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伴有非坏死性肉芽肿性炎症和淋巴结病的食管海绵状癌:临床病理特征及诊断挑战

Esophageal Carcinoma Cuniculatum Associated with Non-Necrotizing Granulomatous Inflammation and Lymphadenopathy: Clinicopathologic Features and Diagnostic Challenges.

作者信息

Dick Toni M, El Hag Mohamed, Mallery J Shawn, Amin Khalid

机构信息

Department of Laboratory Medicine and Pathology, University of Minnesota Medical Center, Minneapolis, MN, USA.

Department of Gastroenterology, University of Minnesota Medical Center, Minneapolis, MN, USA.

出版信息

Am J Case Rep. 2018 Jul 5;19:790-795. doi: 10.12659/AJCR.908116.

DOI:10.12659/AJCR.908116
PMID:29973579
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6064193/
Abstract

BACKGROUND Esophageal carcinoma cuniculatum (CC) is an exceptionally rare, well-differentiated squamous cell carcinoma (SCC) with initial microscopic evaluation often yielding inconclusive diagnoses due to its characteristically bland histomorphologic appearance on superficial endoscopic biopsy. This can lead to delayed diagnosis and pose challenges in further management of these cases. CASE REPORT We present the case of a 52-year-old man with symptoms of dysphagia and odynophagia. The initial chest CT scan showed gastroesophageal (GE) junction wall thickening and regional lymphadenopathy. Esophagogastroduodenoscopy (EGD) revealed an esophageal mass, but the mucosal biopsies were inconclusive. Repeat endoscopic biopsies also failed to yield a definitive diagnosis. Under strong clinical suspicion for malignancy, an esophagogastrectomy was performed, which yielded the diagnosis of CC, and the associated enlarged lymph nodes revealed non-necrotizing granulomatous lymphadenitis. CONCLUSIONS Only 15 cases of esophageal CC have been described in the literature. This particular case is unique due to the associated abundant lymphoplasmacytic and granulomatous inflammation and involvement of regional lymph nodes by non-necrotizing granulomas not previously described.

摘要

背景 兔穴状食管癌(CC)是一种极为罕见的高分化鳞状细胞癌(SCC),由于其在浅表内镜活检时具有典型的平淡组织形态学表现,初始显微镜评估往往无法得出明确诊断。这可能导致诊断延迟,并给这些病例的进一步管理带来挑战。病例报告 我们报告一例52岁男性,有吞咽困难和吞咽痛症状。初始胸部CT扫描显示胃食管(GE)交界处壁增厚和区域淋巴结肿大。食管胃十二指肠镜检查(EGD)发现食管肿物,但黏膜活检结果不明确。重复内镜活检也未能得出明确诊断。在强烈怀疑为恶性肿瘤的情况下,进行了食管胃切除术,确诊为CC,相关肿大淋巴结显示为非坏死性肉芽肿性淋巴结炎。结论 文献中仅描述了15例食管CC病例。该特殊病例具有独特性,因为伴有大量淋巴细胞浆细胞和肉芽肿性炎症,且区域淋巴结受累于非坏死性肉芽肿,此前未见相关描述。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/31a8/6064193/79f368008969/amjcaserep-19-790-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/31a8/6064193/3d57250f1ddf/amjcaserep-19-790-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/31a8/6064193/79f368008969/amjcaserep-19-790-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/31a8/6064193/3d57250f1ddf/amjcaserep-19-790-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/31a8/6064193/79f368008969/amjcaserep-19-790-g002.jpg

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Immunoglobulin G4-related disease (IgG4-RD) affecting the esophagus, stomach, and liver.免疫球蛋白G4相关性疾病(IgG4-RD)累及食管、胃和肝脏。
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Endoscopic submucosal dissection in a rare case of carcinoma cuniculatum of the esophagus initially misdiagnosed as benign squamous papilloma.内镜下黏膜剥离术治疗一例罕见的食管漏斗状癌,该病例最初被误诊为良性鳞状乳头状瘤。
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