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 [1]. 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 [2].

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 [3], it was considered a cancerous lesion. Also, the biopsy showed high grade intraepithelial neoplasia (HGIN), and thus endoscopic submucosal dissection (ESD) was performed.

Fig. 1 Endoscopic views of lesion. a, b A discolored and laterally spread elevated lesion, 2.5 × 3.0 cm in size, in the greater curvature of the upper gastric body. c Indigo carmine dyeing showed clear demarcation line.

Fig. 2 a, b, c No atrophy and intestinal metaplasia in the background gastric mucosa, and no obvious abnormality or Hp infection was observed in biopsies from these areas.

Fig. 3 a, b, c Narrow-band imaging with magnifying endoscopy showed an irregular papillary-shaped microsurface and irregular microvascular pattern.

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) [4].

Fig. 4 a Histological examination showed tumor glands presented papillary or tubular growth with obvious structural atypia. b The tumor cell density was significantly increased.

Fig. 5 Immunohistochemical analysis showed that the tumor cells were positive for MUC5AC, but negative for P53, MUC6, MUC2, CDX-2, and CD10 expression.

The main etiology of this disease remains unclear; it may be associated with lifestyle, gene mutations, virus infection, or an autoimmune disorder [5]. 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.

Endoscopy_UCTN_Code_CCL_1AB_2AD_3AB

Competing interests

The authors declare that they have no conflict of interest.

^* Authors co-share first authorship

• References

• 1 Mizutani T, Araki H, Saigo C. et al. Endoscopic and pathological characteristics of Helicobacter pylori infection-negative early gastric cancer. Dig Dis 2020; 38: 474-483
• 2 Zhang Z, Zhang C, Zhang Z. Primary hypertrophic osteoarthropathy: an update. Front Med 2013; 7: 60-64
• 3 Muto M, Yao K, Kaise M. et al. Magnifying endoscopy simple diagnostic algorithm for early gastric cancer (MESDA-G). Dig Endosc 2016; 28: 379-393
• 4 Sato C, Hirasawa K, Tateishi Y. et al. Clinicopathological features of early gastric cancers arising in Helicobacter pylori uninfected patients. World J Gastroenterol 2020; 26: 2618-2631
• 5 Yamamoto Y, Fujisaki J, Omae M. et al. Helicobacter pylori-negative gastric cancer: characteristics and endoscopic findings. Dig Endosc 2015; 27: 551-561

Publication History

Article published online:
26 January 2022