A rare case of xanthogranuloma of the stomach masquerading as an advanced stage tumor
© Kinoshita et al; licensee BioMed Central Ltd. 2011
Received: 7 January 2011
Accepted: 2 July 2011
Published: 2 July 2011
Xanthogranuloma of the stomach is an extremely rare disease, and this lesion has only been found to coexist with early gastric cancer in 2 cases in the literature.
We report a case of xanthogranuloma of the stomach combined with early gastric cancer that mimicked an advanced stage tumor. A 65-year-old female was referred to our hospital because of epigastralgia. During a physical examination, a defined abdominal mass was palpable in the region of the left hypochondrium. Imaging studies revealed an advanced gastric cancer, which was suspected of having infiltrated the abdominal wall. Total gastrectomy and resection of the regional lymph node and abdominal wall were performed. Histopathologic examination of the resected specimen demonstrated xanthogranuloma combined with early gastric cancer.
Xanthogranuloma presenting as a form of SMT (submucosal tumor) of the stomach is an extremely rare disease, and diagnosing it preoperatively is difficult. Further accumulation and investigation of this entity is necessary.
Keywordsxanthogranuloma early gastric cancer
Xanthogranuloma was first described by Oberling in 1935 . Although it is known to develop in the gall bladder as xanthogranulomatous cholecystitis, xanthogranuloma of the stomach is an extremely rare disease, and only a few cases have been reported. Hence, we report a case of xanthogranuloma combined with early gastric cancer that mimicked an advanced stage tumor.
Xanthogranuloma is a tumor that is macroscopically characterized by the formation of multiple golden yellow or bright yellow nodules, and histologically, the lesion is predominantly composed of foamy histiocytes mixed with acute and chronic inflammatory cells. The pathogenesis of xanthogranuloma has not been fully established, although it is thought to be a chronic lesion associated with infection, immunological disorders, lipid transport, and lymphatic obstruction .
To the best of our knowledge, only seven cases of xanthogranuloma of the stomach have been reported [2–8], and the coexistence of this lesion with early gastric cancer has only been reported in 2 cases. Our histopathological inspection in these cases did not support continuity between the xanthogranuloma and early gastric cancer. Therefore, it is unclear whether early gastric cancer participates in xanthogranuloma.
Pathologically, stromal tumors such as GIST, myogenetic tumors, and neurogenic tumors account for 54 percent of all SMT, followed by heterotopic pancreas, cyst, lipoma, carcinoid, lymphangioma, and hemangioma . There have been no previous cases of preoperatively diagnosed xanthogranuloma as was found in the current case.
In our case, the gastric xanthogranuloma was preoperatively misdiagnosed as an advanced gastric cancer. This occurred for the following reasons: First, a gastrointestinal endoscopic examination demonstrated an elevated lesion close to the anal side of an ulcerated lesion and a moderately or poorly differentiated adenocarcinoma was detected by the endoscopic biopsy. Second, CT indicated that the elevated lesion had invaded the abdominal wall, and a defined abdominal mass was palpable on physical examination. Therefore, the tumor was recognized as an advanced gastric cancer. Biopsy of the elevated lesion should have been carried out preoperatively to obtain a correct diagnosis in consideration of the coexistence of the two lesions.
We report an extremely rare case of gastric xanthogranuloma combined with early gastric cancer. When we find SMT of the stomach, we should bear in mind not only neoplastic tumors but also inflammatory tumors. Further accumulation and investigation of gastric xanthogranuloma cases is necessary.
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
- Oberling C: Retroperitoneal xanthogranuloma. Am J Cancer. 1935, 23: 477-489.View ArticleGoogle Scholar
- Zafisaona G: Inflammatory fibrous histiocytoma of the stomach. Apropos of a case of xanthogranuloma?. Arch Anat Cytol Pathol. 1987, 35: 149-153.PubMedGoogle Scholar
- Zhang L, Huang X, Li J: Xanthogranuloma of the stomach: a case report. Eur J Surg Oncol. 1992, 18: 293-295.PubMedGoogle Scholar
- Guarino M, Reale D, Micoli G, Tricomi P, Cristofori E: Xanthogranulomatous gastritis: association with xanthogranulomatous cholecystitis. J Clin Pathol. 1993, 46: 88-90. 10.1136/jcp.46.1.88.PubMed CentralView ArticlePubMedGoogle Scholar
- Lespi PJ: Gastric xanthogranuloma (inflammatory malignant fibrohistiocytoma). Case report and literature review. Acta Gastroenterol Latinoam. 1998, 28: 309-310.PubMedGoogle Scholar
- Lai HY, Chen JH, Chen CK, Chen YF, Ho YJ, Yang MD, Shen WC: Xanthogranulomatous pseudotumor of stomach induced by perforated peptic ulcer mimicking a stromal tumor. Eur Radiol. 2006, 16: 2371-2372. 10.1007/s00330-006-0188-3.View ArticlePubMedGoogle Scholar
- Kubosawa H, Yano K, Oda K, Shiobara M, Ando K, Nunomura M, Sarashina H: Xanthogranulomatous gastritis with pseudosarcomatous changes. Pathol Int. 2007, 57: 291-295. 10.1111/j.1440-1827.2007.02089.x.View ArticlePubMedGoogle Scholar
- Aikawa M, Ishii T, Nonaka K, Nakao M, Ishikawa K, Arai S, Kita H, Miyazawa M, Koyama I, Motosugi U, Ban S: A case of gastric xanthogranuloma associated with early gastric cancer. Nippon Shokakibyo Gakkai Zasshi. 2009, 106: 1610-1615.PubMedGoogle Scholar
- Polkowski M: Endoscopic ultrasound and endoscopic ultrasound-guided fine-needle biopsy for the diagnosis of malignant submucosal tumors. Endoscopy. 2005, 37: 635-645. 10.1055/s-2005-861422.View ArticlePubMedGoogle Scholar
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