Nephrol Dial Transplant (2003) 18: 2421–2423 DOI: 10.1093/ndt/gfg360 Case Report Myeloperoxidase-antineutrophil cytoplasmic antibody-positive crescentic glomerulonephritis associated with benzylthiouracil therapy: report of the first case Faic¸al Jarraya 1 , Mohamed Abid 2 , Rachid Jlidi 3 , Khaled Mkaouar 1 , Mouna Mnif 2 , Mahmoud Kharrat 1 , Khaled Charfeddine 1 , Khaoula Kammoun 1 , Mohamed Ben Hmida 1 and Jamil Hachicha 1 1 Department of Nephrology, 2 Department of Endocrinology and 3 Department of Pathology, Hedi Chaker University Hospital, Sfax, Tunisia Keywords: acute renal failure; ANCA; benzylthio- uracil; crescentic glomerulonephritis; Grave’s disease Introduction Grave’s disease is a common form of autoimmune thyroiditis which has been successfully treated with anti-thyroid drugs for more than half a century. However, these drugs may cause major complications including agranulocytosis, hepatotoxicity and immu- nological disturbances such as lupus erythematosus syndrome. Anti myeloperoxidase-antineutrophil cytoplasmic antibody (MPO-ANCA) mediated crescentic glomer- ulonephritis in association with anti-thyroid treatment was first described in 1994 [1]. In the literature, 22 cases have been reported to date. All were associated with propylthiouracil (PTU), except two [2–5]. One of the latter was associated with thiamazole (TMZ) [6] and the other with carbimazole (CMZ) therapy [7]. We report here the first case of an anti MPO-ANCA- positive crescentic glomerulonephritis in association with benzylthiouracil (BTU) treatment. Case In 1996, a previously healthy 22-year-old woman was admitted to our Endocrinology Division because of weight loss, thermophobia and neck swelling. Physical examination revealed an acquired exophthalmos with goitre, trembling of the upper extremities and tachycardia. Blood pressure was normal at 120/70 mmHg, sedimentation rate was 30 mm and serum creatinine of 46 mmol/l. There was no haematuria or proteinuria. Free serum thyroxin (FT4) was 24 pmol/l (normal range 11–22 pmol/l) and thyroid-stimulating hormone (TSH) 0.03 mIU/l (normal range 0.2–6 mIU/l). Anti- thyroglobulin and anti-thyroperoxidase antibody titres were, respectively, 328 IU/ml (normal <100 IU/ml) and 369 IU/ml (normal <70 IU/ml). Thyroid scintig- raphy showed a homogeneous fixation. The diagnosis of Grave’s disease was made and anti-thyroid drug BTU started at 250 mg/day for 1 month. The dose was then slightly reduced to 50 mg/day. Symptoms improved and the patient has stayed euthyroid since. She presented again in 1997 because of anorexia, nausea and vomiting of recent onset. She was still taking BTU at 50 mg/day. On admission, blood pres- sure was 110/70 mmHg and pulse rate was 70 beats/min and regular. An enlargement of the thyroid gland and exophthalmos were observed. There was no oedema and urinary output was within normal limits, 1–2 l/24 h. Laboratory findings were: serum creatinine, 1040 mmol/l; proteinuria, 2.8 g/24 h; haematuria of 950 000 red blood cells/min; and erythrocyte sedimentation rate, 150 mm. Serum complement was normal: CH50, 444 IU/l (normal range 23–430 IU/l); C3, 0.96 g/l (normal range 0.7–1.5 g/l); and C4, 0.45 g/l (normal range 0.15–0.45 g/l). Anti MPO-ANCA was found with an ELISA test at three marks (Sanofi diagnostics Pasteur). Chest X-ray was normal. Ultrasound exam- ination showed normal-sized kidneys. Renal biopsy revealed features of a fibrocellular, crescentic glomer- ulonephritis in three of six glomeruli. The other three glomeruli exhibited sclerotic changes. In addition, there Correspondence and offprint requests to: Dr Faic¸ al Jarraya, Nephrology Department, Hedi Chaker University Hospital, Sfax 3029, Tunisia. Email: faijar@excite.com by guest on June 9, 2013 http://ndt.oxfordjournals.org/ Downloaded from