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A rare helicobacter pylori infection-negative early gastric cancer in a young man with primary hypertrophic osteoarthropathy
Helicobacter pylori (Hp) infection-negative gastric cancer is very rare . We describe a rare early gastric cancer in Hp infection-negative gastric mucosa in a young man with primary hypertrophic osteoarthropathy, which is a rare monogenetic disease .
A 30-year-old man was referred to our hospital for endoscopic examination. Endoscopy revealed a whitish and laterally spread elevated lesion in the greater curvature of the upper gastric body. The size of the lesion was approx. 2.5 × 3.0 cm ([Fig. 1]). No atrophy or intestinal metaplasia was observed in the background gastric mucosa. The biopsy from the background mucosa showed no obvious abnormality ([Fig. 2]). Furthermore, both the serum Hp antibody and urea breath test were negative. Narrow-band imaging (NBI) with magnifying endoscopy revealed a papillary-shaped microsurface and irregular microvessels in the lesion with clear demarcation ([Fig. 3], [Video 1]). According to the vascular and surface pattern classification system , it was considered a cancerous lesion. Also, the biopsy showed high grade intraepithelial neoplasia (HGIN), and thus endoscopic submucosal dissection (ESD) was performed.
Video 1 The endoscopic characteristics of a rare foveolar-type early gastric cancer.
Histologically, the tumor glands presented papillary or tubular growth with obvious structural atypia, and the tumor cell density was significantly increased ([Fig. 4]). Immunohistochemically, the neoplasia area was positive for MU5AC but negative for MUC6, MUC2, CD10, and CDX-2 expression, suggesting that the mucin genotype was gastric. The ki-67 labeling index was 10 %. The expression of p53 protein was negative ([Fig. 5]). Finally, the lesion was diagnosed as a foveolar-type adenoma with HGIN according to the World Health Organization (WHO) classification, and as a well-differentiated adenocarcinoma using the Japanese Classification of Gastric Carcinoma (JCGC) .
The main etiology of this disease remains unclear; it may be associated with lifestyle, gene mutations, virus infection, or an autoimmune disorder . In this case, SLCO2A1 mutation was detected. An SLCO2A1 mutation results in increased circulating PGE2 levels, which can stimulate cell proliferation, suggesting a link between primary hypertrophic osteoarthropathy and tumors. Therefore, endoscopy should be performed to monitor the gastric neoplasia lesions in this subset of patients.
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* Authors co-share first authorship
Article published online:
26 January 2022
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