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超乎寻常的病因:原发性上颌前骨结节结节病

Beyond the Usual Suspects: Primary Premaxilla Sarcoidosis.

作者信息

Mittal Pankhuri, Gupta Brijnandan, Kumar Subodh, Rawat Aakanksha, Singh Anshi

机构信息

Department of ENT, All India Institute of Medical Sciences (AIIMS) Gorakhpur, India.

Department of Pathology, AIIMS Gorakhpur, India.

出版信息

Iran J Otorhinolaryngol. 2024 Jan;36(1):365-369. doi: 10.22038/IJORL.2023.73996.3493.

Abstract

INTRODUCTION

Sarcoidosis is an idiopathic systemic granulomatous disorder that can affect multiple organs, including rare extrapulmonary sites like the premaxilla. This case report presents a rare occurrence of premaxillary sarcoidosis, a condition scarcely reported in medical literature.

CASE REPORT

The patient, a 62-year-old male, presented with a progressively enlarging painless swelling on the right cheek over a three-year period. Despite multiple Fine Needle Aspiration Cytology (FNAC) examinations yielding no conclusive diagnosis, a contrast-enhanced computed tomographic (CT) scan revealed an ill-defined lesion in the premaxillary soft tissue. Biopsy and subsequent excision procedures confirmed the presence of non-caseating granulomas with asteroid bodies, indicative of sarcoidosis. With no systemic involvement and complete excision of the disease, further treatment was not necessary.

CONCLUSION

This case highlights the challenges in diagnosing premaxillary (Extrapulmonary Sarcoidosis) sarcoidosis due to its rarity and resemblance to other dental and maxillofacial conditions and granulomatous lesions. Accurate diagnosis requires a high index of suspicion, multidisciplinary approach, involving clinical assessment, histopathological analysis, and imaging modalities. By deepening our understanding of these uncommon presentations, this report aims to enhance clinical awareness and contribute to improved patient outcomes.

摘要

引言

结节病是一种特发性全身性肉芽肿性疾病,可累及多个器官,包括罕见的肺外部位,如前上颌骨。本病例报告呈现了一例罕见的前上颌骨结节病,这种情况在医学文献中鲜有报道。

病例报告

患者为一名62岁男性,在三年时间里右侧脸颊出现逐渐增大的无痛性肿胀。尽管多次细针穿刺抽吸细胞学检查(FNAC)均未得出明确诊断,但增强计算机断层扫描(CT)显示前上颌骨软组织有一个边界不清的病变。活检及后续切除手术证实存在伴有星状体的非干酪样肉芽肿,提示为结节病。由于无全身受累且疾病已完全切除,无需进一步治疗。

结论

本病例凸显了诊断前上颌骨(肺外结节病)结节病的挑战,因其罕见且与其他口腔颌面部疾病及肉芽肿性病变相似。准确诊断需要高度的怀疑指数,采用多学科方法,包括临床评估、组织病理学分析和影像学检查。通过加深我们对这些罕见表现的理解,本报告旨在提高临床认识并有助于改善患者预后。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bcbf/10800137/1fa035316ddb/ijo-36-365-g001.jpg

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