Abstract
A 62-year-old Thai man with a 2-year history of bilateral lymphedema and an unprovoked left axillary vein thrombosis presented with progressive leg, scrotal, and abdominal swelling, and shortness of breath. He denied any gastrointestinal symptoms. His lymphedema had initially been diagnosed as chronic filariasis due to positive blood tests for anti-filarial antibodies; however, treatment with anti-filarial drugs failed to improve his symptoms. Subsequently, he underwent surgical lymphaticovenular anastomosis with scrotal reduction, which proved to be of limited symptomatic relief. Later investigations revealed bilateral chylothorax and chylous ascites, with the presence of metastatic adenocarcinoma. Histopathological examination of the patient’s skin and scrotum biopsy from his previous surgery revealed invasion of the lymphatics by neoplastic cells with signet ring cell formation. Gastroscopy uncovered a gastric mass, and biopsy confirmed the diagnosis of stage IV gastric adenocarcinoma with signet ring cell. He later received palliative chemotherapy. For the management of chyle leakage, he was prescribed a very low-fat diet and supplemented with parenteral nutrition. Despite treatment, he developed cutaneous metastasis and was transitioned to best supportive care. The patient passed away 14 months after diagnosis.
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References
Thrift AP, El-Serag HB. Burden of gastric cancer. Clin Gastroenterol Hepatol. 2020;18:534–42.
Pernot S, Voron T, Perkins G, et al. Signet-ring cell carcinoma of the stomach: Impact on prognosis and specific therapeutic challenge. World J Gastroenterol. 2015;21:11428–38.
Arnold M, Abnet CC, Neale RE, et al. Global burden of 5 major types of gastrointestinal cancer. Gastroenterology. 2020;159:335-49.e15.
Nagano N, Suzuki M, Tamura K, et al. Refractory chylothorax and lymphedema caused by advanced gastric cancer. Intern Med. 2019;58:3143–8.
Hamid DMHA. Lymphatic Filariasis MyHEALTH Portal: Ministry of Health Malaysia; 2016 [updated 13 May 2016. Available from: http://www.myhealth.gov.my/en/lymphatic-filariasis/.
Riley LE, Ataya A. Clinical approach and review of causes of a chylothorax. Respir Med. 2019;157:7–13.
Shibata K, Kitagawa S, Fujimura M, et al. Chylothorax associated with inflammatory carcinoma. Intern Med. 1998;37:538–41.
Gramlich L, Ireton-Jones C, Miles JM, et al. Essential fatty acid requirements and intravenous lipid emulsions. J Parenter Enter Nutr. 2019;43:697–707.
Mogulkoc N, Onal B, Okyay N, et al. Chylothorax, chylopericardium and lymphoedema–the presenting features of signet-ring cell carcinoma. Eur Respir J. 1999;13:1489–91.
Lanznaster G, Adami M, Crivellaro C, et al. Gastric signet-ring cell carcinoma: unilateral lower extremity lymphoedema as the presenting feature. Sci World J. 2007;7:1189–92.
Majoor CJ, Aliredjo RP, Dekhuijzen PN, et al. A rare cause of chylothorax and lymph edema. J Thorac Oncol. 2007;2:247–8.
Kayacan O, Karnak D, Ayşe Can B, et al. Gastric signet-ring cell adenocarcinoma presenting with left arm deep-vein thrombosis and bilateral chylothorax. Clin Appl Thromb Hemost. 2008;14:476–80.
Devaraj U, Ramachandran P, Correa M, et al. Chylothorax in gastric adenocarcinoma: a case report and systematic review of the English literature. Lung India. 2014;31:47–52.
Wu J, Lv L, Zhou K, et al. Chylothorax and lymphedema as the initial manifestations of gastric carcinoma: a case report and review of the literature. Oncol Lett. 2016;11:2835–8.
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We would like to thank the patient, his wife, and the medical team who were involved in the care of the patient.
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SW and PCS conceived the study, treated the patient, drafted, and revised the manuscript. NN, KK, WS, SP, PB, AP, PP, and DW treated the patient and revised the manuscript. All authors reviewed and approved the final manuscript before submission.
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This case report was reviewed and approved by the Human Research Ethics Committee, Faculty of Medicine Ramathibodi Hospital, Mahidol University, approval number COA. MURA2022/137.
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Wongrukmit, S., Ngamphaiboon, N., Kiranantawat, K. et al. Unusual manifestation of gastric adenocarcinoma presenting with lymphedema, chylothorax, and chylous ascites. Clin J Gastroenterol 16, 822–828 (2023). https://doi.org/10.1007/s12328-023-01851-5
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DOI: https://doi.org/10.1007/s12328-023-01851-5