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Propylthiouracil-associated rapidly progressive crescentic glomerulonephritis with double positive anti-glomerular basement membrane and antineutrophil cytoplasmic antibody: the first case report

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Abstract

We report a case of propylthiouracil (PTU)-induced double antineutrophil cytoplasmic antibody (ANCA) and anti-glomerular basement membrane antibody (anti-GBM antibody) disease causing pulmonary-renal syndrome in a 35-year-old Thai woman with 10-year history of PTU treatment for thyrotoxicosis. She developed clinical symptoms of vasculitis upon receiving long-term PTU treatment. Prednisolone treatment and the switching from PTU to methimazole resulted to short-term clinical improvement. Nevertheless following termination of steroid treatment, she developed recurrent pulmonary hemorrhage and rapidly progressive glomerulonephritis. The kidney biopsy showed crescentic glomerulonephritis with linear IgG deposit on the glomerular basement membrane although transbronchial lung biopsy showed no immune deposit along the alveolar basement membrane. Serum testing for p-ANCA was positive and western blot showed positive antibody to the alpha-3 chain of collagen type IV. Both ANCA and anti-GBM antibody may play a role in the development of end organ damage. To facilitate early and specific intervention, clinicians should be aware of the propensity of PTU to cause lupus-like syndromes with renal involvement. In patients with PTU-induced ANCA-associated glomerulonephritis, serum anti-GBM antibody test may be useful in the early diagnosis of double positive antibodies disease and plasmapheresis should be performed without delay.

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Acknowledgments

The authors would like to thank Suchin Worawichawong for interpretation of the immunostaining of the light chains and special stainings of the kidney biopsy.

Conflict of interest

The authors declare that they have no competing interests.

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Authors and Affiliations

  1. Division of Nephrology, Department of Medicine, Ramathibodi Hospital, Mahidol University, Rama VI Road, Phaya Thai, Bangkok, 10400, Thailand

    Pitchaporn Kantachuvesiri, Bunyong Phakdeekitcharoen, Thitima Lothuvachai, Kannika Niticharoenpong, Piyanuch Radinahamed & Surasak Kantachuvesiri

  2. Department of Pathology, Ramathibodi Hospital, Mahidol University, Bangkok, Thailand

    Panas Chalermsanyakorn

  3. Renal and Autoimmunity Group, MRC Centre for Inflammation, University of Edinburgh, Edinburgh, UK

    Neil Turner

Authors
  1. Pitchaporn Kantachuvesiri
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  2. Panas Chalermsanyakorn
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  3. Bunyong Phakdeekitcharoen
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  4. Thitima Lothuvachai
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  5. Kannika Niticharoenpong
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  6. Piyanuch Radinahamed
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  7. Neil Turner
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  8. Surasak Kantachuvesiri
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Correspondence to Surasak Kantachuvesiri.

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Kantachuvesiri, P., Chalermsanyakorn, P., Phakdeekitcharoen, B. et al. Propylthiouracil-associated rapidly progressive crescentic glomerulonephritis with double positive anti-glomerular basement membrane and antineutrophil cytoplasmic antibody: the first case report. CEN Case Rep 4, 180–184 (2015). https://doi.org/10.1007/s13730-014-0163-9

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  • DOI: https://doi.org/10.1007/s13730-014-0163-9

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