Congential unilateral ichthyosiform erythroderma was first described in 1963.1 All reported cases have been associated with other congenital anomalies. Although there has been no successful treatment of this condition, because of the preliminary success of oral isotretinoin for other forms of ichthyosis, our patient was given a trial of this drug. The treatment was a failure, and a complication of reversible corneal deposits occurred.
Report of a Case
A 14-month-old girl, the product of a full-term pregnancy and normal vaginal delivery, was seen and hospitalized at the Albany (NY) Medical Center in January 1977 for evaluation of multiple congenital abnormalities. There was no family history of anomalies. The mother had taken only the normal medications permitted during pregnancy.On physical examination, the child had a lobster-claw deformity of the left hand as well as a malformed left lower extremity. A well-demarcated, crusted, scaling erythroderma extended from the anterior midline
Weiss J, Degnan M, Leupold R, Lumpkin LR. Bilateral Corneal OpacitiesOccurrence in a Patient Treated With Oral Isotretinoin. Arch Dermatol. 1981;117(3):182-183. doi:10.1001/archderm.1981.01650030060020