Literature DB >> 33616883

Selective immunoglobulin M deficiency complicated by systemic lupus erythematosus and antiphospholipid syndrome: a case report and review of literature.

Yoshinosuke Shimamura1, Takuto Maeda2, Koki Abe2, Yayoi Ogawa3, Hideki Takizawa2.   

Abstract

Selective immunoglobulin M deficiency (SIgMD) is the isolated absence of serum immunoglobulin M (IgM) with normal levels of other serum immunoglobulins. SIgMD is associated with infections and autoimmune diseases. While there are few reports on SIgMD complicated by systemic lupus erythematosus (SLE), there are no reports on SIgMD complicated by SLE and antiphospholipid syndrome (APS); we present the first report of this kind. A 61-year-old Japanese woman presented with microscopic hematuria and proteinuria. Clinical investigations revealed an elevated serum creatinine level, an undetectable serum IgM level, and seropositivity of antinuclear antibody, anti-Smith antibody, and double-stranded DNA antibody. Radiological investigations were unremarkable. Renal biopsy revealed focal and segmental mesangial cell proliferation; thickened glomerular capillary walls; and IgG, IgA, C3, and C1q deposition, which indicated class III (A/C) lupus nephritis (Renal Pathology Society/International Society of Nephrology classification). Furthermore, anti-CLβ2GP1 antibody positivity and deep vein thrombosis were noted, which fulfilled the revised Sapporo classification criteria for the diagnosis of APS. Thus, she was diagnosed with SIgMD complicated by SLE and APS. The patient was treated with prednisolone, mycophenolate mofetil, and warfarin. After a 1-year follow-up, she achieved clinical remission of SLE and APS without infectious complications; however, the serum IgM level remained undetectable. In conclusion, SIgMD can be complicated by autoimmune disorders. Although rare, we recommend that SLE and APS be considered in patients with SIgMD who present with hematuria, proteinuria, and deep vein thrombosis. We also recommend measuring the titers of antinuclear antibodies, double-stranded DNA antibodies, and anti-CLβ2GP1 antibodies.
© 2021. Japanese Society of Nephrology.

Entities:  

Keywords:  Antiphospholipid syndrome; Selective IgM deficiency; Systemic lupus erythematosus

Year:  2021        PMID: 33616883     DOI: 10.1007/s13730-021-00583-0

Source DB:  PubMed          Journal:  CEN Case Rep        ISSN: 2192-4449


  4 in total

1.  Selective IgM deficiency in adults: phenotypically and functionally altered profiles of peripheral blood lymphocytes.

Authors:  T Ohno; M Inaba; K Kuribayashi; T Masuda; T Kanoh; H Uchino
Journal:  Clin Exp Immunol       Date:  1987-06       Impact factor: 4.330

2.  IgM deficiency: clinical spectrum and immunologic assessment.

Authors:  M F Guill; D A Brown; H D Ochs; K H Pyun; J E Moffitt
Journal:  Ann Allergy       Date:  1989-06

3.  Functional assessment of T and B lymphocytes in patients with selective IgM deficiency.

Authors:  E G De la Concha; M C Garcia-Rodriguez; J M Zabay; M T Laso; F Alonso; A Bootello; G Fontan
Journal:  Clin Exp Immunol       Date:  1982-09       Impact factor: 4.330

4.  Comprehensive clinical and immunological features of 62 adult patients with selective primary IgM deficiency.

Authors:  Dayna Lee Lucuab-Fegurgur; Sudhir Gupta
Journal:  Am J Clin Exp Immunol       Date:  2019-12-25
  4 in total

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