Views: 1,570 | Downloads:
38
| Responses: 0
| XML | Respond to this article | Alert & updates | Request permissions | Email to a friend |
Case ReportOpen Access
Antineutrophilic Cytoplasmic Antibody - Positive Systemic Vasculitis Associated with Propylthiouracil Therapy : Report of 2 Children with Graves' Disease
Poomthavom P 
,
Mahachoklertwattana P ,
Tapaneya-Olarn W ,
Chuansumrit A ,
Chunharas A
,
Mahachoklertwattana P ,
Tapaneya-Olarn W ,
Chuansumrit A ,
Chunharas A Systemic vasculitis is a rare complication of therapy with antithyroid medication. Antineutrophilic
cytoplasmic antibody (ANCA)-associated vasculitis has been described in patients treated with
propylthiouracil (PTU) and methimazole (MMI). The majority of cases have underlying Graves'
disease. The authors report 2 children who developed ANCA-associated systemic vasculitis during
PTU therapy of Graves' disease. One patient, after PTU treatment for 3 years, developed severe
systemic vasculitis. After 3 weeks of arthritis, she abruptly presented with hematuria, proteinuria and
edema concomitant with anemia. Her serum creatinine was elevated, to 6 mg/dl. Renal biopsy revealed
crescentic glomerulonephritis. After admission, she developed intracerebral hemorrhage and pulmonary
hemorrhage. She had positive perinuclear-ANCA (p-ANCA) with a titer of 1:160. Despite intensive
therapy with immunosuppressive agents and plasmapheresis, as well as discontinuation of PTU,
she died of the complications of severe systemic vasculitis. The other patient developed fever, arthralgia
and leukocytoclastic vasculitis of the skin during treatment with PTU for about 2 years. Her symptoms
and skin lesions disappeared after discontinuation of PTU. However, she has had a persistently
high titer of p-ANCA 1:320 through 17 months follow-up time. Thus, patients who are treated with
PTU can develop ANCA-positive vasculitis in a mild or severe form. Therefore, they should be
carefully followed and monitored, not only for their thyroid status but also the serious complications
of PTU.
Key word : Antineutrophilic Cytoplasmic Antibody, Propylthiouracil, Vasculitis, Graves' Disease, Thyrotoxicosis, Rapidly Progressive Glomerulonephritis
cytoplasmic antibody (ANCA)-associated vasculitis has been described in patients treated with
propylthiouracil (PTU) and methimazole (MMI). The majority of cases have underlying Graves'
disease. The authors report 2 children who developed ANCA-associated systemic vasculitis during
PTU therapy of Graves' disease. One patient, after PTU treatment for 3 years, developed severe
systemic vasculitis. After 3 weeks of arthritis, she abruptly presented with hematuria, proteinuria and
edema concomitant with anemia. Her serum creatinine was elevated, to 6 mg/dl. Renal biopsy revealed
crescentic glomerulonephritis. After admission, she developed intracerebral hemorrhage and pulmonary
hemorrhage. She had positive perinuclear-ANCA (p-ANCA) with a titer of 1:160. Despite intensive
therapy with immunosuppressive agents and plasmapheresis, as well as discontinuation of PTU,
she died of the complications of severe systemic vasculitis. The other patient developed fever, arthralgia
and leukocytoclastic vasculitis of the skin during treatment with PTU for about 2 years. Her symptoms
and skin lesions disappeared after discontinuation of PTU. However, she has had a persistently
high titer of p-ANCA 1:320 through 17 months follow-up time. Thus, patients who are treated with
PTU can develop ANCA-positive vasculitis in a mild or severe form. Therefore, they should be
carefully followed and monitored, not only for their thyroid status but also the serious complications
of PTU.
Key word : Antineutrophilic Cytoplasmic Antibody, Propylthiouracil, Vasculitis, Graves' Disease, Thyrotoxicosis, Rapidly Progressive Glomerulonephritis
Download:
PDF