DOI: 10.12890/2026_006500

From thyroid therapy to systemic crisis: PTU-induced c-ANCA vasculitis accompanied by cold-agglutinin disease and organ failure

Authors

  • Pelagia Kefala-Karli Paphos General Hospital, State Health Organization Services, Paphos, Cyprus
  • Theodosia Christou Paphos General Hospital, State Health Organization Services, Paphos, Cyprus
  • Elisavet Georgiou Paphos General Hospital, State Health Organization Services, Paphos, Cyprus
  • Despina Pavlou Paphos General Hospital, State Health Organization Services, Paphos, Cyprus
  • Andreas Kapouralos Paphos General Hospital, State Health Organization Services, Paphos, Cyprus
  • Kyriakos Panagi Paphos General Hospital, State Health Organization Services, Paphos, Cyprus
  • Konstantina Chalmoukou Paphos General Hospital, State Health Organization Services, Paphos, Cyprus

chalmkon@gmail.com

Keywords

Propylthiouracil, vasculitis, acute renal failure, c-ANCA, cold agglutinin disease

Abstract

Introduction: Propylthiouracil (PTU) is typically used as first line therapy for hyperthyroidism but is associated with a spectrum of adverse effects including vasculitis, that may lead to acute renal failure and exceptionally to pancreatitis, represents the most severe.
Case description: Here we present the case of a 43-year-old woman with a history of Graves’ disease until recently on PTU therapy, who presented with fever and myalgia complicated by cold agglutinin disease, acute renal failure and pancreatitis. Subsequent laboratory studies and thorough physical examination excluding other causes revealed the presence of antibodies against leukocyte proteinase 3. Prompt initiation of immunosuppressive therapy and plasmapheresis led to immediate improvement of the patient’s clinical condition and laboratory tests.
Conclusion: Clinicians should maintain a high index of suspicion for autoimmune phenomena in patients receiving PTU even after cessation of the medication. Early recognition, immediate discontinuation of the drug, and timely initiation of immunosuppressive therapy are essential to prevent irreversible organ damage.

View the entire article

Downloads

References

  1. Azizi F. The safety and efficacy of antithyroid drugs. Expert Opin Drug Saf 2006;5:107-116. doi: 10.1517/14740338.5.1.107
  2. Kitching AR, Anders HJ, Basu N, Brouwer E, Gordon J, Jayne DR, et al. ANCA-associated vasculitis. Nat Rev Dis Primers 2020;6:71. doi: 10.1038/s41572-020-0204-y
  3. Almeida MS, Ramalho C, Gomes F, Ginga MDR, Vilchez J. Propylthiouracil-Induced Skin Vasculitis. Cureus 2022;14:e27073. doi: 10.7759/cureus.27073
  4. Gabbard AP, Booth GS. Cold Agglutinin Disease. Clin Hematol Int 2020;2:95-100. doi: 10.2991/chi.k.200706.001
  5. Weng CH, Liu ZC. Drug-induced anti-neutrophil cytoplasmic antibody-associated vasculitis. Chin Med J (Engl) 2019;132:2848-2855. doi: 10.1097/CM9.0000000000000539
  6. Yamauchi K, Sata M, Machiya J, Osaka D, Wada T, Abe S, et al. Antineutrophil cytoplasmic antibody positive alveolar haemorrhage during propylthiouracil therapy for hyperthyroidism. Respirology 2003;8:532-535. doi: 10.1046/j.1440-1843.2003.00499.x
  7. Khalaf R, Mhesin D, Abu Salim H, Taha S, Abed W. Propylthiouracil-induced vasculitis with alveolar hemorrhage after 31 years of treatment: a case report. Clin Diabetes Endocrinol 2024;10:42. doi: 10.1186/s40842-024-00215-3.
  8. Nakazawa D, Tomaru U, Suzuki A, Masuda S, Hasegawa R, Kobayashi T, et al. Abnormal conformation and impaired degradation of propylthiouracil-induced neutrophil extracellular traps: implications of disordered neutrophil extracellular traps in a rat model of myeloperoxidase antineutrophil cytoplasmic antibody-associated vasculitis. Arthritis Rheum 2012;64:3779-3787. doi: 10.1002/art.34619
  9. Balavoine AS, Glinoer D, Dubucquoi S, Wémeau JL. Antineutrophil Cytoplasmic Antibody-Positive Small-Vessel Vasculitis Associated with Antithyroid Drug Therapy: How Significant Is the Clinical Problem? Thyroid 2015;25:1273-1281. doi: 10.1089/thy.2014.0603
  10. Andrade GC, Maia FCP, Mourão GF, Rosario PW, Calsolari MR. Antineutrophil cytoplasmic antibodies in patients treated with methimazole: a prospective Brazilian study. Braz J Otorhinolaryngol 2019;85:636-641. doi: 10.1016/j.bjorl.2018.05.014
  11. Ishii R, Imaizumi M, Ide A, Sera N, Ueki I, Horie I, et al. A long-term follow-up of serum myeloperoxidase antineutrophil cytoplasmic antibodies (MPO-ANCA) in patients with Graves disease treated with propylthiouracil. Endocr J 2010;57:73-79. doi: 10.1507/endocrj.k09e-230
  12. Mayer B, Bartolmäs T, Yürek S, Salama A. Variability of Findings in Drug-Induced Immune Haemolytic Anaemia: Experience over 20 Years in a Single Centre. Transfus Med Hemother 2015;42:333-339. doi: 10.1159/000440673
  13. Ivkovic V, Windpessl M, Berke I, Geetha D, Callemeyn J, Norouzi S, et al. ANCA-Associated Glomerulonephritis: Diagnosis and Therapy Proceedings of the Henry Shavelle Lectureship. Glomerular Dis 2024;5:26-47. doi: 10.1159/000542925
  14. Iida T, Adachi T, Tabeya T, Nakagaki S, Yabana T, Goto A, et al. Rare type of pancreatitis as the first presentation of anti-neutrophil cytoplasmic antibody-related vasculitis. World J Gastroenterol 2016;22:2383-2390. doi: 10.3748/wjg.v22.i7.2383
  15. Jayne DR, Gaskin G, Rasmussen N, Abramowicz D, Ferrario F, Guillevin L, et al. Randomized trial of plasma exchange or high-dosage methylprednisolone as adjunctive therapy for severe renal vasculitis. J Am Soc Nephrol 2007;18:2180-2188. doi: 10.1681/asn.2007010090
  16. Specks U, Merkel PA, Seo P, Spiera R, Langford CA, Hoffman GS, et al. Efficacy of remission-induction regimens for ANCA-associated vasculitis. N Engl J Med 2013;369:417-427. doi: 10.1056/NEJMoa1213277
  17. Unizony S, Villarreal M, Miloslavsky EM, Lu N, Merkel PA, Spiera R, et al. Clinical outcomes of treatment of anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis based on ANCA type. Ann Rheum Dis 2016;75:1166-1169. doi: 10.1136/annrheumdis-2015-208073

Published

2026-04-22

How to Cite

1.
Kefala-Karli P, Christou T, Georgiou E, Pavlou D, Kapouralos A, Panagi K, et al. From thyroid therapy to systemic crisis: PTU-induced c-ANCA vasculitis accompanied by cold-agglutinin disease and organ failure. EJCRIM [Internet]. 2026 Apr. 22 [cited 2026 Nov. 3];13(5). Available from: https://ejcrim.com/index.php/ejcrim/article/view/6500