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Bilateral Choanal Atresia in a 65-Year-Old Female: A Case Report and Literature Review. 65岁女性双侧后肛门闭锁1例报告并文献复习。
IF 0.4 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-11-05 eCollection Date: 2025-01-01 DOI: 10.1155/crot/5584900
Maraam Al Qout, Abdullah Alkarni, Abdulaziz Alaraifi, Mohammad Almahdi

Background: Choanal atresia (CA) is a congenital condition characterized by occlusion of the nasal airway due to failure of recanalization during embryological development. It is more commonly unilateral and typically presents during infancy. In contrast, bilateral CA is a neonatal emergency that often manifests as respiratory distress at birth. The presentation of bilateral CA in late adulthood is extremely rare.

Case description: This case presents an unusual case of an elderly patient diagnosed with bilateral CA at 65 years. She presented with a primary complaint of lifelong bilateral nasal obstruction and denied any symptoms suggestive of chronic rhinosinusitis. Examination revealed bilateral choanal obstruction with no visible openings in the nasal cavity. The patient underwent endoscopic transseptal repair of bilateral CA, which included perforation of the atretic plate, posterior septectomy, and flap reconstruction. Postoperative evaluation demonstrated bilaterally patent choanae, and the patient remained asymptomatic, with no further complications.

Conclusion: This case highlights a rare presentation of bilateral CA diagnosed in late adulthood. Although typically detected in the neonatal period, bilateral CA can occasionally remain undiagnosed for decades. This patient represents the oldest reported case of bilateral CA in the literature, emphasizing the variability in clinical presentation and the potential for delayed diagnosis.

背景:后肛门闭锁(CA)是一种先天性疾病,其特征是在胚胎发育期间由于再通失败而导致鼻气道闭塞。它更常见的单侧和典型表现在婴儿期。相反,双侧CA是一种新生儿急症,通常表现为出生时呼吸窘迫。在成年后期出现双侧CA是非常罕见的。病例描述:本病例是一位罕见的65岁老年患者,诊断为双侧CA。她以终身双侧鼻塞为主诉,否认有任何提示慢性鼻窦炎的症状。检查发现双侧后鼻孔梗阻,鼻腔内未见开口。患者行内镜下双侧CA经间隔修复术,包括锁骨板穿孔、后隔切除术和皮瓣重建。术后评估显示双侧choanae未闭,患者无症状,无进一步并发症。结论:本病例为罕见的双侧CA,诊断于成年晚期。虽然通常在新生儿期发现,但双侧CA偶尔会在几十年内未被诊断出来。该患者是文献中最早报道的双侧CA病例,强调临床表现的可变性和延迟诊断的可能性。
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引用次数: 0
Pediatric Parapharyngeal Space Mass: Successful Outcome Following Transoral Simple Cyst Resection. 小儿咽旁间隙肿块:经口单纯囊肿切除术后的成功结果。
IF 0.4 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-10-29 eCollection Date: 2025-01-01 DOI: 10.1155/crot/9934709
Christian M Kabongo, Alexander P Marston

Background: Parapharyngeal space masses are rare lesions identified in pediatric patients. The present case describes the clinical history and surgical treatment of a parapharyngeal space in a 32-month-old female.

Methods: The electronic medical record was used for this retrospective case report.

Results: A 32-month-old female had a nonsignificant clinical presentation and a negative flexible laryngoscopy. Upon imaging, a mass was seen in the left parapharyngeal space. Resection of the mass was conducted. The final pathology confirmed a benign simple cyst. The patient did not have any postoperative complications.

Conclusions: This case illustrates that surgical resection can be an adequate treatment method for a parapharyngeal space mass. It also adds to the literature on types of parapharyngeal space masses that may present in pediatric patients.

背景:咽旁间隙肿块是儿科患者中罕见的病变。本病例描述了32个月大的女性咽旁间隙的临床病史和手术治疗。方法:采用电子病历进行回顾性病例报告。结果:一名32个月大的女性,临床表现不明显,柔韧性喉镜检查阴性。影像学显示左侧咽旁间隙有肿块。切除肿块。最终病理证实为良性单纯性囊肿。患者无术后并发症。结论:本病例提示手术切除是咽旁间隙肿物的有效治疗方法。它也增加了文献类型咽旁间隙肿块可能存在于儿科患者。
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引用次数: 0
Diagnostic Pitfalls and Unique Radiological Insights in Thyroid Paraganglioma: A Case Report and Literature Review. 甲状腺副神经节瘤的诊断缺陷和独特的放射学见解:1例报告和文献复习。
IF 0.4 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-10-25 eCollection Date: 2025-01-01 DOI: 10.1155/crot/5395659
Ainulakbar Mughal, Fatima Syed Amanullah, Zubia Ali, Shabbir Akhtar, Sehar Suleman

Paragangliomas (PGLs) are extremely rare endocrine tumors that arise from the autonomic nervous system. Their rarity contributes to their frequent misdiagnosis. Ultrasound and immunohistochemical findings are heavily relied on for correct diagnosis. We present a case of thyroid PGL in a 40-year-old female patient who presented with a 1-year history of anterior neck mass and a 4-month history of hemoptysis. Ultrasound findings showed a solitary, lobulated, hypoechoic, and vascular lesion in the right thyroid lobe. She underwent total thyroidectomy and tracheal end-to-end anastomosis. Histopathology showed cells organized in distinct nests (zellballen) pattern enclosed by a delicate fibrovascular stroma. On immunohistochemical analysis, the tumor was positive for synaptophysin, CD56, GATA-3, and S100. The patient is stable postoperatively and disease-free. The role of ultrasound is vital in forming a correct diagnosis of thyroid PGL preoperatively. We review the current literature regarding diagnostic findings and treatment of thyroid PGLs with the aim of supplementing the findings of the thyroid PGLs that have been previously reported. The creation of a robust imaging and immunohistochemical profile for this entity is needed to combat the frequent misdiagnoses that occur with thyroid PGLs.

副神经节瘤(PGLs)是一种极为罕见的内分泌肿瘤,起源于自主神经系统。它们的罕见性导致它们经常被误诊。超声和免疫组织化学检查结果是正确诊断的重要依据。我们报告一个40岁的女性甲状腺PGL病例,她有1年的前颈部肿块史和4个月的咯血史。超声显示右侧甲状腺叶单发、分叶状、低回声及血管性病变。行甲状腺全切除术及气管端对端吻合。组织病理学显示细胞呈独特的巢状排列,并被精致的纤维血管基质包围。免疫组化分析显示,肿瘤synaptophysin、CD56、GATA-3和S100阳性。患者术后病情稳定,无疾病。术前超声检查对甲状腺PGL的正确诊断至关重要。我们回顾了目前关于甲状腺pgl的诊断结果和治疗的文献,目的是补充先前报道的甲状腺pgl的发现。需要为该实体创建一个强大的成像和免疫组织化学谱,以对抗甲状腺pgl经常发生的误诊。
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引用次数: 0
The Impact of Hybrid Therapy on Langerhans Cell Histiocytosis of the Mandible in an Older Male: A Case Report and Literature Review. 混合疗法对老年男性下颌骨朗格汉斯细胞组织细胞增多症的影响:1例报告及文献复习。
IF 0.4 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-10-22 eCollection Date: 2025-01-01 DOI: 10.1155/crot/2996605
Yoshifumi Matsumoto, Maki Akamatsu, Shinichi Ohba, Fumihiko Matsumoto

Background: The unusual disorder known as Langerhans cell histiocytosis (LCH), which is most frequently observed in children and young adults, is caused by the clonal proliferation of Langerhans cells. This disease is classified into several types depending on the extent of the lesion. Because it is rare diseases, there is no established standard treatment of this disease. In this report, we describe an extremely rare case of LCH that developed in the mandible of an older male. This is the first report in the world of a favorable outcome following surgical resection and local steroids administration.

Case report: The patient was a 75-year-old male who complained of swelling and pain in his mandible; however, there were no abnormal findings upon intraoral examination. Panoramic radiograph, computed tomography (CT), and magnetic resonance imaging (MRI) revealed osteolytic tumors. A diagnosis of LCH was confirmed based on the pathological findings of a cluster of Langerhans cells in a biopsy specimen of a submucosal tumor. Because the patient was elderly and the primary tumor was in the mandible, radiation therapy, chemotherapy, and systemic steroid administration were difficult to tolerate due to side effects. We performed combined treatment with surgical resection and local steroids injection. He was discharged from the hospital 1 week after the operation; the intraoral wound healed after 2 months, and the pain improved.

Conclusions: This report suggests that a combination of surgical resection and local steroids administration is effective in treating LCH of the mandible in elderly patients.

背景:朗格汉斯细胞组织细胞增生症(LCH)是一种罕见的疾病,最常见于儿童和年轻人,由朗格汉斯细胞的克隆性增殖引起。这种疾病根据病变的程度分为几种类型。由于这是一种罕见的疾病,目前还没有确定的标准治疗方法。在这个报告中,我们描述了一个极其罕见的LCH的情况下,发展在一个老年男性下颌骨。这是世界上首次报道手术切除和局部类固醇治疗后的良好结果。病例报告:患者为75岁男性,主诉下颌骨肿胀疼痛;但口腔内检查未见异常。全景x线摄影、计算机断层扫描(CT)和磁共振成像(MRI)显示溶骨性肿瘤。LCH的诊断是根据粘膜下肿瘤活检标本中朗格汉斯细胞群的病理结果确定的。由于患者年龄较大,原发肿瘤在下颌骨,放疗、化疗和全身类固醇治疗因副作用难以耐受。我们采用手术切除和局部注射类固醇联合治疗。术后1周出院;术后2个月口内创面愈合,疼痛减轻。结论:本报告提示手术切除结合局部类固醇治疗老年下颌骨LCH患者是有效的。
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引用次数: 0
A Case of Follicular Dendritic Cell Sarcoma of the Palatine Tonsil That Developed as a Radiation-Induced Sarcoma. 放射诱导的腭扁桃体滤泡树突状细胞肉瘤1例。
IF 0.4 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-10-19 eCollection Date: 2025-01-01 DOI: 10.1155/crot/8861715
Alyssa Yoshida, Takuya Mikoshiba, Mariko Sekimizu, Shintaro Nakamura, Ryoto Nagai, Miho Kawaida, Katsura Emoto, Hiroyuki Ozawa

Follicular dendritic cell sarcoma is a rare malignant tumor originating from follicular dendritic cells. We present the first report of such a sarcoma of the palatine tonsils that developed as a radiation-induced sarcoma. A 78-year-old man, who had undergone chemoradiotherapy for hypopharyngeal and esophageal cancer 7 years prior, presented with discomfort during swallowing. Endoscopic pharyngeal examination revealed a tumor in the right palatine tonsil which was histopathologically diagnosed as a follicular dendritic cell sarcoma. The tumor also met the diagnostic criteria for radiation-induced sarcoma. Positron emission tomography/computed tomography revealed abnormal uptake in the right palatine tonsil and bilateral cervical lymph nodes; no distant metastases were detected. The patient underwent tumor resection using the mandibular swing approach, pharyngeal reconstruction using an anterolateral thigh flap, bilateral neck lymph node dissection, and tracheostomy. Postoperative radiotherapy was not administered because of previous irradiation; however, no apparent recurrence or metastasis was observed 4 years after surgery. Follicular dendritic cell sarcoma can develop over an extended period after previous irradiation. Extended resection with a negative margin may be pivotal in treating follicular dendritic cell sarcoma when postoperative radiotherapy cannot be administered.

滤泡树突状细胞肉瘤是一种罕见的起源于滤泡树突状细胞的恶性肿瘤。我们提出的第一个报告,这种肉瘤的腭扁桃体发展为辐射诱导肉瘤。78岁男性,7年前因下咽癌和食管癌接受放化疗,吞咽时出现不适。咽镜检查发现右腭扁桃体肿瘤,经组织病理学诊断为滤泡树突状细胞肉瘤。该肿瘤也符合放射性肉瘤的诊断标准。正电子发射断层扫描/计算机断层扫描显示右侧腭扁桃体和双侧颈部淋巴结摄取异常;未发现远处转移。患者采用下颌骨摆动入路切除肿瘤,采用股前外侧皮瓣重建咽部,双侧颈部淋巴结清扫和气管切开术。术后未行放疗,因既往放疗;术后4年未见明显复发或转移。滤泡树突状细胞肉瘤可在先前照射后的较长时间内发展。当术后不能进行放射治疗时,扩大切除阴性切缘可能是治疗滤泡树突状细胞肉瘤的关键。
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引用次数: 0
Nonsurgical Management of Sinonasal Teratocarcinosarcoma With Chemoradiotherapy: A Case Report. 鼻窦畸胎瘤放化疗的非手术治疗1例报告。
IF 0.4 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-10-09 eCollection Date: 2025-01-01 DOI: 10.1155/crot/1109193
Yuki Ban, Junko Tsuda, Yosuke Okinaka, Youhei Yamamoto, Mei Sakamoto, Tomoyasu Yamagata, Yosuke Takemoto, Makoto Hashimoto, Yoshinobu Hoshii, Kazuma Sugahara

Sinonasal teratocarcinosarcoma (SNTCS) is a rare, aggressive malignancy with epithelial, mesenchymal, and neuroectodermal components. We report the case of a 66-year-old man with right nasal obstruction and epistaxis. Imaging revealed a mass in the right nasal cavity and ethmoid sinus. Histopathology revealed immature squamous nests with clear cytoplasm, and immunohistochemistry confirmed multiphenotypic differentiation. Therefore, SNTCS was diagnosed. Owing to inoperability, the patient underwent chemoradiotherapy, which resulted in stable disease at 6 months. This case highlights the diagnostic complexity of SNTCS and suggests that nonsurgical management may be effective in select cases.

鼻窦畸形瘤肉瘤是一种罕见的侵袭性恶性肿瘤,具有上皮、间充质和神经外胚层成分。我们报告的情况下,66岁的男子右鼻塞和鼻出血。影像显示右鼻腔及筛窦有肿块。组织病理学显示未成熟的鳞状巢,细胞质清晰,免疫组织化学证实多表型分化。因此诊断为SNTCS。由于不能手术,患者接受了放化疗,6个月时病情稳定。该病例突出了SNTCS诊断的复杂性,并提示非手术治疗可能对某些病例有效。
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引用次数: 0
Case Report and Literature Review on Tracheostomal Myiasis: Clinical Presentation, Challenges, and Treatment. 气管造口蝇蛆病病例报告及文献回顾:临床表现、挑战及治疗。
IF 0.4 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-09-27 eCollection Date: 2025-01-01 DOI: 10.1155/crot/1555107
Yazieed M Albarrak, Ali M Alsudays, Mohammed A Alwabili, Khaled A Almanea, Fareed R Alghamdi, Reema A Aldawish

Tracheostomal myiasis, the infestation of a tracheostomy site by fly larvae, is an uncommon and challenging condition, with limited cases reported in the medical literature. This case report aims to describe both the clinical presentation and management of tracheostomal myiasis in a patient with multiple comorbidities. The patient presented with foul-smelling discharge and visible maggots at the tracheostomy site. Management involved manual removal of larvae, surgical debridement, irrigation with iodine solution, and sealing the stoma with paraffin ointment. This case highlights the need for clinician awareness of this rare complication, particularly in patients with compromised health, and highlights the importance of preventive care and early intervention. As a single case report, it reflects the experience within a broader context of managing tracheostomy-related complications.

气管造口蝇蛆病是一种罕见且具有挑战性的疾病,在医学文献中报道的病例有限。本病例报告的目的是描述气管造口蝇蛆病的临床表现和管理的病人有多种合并症。患者在气管切开术部位有恶臭的分泌物和可见的蛆。处理方法包括人工清除幼虫,手术清创,碘溶液冲洗,并用石蜡软膏密封口。这一病例突出了临床医生对这一罕见并发症的认识的必要性,特别是在健康受损的患者中,并突出了预防保健和早期干预的重要性。作为一个单一的病例报告,它反映了在更广泛的背景下处理气管切开术相关并发症的经验。
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引用次数: 0
Primary Posterior Neck Hydatid Cyst: A Case Report and Review of the Literature. 原发性后颈部包虫病1例报告及文献复习。
IF 0.4 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-09-26 eCollection Date: 2025-01-01 DOI: 10.1155/crot/6909432
Gibran Atwi, Charbel Saad, Samer Serhal, Rami Saade

Introduction: Hydatid cyst disease is an endemic parasitic infection caused by Echinococcus granulosus. Hydatid cysts occur mainly in the liver and lungs and are rare in the head and neck region, even in endemic areas. Due to their nonspecific clinical manifestations, diagnostic challenges are encountered in such atypical presentations. Case Presentation: We report the case of a 6-year-old girl who presented with a progressively enlarging, painless left postauricular mass. Radiological assessment revealed a well-circumscribed, unilocular cystic lesion at level V of the neck. Surgical resection was performed, and histopathology confirmed a hydatid cyst. Postoperatively, the patient was treated with albendazole. No recurrence was observed at follow-up. Clinical Discussion: Hydatid cysts in the cervical region are exceptionally rare, with only a few cases documented in the literature. The disease can remain asymptomatic for years, often presenting as a slow-growing mass. Imaging with ultrasound and CT is critical in preoperative diagnosis. The gold standard of treatment remains complete surgical excision, reinforced by anthelmintic therapy to prevent recurrence. Conclusion: Although rare, hydatid cyst disease should remain among the differential diagnoses of cystic neck masses, especially in endemic areas. Early diagnosis and intervention are essential in evading complications of the disease such as anaphylaxis or recurrence. Ultimately, prevention remains the most effective strategy, emphasizing the need for community awareness, hygienic practices, and coordinated control efforts to break the parasite's life cycle.

简介:包虫病是由细粒棘球绦虫引起的地方性寄生虫感染。包虫囊肿主要发生在肝脏和肺部,即使在流行地区,在头颈部也很少见。由于其非特异性的临床表现,在这种非典型的表现中遇到了诊断上的挑战。病例介绍:我们报告一个6岁的女孩,她表现出一个逐渐扩大的无痛左耳后肿块。影像学检查显示颈部V级有一界限清楚的单眼囊性病变。手术切除,组织病理学证实为包虫囊肿。术后给予阿苯达唑治疗。随访未见复发。临床讨论:在宫颈区域包虫病是非常罕见的,只有少数病例记录在文献中。这种疾病可以保持多年无症状,通常表现为缓慢增长的肿块。超声和CT成像在术前诊断中至关重要。治疗的金标准仍然是完全的手术切除,并辅以驱虫药治疗以防止复发。结论:包虫病虽罕见,但仍应作为囊性颈部肿块的鉴别诊断之一,特别是在流行地区。早期诊断和干预是必不可少的,以避免疾病并发症,如过敏反应或复发。最终,预防仍然是最有效的战略,强调需要社区意识、卫生习惯和协调控制努力,以打破寄生虫的生命周期。
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引用次数: 0
A Sinonasal Myxoma Arising From the Inferior Nasal Concha: Report of a Rare Case and Review of the Literature. 下鼻甲发生鼻窦黏液瘤1例报告及文献复习。
IF 0.4 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-09-23 eCollection Date: 2025-01-01 DOI: 10.1155/crot/5590381
Hesam Jahandideh, Maryam Arab, Maryam Roomiani

Head and neck myxomas are extremely rare neoplasms, and only a few reports have been in the literature. Specifically, reports of myxomas of the nose and paranasal sinuses are sporadic. Here, we present a rare case of a 47-year-old female with progressive nasal obstruction who was found to have a myxoma arising within the inferior nasal concha-the first reported case of its kind. The diagnosis was confirmed by biopsy, and endoscopic transnasal resection (ETNR) was conducted. ETNR proved effective in resolving symptoms of nasal myxoma, with complete removal of the tumor and no recurrence on follow-up. The operation is described, and the literature on the subject is reviewed.

头颈部黏液瘤是一种极为罕见的肿瘤,文献中仅有少数报道。具体地说,鼻窦和副鼻窦黏液瘤的报告是零星的。在此,我们报告一例罕见的47岁女性进行性鼻塞病例,她被发现下鼻甲壳内有粘液瘤,这是此类病例的首次报道。活检证实诊断,并进行内镜经鼻切除术(ETNR)。经证实,ETNR能有效缓解鼻黏液瘤的症状,在随访中肿瘤完全切除且无复发。描述了手术过程,并回顾了有关该主题的文献。
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引用次数: 0
Laryngeal Mycobacterium bovis: A Unique Cause of Airway Compromise in a 27-Year-Old Male With Down Syndrome. 牛喉分枝杆菌:27岁男性唐氏综合症患者气道受损的独特原因。
IF 0.4 Q4 OTORHINOLARYNGOLOGY Pub Date : 2025-09-11 eCollection Date: 2025-01-01 DOI: 10.1155/crot/6485801
Morgan Davis Mills, Michael Oca, Michelle Don, Andrew M Vahabzadeh-Hagh

Introduction: Laryngeal tuberculosis (TB) due to Mycobacterium bovis is an extremely rare cause of airway obstruction. This case report describes a unique instance of acute airway obstruction in an immunocompetent 27-year-old male with down syndrome caused by laryngeal Mycobacterium bovis, shedding light on the challenges of diagnosis and treatment. Case: A 27-year-old male with trisomy 21 presented with progressive shortness of breath, productive cough, dysphonia, and dysphagia. After a failed workup for pneumonia and other conditions, imaging revealed likely epiglottitis and a right upper lung lesion. A tracheostomy was performed due to worsening airway compromise. Biopsy results confirmed granulomatous inflammation and identified Mycobacterium bovis, which was resistant to pyrazinamide. The patient was treated with a modified RIPE regimen and successfully decannulated 2 months later. Conclusion: This case emphasizes the importance of a comprehensive diagnostic approach, including tissue biopsy and culture, in patients with airway compromise of unclear etiology. Mycobacterium bovis, though rare, should be considered in the differential diagnosis of laryngeal TB, especially in cases with progressive symptoms and atypical findings. Early recognition and tailored treatment are critical for favorable outcomes.

由牛分枝杆菌引起的喉部结核(TB)是一种极为罕见的气道阻塞原因。本病例报告描述了一个独特的急性气道阻塞的情况下,免疫功能正常的27岁男性唐氏综合症由喉部牛分枝杆菌引起的,揭示了诊断和治疗的挑战。病例:一名27岁男性21三体患者,表现为进行性呼吸短促、咳嗽、发音困难和吞咽困难。在肺炎和其他疾病检查失败后,影像学显示可能是会厌炎和右上肺病变。由于气道损伤加重,行气管切开术。活检结果证实肉芽肿性炎症,并鉴定出对吡嗪酰胺耐药的牛分枝杆菌。患者接受改良的RIPE方案治疗,2个月后成功脱管。结论:本病例强调了综合诊断方法的重要性,包括组织活检和培养,对病因不明的气道损害患者。牛分枝杆菌虽然罕见,但在喉部结核的鉴别诊断中应予以考虑,特别是在有进展症状和非典型表现的病例中。早期识别和量身定制的治疗对于良好的结果至关重要。
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引用次数: 0
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