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Inflammatory Linear Verrucous Epidermal Nevus: a Case Report and Short Review of the Literature

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Inflammatory Linear Verrucous Epidermal Nevus: a Case Report and Short Review of the Literature CONTINUING MEDICAL EDUCATION Inflammatory Linear Verrucous Epidermal Nevus: A Case Report and Short Review of the Literature Amor Khachemoune, MD, CWS; Shahbaz A. Janjua, MD; Kjetil Kristoffer Guldbakke, MD GOAL To understand inflammatory linear verrucous epidermal nevus (ILVEN) to better manage patients with the condition OBJECTIVES Upon completion of this activity, dermatologists and general practitioners should be able to: 1. Describe the presenting characteristics of ILVEN. 2. Explain the differential diagnosis of ILVEN. 3. Discuss the treatment options for ILVEN. CME Test on page 248. This article has been peer reviewed and approved Einstein College of Medicine is accredited by by Michael Fisher, MD, Professor of Medicine, the ACCME to provide continuing medical edu- Albert Einstein College of Medicine. Review date: cation for physicians. September 2006. Albert Einstein College of Medicine designates This activity has been planned and imple- this educational activity for a maximum of 1 AMA mented in accordance with the Essential Areas PRA Category 1 CreditTM. Physicians should only and Policies of the Accreditation Council for claim credit commensurate with the extent of their Continuing Medical Education through the participation in the activity. joint sponsorship of Albert Einstein College of This activity has been planned and produced in Medicine and Quadrant HealthCom, Inc. Albert accordance with ACCME Essentials. Drs. Khachemoune, Janjua, and Guldbakke report no conflict of interest. The authors discuss off-label use of 5-fluorouracil, acitretin, calcipotriol, corticosteroids, dithranol, pimecrolimus, and tretinoin. Dr. Fisher reports no conflict of interest. Inflammatory linear verrucous epidermal nevus in a 4-year-old boy and provide a short review of (ILVEN) is a unilateral, persistent, linear, pruritic the literature, with emphasis on our current under- eruption that usually appears on an extremity in standing of the etiology, clinical presentation, infancy or childhood. We present a case of ILVEN diagnosis, and management of ILVEN. Cutis. 2006;78:261-267. Case Report Accepted for publication November 28, 2005. Dr. Khachemoune is Assistant Professor, Ronald O. Perelman A 4-year-old boy presented for evaluation of an Department of Dermatology, New York University School of extremely pruritic linear plaque involving his right Medicine, New York. Dr. Janjua is a specialist, Ayza Skin and groin and right thigh since the age of one year Research Center, Lalamusa, Pakistan. Dr. Guldbakke currently (Figure 1). The plaque first appeared on the right is serving in the military in Norway. buttock (Figure 2) and slowly enlarged, extending Reprints: Amor Khachemoune, MD, CWS, Ronald O. Perelman Department of Dermatology, New York University School of down spirally to involve the right inguinal region Medicine, 530 First Ave, Suite 7R, New York, NY 10016 and upper inner thigh. There was no family history (e-mail: [email protected]). of similar dermatoses. VOLUME 78, OCTOBER 2006 261 Inflammatory Linear Verrucous Epidermal Nevus Results of a physical examination revealed mul- tiple erythematous warty scaling papules coalesced to form a linear, verrucous, hyperpigmented plaque. The plaque extended from the right buttock and inguinal region to the right upper medial thigh following the lines of Blaschko. The rest of the physical examina- tion results were unremarkable, and no associated physical anomaly was found. The eruption and the associated pruritus did not respond to either oral anti- histamines or topical high potency steroids. Results of a biopsy specimen taken from the lesion revealed hyperkeratosis, parakeratosis, acan- thosis, and a decreased granular layer. A perivascular infiltrate of lymphocytes also was evident in the upper dermis. A diagnosis of inflammatory linear verrucous epidermal nevus (ILVEN) was made based on clinical and histopathologic grounds. Comment The condition later known as ILVEN was first described by Unna in 1896.1 However, it was not until 1971 that the disorder was described and clearly defined as a dis- tinct entity by Altman and Mehregan2 in a case series of 25 patients. The authors clearly delineated ILVEN as a clinical and histopathologic variety of linear ver- rucous nevus that clinically appears inflammatory and histopathologically appears psoriasiform. The etiology of ILVEN remains unknown. It is considered a variant of keratinocytic epidermal nevus. Most cases are sporadic, but a familial case, with the condition occurring in a mother and her daughter, has been described.3 Because ILVEN bears some his- Figure 1. Well-demarcated erythematous scaly papules coalescing into a linear plaque involving the right groin tologic resemblance to psoriasis, some authors believe and medial thigh. that the 2 conditions share a common pathogenesis, possibly mediated by cytokines.4 There is some evi- dence that interleukins 1 and 6, tumor necrosis factor (16 patients) were male, and extension beyond the α, and intercellular adhesion molecule-1 are upregu- original margins occurred in 6 patients (26%).7 lated in ILVEN, similar to psoriasis.4 It also has been Altman and Mehregan2 described 6 charac- proposed that activation of an autosomal-dominant teristic features of ILVEN: (1) early age of onset, lethal mutation that survives by mosaicism may be (2) predominance in females (4:1 female-male the cause. The mutated cells might survive in the ratio), (3) frequent involvement of the left leg, vicinity of the normal cells.5 (4) pruritus, (5) marked refractoriness to therapy, ILVEN usually appears in infancy or early child- and (6) a distinctive psoriasiform and inflamma- hood but may be present at birth; the condition is tory histologic appearance. very rarely present in adulthood.6 ILVEN occurs pre- In a few children, ILVEN has been found to occur dominantly in females (female-male ratio, 4:1), and in association with musculoskeletal or other abnor- no racial predominance has been noted. About 6% malities, including supernumerary digits and strabis- of patients with epidermal nevi had ILVEN.6 mus,8 congenital bony anomalies of the ipsilateral ILVEN typically presents with multiple, discrete, extremities,9 congenital dislocation of the ipsilateral erythematous, slightly warty and scaly papules that hip and Fallot tetralogy of the heart,10 autoimmune tend to coalesce into linear plaques. In a retrospec- thyroiditis,11 lichen amyloidosis,12 nevus depigmen- tive study of 23 patients with ILVEN, Lee and tosus,13 arthritis14 and melanodontia.15 More recently, Rogers7 documented a predilection for the buttock ILVEN was associated with ipsilateral undescended and legs, and most cases were unilateral. Onset usu- testicle.16 However, this finding was disputed by ally was within the first 6 months of life, most patients Happle,17 who interpreted the case as an epidermal 262 CUTIS® Inflammatory Linear Verrucous Epidermal Nevus Differential Diagnosis for Inflammatory Linear Verrucous Epidermal Nevus* Clinical Condition Population Group Characteristics Pathology Therapy Nevoid psoriasis Usually children Erythematous scaly Hyperkeratosis, Topical corti- in a Blaschko plaques parakeratosis, costeroids, distribution absence of vitamin D3 granular cell analogues, layer, elongation systemic of rete ridges, antipsoriatic suprapapillary medications, thinning, Munro phototherapy microabscesses Epidermal nevi Present at birth or The lesion may be Variable Investigate during infancy warty, smooth, or for any asso- barely palpable of ciated features variable color±systemic abnormalities Ichthyosiform Infancy, female Unilateral, waxy, scaling Acanthosis with Emollients, nevus of CHILD predominance erythematous plaques alternating topical kerato- syndrome with a sharp midline orthokeratosis, lytics, man- demarcation±systemic parakeratosis, agement of abnormalities and patchy any associated hypergranulosis abnormality Linear lichen Mainly children Violaceous, shiny, Irregular Topical corti- planus polygonal papules of acanthosis, costeroids, varying sizes, often colloid bodies, antihistamines with prominent PIH bandlike infiltrate of lymphocytes and histiocytes in the upper dermis Linear Usually presents Grouped, linearly Tightly packed Topical porokeratosis during childhood, arranged, annular parakeratotic cells 5-fluorouracil, no sex predilection papules and plaques, in the coronoid vitamin D3 with a raised peripheral lamella, lympho- analogues, ridge cytic infiltrate in imiquimod, the papillary retinoids, close dermis, central monitoring for atrophy malignant change Nevoid BCC Childhood/Early Multiple BCCs, pitting Indistinguishable Radiation, syndrome adolescence, of palms and soles, from BCC 5-fluorouracil, no sex predilection jaw cysts±systemic retinoids, PDT, abnormalities surgery TABLE CONTINUED ON PAGE 264 VOLUME 78, OCTOBER 2006 263 Inflammatory Linear Verrucous Epidermal Nevus (continued) Clinical Condition Population Group Characteristics Pathology Therapy Basaloid More common in Hypopigmented, Folliculocentered Close follow-up follicular children smooth, or lesions with small for any change hamartoma striaelike areas, basaloid cells in appearance with later appearance admixed with of pigmented papules squamous cells and tumors, with or in a loose fibrous without or ulceration stroma Linear lichen Usually in adults, Erythematous, scaly, Hyperkeratosis, Topical corti- simplex more common well-demarcated, acanthosis, costeroids, chronicus in women lichenified, firm, spongiosis, patches tacrolimus, rough plaques with
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