Bicytopenia and leukoerythroblastosis: a rare initial presentation of signet ring cell gastric adenocarcinoma
Vilma Takayasu; Edna Harumi Goto; Mayra Zanon Casagrande; Paulo Guilherme de Arruda Miranda; Gabriela Bezerra de Freitas Diniz; Micaela Frasson Monteiro; Aloisio Felipe-Silva
Gastric adenocarcinoma is a common neoplasia and is responsible for up to 30% of the overall deaths due to cancer. Advanced disease is mostly characterized by peritoneum, liver, and lung involvement. The spread of the disease to the bone is rare, and bone marrow dissemination is even rarer. In this setting, leukoerythroblastosis may be the initial manifestation of the disease. The authors report the case of a 64-year-old Caucasian man who sought medical care complaining of back pain, weakness, and weight loss. The physical examination revealed pallor, and the laboratory work-up depicted severe anemia and thrombocytopenia; the peripheral blood smear was consistent with leukoerythroblastosis. The ongoing investigation through a bone marrow biopsy showed massive involvement of the bone marrow by a signet ring cell adenocarcinoma. During hospitalization, the patient presented melena, and an upper digestive endoscopy depicted an ulcerated and infiltrative lesion in the cardia, upon which the histological examination revealed a signet ring cell adenocarcinoma. This case highlights the bone marrow invasion represented by bicytopenia and leukoerythroblastosis as the initial manifestation of this histological type of gastric cancer. Although treatment attempts were made with chemotherapy and radiotherapy, the patient died early on, showing the aggressive behavior of this form of tumoral presentation.
1. Ferlay J, Soerjomataram I, Dikshit R, et al. Cancer incidence and mortality worldwide: sources, methods and major patterns in GLOBOCAN 2012. Bray F. Int. J. Cancer. 2015;136(5):E359-86. PMid:25220842. http://dx.doi.org/10.1002/ijc.29210.
2. Pinheiro NC, Rodrigues J, Pereira J, Silva AM. Signet ring cell carcinoma’s myelophthisis. BMJ Case Rep. 2014;2014(1):bcr2014203662. PMid:24623544. http://dx.doi.org/10.1136/bcr-2014-203662.
3. Dittus C, Mathew H, Malek A, Negroiu A. Bone marrow infiltration as the initial presentation of gastric signet ring cell adenocarcinoma. J Gastrointest Oncol. 2014;5(6):E113-6. PMid:25436133.
4. Ekinci AŞ, Bal Ö, Özatlı T, et al. Gastric carcinoma with bone marrow metastasis: a case series. J J Gastric Cancer. 2014;14(1):54-7. PMid:24765538. http://dx.doi.org/10.5230/jgc.2014.14.1.54.
5. Instituto Nacional de Câncer (INCA). Estimativa 2016: incidência de câncer no Brasil. Rio de Janeiro: INCA; 2015.
6. Zilberstein B, Malheiros C, Lourenço LC, et al. Brazilian consensus in gastric cancer: guidelines for gastric cancer in Brazil. ABCD Arq Bras Cir Dig. 2013;26(1):2-6. PMid:23702862. http://dx.doi.org/10.1590/S0102-67202013000100002.
7. Mori W, Adachi Y, Okabe H, et al. An analysis of 799 autopsied cases of malignant tumors: a statistical study of their metastasis. Gan No Rinsho. 1963;9:351-74.
8. Nishidoi H, Koga S. Clinicopathological study of gastric cancer with bone metastasis. Gan To Kagaku Ryoho. 1987;14(5 Pt 2):1717-22. PMid:3592711.
9. Silvestris N, Pantano F, Ibrahim T, et al. Natural history of malignant bone disease in gastric cancer: final results of a multicenter bone metastasis survey. PLoS One. 2013;8(10):e74402. PMid:24204569. http://dx.doi.org/10.1371/journal.pone.0074402.
10. Yamamura Y, Kito T, Yamada E. Clinical evaluation on bone and bone marrow metastasis of gastric carcinoma. Jpn J Gastroenterol Surg. 1985;18(11):2288-93. http://dx.doi.org/10.5833/jjgs.18.2288.
11. Yoshikawa K, Kitaoka H. Bone metastasis of gastric cancer. Jpn J Surg. 1983;13(3):173-6. PMid:6632389. http://dx.doi.org/10.1007/BF02469472.
12. Park JM, Song KY, O JH, Kim WC, Choi MG, Park CH. Bone recurrence after curative resection of gastric cancer. Gastric Cancer. 2013;16(3):362-9. PMid:22961057. http://dx.doi.org/10.1007/s10120-012-0193-y.
13. Iguchi H. Recent aspects for disseminated carcinomatosis of the bone marrow associated with gastric cancer: What has been done for the past, and what will be needed in future? World J Gastroenterol. 2015;21(43):12249-60. PMid:26604634. http://dx.doi.org/10.3748/wjg.v21.i43.12249.
14. Kim HS, Yi SH, Jun HJ, et al. Clinical outcome of gastric cancer patients with bone marrow metastases. Oncology. 2007;73(3-4):192-7. PMid:18418012. http://dx.doi.org/10.1159/000127386.
15. Kwon JY, Yun J, Kim HJ, et al. Clinical outcome of gastric cancer patients with bone marrow metastases. Cancer Res Treat. 2011;43(4):244-9. PMid:22247710. http://dx.doi.org/10.4143/crt.2011.43.4.244.
16. Anner RM, Drewinko B. Frequency and significance of bone marrow involvement by metastatic solid tumors. Cancer. 1977;39(3):1337-44. PMid:912663. http://dx.doi.org/10.1002/1097-0142(197703)39:3<1337::AID-CNCR2820390349>3.0.CO;2-X.
17. Mehdi SR, Bhatt ML. Metastasis of solid tumors in bone marrow: a study from northern India. Indian J Hematol Blood Transfus. 2011;27(2):93-5. PMid:22654299. http://dx.doi.org/10.1007/s12288-011-0069-z.
18. Shiozawa Y, Eber MR, Berry JE, Taichman RS. Bone marrow as a metastatic niche for disseminated tumor cells from solid tumors. Bonekey Rep. 2015;4:689. PMid:26029360. http://dx.doi.org/10.1038/bonekey.2015.57.
19. Kusumoto H, Haraguchi M, Nozuka Y, Oda Y, Tsuneyoshi M, Iguchi H. Characteristic features of disseminated carcinomatosis of the bone marrow due to gastric cancer: the pathogenesis of bone destruction. Oncol Rep. 2006;16(4):735-40. PMid:16969487.
20. Zhou YN, Xu CP, Han B, et al. Expression of E-cadherin and beta-catenin in gastric carcinoma and its correlation with the clinicopathological features and patient survival. World J Gastroenterol. 2002;8(6):987-93. PMid:12439911. http://dx.doi.org/10.3748/wjg.v8.i6.987.
21. Humar B, Blair V, Charlton A, More H, Martin I, Guilford P. E-cadherin deficiency initiates gastric signet ring cell carcinoma in mice and man. Cancer Res. 2009;69(5):2050-6. PMid:19223545. http://dx.doi.org/10.1158/0008-5472.CAN-08-2457.
22. Turkoz FP, Solak M, Kilickap S, et al. Bone Metastasis from Gastric Cancer: The Incidence, Clinicopathological Features, and Influence on Survival. J Gastric Cancer. 2014;14(3):164-72. PMid:25328761. http://dx.doi.org/10.5230/jgc.2014.14.3.164.
24. Nebuloni DR, Mak MP, Souza FH, et al. Modified FLOX as first-line chemotherapy for metastatic colorectal cancer patients in the public health system in Brazil: Effectiveness and cost-utility analysis. Mol Clin Oncol. 2013;1(1):175-9. PMid:24649143.