{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,22]],"date-time":"2026-10-22T16:55:25Z","timestamp":1792688125659,"version":"4.5.0"},"reference-count":17,"publisher":"Walter de Gruyter GmbH","issue":"1","license":[{"start":{"date-parts":[[2022,3,1]],"date-time":"2022-03-01T00:00:00Z","timestamp":1646092800000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/3.0"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,3,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>We herein report the first case of lupus-related protein-losing enteropathy associated with pseudo-pseudo Meigs\u2019 syndrome. Lupus-related protein-losing enteropathy and pseudo-pseudo Meigs\u2019 syndrome are extremely rare complications in patients with systemic lupus erythematosus, Both have a similar clinical course characterized by producing marked ascites, and respond to steroids in typical cases. However, in our case, steroid monotherapy was inadequate and the addition of hydroxychloroquine was effective for their treatment. Furthermore, no reports have previously confirmed elevated CA 125 levels with lupus-related protein-losing enteropathy or increased 99mTc-HSA activity with pseudo-pseudo Meigs\u2019 syndrome. In addition, we are the first to report an evaluation of the histopathology of lupus-related protein-losing enteropathy. Previously reported cases have been described as being caused by either pseudo-Meigs\u2019s syndrome or lupus-related protein-losing enteropathy as the cause of the rare pathology that causes marked pleural effusion and ascites in patients with systemic lupus erythematosus, but it has not been evaluated whether the other is co-occurring. Our case highlights that there is a potential case of overlapping lupus-related protein-losing enteropathy and pseudo-Pseudo-Meigs\u2019s syndrome. Furthermore, it is possible that patients with marked ascites with elevated CA 125 levels were mistakenly diagnosed with Meigs\u2019s syndrome or pseudo-Meigs\u2019s syndrome associated with malignant or benign ovarian tumors and underwent surgery. Clinicians should not forget SLE with pseudo-Pseudo-Meigs\u2019s syndrome as one of the differential diagnoses for marked ascites with elevated CA 125 levels.<\/jats:p>","DOI":"10.2478\/rjim-2021-0032","type":"journal-article","created":{"date-parts":[[2021,8,1]],"date-time":"2021-08-01T19:07:12Z","timestamp":1627844832000},"page":"85-89","source":"Crossref","is-referenced-by-count":4,"title":["Lupus-related protein-losing enteropathy associated with pseudo-pseudo Meigs\u2019 syndrome and successfully treated with hydroxychloroquine"],"prefix":"10.2478","volume":"60","author":[{"given":"Taro","family":"Horino","sequence":"first","affiliation":[{"name":"Department of Endocrinology, Metabolism and Nephrology , Kochi Medical School, Kochi University , Kohasu, Oko-cho, Nankoku , Kochi , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masami","family":"Ogasawara","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Metabolism and Nephrology , Kochi Medical School, Kochi University , Kohasu, Oko-cho, Nankoku , Kochi , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Takeshi","family":"Kashio","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Metabolism and Nephrology , Kochi Medical School, Kochi University , Kohasu, Oko-cho, Nankoku , Kochi , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Satoshi","family":"Inotani","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Metabolism and Nephrology , Kochi Medical School, Kochi University , Kohasu, Oko-cho, Nankoku , Kochi , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masayuki","family":"Ishihara","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Kochi Medical School , Kochi University , Kohasu, Oko-cho, Nankoku , Kochi , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroshi","family":"Ohnishi","sequence":"additional","affiliation":[{"name":"Department of Hematology and Respiratory Medicine , Kochi Medical School, Kochi University , Kohasu, Oko-cho, Nankoku , Kochi , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masahiro","family":"Komori","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology , Kochi Medical School, Kochi University , Kohasu, Oko-cho, Nankoku , Kochi , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Osamu","family":"Ichii","sequence":"additional","affiliation":[{"name":"Laboratory of Anatomy , Department of Basic Veterinary Sciences, Faculty of Veterinary Medicine , Hokkaido University , Kita 18, Nishi 9, Kita-Ku , Sapporo , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yoshio","family":"Terada","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Metabolism and Nephrology , Kochi Medical School, Kochi University , Kohasu, Oko-cho, Nankoku , Kochi , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"374","published-online":{"date-parts":[[2022,3,17]]},"reference":[{"key":"2026042915235923684_j_rjim-2021-0032_ref_001","unstructured":"1. ARINGER M., COSTENBADER K., DAIKH D., BRINKS R., MOSCA M., RAMSEY-GOLDMAN R., et al. 2019 European League Against Rheumatism\/American College of Rheumatology classification criteria for systemic lupus erythematosus. Ann Rheum Dis. 2019;78(9):1151\u20139.10.1136\/annrheumdis-2018-21481931383717"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_002","doi-asserted-by":"crossref","unstructured":"2. AL-MOGAIREN SM. Lupus protein-losing enteropathy (LUPLE): a systematic review. Rheumatol Int. 2011;31(8):995\u20131001.10.1007\/s00296-011-1827-921344315","DOI":"10.1007\/s00296-011-1827-9"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_003","doi-asserted-by":"crossref","unstructured":"3. TIALMA WA. Ascites, pleural effusion, and CA 125 elevation in an SLE patient, either a Tjalma syndrome or, due to the migrated Filshie clips, a pseudo-Meigs syndrome. Gynecol Oncol. 2005;97(1):288\u201391.10.1016\/j.ygyno.2004.12.02215790480","DOI":"10.1016\/j.ygyno.2004.12.022"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_004","doi-asserted-by":"crossref","unstructured":"4. HOCHBERG MC. Updating the American College of Rheumatology revised criteria for the classification of systemic lupus erythematosus. Arthritis Rheum. 1997;40(9):1725.10.1002\/art.17804009289324032","DOI":"10.1002\/art.1780400928"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_005","unstructured":"5. PETRI M., ORBAI AM., ALARCON GS., GORDON C., MERRILL JT., FORTIN PR., et al. Derivation and validation of the Systemic Lupus International Collaborating Clinics classification criteria for systemic lupus erythematosus. Arthritis Rheum. 2012;64(8):2677\u201386.10.1002\/art.34473340931122553077"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_006","doi-asserted-by":"crossref","unstructured":"6. YOSHIDA M., MIYATA M., SAKA M., SASAJIMA T., SATO H., TAKIGUCHI J., et al. Protein-losing enteropathy exacerbated with the appearance of symptoms of systemic lupus erythematosus. Intern Med. 2001;40(5):449\u201353.10.2169\/internalmedicine.40.44911393422","DOI":"10.2169\/internalmedicine.40.449"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_007","doi-asserted-by":"crossref","unstructured":"7. CHUNG U., OKA M., NAKAGAWA Y., NISHISHITA T., SEKINE N., TANAKA Y., et al. A patient with protein-losing enteropathy associated with systemic lupus erythematosus. Intern Med. 1992;31(4):521\u20134.10.2169\/internalmedicine.31.5211633362","DOI":"10.2169\/internalmedicine.31.521"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_008","doi-asserted-by":"crossref","unstructured":"8. WERNER DE CASTRO GR., APPENZRLLER S., BERTOLO MB., COSTALLAT LT. Protein-losing enteropathy associated with systemic lupus erythematosus: response to cyclophosphamide. Rheumatol Int. 2005;25(2):135\u20138.10.1007\/s00296-004-0483-815249982","DOI":"10.1007\/s00296-004-0483-8"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_009","doi-asserted-by":"crossref","unstructured":"9. TANSIR G., KUMAR P., PIUS A., SUNNY SK., SONEJA M. Pseudo-pseudo Meigs\u2019 syndrome: a rare presentation of systemic lupus erythematosus. Reumatismo. 2019;71(2):108\u201312.10.4081\/reumatismo.2019.114031309785","DOI":"10.4081\/reumatismo.2019.1140"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_010","unstructured":"10. YANG Z., LIANG Y., LI C., ZHONG R. Serum CA125 elevation is independently associated with serositis in SLE patients. Clin Exp Rheumatol. 2012;30(1):93\u20138."},{"key":"2026042915235923684_j_rjim-2021-0032_ref_011","doi-asserted-by":"crossref","unstructured":"11. ZEIMET AG., MARTH C., OFFNER FA., OBRIST P, UHL-STEIDL M., FEICHTINGER H. Human peritoneal mesothelial cells are more potent than ovarian cancer cells in producing tumor marker CA-125. Gynecol Oncol. 1996;62(3):384\u20139.10.1006\/gyno.1996.02538812537","DOI":"10.1006\/gyno.1996.0253"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_012","doi-asserted-by":"crossref","unstructured":"12. LI T, XIE QB. A case report of pseudo-pseudo Meigs\u2019 syndrome. Chin Med J (Engl). 2019;132(12):1497\u20138.10.1097\/CM9.0000000000000231662932231205113","DOI":"10.1097\/CM9.0000000000000231"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_013","doi-asserted-by":"crossref","unstructured":"13. FAVA A, PETRI M. Systemic lupus erythematosus: diagnosis and clinical management. J Autoimmun. 2019;96:1\u201313.10.1016\/j.jaut.2018.11.001631063730448290","DOI":"10.1016\/j.jaut.2018.11.001"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_014","unstructured":"14. Health and Labor Sciences Research Grant Subsidy for Intractable Disease Policy Research. Project, Research on autoimmune diseases, Japan college of Rheumatology. Guideline for the management of systemic lupus erythematosus 2019;2019:226-30 (in Japanese)"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_015","doi-asserted-by":"crossref","unstructured":"15. FANOURIAKIS A., KOSTOPOULOU M., ALUNNO A., ARINGER M., BAJEMA I., BOLETIS JN. 2019 update of the EULAR recommendations for the management of systemic lupus erythematosus. Ann Rheum Dis. 2019;78(6):736\u201345.10.1136\/annrheumdis-2019-21508930926722","DOI":"10.1136\/annrheumdis-2019-215089"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_016","doi-asserted-by":"crossref","unstructured":"16. HANAOKA H., IIDA H., KIYOKAWA T., TAKAKUWA Y., KAWAHATA K. Hydroxychloroquine improves the disease activity and allows the reduction of the corticosteroid dose regardless of background treatment in Japanese patients with systemic lupus erythematosus. Intern Med. 2019;58(9):1257\u201362.10.2169\/internalmedicine.1999-18654322930626831","DOI":"10.2169\/internalmedicine.1999-18"},{"key":"2026042915235923684_j_rjim-2021-0032_ref_017","doi-asserted-by":"crossref","unstructured":"17. MIYAGAWA I., NAKANO K., NAKAYAMADA S., IWATA S., HANAMI K., FUKUYO S. Effectiveness and safety of hydroxychloroquine therapy with or without corticosteroid in patients with systemic lupus erythematosus. Int J Rheum Dis. 2019;22(3):434\u201342.10.1111\/1756-185X.1338730338639","DOI":"10.1111\/1756-185X.13387"}],"container-title":["Romanian Journal of Internal Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/reference-global.com\/pdf\/10.2478\/rjim-2021-0032","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,29]],"date-time":"2026-04-29T19:13:24Z","timestamp":1777490004000},"score":1,"resource":{"primary":{"URL":"https:\/\/reference-global.com\/article\/10.2478\/rjim-2021-0032"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,3,1]]},"references-count":17,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2022,3,17]]},"published-print":{"date-parts":[[2022,3,1]]}},"alternative-id":["10.2478\/rjim-2021-0032"],"URL":"https:\/\/doi.org\/10.2478\/rjim-2021-0032","relation":{},"ISSN":["2501-062X"],"issn-type":[{"value":"2501-062X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,3,1]]}}}