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Answer: Can you identify this condition?

Lina Abdullah and Ossama Abbas
Canadian Family Physician March 2012, 58 (3) 286;
Lina Abdullah
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Ossama Abbas
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Answer to Dermacase continued from page 285

2. Inflammatory linear verrucous epidermal nevus

First described in 1971, inflammatory linear verrucous epidermal nevus (ILVEN) is a distinct, uncommon epidermal nevus variant that typically presents in early childhood, although adult onset has also been reported.1–4 Clinically, it is characterized by persistent, intensely pruritic, unilateral, erythematous, verrucous, and scaly papules arranged in a Blaschko-linear distribution most commonly involving the lower extremities, pelvis, or buttocks.1–4 Although familial cases have been reported, most ILVEN cases are sporadic.2–5 Histopathologically, ILVEN exhibits hyperkeratosis with parakeratotic foci, papillomatosis, psoriasiform epidermal hyperplasia with elongation and thickening of the rete ridges, occasional slight spongiosis with lymphocyte exocytosis, and a mild to moderate perivascular lymphohistiocytic infiltrate.2–4 A biopsy from our patient had similar characteristic features.

The etiopathogenesis of the condition is not clear. Most ILVEN cases typically occur as isolated findings, and although there have been infrequent reports of associated disorders such as autoimmune thyroiditis and arthritis, these most likely represent coincidental findings.2–4,6,7 While noninflammatory epidermal nevi are commonly associated with central nervous system, ocular, and skeletal abnormalities, ILVEN has been occasionally reported in association with ipsilateral skeletal and renal abnormalities. There has even been one report of ipsilateral undescended testes associated with ILVEN involving the left scrotal sac and penis.2,8,9 Our patient had no associated abnormality.

Differential diagnosis

Clinical differential diagnosis of ILVEN includes noninflammatory epidermal nevi, linear psoriasis, and lichen striatus.2,9,10 Noninflammatory epidermal nevi are typically asymptomatic, are usually associated with other abnormalities, and commonly do not have a predilection to involve the pelvis, buttocks, or lower extremities.2,9 Linear psoriasis is usually asymptomatic and, unlike ILVEN, typically responds completely to antipsoriatic treatment.2 Lichen striatus is usually asymptomatic, tends to resolve spontaneously, and has a different histopathologic picture.2,10

Management

The natural history of ILVEN suggests that it has no tendency to remit or improve with time.2,3 Typically, it is resistant to therapy. Therapeutic options including topical and intralesional glucocorticoids, surgical excision, cryotherapy, and laser therapy have been tried with variable benefit.2,3

Footnotes

  • Competing interests

    None declared

  • Copyright© the College of Family Physicians of Canada

References

  1. ↵
    1. Altman J,
    2. Mehregan AH
    . Inflammatory linear verrucose epidermal nevus. Arch Dermatol 1971;104(4):385-9.
    OpenUrlCrossRefPubMed
  2. ↵
    1. Lee SH,
    2. Rogers M
    . Inflammatory linear verrucous epidermal naevi: a review of 23 cases. Australas J Dermatol 2001;42(4):252-6.
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    1. Morag C,
    2. Metzker A
    . Inflammatory linear verrucous epidermal nevus: report of seven new cases and review of the literature. Pediatr Dermatol 1985;3(1):15-8.
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  4. ↵
    1. Kawaguchi H,
    2. Takeuchi M,
    3. Ono H,
    4. Nakajima H
    . Adult onset of inflammatory linear verrucous epidermal nevus. J Dermatol 1999;26(9):599-602.
    OpenUrlPubMed
  5. ↵
    1. Alsaleh QA,
    2. Nanda A,
    3. Hassab-El-Naby HM,
    4. Sakr MF
    . Familial inflammatory linear verrucous epidermal nevus (ILVEN). Int J Dermatol 1994;33(1):52-4.
    OpenUrlPubMed
  6. ↵
    1. Dereure O,
    2. Paillet C,
    3. Bonnel F,
    4. Guilhou JJ
    . Inflammatory linear verrucous epidermal naevus with auto-immune thyroiditis: coexistence of two auto-immune epithelial inflammations? Acta Derm Venereol 1994;74(3):208-9.
    OpenUrlPubMed
  7. ↵
    1. Al-Enezi S,
    2. Huber AM,
    3. Krafchik BR,
    4. Laxer RM
    . Inflammatory linear verrucous epidermal nevus and arthritis: a new association. J Pediatr 2001;138(4):602-4.
    OpenUrlPubMed
  8. ↵
    1. Oskay T,
    2. Kutluay L
    . Inflammatory linear verrucous epidermal naevus associated with ipsilateral undescended testicle. Clin Exp Dermatol 2003;28(5):557-8.
    OpenUrlPubMed
  9. ↵
    1. Rogers M
    . Epidermal nevi and the epidermal nevus syndromes: a review of 233 cases. Pediatr Dermatol 1992;9(4):342-4.
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  10. ↵
    1. Kennedy D,
    2. Rogers M
    . Lichen striatus. Pediatr Dermatol 1996;13(2):95-9.
    OpenUrlPubMed
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Canadian Family Physician: 58 (3)
Canadian Family Physician
Vol. 58, Issue 3
1 Mar 2012
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