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Dedifferentiated Leiomyosarcoma of the Auricle with Heterologous Osteosarcoma Component: Case Report and Literature Review 伴有异体骨肉瘤成分的耳廓去分化平滑肌肉瘤:1例报告及文献复习
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2022-05-31 DOI: 10.1155/2022/3684461
Andrea Dekanić, N. Jonjić, Anita Savić Vuković
Leiomyosarcomas are rare malignant tumors of smooth muscles. Head and neck involvement by this disease is very rare, and cutaneous leiomoysarcomas of the ear are even rarer. This is way clinically they are usually mistaken for either squamous or basal cell carcinomas, as was the case in an 85-year-old male patient presented in this report. However, the final diagnosis was even more interesting considering that it was a dedifferentiated leiomyosarcoma of the auricle with a heterologous component of osteosarcoma. The auricular cutaneous malignancies have a much higher rate of recurrence than the corresponding malignancy in other regions of the head and neck, even when resected with negative surgical margins, and dedifferentiated leiomyosarcoma is clinically even more aggressive. Thus, the treatment of choice is a total auriculectomy and great attention should be paid to appropriate margins.
摘要平滑肌肉瘤是一种罕见的平滑肌恶性肿瘤。这种疾病累及头颈部非常罕见,耳皮肤平滑肌肉瘤更罕见。这是临床上常被误认为鳞状细胞癌或基底细胞癌的原因,正如本报告中一位85岁男性患者的病例。然而,最后的诊断更加有趣,因为这是一种耳廓去分化平滑肌肉瘤,伴有骨肉瘤的异源成分。耳廓皮肤恶性肿瘤的复发率远高于头颈部其他部位的相应恶性肿瘤,即使在手术切缘阴性的情况下也是如此,而去分化平滑肌肉瘤在临床上更具侵袭性。因此,治疗的选择是全耳切除术,并应注意适当的边缘。
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引用次数: 0
The Youngest Case of Metachronous Bilateral Acinic Cell Carcinoma of the Parotid Gland: A Case Report and Literature Review 最年轻的双侧异时性腮腺腺泡细胞癌1例报告并文献复习
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2022-05-24 DOI: 10.1155/2022/8474741
Raid Alhayaza, M. Dababo, S. Velagapudi
Introduction Acinic cell carcinoma (ACC) is a low-grade malignant salivary neoplasm that represents 17% of all salivary gland malignancies. It has a tendency to affect young individuals, especially females. ACC mainly originates in the parotid gland and has a potential for recurrence and metastases. Rarely, ACC can affect both parotid glands in a single individual. A bilateral ACC of the parotid gland could either present as a synchronous or a metachronous tumor. Case Report. Our patient is a 19-year-old female known case of ACC of the right parotid gland. The tumor was resected in December 2017. After 3 years, she presented with a left parotid pain and swelling, which raised the suspicion of a contralateral metachronous tumor of the left parotid gland. In September 30, 2020 we proceeded with ultrasound-guided fine needle aspiration of the left intraparotid lesion, and the results turned out to be consistent with ACC. Here, we report a case of a 19-year-old female presenting with metachronous bilateral ACC of the parotid gland with an interval of 3 years, which is the 6th of its kind in the literature and the youngest amongst them. Conclusion Despite the rareness of metachronous occurrence of bilateral ACC of the parotid gland, it is still encountered in the medical practice. Here, we are highlighting the importance of follow-up with a periodic clinical and radiological examinations, bearing in mind the contralateral nonaffected parotid gland.
腺泡细胞癌(ACC)是一种低级别恶性涎腺肿瘤,占所有涎腺恶性肿瘤的17%。它倾向于影响年轻人,尤其是女性。ACC主要起源于腮腺,有复发和转移的可能。罕见的是,ACC可以同时影响一个人的两个腮腺。双侧腮腺ACC可表现为同步或异时性肿瘤。病例报告。我们的病人是一名19岁的女性,已知右腮腺ACC病例。该肿瘤于2017年12月切除。3年后,她出现左侧腮腺疼痛和肿胀,这引起了对侧左腮腺异时性肿瘤的怀疑。我们于2020年9月30日行超声引导下左侧腮腺内病变细针穿刺,结果与ACC一致。在此,我们报告一例19岁女性腮腺异时性双侧ACC,间隔3年,这是文献中第6例,也是其中最年轻的一例。结论腮腺双侧ACC异时性发生虽少见,但在医疗实践中仍会遇到。在这里,我们强调随访的重要性,定期临床和放射检查,记住对侧未受影响的腮腺。
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引用次数: 0
Frontal Balloon Sinuplasty in Complicated Acute Pediatric Rhinosinusitis (ARS) 额窦球囊成形术治疗小儿复杂急性鼻窦炎
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2022-05-14 DOI: 10.1155/2022/7232588
Smrithi Chidambaram, B. Wahle, D. Leonard
Utilization of frontal balloon sinuplasty in pediatric complicated acute rhinosinusitis (ARS) is demonstrated to be a safe and expedient alternative to other procedures such as trephination or functional endoscopic sinus surgery (FESS) in this case series. We performed a retrospective review of six pediatric cases of frontal balloon sinuplasty for ARS with intracranial complications at a tertiary academic center. Patients underwent unilateral (n = 5) or bilateral dilation (n = 1) in addition to functional endoscopic sinus surgery (FESS) including anterior ethmoidectomy (n = 5) and maxillary antrostomy (n = 6). This technique effectively addressed frontal sinus obstruction and served as an alternative to procedures such as trephination or functional endoscopic sinus surgery. No immediate or short-term complications of balloon dilation were observed in these cases. A larger cohort and extended follow-up are necessary to determine the use and long-term impact of this technique.
在本病例系列中,使用额叶球囊鼻窦成形术治疗儿童复杂的急性鼻窦炎(ARS)被证明是一种安全、方便的替代方法,可以替代其他手术,如穿刺或功能性内窥镜鼻窦手术(FESS)。我们对在某三级学术中心接受额叶球囊窦成形术治疗ARS合并颅内并发症的6例患儿进行回顾性分析。患者接受单侧(n = 5)或双侧扩张(n = 1),以及功能性内窥镜鼻窦手术(FESS),包括前筛窦切除术(n = 5)和上颌窦口造口术(n = 6)。这项技术有效地解决了额窦阻塞,并作为其他手术的替代方案,如穿甲术或功能性内窥镜鼻窦手术。在这些病例中没有观察到球囊扩张的立即或短期并发症。为了确定这项技术的使用和长期影响,有必要进行更大的队列研究和长期随访。
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引用次数: 1
Perilymph Fistula as a Complication of Eustachian Tube Dilation and Tympanoplasty 咽鼓管扩张和鼓室成形术的并发症——淋巴周围瘘
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2022-05-07 DOI: 10.1155/2022/5978757
R. Kim, L. Scholtz, R. Jadeed, C. Pfeiffer, H. Sudhoff, I. Todt
Eustachian tube dilation (ETD) is an established, minimally invasive therapeutic approach for chronic eustachian tube dysfunction. The complications associated with performing a ETD are rare. A 22-year-old female patient presented with chronic otitis media on the right side and chronic obstructive tube dilation disorder on both sides. A type I tympanoplasty was performed on the right side because of a tympanic membrane perforation after a ETD on both sides without apparent complications. On the 5th postoperative day, she presented with headache, dizziness and hearing loss on the right side. There was a decrease of hearing threshold on the right side in the pure-tone audiogram and vHIT, cVEMP, and SVV were irregular. The β-2-transferrin test was positive. Since a right-sided perilymph fistula was suspected, an emergency tympanotomy was performed with a round window membrane cover with fascia on the right side. Intraoperatively, a regular, intact ossicular chain was found with a slightly moist middle ear mucosa. The round window membrane was covered by the promontorial lip. Under these measures, the patient's dizziness regressed. The right ear pure-tone threshold vHIT, cVEMP, and SVV normalized.
耳咽管扩张(ETD)是一种成熟的微创治疗慢性耳咽管功能障碍的方法。与ETD相关的并发症是罕见的。一位22岁的女性患者表现为右侧慢性中耳炎和两侧慢性阻塞性管道扩张障碍。由于双侧ETD后鼓膜穿孔,右侧行I型鼓室成形术,无明显并发症。术后第5天,患者出现头痛、头晕、右侧听力下降。纯音听力图右侧听阈降低,vHIT、cvpp、SVV不规则。β-2转铁蛋白试验呈阳性。由于怀疑右侧淋巴周围瘘管,我们在右侧进行了一个圆形膜覆盖的紧急鼓室切开术。术中发现规则完整的听骨链,中耳黏膜微湿。圆形的窗膜被突起的唇盖住。在这些措施下,病人的头晕消退了。右耳纯音阈值vHIT、cVEMP和SVV归一化。
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引用次数: 0
Actinomyces Acute Rhinosinusitis Complicated by Subperiosteal Abscess in an Immunocompromised 12-Year-Old: Case Report and Literature Review 12岁儿童放线菌性急性鼻窦炎并发骨膜下脓肿:病例报告及文献复习
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2022-04-11 DOI: 10.1155/2022/7058653
S. Nimmagadda, Li-Xing Man, Margo K McKenna, J. Faria, I. Schmale
Objective To describe a rare case of pediatric actinomycotic rhinosinusitis with orbital subperiosteal abscess and review the current literature to assess methods of diagnosis, treatment modalities, and outcomes with appropriate treatment. Methods A case report and a review of the literature. Results A 12-year-old patient with Crohn's disease on infliximab presented with rhinosinusitis with orbital subperiosteal abscess formation. Endoscopic sinus surgery was performed and cultures grew actinomyces. A prolonged course of antibiotics was started, resulting in the complete resolution of the infection. In a literature review, all cases of uncomplicated and complicated actinomyces rhinosinusitis managed with appropriate surgery and prolonged antibiotics resulted in a cure. Our case is the first reported in a pediatric patient and the first taking immunosuppressive medication. Overall, only 3 cases of actinomyces rhinosinusitis in immunosuppressed individuals have been reported, each with uncontrolled diabetes and each also responded well to surgery and appropriate antibiotics. Conclusion Actinomycosis of the paranasal sinuses poses a diagnostic challenge, with infections varying widely in presentation and extent of disease. A high index of suspicion, appropriate testing, and early aggressive treatment are critical in managing patients with this infection. Our case and prior published studies show that actinomyces rhinosinusitis can be successfully managed with endoscopic sinus surgery, abscess drainage as necessary, and a prolonged course of antibiotics, even in immunocompromised and pediatric populations.
目的报道1例罕见的小儿放线菌性鼻窦炎合并眼眶骨膜下脓肿病例,回顾文献资料,探讨其诊断、治疗方法及治疗效果。方法结合病例报告和文献复习。结果1例12岁的克罗恩病患者接受英夫利昔单抗治疗后表现为鼻窦炎伴眶骨膜下脓肿形成。内镜下鼻窦手术,培养出放线菌。开始了一个延长疗程的抗生素治疗,导致感染完全消失。在一篇文献综述中,所有简单和复杂放线菌性鼻窦炎的病例都通过适当的手术和长期的抗生素治疗而治愈。我们的病例是第一例报道的儿科患者,也是第一例服用免疫抑制药物的患者。总体而言,仅报道了3例免疫抑制个体的放线菌性鼻窦炎,每个人都有不受控制的糖尿病,每个人都对手术和适当的抗生素反应良好。结论鼻窦放线菌病的诊断具有挑战性,其感染在表现和疾病程度上差异很大。高度的怀疑指数、适当的检测和早期积极治疗对于管理这种感染的患者至关重要。我们的病例和先前发表的研究表明,放线菌性鼻窦炎可以通过内镜鼻窦手术、必要时脓肿引流和延长抗生素疗程来成功治疗,即使在免疫功能低下和儿童人群中也是如此。
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引用次数: 1
Giant Nonfunctioning Parathyroid Cyst: A Case Report and Review of the Literature 巨大无功能甲状旁腺囊肿1例报告及文献复习
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2022-04-09 DOI: 10.1155/2022/6388749
E. Cleere, M. Corbett, A. Quinn, T. Subramaniam
Parathyroid cysts are a rare clinical entity that may arise in the neck or mediastinum. They are more common in women and generally present in the fourth and fifth decades of life. Diagnosis of parathyroid cysts is challenging, and despite thorough radiological and cytological investigation, they are often mistaken for thyroid pathology. Definitive diagnosis is often only confirmed following complete surgical resection and histopathological analysis. We present the case of a woman who was referred to our outpatient clinic with a left-sided neck mass and associated compressive symptoms. Initial examination and investigation appeared consistent with a large thyroid nodule. Following surgical resection, the lesion was found to be a parathyroid cyst. Subsequently, we review the available literature on parathyroid cysts with particular emphasis on the diagnostic challenge they pose to clinicians.
甲状旁腺囊肿是一种罕见的临床实体,可能出现在颈部或纵隔。它们在女性中更为常见,通常出现在生命的第四和第五十年。甲状旁腺囊肿的诊断是具有挑战性的,尽管进行了彻底的放射学和细胞学检查,但它们经常被误认为甲状腺病理。明确的诊断通常只有在完全手术切除和组织病理学分析后才能得到证实。我们提出的情况下,一名妇女谁被转介到我们的门诊与左侧颈部肿块和相关的压缩症状。初步检查和调查显示为甲状腺大结节。手术切除后,发现病变为甲状旁腺囊肿。随后,我们回顾了甲状旁腺囊肿的现有文献,特别强调了他们对临床医生的诊断挑战。
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引用次数: 2
A Case of Isolated Congenital Stapedial Suprastructure Fixation 孤立性先天性镫骨上结构固定1例
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2022-03-19 DOI: 10.1155/2022/8620738
J. Lee, H. Lee, Sung Huhn Kim
We report a case of conductive hearing loss caused by isolated congenital stapedial suprastructure fixation with normal footplate mobility. A 60-year-old woman visited the clinic for right-sided mixed hearing loss. Exploratory tympanotomy revealed a bony synostosis between the stapedial suprastructure and promontory, while all the ossicles were present and normally shaped. As the bony synostosis was separated, the stapes became mobile. This is the first report in the medical literature of this congenital ear anomaly. This case also illustrates that stapedial fixation can occur in the suprastructure as well as in the footplate; thus, one must be mindful of this when performing exploratory tympanotomy for stapes fixation.
我们报告一例传导性听力损失是由孤立的先天性镫骨上结构固定引起的。一名60岁妇女因右侧混合性听力损失就诊。探索性鼓室切开术显示镫骨上结构和海岬之间有骨结合,而所有小骨均存在且形状正常。随着骨性结膜的分离,镫骨开始活动。这是医学文献中首次报道这种先天性耳畸形。该病例还表明,镫骨固定既可发生在足跖,也可发生在上部结构;因此,在进行探索性鼓室切开术进行镫骨固定时,必须注意这一点。
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引用次数: 2
Combination Immunosuppressive Therapy in Primary Autoimmune Inner Ear Disease in Pregnancy 联合免疫抑制治疗妊娠期原发性自身免疫性内耳疾病
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2022-03-18 DOI: 10.1155/2022/9210780
Saikrishna Ananthapadmanabhan, J. Jabbour, David Brown, V. Sivapathasingam
Objective Autoimmune inner ear disease (AIED) is a rare disorder characterized by rapidly progressive, sensorineural hearing loss that demonstrates good responsiveness to corticosteroid and immunosuppressive therapy. The pathophysiology is likely driven by chronic trafficking of immune cells into the inner ear, targeting inner ear proteins to coordinate inflammation. Suppression or modulation of the immune response can minimize cochleitis allowing for potential recovery of hearing. It is an otologic emergency requiring a multidisciplinary approach to management to commence immunosuppressive therapy. This can be achieved using steroids, immunomodulators, plasmapheresis, intravenous immunoglobulin, or biologic agents. Treatment decisions are further complicated in pregnancy and require supervision by an obstetrician and maternal-fetal medicine (MFM) specialist. Concerns include safe dosing of steroids and potential for transplacental migration of immune complexes. We provide the first comprehensive literature review on AIED and its implications in pregnancy. We frame our discussion in the context of the second reported case of primary AIED in pregnancy and the first to show excellent response to immunosuppressive therapy. Methods We reviewed the presented case and literature on AIED. Results A 27-year-old, pregnant, HSP-70 positive woman was diagnosed with AIED and had excellent recovery of hearing and balance following a combination of steroid treatment, augmented by oral immunomodulators, plasmapheresis, and IVIG. Conclusion AIED is a diagnostic challenge, and treatment considerations are complex when encountered in pregnancy. Management requires multidisciplinary involvement between otolaryngologists, immunologists, and obstetricians to balance maternal and fetal health outcomes.
目的自身免疫性内耳疾病(AIED)是一种罕见的疾病,其特征是快速进展的感觉神经性听力损失,对皮质类固醇和免疫抑制治疗表现出良好的反应性。病理生理可能是由免疫细胞进入内耳的慢性运输驱动,以内耳蛋白质为目标来协调炎症。抑制或调节免疫反应可以使耳蜗炎最小化,从而使听力的潜在恢复。这是一个耳科急症,需要多学科的方法来管理,开始免疫抑制治疗。这可以通过类固醇、免疫调节剂、血浆置换、静脉注射免疫球蛋白或生物制剂来实现。妊娠期的治疗决定更加复杂,需要产科医生和母胎医学(MFM)专家的监督。担忧包括类固醇的安全剂量和免疫复合物经胎盘迁移的可能性。我们提供了第一个全面的文献综述AIED及其在妊娠中的意义。我们在第二例报道的妊娠期原发性AIED病例和第一例对免疫抑制治疗有良好反应的病例的背景下进行讨论。方法回顾已报道的AIED病例及相关文献。结果一名27岁的孕妇,HSP-70阳性,诊断为AIED,在类固醇治疗、口服免疫调节剂、血浆置换和IVIG联合治疗后,听力和平衡恢复良好。结论妊娠期AIED诊断困难,治疗考虑复杂。管理需要耳鼻喉科医生、免疫学家和产科医生之间的多学科参与,以平衡孕产妇和胎儿的健康结果。
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引用次数: 0
Persistent Stapedial Artery Encountered during Cochlear Implantation 人工耳蜗植入过程中遇到持续性镫骨动脉
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2022-02-22 DOI: 10.1155/2022/8179062
H. Jones, J. Hintze, A. Gendre, C. Wijaya, F. Glynn, L. Viani, P. Walshe
Objectives Persistent stapedial artery (PSA) is a rare congenital anomaly that can complicate middle ear surgery. Methods We present the case of a 25-year-old male who underwent right-sided cochlear implantation. A PSA was encountered lying over the middle promontory intraoperatively. Results The PSA was carefully lifted off the middle ear promontory using a Hughes elevator to divide adhesions and delineate the artery. The implant electrode was placed through the round window niche in the usual fashion. Tragal cartilage and fibrin glue were used to control the trajectory of the electrode. Conclusion Cochlear implantation can be performed safely in patients with PSA.
目的持续性镫骨动脉(PSA)是一种罕见的先天性异常,可使中耳手术复杂化。方法我们报告一例25岁男性接受右侧人工耳蜗植入的病例。术中在中岬处发现PSA。结果使用休斯升降机将PSA小心地从中耳峡部提起,分离粘连并划定动脉。植入电极通常通过圆形窗口置入。用耳膜软骨和纤维蛋白胶控制电极的运动轨迹。结论对PSA患者行人工耳蜗植入术是安全的。
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引用次数: 1
Ipsilateral Vestibular Schwannoma after Cochlear Implantation 人工耳蜗植入术后同侧前庭神经鞘瘤
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2022-02-18 DOI: 10.1155/2022/4918785
S. Tüpker, N. Ay, L. Scholtz, H. Gehl, V. Mautner, P. Goon, H. Sudhoff, I. Todt
Objective The vestibular schwannoma incidence rate is approximately 4.2 per 100,000/year. Thus far, about 700,000 cochlear implantations have been performed worldwide; therefore, the occurrence of vestibular schwannoma postcochlear implantations can be assumed to be infrequent. Recent developments allow safe observation and surveillance of the implanted-side internal auditory canal (IAC) and cochlea by magnetic resonance imaging (MRI), even after cochlear implantation. Patients. A 71-year-old woman with sudden hearing loss and a contralateral vestibular schwannoma without clinical and genetic signs of neurofibromatosis type II. Intervention(s). Ipsilateral cochlear implantation and contralateral vestibular schwannoma extirpation with regular tumor follow-up. Main Outcome Measure(s). Comparison of ipsilateral pre and postcochlear implantation 3T MRI T1 GAD. Results We observed a tumor growing at the fundus of the internal auditory canal 1 year after cochlear implantation on the ipsilateral side. Although first detected after cochlear implantation beside a known vestibular schwannoma on the contralateral side, a scan slice thickness of 2 mm cannot fully exclude the preoperative persistence of a small tumor. Based on the clinical findings and after genetic exclusion of NFII, the patient was classified as a NFII mosaic type. Conclusion Even after cochlear implantation, tumors in the IAC causing vertigo, facial palsy, and affecting the audiologic outcome can be detected by MRI. The MRI slice thickness used before cochlear implantation should be under 2 mm.
目的前庭神经鞘瘤的发病率约为4.2 / 10万/年。到目前为止,全世界约有70万例人工耳蜗植入手术;因此,耳蜗植入术后前庭神经鞘瘤的发生可以认为是罕见的。最近的发展允许通过磁共振成像(MRI)对植入侧内耳道(IAC)和耳蜗进行安全观察和监测,即使在人工耳蜗植入后也是如此。病人。71岁女性,突发性听力丧失,对侧前庭神经鞘瘤,无II型神经纤维瘤病的临床和遗传征象。干预(s)。同侧人工耳蜗植入及对侧前庭神经鞘瘤切除伴肿瘤定期随访。主要结果测量。同侧人工耳蜗植入前后3T MRI T1 GAD比较。结果在同侧人工耳蜗植入术1年后,观察到一肿瘤在内耳道底部生长。虽然在耳蜗植入后首次在对侧前庭神经鞘瘤旁发现,但2毫米的扫描层厚度不能完全排除术前持续存在的小肿瘤。根据临床表现和基因排除NFII后,将患者分类为NFII马赛克型。结论人工耳蜗植入术后,腹腔内肿瘤仍可引起眩晕、面瘫,影响听力学预后。人工耳蜗植入前使用的MRI切片厚度应在2mm以下。
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引用次数: 0
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Case Reports in Otolaryngology
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