Adult onset atopic dermatitis with secondary follicular mucinosis with cyclosporine induced spiny follicular hyperkeratosis and hair casts

Abstract

Secondary follicular mucinosis developing in adult patient with atopic dermatitis (AD) was observed on histology. Due to severity of disease, patient was started on oral cyclosporine 300 mg daily in 3 divided doses. Two weeks later patient started developing multiple spiny follicular keratoses mainly on face but also on extremities and trunk along with hair casts on scalp. Repeat biopsy from such keratotic lesions confirmed diagnosis spiny follicular hyperkeratosis (SFH). On investigations no evidence of multiple myeloma or any other malignancy was found. Cyclosporine was stopped and lesions cleared over 2 weeks. We report this case due to rare association of AD with follicular mucinosis and cyclosporine induced SFH with hair casts.

Follicular mucinosis: a review

“…37 In their recent case series, Muñoz- and TNF, and fibroblast stimulation by the HIV infection, may also play a role. 38 Other cutaneous conditions rarely associated with secondary FM include angiolymphoid hyperplasia with eosinophilia, 39 atopic dermatitis, 40 childhood alopecia universalis, 41 systemic lupus erythematosus, 42 and following radiation therapy. 43 In such cases of secondary FM, treatment of the underlying condition is required.…”

Sorafenib-Induced Spiny Follicular Hyperkeratosis: A Case Report with Review of Literature

“…13 28 Other known associations are Crohn's disease, hypovitaminosis A, chronic renal failure, human immunodeficiency virus infection, cryoglobulinemia, Sezary syndrome, and lymphoma. 13 14 28…”

“…Treatment of SFH with topical retinoids, fluocinolone acetonide gel, and 12% lactic acid cream has been tried in the past with little benefit. 14 28 Discontinuation of the causative drug or management of the underlying condition is considered the definitive treatment. 13 28 Among the 11 cases of sorafenib-induced SFH in literature, six patients showed complete clearance of the rash on discontinuing sorafenib, with recurrence seen in three patients on reinstitution of treatment.…”

“…Besides sorafenib, SFH is linked to various diseases, few drugs like vemurafenib, glasdegib, cyclosporine, acitretin, and can also be idiopathic. [12][13][14][15][16]28 Paraproteinemia, particularly multiple myeloma, is the earliest known association and most frequently linked to SFH in literature. 13,28 Other known associations are Crohn's disease, hypovitaminosis A, chronic renal failure, human immunodeficiency virus infection, cryoglobulinemia, Sezary syndrome, and lymphoma.…”

“…13,28 Other known associations are Crohn's disease, hypovitaminosis A, chronic renal failure, human immunodeficiency virus infection, cryoglobulinemia, Sezary syndrome, and lymphoma. 13,14,28 Unlike HFSR, SFH is not a dose-limiting AE. Treatment of SFH with topical retinoids, fluocinolone acetonide gel, and 12% lactic acid cream has been tried in the past with little benefit.…”