PMID- 35630067
OWN - NLM
STAT- MEDLINE
DCOM- 20220531
LR  - 20220716
IS  - 1648-9144 (Electronic)
IS  - 1010-660X (Print)
IS  - 1010-660X (Linking)
VI  - 58
IP  - 5
DP  - 2022 May 11
TI  - Late Onset of ANCA Vasculitis as a Side Effect of Levamisole Treatment in 
      Nephrotic Syndrome.
LID - 10.3390/medicina58050650 [doi]
LID - 650
AB  - Levamisole is effectively used in steroid-dependent nephrotic syndrome and the 
      more frequent side effects reported are cytopenia and liver enzymes alterations. 
      Several studies have demonstrated that this drug can induce high titers of 
      circulating perinuclear antineutrophil cytoplasmic autoantibodies (ANCA) and 
      vasculitis, most of them occurring in the case of prolonged use. A four-year-old 
      boy that was affected with steroid-dependent nephrotic syndrome was treated with 
      Levamisole as a steroid-sparing agent at a dose of 2 mg/kg/48 h. After initiation 
      of the treatment, the number of relapses drastically decreased, enabling a 
      significant reduction in the cumulative steroid dose. Levamisole was well 
      tolerated, and was therefore administered for several years. At the age of 15, he 
      was also diagnosed with celiac disease. After nine years of continuous Levamisole 
      treatment, he presented with a high fever, hand and foot joint arthritis, and 
      increased levels of total and direct bilirubin. Since the symptoms started two 
      days after the injection of the second dose of the COVID-19 vaccine, it was 
      initially concluded that these manifestations were rare vaccination side effects. 
      Therefore, he did not receive any specific treatments, and Levamisole was 
      continued at the same dose. After an initial improvement, two months later, the 
      patient presented with the same symptoms. Suspecting Levamisole-induced 
      vasculitis, an ANCA titer was measured and this returned positive. Clinical 
      manifestations and double positivity for both myeloperoxidase (MPO) and 
      anti-proteinase 3 (PR3) antibodies argued against the fact that that these 
      findings were secondary to vaccination, cocaine abuse, or celiac disease. 
      Assuming that we were facing a rare drug reaction, Levamisole was promptly 
      interrupted. This resulted in a rapid remission of fever and arthritis 
      improvement, and a decrease in ANCA titers. By reporting this case, we want to 
      raise awareness among clinicians regarding a rare complication of treatment with 
      Levamisole that is often misdiagnosed due to the fact that the current literature 
      lacks univocal guidelines regarding the precise timing of ANCA titrations and the 
      duration of the treatment.
FAU - Bernardi, Silvia
AU  - Bernardi S
AD  - School of Nephrology, Universita degli Studi di Milano, ASST Papa Giovanni XXIII, 
      24127 Bergamo, Italy.
FAU - Innocenti, Samantha
AU  - Innocenti S
AD  - School of Nephrology, Universita degli Studi di Firenze, AOU Meyer, 50139 
      Firenze, Italy.
FAU - Charbit, Marina
AU  - Charbit M
AD  - Nephrologie Pediatrique, Centre de Reference du Syndrome Nephrotique Idiopathique 
      de l'Enfant et de l'Adulte, Hopital Necker-Enfants Malades, AP-HP, 75015 Paris, 
      France.
FAU - Boyer, Olivia
AU  - Boyer O
AUID- ORCID: 0000-0002-3957-1359
AD  - Nephrologie Pediatrique, Centre de Reference du Syndrome Nephrotique Idiopathique 
      de l'Enfant et de l'Adulte, Hopital Necker-Enfants Malades, AP-HP, 75015 Paris, 
      France.
AD  - INSERM U1163, Institut Imagine, Universite Paris Cite, 75015 Paris, France.
LA  - eng
PT  - Case Reports
DEP - 20220511
PL  - Switzerland
TA  - Medicina (Kaunas)
JT  - Medicina (Kaunas, Lithuania)
JID - 9425208
RN  - 0 (Antibodies, Antineutrophil Cytoplasmic)
RN  - 0 (COVID-19 Vaccines)
RN  - 2880D3468G (Levamisole)
SB  - IM
MH  - Antibodies, Antineutrophil Cytoplasmic
MH  - *Arthritis
MH  - *COVID-19
MH  - COVID-19 Vaccines
MH  - *Celiac Disease
MH  - Child, Preschool
MH  - Humans
MH  - Levamisole/adverse effects
MH  - Male
MH  - Neoplasm Recurrence, Local
MH  - *Nephrotic Syndrome
MH  - *Vasculitis/chemically induced
PMC - PMC9147217
OTO - NOTNLM
OT  - ANCA-vasculitis
OT  - Levamisole
OT  - case report
OT  - children
OT  - nephrotic syndrome
COIS- The authors declare no conflict of interest.
EDAT- 2022/05/29 06:00
MHDA- 2022/06/01 06:00
PMCR- 2022/05/11
CRDT- 2022/05/28 01:31
PHST- 2022/04/03 00:00 [received]
PHST- 2022/05/02 00:00 [revised]
PHST- 2022/05/08 00:00 [accepted]
PHST- 2022/05/28 01:31 [entrez]
PHST- 2022/05/29 06:00 [pubmed]
PHST- 2022/06/01 06:00 [medline]
PHST- 2022/05/11 00:00 [pmc-release]
AID - medicina58050650 [pii]
AID - medicina-58-00650 [pii]
AID - 10.3390/medicina58050650 [doi]
PST - epublish
SO  - Medicina (Kaunas). 2022 May 11;58(5):650. doi: 10.3390/medicina58050650.