PMID- 31893199
OWN - NLM
STAT- PubMed-not-MEDLINE
LR  - 20220412
IS  - 2284-2594 (Electronic)
IS  - 2284-2594 (Linking)
VI  - 6
IP  - 12
DP  - 2019
TI  - Benzylthiouracil-induced ANCA-associated Vasculitis: A Case Report and Literature 
      Review.
PG  - 001283
LID - 10.12890/2019_001283 [doi]
LID - 001283
AB  - Iatrogenic antineutrophil cytoplasm antibody (ANCA)-associated vasculitis (AAV) 
      is not exceptional. Many cases of small vessel vasculitis induced by anti-thyroid 
      drugs (ATD), mainly propylthiouracil (PTU), have been reported. We present a case 
      of AAV related to another ATD: benzylthiouracil (BTU) and review the literature. 
      An 84-year-old man with a 4-year history of multinodular goitre with 
      hyperthyroidism was treated with BTU. He presented an acute syndrome with 
      weakness, fever, epigastric pain and abdominal distension. Lactate and lipase 
      tests were normal. An abdominal scan showed a thrombosis of the splenic artery 
      with splenic infarction. We excluded a hypothesis of associated embolic 
      aetiology: atrial fibrillation, atrial myxoma, intraventricular thrombus or 
      artery aneurysm. Exploration of a possible prothrombotic state (complete blood 
      count, haemostasis tests, activated protein C resistance, factor V Leiden, 
      protein C, S, antithrombin III) gave normal results. Tests for antinuclear 
      antibodies (ANA) and antiphospholipid antibodies (APL) were negative. However, 
      testing for p-ANCA, with antimyeloperoxidase (MPO) specificity, was positive: 
      120.6 CU (N<20.0). We did not find other systemic manifestations, except a 
      non-specific kidney failure. BTU was discontinued without steroids or 
      immune-modulating drugs. Subsequently, symptoms disappeared progressively and 
      titres of ANCA fell until normalization, 4 months later. Many patients treated 
      with BTU present a high prevalence of ANCA, mainly, but not exclusively, directed 
      against MPO. Vasculitis, however, remains an uncommon complication. The mechanism 
      of this anomaly remains to be elucidated. Some studies suggest the possibility of 
      an autoimmune reaction initiated by drug bioactivation mediated by 
      neutrophil-derived MPO. The present observation is particular because the 
      involved drug was BTU and clinical expression was unusual. LEARNING POINTS: 
      ANCA-associated vasculitis related to anti-thyroid drugs is not exceptional, 
      particularly in patients receiving long-term therapy with thioamides.Clinical 
      manifestations are highly variable.Treatment consists firstly of stopping the 
      anti-thyroid drug. Introduction of steroids and/or immunosuppressive therapy 
      depends on the severity of organic impairments. Prognosis is less severe than 
      primary ANCA vasculitis.
CI  - (c) EFIM 2019.
FAU - Bensiradj, Fatima
AU  - Bensiradj F
AD  - Federation de Medecine Geriatrie Therapeutique, Rouen, France.
FAU - Hignard, Mathilde
AU  - Hignard M
AD  - Departement de Pharmacie, CHU de Rouen, Rouen, France.
FAU - Nakkash, Rand
AU  - Nakkash R
AD  - Federation de Medecine Geriatrie Therapeutique, Rouen, France.
FAU - Proux, Alice
AU  - Proux A
AD  - Federation de Medecine Geriatrie Therapeutique, Rouen, France.
FAU - Massy, Nathalie
AU  - Massy N
AD  - Centre Regional de Pharmacovigilance, Rouen, France.
FAU - Kadri, Nadir
AU  - Kadri N
AD  - Federation de Medecine Geriatrie Therapeutique, Rouen, France.
FAU - Doucet, Jean
AU  - Doucet J
AD  - Federation de Medecine Geriatrie Therapeutique, Rouen, France.
FAU - Landrin, Isabelle
AU  - Landrin I
AD  - Federation de Medecine Geriatrie Therapeutique, Rouen, France.
LA  - eng
PT  - Case Reports
DEP - 20191210
PL  - Italy
TA  - Eur J Case Rep Intern Med
JT  - European journal of case reports in internal medicine
JID - 101648453
PMC - PMC6936924
OTO - NOTNLM
OT  - ANCA vasculitis
OT  - Anti-thyroid drugs
OT  - benzylthiouracil
OT  - hyperthyroidism
COIS- Conflicts of Interests: The Authors declare that there are no competing interest
EDAT- 2020/01/02 06:00
MHDA- 2020/01/02 06:01
PMCR- 2019/12/10
CRDT- 2020/01/02 06:00
PHST- 2019/09/20 00:00 [received]
PHST- 2019/09/23 00:00 [accepted]
PHST- 2020/01/02 06:00 [entrez]
PHST- 2020/01/02 06:00 [pubmed]
PHST- 2020/01/02 06:01 [medline]
PHST- 2019/12/10 00:00 [pmc-release]
AID - 1283-1-10391-2-10-20191203 [pii]
AID - 10.12890/2019_001283 [doi]
PST - epublish
SO  - Eur J Case Rep Intern Med. 2019 Dec 10;6(12):001283. doi: 10.12890/2019_001283. 
      eCollection 2019.