Long-term immunosuppressive treatment of a child with Takayasu's arteritis and high IgE immunoglobulins.
A 7-year-old child presented with a severe form of Takayasu's arteritis, with two consecutive episodes involving the right testis and then the left kidney 6 months later. The renal artery obstruction was accompanied by severe hypertension. An aortography showed a complete obstruction of the left renal artery and a narrowing of the right subclavian artery. Plasma renin activity was high. Serum immunoglobulins were within the normal range, except for an increase in IgE (880 mu/l). Despite 4 months', treatment with antihypertensive drugs, prednisone, cyclophosphamide, and anticoagulant, the blood pressure never returned to normal and the left renal function remained completely absent. A nephrectomy was performed which immediately normalized plasma renin activity and blood pressure. The child was subsequently treated with alternate-day prednisone for 3 months, alternating with 3 months of cyclophosphamide or, later, azathioprine. Persantine (dipyridamole) and acetylsalicylic acid were administered continuously. The right radial pulse returned to normal within 2 years. An 8-year follow-up failed to detect any new episode of arteritis. The right kidney showed signs of compensatory hypertrophy. Finally, a recent arteriography demonstrated not only a normal right renal artery blood flow but almost total disappearance of the right subclavian artery obstruction. However, the IgE remained abnormally high (2,023 micrograms/l).
Brunette
M G
MG
Maisonneuve-Rosemont Hospital, Montreal, Canada.
Bonny
Y
Y
Spigelblatt
L
L
Barrette
G
G
D000925:Anticoagulants / Q000627:therapeutic use; D002648:Child; D006801:Humans; D007073:Immunoglobulin E / Q000378:metabolism*; D007166:Immunosuppressive Agents / Q000627:therapeutic use*; D008297:Male; D012039:Regional Blood Flow / Q000502:physiology; D012077:Renal Artery / Q000473:pathology; D012079:Renal Circulation / Q000502:physiology; D013625:Takayasu Arteritis / Q000188:drug therapy* / Q000276:immunology / Q000473:pathology; D013737:Testis / Q000098:blood supply
D000925:Anticoagulants; D007166:Immunosuppressive Agents; D007073:Immunoglobulin E