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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
Coexisting Thyroglossal Duct Cyst with Papillary Thyroid Cancer: A Case Report and Literature Review. 甲状腺舌管囊肿合并甲状腺乳头状癌1例报告并文献复习。
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2021-12-26 eCollection Date: 2021-01-01 DOI: 10.1155/2021/6111308
Abdullah A Alarfaj, Ahmed Zekri, Ibrahim Alyaeesh, Ahmed Alomairin, Abdulrahman Al Naim

Thyroglossal duct cysts (TGDCs) are common developmental anomalies in which the thyroglossal duct is not obliterated. Coexisting papillary thyroid cancer and TGDC are uncommon and should be investigated thoroughly to rule out TGDC carcinoma. We report a rare case of coexisting papillary thyroid cancer and TGDC in a 48-year-old man, who presented with a history of recurrent mild painful midline neck swelling, and ultrasound (US) revealed a TGDC that was subsequently managed conservatively. On follow-up after 1.6 years, a thyroid US and a fine-needle aspiration (FNA) biopsy were performed, which showed malignant papillary thyroid carcinoma. Total thyroidectomy, the Sistrunk procedure, and central neck dissection were implemented. After three days, the patient was discharged on 150 mg of levothyroxine. Follow-up was unremarkable with no complications. The authors would like to stress the importance of regular TGDC and thyroid gland follow-ups for early detection and diagnosis of thyroid malignancy via clinical examination and US.

甲状舌管囊肿(TGDCs)是常见的发育异常,甲状舌管未完全消失。甲状腺乳头状癌与TGDC共存并不常见,应彻底检查以排除TGDC癌。我们报告一例罕见的乳头状甲状腺癌和TGDC共存的病例,患者为48岁男性,其表现为复发性轻度疼痛颈部中线肿胀史,超声(US)显示TGDC,随后进行保守治疗。在1.6年后的随访中,进行了甲状腺US和细针穿刺(FNA)活检,显示为恶性甲状腺乳头状癌。实施甲状腺全切除术、Sistrunk手术和中央颈部清扫术。三天后,患者给予150毫克左甲状腺素出院。随访无显著差异,无并发症。作者想强调定期TGDC和甲状腺随访对于通过临床检查和超声早期发现和诊断甲状腺恶性肿瘤的重要性。
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引用次数: 0
Delayed-Onset Neuropathic Pain after Septoplasty. 鼻中隔成形术后的延迟性神经性疼痛。
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2021-12-22 eCollection Date: 2021-01-01 DOI: 10.1155/2021/9966318
Foteini-Stefania Koumpa, Mark Ferguson, Hesham Saleh

Postoperative pain following a septoplasty is expected to be mild and limited to a few days after the operation. Chronic pain following the procedure is rare. No cases of delayed-onset neuropathic pain or allodynia have been described in the literature. This paper presents a case of delayed-onset neuropathic pain after septoplasty in a previously pain-free asthmatic patient that was successfully managed by administration of intranasal local anaesthesia. Physical examination and imaging excluded any other cause of neuralgia. A literature review revealed reports of chronic pain in patients following septoplasty if there were nasal contact or compression points or nasal tumours. Separately, acute postseptoplasty allodynia is documented in iatrogenic maxillary nerve damage. However, delayed-onset neuralgic pain, exacerbated by certain environmental triggers, has not been previously described. Facial pain can be debilitating; successfully managing this neuralgic pain with administration of intranasal local anaesthetic had a substantial effect on the patient's quality of life.

鼻中隔成形术后的术后疼痛预计是轻微的,并限制在手术后几天。手术后的慢性疼痛是罕见的。文献中没有迟发性神经性疼痛或异常性疼痛的病例。这篇论文提出了一个延迟发作神经性疼痛后的鼻中隔成形术在一个以前无痛的哮喘患者成功地管理管理的鼻内局部麻醉。体格检查和影像学检查排除了任何其他神经痛的原因。一篇文献综述揭示了鼻中隔成形术后患者慢性疼痛的报告,如果有鼻接触或压迫点或鼻肿瘤。另外,急性鼻中隔成形术后异常痛症在医源性上颌神经损伤中也有记载。然而,延迟性神经痛,某些环境诱因加剧,以前没有描述。面部疼痛会使人虚弱;成功地管理这种神经痛与鼻内局部麻醉的管理对患者的生活质量有实质性的影响。
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引用次数: 0
A Rare Complication of Fine-Needle Aspiration of Neck Structures. 颈部结构细针抽吸的罕见并发症。
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2021-12-15 eCollection Date: 2021-01-01 DOI: 10.1155/2021/8944119
Yazeed M Qadadha, Nainika Nanda, Chad Ennis, Timothy McCulloch

Fine-needle aspiration (FNA) is a generally accepted tool for safe diagnostic evaluation in the workup of lesions and masses. Aside from the commonly discussed risks of infection and minor bleeding related to skin puncture, other more serious complications have been reported sparingly. We present two cases of pneumothorax from FNA of neck structures, which have been theorized but not previously reported to our knowledge. Discussion of cases of this complication rather than solely a theoretical understanding of it will aid in diagnosis and management of this complication.

细针穿刺(FNA)是一种普遍接受的工具,用于安全的诊断评估病变和肿块的工作。除了经常讨论的感染风险和与皮肤穿刺有关的轻微出血外,其他更严重的并发症很少有报道。我们提出了两例由颈部结构的FNA引起的气胸,这是理论性的,但据我们所知,以前没有报道过。讨论这种并发症的病例,而不仅仅是理论上的理解,将有助于这种并发症的诊断和管理。
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引用次数: 0
Importance of Imaging in Congenital Unilateral Vocal Fold Paralysis: A Case of Neck Neuroblastoma Presenting with Unilateral Vocal Fold Paralysis. 影像学在先天性单侧声带麻痹中的重要性:一例颈部神经母细胞瘤表现为单侧声带麻痹。
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2021-12-06 eCollection Date: 2021-01-01 DOI: 10.1155/2021/7368567
W X Yeo, C Y Chan, K K H Tan

Congenital vocal fold paralysis (VFP) is an important cause of respiratory compromise in infants. It can either be unilateral or bilateral, while imaging is routinely performed for bilateral VFP to evaluate for potential neurological causes, and such a practice may not be routine for unilateral VFP. While many of the unilateral VFP cases are idiopathic, the cause may occasionally be more sinister in nature, such as tumors. Therefore, unless an obvious cause of unilateral VFP is present (such as cardiac surgery or birth trauma), routine imaging of the brain, neck, and mediastinum should be performed for congenital unilateral VFP. We describe a rare case of a cervical neuroblastoma presenting with unilateral VFP that was detected only on imaging, thus highlighting its value and importance.

先天性声带麻痹(VFP)是导致婴幼儿呼吸系统损伤的重要原因。它可以是单侧或双侧,而成像是常规的双侧VFP,以评估潜在的神经系统原因,这种做法可能不是单侧VFP的常规做法。虽然许多单侧VFP病例是特发性的,但有时病因可能更险恶,如肿瘤。因此,除非存在单侧VFP的明显原因(如心脏手术或出生创伤),否则应对先天性单侧VFP进行常规脑、颈部和纵隔成像。我们描述了一个罕见的宫颈神经母细胞瘤表现为单侧VFP,仅在影像学上被检测到,从而强调其价值和重要性。
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引用次数: 0
Bilateral Recurrent Laryngeal Nerve Palsy following Total Thyroidectomy in Triple A Syndrome, an Unexpected but Critical Complication. 全甲状腺切除术后双侧喉返神经麻痹的三重A综合征,一个意想不到的但关键的并发症。
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2021-11-19 eCollection Date: 2021-01-01 DOI: 10.1155/2021/1315117
Mathieu Chamberland, Marc-Antoine Poulin, Danielle Beaudoin

Introduction: Triple "A" syndrome (TAS) is a rare autosomal recessive disorder that presents in childhood with achalasia cardia, alacrima, ACTH-resistant adrenal insufficiency, with sensorimotor and autonomic polyneuropathy developing later in the course of the disease. Case Presentation. An adult white male affected by this syndrome underwent an uneventful total thyroidectomy for malignancy and suffered delayed bilateral recurrent laryngeal nerve palsy in the early postoperative hours. The palsy spontaneously resolved after a five-week course.

Conclusion: Given the rarity of this severe condition and the absence of surgical or medical causes identifiable, there is possibility that it is the neurological involvement caused by TAS that predisposed the patient to this adverse outcome, precipitated by standard manipulations during surgery.

简介:三“A”综合征(TAS)是一种罕见的常染色体隐性遗传病,儿童期表现为贲门失弛缓症、肺水肿、acth抵抗性肾上腺功能不全,在病程后期出现感觉运动和自主神经病变。案例演示。一个成年白人男性受此综合征的影响,接受了良性甲状腺全切除术,术后早期遭受延迟性双侧喉返神经麻痹。经过五周的治疗,麻痹自行消退。结论:鉴于这种严重疾病的罕见性和缺乏可识别的外科或医学原因,有可能是由TAS引起的神经系统受累使患者易患这种不良结果,并在手术期间通过标准操作引起。
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引用次数: 0
INI1-Intact Sinonasal Carcinoma with Rhabdoid Features. 完整鼻窦癌伴横纹肌样特征。
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2021-11-18 eCollection Date: 2021-01-01 DOI: 10.1155/2021/6075130
Erin Mulry, Danielle M Blake, Poornima Hegde, Todd E Falcone

Sinonasal malignancies are known for their associated poor prognosis and diversity of histologic features. While poor prognosis is largely due to advanced disease at presentation, histologic features also play a significant role. Therefore, accurate pathologic diagnosis is of utmost importance. Here, we describe a 63-year-old male with chronic left-sided nasal obstruction and left-sided epistaxis who was found to have a large mass occupying most of the nasal cavity extending through the nasopharynx to just below the nasopharyngeal surface of the soft palate. During surgical excision, the mass was noted to originate from the floor of the maxillary sinus with erosion of the medial wall of the maxillary sinus. Pathology revealed a diagnosis of INI1-intact poorly differentiated composite carcinoma with rhabdoid phenotype and sarcomatoid and squamous cell carcinoma foci arising within an inverted papilloma. Included in this report is a detailed description of both the patient's medical course and this pathologically novel sinonasal neoplasm. We aim to elucidate this rare tumor's complex features in order to improve future diagnosis and stimulate prospective research on sinonasal malignancies with complex histology.

鼻窦恶性肿瘤以其预后差和组织学特征的多样性而闻名。虽然预后不良主要是由于疾病晚期,但组织学特征也起着重要作用。因此,准确的病理诊断至关重要。在这里,我们描述了一个63岁的男性慢性左侧鼻塞和左侧鼻出血,他被发现有一个很大的肿块占据了大部分鼻腔,穿过鼻咽部延伸到鼻咽部软腭表面的下方。在手术切除时,发现肿块起源于上颌窦底部,并侵蚀上颌窦内侧壁。病理诊断为ini1完整的低分化复合型癌,横纹肌样表型,肉瘤样癌和鳞状细胞癌灶出现在倒置乳头状瘤内。包括在这个报告是一个详细的描述,这两个病人的医疗过程和病理新颖的鼻窦肿瘤。我们的目的是阐明这种罕见肿瘤的复杂特征,以提高未来的诊断和促进对复杂组织学的鼻窦恶性肿瘤的前瞻性研究。
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引用次数: 0
ALK-Positive Anaplastic Large Cell Lymphoma: A Diagnostic Dilemma for the Otolaryngologist in a Resource Poor Setting. alk阳性间变性大细胞淋巴瘤:一个诊断困境的耳鼻喉科医生在资源贫乏的设置。
IF 0.6 Q4 OTORHINOLARYNGOLOGY Pub Date : 2021-11-01 eCollection Date: 2021-01-01 DOI: 10.1155/2021/3756742
Nicholas Figaro, Rickhi Ramoutar, Rodolfo Arozarena, Dawn Meyers, Solaiman Juman

Anaplastic large cell lymphoma is a rare subtype of non-Hodgkin's lymphoma. The morphological diversity with which its anaplastic cells confer make the diagnosis of this hematological entity extremely challenging to the pathologist in a resource poor setting. We present a case of a 35-year-old male with a nasopharyngeal mass and cervical lymphadenopathy and the adversities faced by out otolaryngology department with obtaining the diagnosis of ALK-positive anaplastic large cell lymphoma.

间变性大细胞淋巴瘤是一种罕见的非霍奇金淋巴瘤亚型。其间变性细胞赋予的形态多样性使这种血液学实体的诊断在资源贫乏的情况下对病理学家极具挑战性。我们报告一个35岁男性鼻咽肿块及颈部淋巴结肿大的病例,以及耳鼻喉科在获得alk阳性间变性大细胞淋巴瘤诊断时所面临的困难。
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引用次数: 0
期刊
Case Reports in Otolaryngology
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