NDT Advance Access originally published online on March 22, 2006
Nephrology Dialysis Transplantation 2006 21(8):2338-2339; doi:10.1093/ndt/gfl109
| ||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
Henoch–Schönlein purpura associated with propylthiouracil overdose
Email: hayoung.oh{at}samsung.comSir,
Propylthiouracil is commonly used to treat hyperthyroidism. It has been well-documented that propylthiouracil could induce antineutrophil cytoplasmic antibody (ANCA)-positive vasculitis, which predominantly affects small vessels [1,2]. Small vessel vasculitis includes a variety of aetiologic diagnoses with indistinguishable clinical presentations [3]. We describe here a patient who developed Henoch–Schönlein purpura (HSP) after a propylthiouracil overdose.
Case
A 16-year-old girl was hospitalized for progressive abdominal pain and haematochezia over 10 days. She complained of a colicky abdominal pain that was associated with vomiting which aggravated after eating. Fourteen months earlier, she had been diagnosed as having Graves’ disease and had been prescribed propylthiouracil (200 mg/day). Seventeen days prior to admission, she purposely took 100 pills and subsequently suffered from severe nausea and vomiting for 1 day. Thereafter, she stopped the drug. On admission, she denied taking any other drug. On physical examination, her blood pressure was 120/88 mmHg and pulse rate 112/min. Her tongue was dehydrated and she had a symmetric palpable purpuric rash on her lower extremity. Her abdomen was tender. Laboratory findings were as follows: white blood cell count, 14.4 x 109/l; haemoglobin, 15.8 g/dl; platelet count, 266 x 109/l; erythrocyte sedimentation rate, 22 mm/h; C-reactive protein, 56 mg/dl; serum urea, 11 mg/dl; creatinine, 0.4 mg/dl; uric acid, 15 mg/dl; protein, 5.4 g/dl and albumin, 2.9 mg/dl. Urinalysis showed a specific gravity of 1.030, albumin (±), ketone (3+) and no haematuria. The ANCA was positive with a perinuclear pattern at a titre of 1:80. However, neither anti-myeloperoxidase nor anti-proteinase 3 activities were detected. Anti-nuclear antibody and anti-cardiolipin antibody were negative and complement levels were normal. Her chest X-ray was also normal.
Abdominal computed tomography showed bowel wall thickening of multiple jejunal and ileal loops, which were interpreted as gastrointestinal vasculitis. A skin biopsy revealed leucocytoclastic vasculitis with no IgA or C3 deposits by immunofluorescent staining.
She received supportive care including intravenous hydration and nutrition. After a short improvement, the abdominal pain became aggravated. Thereafter, she was treated with intravenous methylprednisolone (20 mg/day, hospital day 15) and her symptoms resolved slowly and skin lesions improved. However, on hospital day 22, repeat urinalysis showed proteinuria and microscopic haematuria; her random urine protein–creatinine ratio had increased up to 2.94 gm/gm, though serum creatinine was stable at 0.4mg/dl. A percutaneous renal biopsy (hospital day 29) was performed. Focal mesangial proliferation was observed in less than half of glomeruli by light microscopy, and immunofluorescence revealed strong mesangial staining for IgA and C3. Three months after the propylthiouracil overdose, she was clinically well and urinary protein excretion was decreasing with steroid tapering.
Comment
Our initial impression of this case was ANCA-positive vasculitis based on purpura and a history of propylthiouracil ingestion. However, the dominant gastrointestinal symptom raised the possibility of HSP. Negative anti-myeloperoxidase activity and a renal pathology finally precipitated a diagnosis of HSP. We were unable to obtain proof of the true causal relationship between propylthiouracil overdose and HSP. However, based on the temporal relationship and the absence of other possible causes, we considered that HSP might have been induced by the propylthiouracil. To our knowledge, this report is the first case of HSP associated with propylthiouracil. We wonder whether HSP would have occurred if she had maintained the prescribed dosage. However, it is likely that no dose-response relationship exists, as drug-induced HSP is considered a type of hypersensitivity reaction [4]. This case suggests that HSP should be included in the differential diagnoses of patients with Graves’ disease treated with propylthiouracil who develop purpura.
Conflict of interest statement. None declared.
Department of Medicine Samsung Medical Center Sungkyunkwan University School of Medicine 50 Irwon-dong Gangnam-gu Seoul, Korea 135-710
References
- Dolman KM, Gans RO, Vervaat TJ, et al. Vasculitis and antineutrophil cytoplasmic autoantibodies associated with propylthiouracil therapy. Lancet 1993; 342: 651–652[CrossRef][Web of Science][Medline]
- Harper L, Cockwell P, Savage CO. Case of propylthiouracil-induced ANCA associated small vessel vasculitis. Nephrol Dial Transplant 1998; 13: 455–458[Web of Science][Medline]
- Jennette JC, Falk RJ. Small-vessel vasculitis. N Engl J Med 1997; 337: 1512–1523
[Free Full Text] - ten Holder SM, Joy MS, Falk RJ. Cutaneous and systemic manifestations of drug-induced vasculitis. Ann Pharmacother 2002; 36: 130–147[Abstract]
CiteULike 
Connotea 
Del.icio.us What's this?
| ||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||