Liu Xiuli, Yang Dennis, Zhang Xuefeng, Oduntan Olusola
Department of Pathology, Immunology, and Laboratory Medicine, College of Medicine, University of Florida, Gainesville, FL, USA.
Department of Gastroenterology, Hepatology, and Nutrition, College of Medicine, University of Florida, Gainesville, FL, USA.
Gastroenterology Res. 2020 Feb;13(1):44-51. doi: 10.14740/gr1262. Epub 2020 Feb 1.
Esophageal carcinoma cuniculatum is a rare variant of squamous cell carcinoma characterized by a unique and common histologic pattern including hyperkeratosis, acanthosis, dyskeratosis, deep keratinization, intraepithelial neutrophils, neutrophilic microabscess, focal cytologic atypia, koilocyte-like cells, and keratin-filled cyst/burrows observed in the resection specimens. Preoperative diagnosis can be extremely difficult. A semiquantitative histologic scoring system has been previously proposed for mucosal biopsies, which has been associated with improved diagnostic yield. However, this histologic schema for the diagnosis of carcinoma cuniculatum has not been applied prospectively. Herein, we describe two cases of esophageal carcinoma cuniculatum in patients presenting with progressive dysphagia and esophageal mass. Presurgical endoscopic mucosal biopsies showed features consistent with carcinoma cuniculatum, and a preoperative diagnosis was achieved by applying the aforementioned semiquantitative histologic schema. Both patients underwent neoadjuvant chemoradiation followed by esophagectomy. Both esophagectomy specimens showed residual adventitia-invading carcinoma cuniculatum, negative lymph nodes, marked tumor regression, and an exuberant histiocytic and giant response. To our best knowledge, these represent the first two cases of esophageal carcinoma cuniculatum diagnosed by applying this semiquantitative histologic schema to mucosal biopsies. Large studies are needed to further confirm these preliminary findings and validate this histologic scoring system.
食管穴状癌是鳞状细胞癌的一种罕见变体，其特征是具有独特且常见的组织学模式，包括角化过度、棘层肥厚、异常角化、深部角化、上皮内中性粒细胞、嗜中性微脓肿、局灶性细胞学异型性、空泡细胞样细胞，以及在切除标本中观察到的充满角蛋白的囊肿/隧道。术前诊断可能极其困难。此前已提出一种用于黏膜活检的半定量组织学评分系统，该系统与提高诊断率相关。然而，这种用于诊断穴状癌的组织学模式尚未被前瞻性应用。在此，我们描述了两例表现为进行性吞咽困难和食管肿物的食管穴状癌患者。术前内镜黏膜活检显示出与穴状癌一致的特征，通过应用上述半定量组织学模式实现了术前诊断。两名患者均接受了新辅助放化疗，随后进行了食管切除术。两份食管切除标本均显示残留有侵犯外膜的穴状癌、阴性淋巴结、明显的肿瘤退缩以及旺盛的组织细胞和巨细胞反应。据我们所知，这是通过将这种半定量组织学模式应用于黏膜活检诊断出的首例两例食管穴状癌病例。需要进行大型研究以进一步证实这些初步发现并验证这种组织学评分系统。
