[Successful treatment with infliximab of a patient with refractory sarcoidosis].
A 35-year-old patient had persistent, refractory symptomatic pulmonary sarcoidosis, complicated by a rare congenital muscular disease: McArdle's disease (a glycogen storage disease caused by myophosphorylase deficiency). As the desaturations during mild exercise caused by the sarcoidosis aggravated the negative consequences of his muscle disease and he failed to respond adequately to corticosteroids and methotrexateimmunosuppressive agents, the patient was successfully treated experimentally with infliximab, a monoclonal antibody and specific tumour necrosis factor alpha (TNF-alpha) inhibitor. The results were favourable: after 17, 21 and 36 months there was an improvement in various lung function parameters, his fatigue was reduced and the patient had been able to resume his work as a taxi driver. TNF-alpha appears to be an important mediator of clinical disease in sarcoidosis and infliximab could be a promising therapy for patients with refractory sarcoidosis.
Fouchier
S M
SM
Academisch Ziekenhuis, Postbus 5800, 6202 AZ Maastricht.
Möller
G M
GM
Van Santen-Hoeufft
M
M
Faber
C G
CG
Smeenk
F W
FW
Drent
M
M
D000328:Adult; D000911:Antibodies, Monoclonal / Q000627:therapeutic use*; D002908:Chronic Disease; D004351:Drug Resistance; D006012:Glycogen Storage Disease Type V / Q000150:complications / Q000503:physiopathology; D006801:Humans; D000069285:Infliximab; D008297:Male; D012129:Respiratory Function Tests; D017565:Sarcoidosis, Pulmonary / Q000150:complications / Q000188:drug therapy* / Q000503:physiopathology; D016896:Treatment Outcome; D014409:Tumor Necrosis Factor-alpha / Q000037:antagonists & inhibitors* / Q000276:immunology
D000911:Antibodies, Monoclonal; D014409:Tumor Necrosis Factor-alpha; D000069285:Infliximab