[Vasculitis with renal involvement and antineutrophil cytoplasmic antibodies (ANCA) in a child receiving benzylthiouracil].

Abstract

Vasculitis associated to antineutrophil cytoplasmic antibodies (ANCA) is a rare complication of therapy with antithyroid medication. They were mainly described in patients treated with propylthiouracil (PTU), carbimazole, methimazole and rarely by benzylthiouracil (Basden). We report a case of 12-years-old girl treated by benzylthiouracil for Grave's disease who developed after 2 years vasculitis associated with cutaneous involvement (generalized ulcer necrotic purpura) and glomerulonephritis with proteinuria of 24 hours at 26 mg/kg/day, microscopic hematuria and renal failure with creatinemia level at 135 micromol/l. The ANCA type antiMPO (myeloperoxidase) was positive. The histology study of the renal needle biopsy was in favour with focal necrotizing glomerulonephritisand crescents with different evolutive stages. The discontinuation of benzylthiouracil and the treatment by the corticoids involved a disappearance of cutaneous lesions, a negative result of proteinuria, a normalization of the renal function (creatinemia=84 micromol/l) and a disappearance of hematuria and ANCA. These results permitted to announce hypothesis that benzylthiouracil was implicated in development of vasculitis associated to ANCA.

• Authors: Mongia Hachicha, Thouraya Kammoun, Wafa Ben Romdhane, Rim Ben Abdallah, Abdelmajid Mahfoudh, Khaoula Kammoun, Jamil Hachicha, Ali Triki
• Journal: Nephrologie & therapeutique (Nephrol Ther) Vol. 3 Issue 4 Pg. 147-51 (Jul 2007) ISSN: 1769-7255 [Print] France
• Vernacular Title: Vascularite avec atteinte rénale et anticorps anticytoplasme des polynucléaires neutrophiles (ANCA) après prise de benzylthio-uracile chez l'enfant.
• PMID: 17658441 (Publication Type: Case Reports, English Abstract, Journal Article)

Chemical References

• Antibodies, Antineutrophil Cytoplasmic
• Antithyroid Agents
• Thiouracil
• Uridine Phosphorylase
• benzylthiouracil

Topics

• Antibodies, Antineutrophil Cytoplasmic (blood, drug effects)
• Antithyroid Agents (adverse effects)
• Child
• Female
• Graves Disease (drug therapy)
• Hematuria (chemically induced)
• Humans
• Kidney Diseases (chemically induced, pathology)
• Thiouracil (adverse effects, analogs & derivatives)
• Treatment Outcome
• Uridine Phosphorylase (antagonists & inhibitors)
• Vasculitis (chemically induced, pathology)