TY - JOUR
T1 - Childhood-onset porphyria cutanea tarda
T2 - Successful therapy with low- dose hydroxychloroquine (Plaquenil)
AU - Bruce, A. J.
AU - Ahmed, I.
N1 - Funding Information:
This article is made possible through an educational grant from Ortho Dermatological.
PY - 1998
Y1 - 1998
N2 - We describe a 4-year-old girl with a spontaneous blistering disorder that was consistent with porphyria cutanea tarda (PCT). There was no familial history of the disease or any obvious causative factors present. Oral hydroxychloroquine (3 mg/kg) was given twice weekly along with vitamin E (200 U/d) as an antioxidant. Within 6 weeks, marked decreased blistering occurred and by 12 weeks no blistering was evident. Despite clinical improvement and tolerance of hydroxychloroquine, urinary uroporphyrin, asparate amino- transferase, and ferritin levels continued to rise reaching peak levels at 16 weeks of therapy. Near total biochemical remission was observed at 40 weeks and all therapy was discontinued at 60 weeks.
AB - We describe a 4-year-old girl with a spontaneous blistering disorder that was consistent with porphyria cutanea tarda (PCT). There was no familial history of the disease or any obvious causative factors present. Oral hydroxychloroquine (3 mg/kg) was given twice weekly along with vitamin E (200 U/d) as an antioxidant. Within 6 weeks, marked decreased blistering occurred and by 12 weeks no blistering was evident. Despite clinical improvement and tolerance of hydroxychloroquine, urinary uroporphyrin, asparate amino- transferase, and ferritin levels continued to rise reaching peak levels at 16 weeks of therapy. Near total biochemical remission was observed at 40 weeks and all therapy was discontinued at 60 weeks.
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U2 - 10.1016/s0190-9622(98)70464-5
DO - 10.1016/s0190-9622(98)70464-5
M3 - Article
C2 - 9591792
AN - SCOPUS:0031924970
SN - 0190-9622
VL - 38
SP - 810
EP - 814
JO - Journal of the American Academy of Dermatology
JF - Journal of the American Academy of Dermatology
IS - 5 II
ER -