Porokeratotic eccrine and hair follicle nevus: a report of two cases and review of the literature*

Agulló-Pérez, Alfredo Daniel; Resano-Abarzuza, Miguel Ángel; Córdoba-Iturriagagoitia, Alicia; Yanguas-Bayona, Juan Ignacio

doi:10.1590/abd1806-4841.20176536

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Case Report • An. Bras. Dermatol. 92 (5 Suppl 1) • 2017 • https://doi.org/10.1590/abd1806-4841.20176536 copy

Porokeratotic eccrine and hair follicle nevus: a report of two cases and review of the literature^* * Study conducted at the Dermatology and Pathology services, Complejo Hospitalario de Navarra, Pamplona, Navarra, Spain.

Authorship SCIMAGO INSTITUTIONS RANKINGS

Abstract:

Porokeratotic eccrine and hair follicle nevus is a very rare non-hereditary disorder of keratinization with eccrine and hair follicle involvement with only 9 cases described in the literature. In 2009 the term porokeratotic anexial ostial nevus was proposed to comprehend porokeratotic eccrine and hair follicle nevus and a related and more common process without follicular involvement: porokeratotic eccrine ostial and dermal duct nevus Recent findings suggest that both entities may be produced by a mutation in GJB2 gene, which is associated to KID syndrome. Herein we report 2 cases of porokeratotic eccrine and hair follicle nevus and review the existing cases in the Spanish and English literature.

Keywords:
Hamartoma; Keratinocytes; Nevus; Pediatrics

Case	Author/ year	Age of onset/ Gender	U or B	Distribution	Morphology	Clinical course	Treatment/Response to therapy
1 (1)	Coskey/ 1982	2 Y/F	U	Forearm, wrist, palms	Linear erythematous, slightly scaly plaque and filiform papules with whitish-colored keratin at their center.	Increased in size proportionately as patient grew	_
2 (6)	Kroumpouzos/ 1999	Birth/F	B	Scalp, neck, trunk, extremities (Generalized)	Yellow to brown keratotic plaques over patches of hair loss. Hyperkeratotic plaques. Red to brown, scaly, verrucous plaques in a symmetrical-linear and in random patterns	Increased in size proportionately as patient grew	_
3 (3)	Goddard/ 2009	Birth/F	B	Face, neck, trunk, arms, but-tocks, groin, legs, back of hands and feet, palms	Erythematous and atrophic erosions and fissures in Blasch-ko linear distribution	Spreading, becoming more hyperkeratotic with fragile atrophic plaques and less ery-thematous component	Pulsed dyed laser, tazarotene 0.1% cream, 5-fluorouracil 5% cream/ No effect
4 (3)	Goddard/ 2009	Birth/F	B	Trunk. Arms and legs. Fourth finger, nail and plantar aspect of r foot	Hiperkertatotic linear plaques with central hypopigmented atrophic plaques. Erythematous papules and tense bullae within areas of hyperpigmentation	Stable till age of 32, when developed ery-thema, pruritic blisters within plaques later ulcers, and finally SCC in lesional skin	Minocycline 100mg, Tretionin 0,1%, Tazaro-ten 0,05%, cryotherapy, fluocinonide/ Minimal improvement
5 (3)	Goddard/ 2009	Birth/F	B	Inguinal area, r arm, l inner thigh, l hand	Rugged pink to gray-brown linear lichenified plaques with peripheral rim of scale, in Blas-chko linear distribution and annular macules with atrophic centers	Thickened over time, transitioning from ery-thema to gray-brown color	Mometasone cream/ slightly improvement, irritation
6 (7)	Martorell-Cala-tayud/ 2010	Birth/M	U	R forearm, r wrist, upper r trunk	Hyperkeratotic, whorled, linear plaques with hypopigmented atrophic center	Increased in size proportionately as patient grew	-
7 (2)	Criscione/ 2010	15 Y/F	B	Legs, trunk, cheeks	Scattered brown keratotic pa-pules, multiple tiny yellow and skin-colored filiform papules, randomly distributed	Gradual spreading of facial papules, trunk and leg papules coalesced into thin plaques	Cryotherapy, t retinoids, steroids, salicyclic acid & urea/ ineffective. Carbon dioxide laser/ moderate improvement
8 (5)	Mayorga-Velás-quez/ 2013	21/M	B	Cheeks	Brown verrucous plaques	Gradual growing over 6 months	-
9 (4)	Llamas-Velasco / 2015	2 Y/M	B	Axillae, groins, thighs	Plaques composed of multiple slightly erythematous keratotic papules, some with whitish central spires vertically projec-ted from their pits	-	Carbon dioxide laser/ moderate improve-ment, discrete recurren-ce after treatment
10	Agulló-Pérez/ 2016	Birth/F	U	Scalp, neck, trunk, right upper and lower limbs. R sole	Erythematous keratotic papu-les and plaques in Blaschko linear distribution. Filiform brown papules with whitish colored keratin at their center	Some lesions experi-mented gradual invo-lution, other become more hyperkeratotic with less erythema-tous component	T tazarotene/No response
11	Agulló-Pérez/ 2016	5 Y/M	U	Axillae	Red to Pink keratotic coa-lescent papules, some with whitish central spires	Stable	Pimecrolimus 0,1%, methylprednisolone/ No effect

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E-mail: revista@sbd.org.br

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[1] Mailing address: Alfredo Daniel Agulló Pérez, Centro Especialidades Principe de Viana, Irunlarrea Nº3 Planta Baja 31008 Pamplona, Spain. Email: tegatreides@gmail.com

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Porokeratotic eccrine and hair follicle nevus: a report of two cases and review of the literature* * Study conducted at the Dermatology and Pathology services, Complejo Hospitalario de Navarra, Pamplona, Navarra, Spain.

Abstract:

Porokeratotic eccrine and hair follicle nevus: a report of two cases and review of the literature^* * Study conducted at the Dermatology and Pathology services, Complejo Hospitalario de Navarra, Pamplona, Navarra, Spain.