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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 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 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 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
Alobar Holoprosencephaly with Cebocephaly in a Neonate Born to an HIV-Positive Mother in Eastern Uganda. 乌干达东部一名艾滋病毒阳性母亲所生的新生儿无前脑畸形伴头畸形。
IF 0.6 Pub Date : 2021-10-25 eCollection Date: 2021-01-01 DOI: 10.1155/2021/7282283
Franck Katembo Sikakulya, Sonye Magugu Kiyaka, Robert Masereka, Robinson Ssebuufu

Background: Holoprosencephaly (HPE) is a rare cerebrofacial abnormality resulting from the complete or partial failure of the diverticulation and cleavage of the primitive forebrain. It has an incidence at birth of 1:16000. Case Presentation. We report a case of a 2600 g newborn female delivered by an HIV-infected mother in whom an antenatal ultrasound scan at 34 weeks' gestation reported features of fetal alobar holoprosencephaly. The neonate was born with cebocephaly, a monkey-like head, and did not survive for more than 30 minutes following delivery by caesarian section despite oxygen therapy.

Conclusion: Alobar HPE with cebocephaly remains incompatible with life. In this resource-limited setting, the diagnosis was made clinically, and only an ultrasound scan was performed to confirm the diagnosis. Chromosomal analysis could have given more information.

背景:全前脑畸形(HPE)是一种罕见的脑面部异常,由原始前脑的憩室和卵裂完全或部分失败引起。它在出生时的发病率是1:16000。案例演示。我们报告了一个2600克的新生儿由艾滋病毒感染的母亲分娩的情况下,产前超声扫描在妊娠34周报告胎儿无前脑畸形的特征。这名新生儿出生时就有头畸形,长得像猴子的头,尽管进行了氧气治疗,但在剖腹产后没有存活超过30分钟。结论:Alobar型HPE伴头畸形仍与生活不相容。在这种资源有限的情况下,诊断是在临床上做出的,只有超声扫描来确认诊断。染色体分析可以提供更多的信息。
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引用次数: 3
Deep Neck Infection: Atypical Presentation of Papillary Thyroid Cancer. 深颈部感染:甲状腺乳头状癌的不典型表现。
IF 0.6 Pub Date : 2021-10-18 eCollection Date: 2021-01-01 DOI: 10.1155/2021/1479201
Apichana Mahattanapreut, Rangsima Aroonroch, Chalermchai Chintrakarn, Chutintorn Sriphrapradang

Deep neck infection is defined as an infectious process in the potential spaces and fascial plane of the neck which may result in a fatal complication. Prompt drainage and broad-spectrum antibiotics are the mainstays of treatment. Deep neck infection as the initial presentation of primary head and neck cancer is not common. Nevertheless, head and neck squamous cell carcinoma is the most common primary head and neck cancer, which could present with cervical metastasis and subsequently becomes infected. Papillary thyroid cancer has a naturally indolent course, and most patients present with a thyroid nodule. However, deep neck infection could be an uncommon presentation of papillary thyroid cancer which may obscure the diagnosis of underlying malignancy. This case report aims to present a rare presentation of papillary thyroid cancer which needs meticulous evaluation. Moreover, the pathological examination should be performed in all cases of deep neck infection for early detection and management of underlying papillary thyroid cancer.

深颈部感染是指颈部潜在间隙和筋膜平面的感染过程,可导致致命的并发症。及时引流和广谱抗生素是治疗的主要手段。深颈部感染作为原发性头颈部癌的最初表现并不常见。然而,头颈部鳞状细胞癌是最常见的原发性头颈部癌症,它可能出现宫颈转移,随后发生感染。甲状腺乳头状癌病程自然无痛,多数患者表现为甲状腺结节。然而,深颈部感染可能是一种罕见的乳头状甲状腺癌的表现,这可能会掩盖潜在恶性肿瘤的诊断。本病例报告的目的是提出一个罕见的表现乳头状甲状腺癌,需要细致的评估。此外,所有深颈部感染病例都应进行病理检查,以便早期发现和处理潜在的甲状腺乳头状癌。
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引用次数: 1
A Case of Nasopharyngeal Mycobacteriosis with Bony Erosion of the External Skull Base. 鼻咽分枝杆菌病合并外颅底骨侵蚀1例。
IF 0.6 Pub Date : 2021-10-15 eCollection Date: 2021-01-01 DOI: 10.1155/2021/7500273
Kohei Matsuo, Satoshi Tanaka, Masayuki Sakata, Hiroki Takeda, Akihiro Nagata, Masashi Mori, Rie Ito, Yoshifumi Yamamoto, Kiyonobu Ueno, Atsuhiko Uno

Primary nasopharyngeal mycobacteriosis is a rare disease. We present a case in which skull base bone erosion appeared and was alleviated during the course of the treatment. Bone complications occur in osteoarticular mycobacteriosis, but their occurrence in primary nasopharyngeal mycobacteriosis has not been reported. A 77-year-old immunocompromised Asian woman presented with a right occipitotemporal headache. An ulcerative mass covered with a thick yellowish discharge was found in the roof and posterior walls of the right nasopharynx. Because histopathological examination indicated the presence of mycobacterial infection, we began using antituberculosis medication for the treatment because of the possibility of primary nasopharyngeal tuberculosis. However, this was followed by glossopharyngeal and vagus nerve paralysis. Computed tomography (CT) showed a diffuse enhancing mucosal irregularity in the nasopharynx with bony erosion of the external skull base. Deep tissue biopsy was repeated to differentiate it from malignant lesions, and drainage of pus from the right nasopharynx was confirmed. Subsequently, the headache, neurological findings, and the yellowish discharge disappeared, and the bony erosion of the external skull base was alleviated. Surgical intervention should also be considered for drug-resistant mycobacteriosis. We concluded that mycobacteriosis should also be considered apart from carcinoma even if CT shows a diffuse enhancing mucosal irregularity with bone destruction in the nasopharynx.

原发性鼻咽分枝杆菌病是一种罕见的疾病。我们提出一个病例,其中颅底骨侵蚀出现,并在治疗过程中得到缓解。骨并发症发生在骨关节分枝杆菌病,但其发生在原发性鼻咽分枝杆菌病尚未见报道。77岁免疫功能低下的亚洲女性表现为右侧枕颞头痛。在右鼻咽顶部和后壁发现一溃疡性肿块,上面有厚的淡黄色分泌物。由于组织病理学检查显示存在分枝杆菌感染,我们开始使用抗结核药物进行治疗,因为可能是原发性鼻咽结核。然而,紧随其后的是舌咽神经和迷走神经麻痹。计算机断层扫描(CT)显示鼻咽部弥漫性强化粘膜不规则,外颅底骨侵蚀。反复进行深部组织活检以鉴别其与恶性病变,并证实右侧鼻咽部有脓渗出。随后,头痛、神经学症状和淡黄色分泌物消失,外颅底骨侵蚀减轻。对于耐药分枝杆菌病也应考虑手术干预。我们的结论是,即使CT显示鼻咽部弥漫性强化粘膜不规则并伴有骨破坏,也应考虑分枝杆菌病。
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引用次数: 0
Arteriovenous Malformation in the Auricle in a 59-Year-Old Woman. 59岁女性耳廓动静脉畸形1例。
IF 0.6 Pub Date : 2021-10-08 eCollection Date: 2021-01-01 DOI: 10.1155/2021/7438571
Junhui Jeong

Arteriovenous malformation (AVM) is a vascular lesion with a direct communication between an artery and a vein without a capillary system. AVM primarily occurs in the intracranial area, but can also occur in the extracranial area. If there is a tender mass with pulsation or recurrent bleeding in the auricle, AVM should be considered even though it rarely occurs in the auricle. AVM in the ear should be managed carefully because the skin is thin in the ear, the cartilage could be involved, and progressive growth or inappropriate management could cause bleeding, infection, and cosmetic problems such as deformity. I present a case of a 59-year-old woman with AVM in the auricle.

动静脉畸形(AVM)是一种动脉和静脉之间没有毛细血管系统直接交流的血管病变。AVM主要发生在颅内,但也可发生在颅外。如果有一个柔软的肿块与搏动或反复出血在耳廓,应考虑AVM,即使它很少发生在耳廓。耳内AVM应谨慎处理,因为耳内皮肤薄,可能累及软骨,进行性生长或处理不当可能导致出血、感染和畸形等美容问题。我报告一个59岁女性耳廓AVM的病例。
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引用次数: 1
Survival of Toddler with Aortoesophageal Fistula after Button Battery Ingestion. 儿童误食纽扣电池后主动脉食管瘘的存活率。
IF 0.6 Pub Date : 2021-10-05 eCollection Date: 2021-01-01 DOI: 10.1155/2021/5557054
Hannah Gibbs, Rishabh Sethia, Patrick I McConnell, Jennifer H Aldrink, Toshiharu Shinoka, Kent Williams, Kris R Jatana
Button batteries (BBs) are found in many households and are a source of esophageal foreign body in the pediatric population. Upon ingestion, significant caustic injury can occur within 2 hours leading to tissue damage and severe, potentially fatal sequelae. Aortoesophageal fistula (AEF) is a rare complication that nearly always results in mortality. We report a rare case of a toddler who developed an AEF after BB ingestion and survived following staged aortic repair. There should be a high index of suspicion for this complication with the history of BB ingestion and presence of hematemesis, hemoptysis, or melena.
纽扣电池(BBs)在许多家庭中被发现,是儿童食道异物的来源。摄入后,可在2小时内造成严重的腐蚀性损伤,导致组织损伤和严重的、可能致命的后遗症。主动脉食管瘘(AEF)是一种罕见的并发症,几乎总是导致死亡。我们报告一个罕见的情况下,一个幼儿谁发展为AEF后,摄取BB和生存后,分阶段主动脉修复。有BB摄入史和出现呕血、咯血或黑黑的患者应高度怀疑该并发症。
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引用次数: 5
A Case of Amelanotic Malignant Melanoma of the Lingual Base That Was Diagnosed Based on a Biopsy of Late Metastasis to a Lumbar Vertebra after Being Misdiagnosed as HPV-Positive Oropharyngeal Anterior Wall Squamous Cell Carcinoma. 1例口咽前壁鳞状细胞癌误诊为HPV阳性,经腰椎晚期转移活检诊断为舌根无毛恶性黑色素瘤。
IF 0.6 Pub Date : 2021-09-30 eCollection Date: 2021-01-01 DOI: 10.1155/2021/7139280
Takumi Okuda, Shinsuke Ide, Kei Kajihara, Tetsuya Tono

We report a case of amelanotic malignant melanoma (AMM) in a 66-year-old female. AMM of the lingual base was diagnosed based on a biopsy of late metastasis to the bone marrow of the L4 lumbar vertebra. The patient was initially treated with chemoradiotherapy after being misdiagnosed with poorly differentiated human papillomavirus- (HPV-) related squamous cell carcinoma of the oropharyngeal anterior wall. p16 immunostaining is used to diagnose HPV-related oropharyngeal cancer. However, while p16 expression is used as a surrogate marker of HPV infection, it is important to be aware that p16 protein overexpression can also be caused by other factors. Malignant melanoma is known to express the p16 protein. Morphologically differentiating between AMM and poorly differentiated squamous cell carcinoma based on hematoxylin-eosin staining is difficult. Therefore, in cases that are pathologically diagnosed as p16-positive poorly differentiated oropharyngeal squamous cell carcinoma, it is important to rule out AMM.

我们报告一例无黑色素瘤恶性黑色素瘤(AMM)在一个66岁的女性。舌底AMM的诊断是基于对L4腰椎骨髓晚期转移的活检。在被误诊为低分化人乳头瘤病毒(HPV)相关口咽前壁鳞状细胞癌后,患者最初接受放化疗。p16免疫染色用于hpv相关口咽癌的诊断。然而,虽然p16表达被用作HPV感染的替代标志物,但重要的是要意识到p16蛋白过表达也可能由其他因素引起。恶性黑色素瘤已知表达p16蛋白。基于苏木精-伊红染色在形态学上区分AMM和低分化鳞状细胞癌是困难的。因此,在病理诊断为p16阳性低分化口咽鳞状细胞癌的病例中,排除AMM很重要。
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引用次数: 1
Presence of Two Separate Hairy Polyps with Meningothelial Elements in a 15-Month-Old Child. 一个15个月大的婴儿出现两个独立的毛状息肉并伴有脑膜上皮成分。
IF 0.6 Pub Date : 2021-09-21 eCollection Date: 2021-01-01 DOI: 10.1155/2021/1844244
Ahmad Saeed A Alghamdi, Nasser Almutairi, Ghassan Alokby
Hairy polyps are benign embryological tumors of the head and neck region that are derived from two germinal layers, the ectoderm and mesoderm. At an incidence of 1 : 40000, hairy polyps are considered rare growths. Presenting symptoms of hairy polyps can vary greatly depending on the size and location of the tumor. To the best of our knowledge, our reported case is the first in the literature to highlight an extremely rare occurrence of two separate hairy polyps being simultaneously present in one patient, in the clivus and the nasion, with the presence of meningothelial cells within both tumors, histopathologically. With surgical resection as the management of choice, the approach of surgery differs greatly depending on many factors. Since the patient in our reported case had a cleft palate, we used a combined transnasal and transoral approach to fully release the clival mass and a direct skin incision for the nasion mass. Recurrence after complete surgical resection is rare, and if happens, it should rise suspicion of histopathologically misdiagnosed tumor. We amplified the importance of advanced radiological investigations along with proper multidisciplinary teamwork to exclude CNS connections and other histologically malignant tumors and to early pick up a possible simultaneous lesion.
毛状息肉是头颈部的良性胚胎肿瘤,起源于两个生发层,外胚层和中胚层。毛状息肉的发病率为1:40 000,被认为是罕见的生长。根据肿瘤的大小和位置不同,毛状息肉的症状也会有很大的不同。据我们所知,我们报告的病例是文献中第一个强调在一个患者中同时出现两个独立的毛状息肉的极其罕见的病例,在斜坡和鼻腔,在两个肿瘤中都存在脑膜上皮细胞,组织病理学。手术切除作为治疗的选择,手术的方法因许多因素而有很大的不同。由于我们报告的病例患者有腭裂,我们采用经鼻和经口联合入路来完全释放斜坡肿块,并直接皮肤切口切除鼻腔肿块。完全手术切除后复发是罕见的,如果发生,应引起组织病理学误诊肿瘤的怀疑。我们强调了先进的放射学检查以及适当的多学科合作的重要性,以排除中枢神经系统连接和其他组织学上的恶性肿瘤,并早期发现可能同时发生的病变。
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引用次数: 0
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Case Reports in Otolaryngology
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