫组化(IHC)结果证实了SNMM的诊断。结论头颈部snmm可表现为非特异性症状。诊断通常需要结合临床、病理和免疫组化研究。
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引用次数: 0
An unusual laryngeal mass: primary tumoral calcinosis of the larynx in a child 一个不寻常的喉部肿块:儿童喉部原发性肿瘤钙质沉着症
IF 0.3 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-11-01 DOI: 10.1016/j.xocr.2025.100710
Miles J. Klimara , Eirene Alexandrou , Anand Kanagasabapathy , Matthew R. Hoffman

Background

Tumoral calcinosis (TC) is a rare, benign calcium hydroxyapatite deposition typically affecting periarticular tissue of large joints, which may occur sporadically or in association with hyperphosphatemia. Laryngeal TC has been reported in several patients, uniformly in the fifth and sixth decade of life. Presented is a case of sporadic combined intra-laryngeal and extra-laryngeal TC in a child.

Case discussion

A 10-year-old female presented with a right neck mass and stridor with activity. Flexible laryngoscopy revealed fullness of the right false vocal fold without intraluminal mass, and computed tomography showed a large, calcified, intra- and extra-laryngeal mass passing through the thyrohyoid space. Open resection was performed. Surgical pathology revealed TC. Laboratory and endocrine evaluation did not reveal any underlying metabolic etiology, supporting a diagnosis of primary laryngeal TC. Airway management, approach to resection, and published cases of laryngeal TC are reviewed.

Conclusion

TC of the larynx is an exceedingly rare entity. Given benign nature of this disease process, a conservative approach is favored when resection is pursued, with preservation of normal structures in order to minimize morbidity. Upon diagnosis, multidisciplinary evaluation should be pursued to rule out underlying endocrine, metabolic, and genetic disorders.
肿瘤性钙质沉着症(TC)是一种罕见的良性羟基磷灰石钙沉积,通常影响大关节的关节周围组织,可能偶尔发生或与高磷血症有关。喉部TC在一些患者中有报道,一致发生在生命的第五和第六十年。本文报告一例儿童散发性合并喉内喉外TC。病例讨论一名10岁女性,表现为右颈部肿块和活动性喘鸣。软性喉镜检查显示右侧假声带丰满,无腔内肿块,计算机断层扫描显示一个大的,钙化的喉内和喉外肿块穿过甲状腺舌骨间隙。行切开切除。手术病理显示为TC。实验室和内分泌评估未发现任何潜在的代谢病因,支持原发性喉部TC的诊断。气道管理,入路切除,并发表的病例喉TC回顾。结论喉部tc是一种极为罕见的疾病。考虑到本病过程的良性性质,在进行切除时,保守的方法是可取的,同时保留正常的结构,以尽量减少发病率。诊断后,应进行多学科评估,以排除潜在的内分泌、代谢和遗传疾病。
{"title":"An unusual laryngeal mass: primary tumoral calcinosis of the larynx in a child","authors":"Miles J. Klimara ,&nbsp;Eirene Alexandrou ,&nbsp;Anand Kanagasabapathy ,&nbsp;Matthew R. Hoffman","doi":"10.1016/j.xocr.2025.100710","DOIUrl":"10.1016/j.xocr.2025.100710","url":null,"abstract":"<div><h3>Background</h3><div>Tumoral calcinosis (TC) is a rare, benign calcium hydroxyapatite deposition typically affecting periarticular tissue of large joints, which may occur sporadically or in association with hyperphosphatemia. Laryngeal TC has been reported in several patients, uniformly in the fifth and sixth decade of life. Presented is a case of sporadic combined intra-laryngeal and extra-laryngeal TC in a child.</div></div><div><h3>Case discussion</h3><div>A 10-year-old female presented with a right neck mass and stridor with activity. Flexible laryngoscopy revealed fullness of the right false vocal fold without intraluminal mass, and computed tomography showed a large, calcified, intra- and extra-laryngeal mass passing through the thyrohyoid space. Open resection was performed. Surgical pathology revealed TC. Laboratory and endocrine evaluation did not reveal any underlying metabolic etiology, supporting a diagnosis of primary laryngeal TC. Airway management, approach to resection, and published cases of laryngeal TC are reviewed.</div></div><div><h3>Conclusion</h3><div>TC of the larynx is an exceedingly rare entity. Given benign nature of this disease process, a conservative approach is favored when resection is pursued, with preservation of normal structures in order to minimize morbidity. Upon diagnosis, multidisciplinary evaluation should be pursued to rule out underlying endocrine, metabolic, and genetic disorders.</div></div>","PeriodicalId":37154,"journal":{"name":"Otolaryngology Case Reports","volume":"37 ","pages":"Article 100710"},"PeriodicalIF":0.3,"publicationDate":"2025-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145519598","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
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Otolaryngology Case Reports
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