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Rheumatology International

, Volume 39, Issue 7, pp 1285–1289 | Cite as

Elevated CA-125 in IgG4 mesenteritis: a red herring or a disease biomarker? Case report and literature review

  • Mailis Michaud Maturana
  • Ismini Panayotidis
  • Savvas PsarelisEmail author
  • George Nakos
  • Elena Nikiphorou
Cases with a Message
  • 170 Downloads

Abstract

Mesenteric panniculitis (MP) is a rare chronic disease characterized by inflammation and subsequently fibrosis of adipose tissue of the omentum. Only recently it has been associated with IgG4-related disease. Cancer antigen 125 (CA-125) is a high-molecular mass glycoprotein, traditionally associated with ovarian cancer, although it can be elevated in other conditions. Herein we describe a case of a 56-year-old man with IgG4 related mesenteric panniculitis associated with very high levels of CA-125 at the onset of disease. The CA-125 levels corresponded to clinical disease activity and improved with steroid therapy and rituximab. A literature review was performed concerning possible association of MP, IgG4-related disease and CA-125. The review of literature suggests that high levels of CA-125 can be raised in non-malignant, inflammatory conditions including IgG4-related mesenteritis and can improve with treatment.

Keywords

Panniculitis, peritoneal CA-125 antigen Mesentery Peritoneum Inflammation Immunoglobulin G Biomarkers, tumor 

Notes

Acknowledgements

The authors thank Aristi Kourri (radiologist) for providing the images.

Author contributions

SP and GN were directly involved in the care of the patient. SP and EN conceived the idea to write the specific case as a case report and literature review. MMM and IP designed and executed the literature search, and drafted the first version of the manuscript with guidance from EN and SP. All authors discussed the results and commented on the manuscript.

Funding

No specific funding was received from any bodies in the public, commercial or not-for-profit sectors to carry out the work described in this manuscript.

Compliance with ethical standards

Conflict of interest

The authors have declared no conflicts of interest.

Informed consent

The patient consented to publication of his case.

References

  1. 1.
    Moss EL, Hollingworth J, Reynolds TM (2005) The role of CA125 in clinical practice. J Clin Pathol 58(3):308–312CrossRefGoogle Scholar
  2. 2.
    Minato H, Shimizu J, Arano Y, Saito K, Masunaga T, Sakashita T et al (2012) IgG4-related sclerosing mesenteritis: a rare mesenteric disease of unknown etiology. Pathol Int 62(4):281–286CrossRefGoogle Scholar
  3. 3.
    Stone JH, Zen Y, Deshpande V (2012) IgG4-related disease. N Engl J Med [Internet] 366(6):539–551.  https://doi.org/10.1056/NEJMra1104650 (Massachusetts Medical Society)CrossRefGoogle Scholar
  4. 4.
    Tong X, Bai M, Wang W, Han Q, Tian P, Fan H (2017 Aug) IgG4-related disease involving polyserous effusions with elevated serum interleukin-6 levels: a case report and literature review. Immunol Res US 65(4):944–950CrossRefGoogle Scholar
  5. 5.
    Khosla M, Le N, Harry R, Kim HJ, Sachdev M (2016) Autoimmune pancreatitis masquerading as malignancy. In: 81st Annual Scientific Meeting of the American College of Gastroenterology. Nature Publishing Group, Las VegasGoogle Scholar
  6. 6.
    Thomas AS, Bunin V, Powell S (2015) Autoimmune hepatitis with primary sclerosing cholangitis and co-existent necrotizing myopathy: a rare association. In: 80th Annual Scientific Meeting of the American College of Gastroenterology. Nature Publishing Group, HonoluluGoogle Scholar
  7. 7.
    Zhou J, Li X, Zeng Q (2014) IgG4-related lung disease with atypical CT imaging: a case report. J Thorac Dis 6(12):E276–E280Google Scholar
  8. 8.
    Fengqing W, Qiang Y, Feng G, Zehao Z, Yuefei M, Jing C et al (2014) Serum trypsin and TCR as novel markers for predicting disease activity in IgG4-related disease. Cent Eur J Immunol 39(2):193–197CrossRefGoogle Scholar
  9. 9.
    Cheng X, Zhou D, Wei J, Zheng S (2013) Regional portal hypertension, systemic lymphadenopathy, and splenomegaly associated with autoimmune pancreatitis. Clin Res Hepatol Gastroenterol 37(3):75–80CrossRefGoogle Scholar
  10. 10.
    Liu Y, Sun Y, Liu X, Feng R (2012) IGG4-related lung disese: a case report. In: CHEST 2012. American College of Chest Physicians, AtlantaGoogle Scholar
  11. 11.
    Graham A, Harvin G (2016) Sclerosing mesenteritis: a rare cause of small bowel obstruction. Case Rep Gastroenterol 10:63–67CrossRefGoogle Scholar
  12. 12.
    Ghadir M-R, Sheikhesmaili F, Attari F, Safdari R, Amirhossein G, Vaez-javadi M (2012) Autoimmune pancreatitis mimicking carcinoma of the head of the pancreas: a case report. J Med Case Rep 6:11CrossRefGoogle Scholar
  13. 13.
    Dogaru I, Avram M, Dumitru E, Achim A, Bulbuc I, Chelaru A et al (2015) Extensive retractile mesenteritis of the rectum and left colon associated with ischemic colitis. Arch Balk Med Union 50(4):613–615Google Scholar
  14. 14.
    Büyükbayrak EE, Yasemin A, Karşidağ K, Kars B, Gül A, Alper Ö et al (2011) Sclerosing mesenteritis mimicking ovarian tumor: a very rare case. Gynaecol Obstet Reprod Med 17(2):126–128Google Scholar
  15. 15.
    Malter W, Behrens A, Bach D, Gokel J, Baltzer J (2007) Mesenteriale pannikulitis unter dem klinischen bild eines ovarialkarzinoms mit aszitesbildung. Geburtshilfe Frauenheilkd 67(3):268–271CrossRefGoogle Scholar
  16. 16.
    Xiaoting L, Gao F, Zhang S, Huang Z, Zhu Y, Zong H et al (2018) Clinical and pathological characteristics of IgG4-related interstitial lung disease. Exp Ther Med 15:1465–1473Google Scholar
  17. 17.
    Nyberg L, Björk J, Björkdahl P, Ekberg O, Sjöberg K, Vigren L (2017) Sclerosing mesenteritis and mesenteric panniculitis—clinical experience and radiological features. BMC Gastroenterol BMC Gastroenterology 17(1):1–7CrossRefGoogle Scholar
  18. 18.
    Sevinc A, Adli M, Kalender ME, Camci C (2007) Benign causes of increased serum CA-125 concentration. Lancet Oncol Elsevier Ltd 8(12):1054–1055CrossRefGoogle Scholar
  19. 19.
    Miralles C, Orea M, España P, Provencio M, Sánchez A, Cantos B et al (2003) Cancer antigen 125 associated with multiple benign and malignant pathologies. Ann Surg Oncol 10(2):150–154CrossRefGoogle Scholar
  20. 20.
    Silberstein LB, Rosenthal AN, Coppack SW, Noonan K, Jacobs IJ (2001) Ascites and a raised serum Ca 125—confusing combination. J R Soc Med 94:581–582CrossRefGoogle Scholar

Copyright information

© Springer-Verlag GmbH Germany, part of Springer Nature 2018

Authors and Affiliations

  1. 1.Medical SchoolUniversity College LondonLondonUK
  2. 2.Rheumatology DepartmentNicosia General HospitalNicosiaCyprus
  3. 3.Medical SchoolUniversity of CyprusNicosiaCyprus
  4. 4.Histopathology DepartmentNicosia General HospitalNicosiaCyprus
  5. 5.Department of Academic RheumatologyKing’s College LondonLondonUK
  6. 6.Department of Inflammation Biology, School of Immunology and Microbial Sciences, Faculty of Life Sciences & MedicineKing’s College LondonLondonUK

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