Ann Dermatol.  2017 Feb;29(1):74-78. 10.5021/ad.2017.29.1.74.

Tumor Necrosis Factor Alpha Blocker-Induced Erythrodermic Sarcoidosis in with Juvenile Rheumatoid Arthritis: A Case Report and Review of the Literature

Affiliations
  • 1Department of Dermatology, Chonbuk National University Medical School, Jeonju, Korea. airmd@jbnu.ac.kr
  • 2Department of Pediatrics, Chonbuk National University Medical School, Jeonju, Korea.
  • 3Research Institute of Clinical Medicine of Chonbuk National University-Biomedical Research Institute of Chonbuk National University Hospital, Jeonju, Korea.

Abstract

The development of cutaneous sarcoidosis as a paradoxical adverse event of tumor necrosis factor alpha (TNF-α) blockers has been reported in the literature; however, an erythrodermic form of cutaneous sarcoidosis during anti-TNF-α therapy has not yet been reported. Herein, we report the first case of an erythrodermic form of cutaneous sarcoidosis during anti-TNF-α therapy and review previous studies of cutaneous sarcoidosis. A 6-year-old Korean girl who had been suffering from juvenile rheumatoid arthritis presented with generalized erythematous skin eruption involving more than about 90% of her body surface area. After 14 months of etanercept treatment, the new erythematous skin eruption had developed and progressed into generalized erythroderma. Exclusion of suspected co-medication had been performed based on medication history. She had no other systemic symptoms, and ophthalmologic and neurologic examinations were normal. Histopathologic findings of the skin lesion revealed diffuse non-caseating granulomatous infiltrates composed of epithelioid histiocytes with sparse lymphocytes involving the entire dermis. Periodic-acid-Schiff and acid-fast stains were negative, and acid-fast bacilli was not detected by polymerase chain reaction of the skin biopsy. Based on clinicopathologic findings, she was diagnosed with etanercept-induced sarcoidal granuloma. After discontinuation of the suspected agent, the lesions spontaneously disappeared.

Keyword

Child; Erythroderma; Etanercept; Sarcoidosis; Tumor necrosis factor-alpha

MeSH Terms

Arthritis, Juvenile*
Biopsy
Body Surface Area
Child
Coloring Agents
Dermatitis, Exfoliative
Dermis
Etanercept
Female
Granuloma
Histiocytes
Humans
Lymphocytes
Neurologic Examination
Polymerase Chain Reaction
Sarcoidosis*
Skin
Tumor Necrosis Factor-alpha*
Coloring Agents
Etanercept
Tumor Necrosis Factor-alpha

Figure

  • Fig. 1 (A) Monomorphous follicular dull reddish papules fused into generalized erythroderma involving the face, trunk, and both upper and lower extremities. (B) Close-up view of the face and chest with scaly follicular hyperkeratotic papules forming erythrodermic patches. (C) Close-up view of the face and chest at 5 months after cessation of etanercept therapy, showing significant improvement

  • Fig. 2 (A) Lower-magnification view showing diffuse involvement with non-caseating granulomatous infiltrates in the entire dermis (H&E, ×40). (B) Higher-magnification view of dermal infiltrate showing epithelioid cells with sparse lymphocytes (H&E, ×200).


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