10.5826/dpc.1302a107
text/xml
2160-9381
2778
en
Gaetano Licata
https://creativecommons.org/licenses/by-nc/4.0
Dermatology Practical & Conceptual
non-invasive technology
Inflammatory Linear Verrucous Epidermal Nevus Assessment With in Vivo Reflectance Confocal Microscopy: An Easy Option to Consider
Text.Serial.Journal
https://dpcj.org/index.php/dpc
Research Letter
2023-04-29
2022-08-23
2023-04-29
2023-06-05
e2023107
Dermatol Pract Concept
Gaetano Licata
2023/04/29
10.5826/dpc.1302a107
e2023107
Department of Dermatology, University of Brescia, ASST Spedali Civili Brescia, Brescia,Italy
2160-9381
non-invasive technology
en
e2023107
Ko RF, Smidt AC, Durkin JR. Reflectance confocal microscopy in pediatric dermatology: A state-of-the-art review. Pediatr Dermatol. 2021;38(6):1488-1499. DOI: 10.1111/pde.14837. PMID: 34651341.
Inflammatory Linear Verrucous Epidermal Nevus Assessment With in Vivo Reflectance Confocal Microscopy: An Easy Option to Consider
https://dpcj.org/index.php/dpc/article/download/2778/2078
    Inflammatory Linear Verrucous Epidermal Nevus
     Assessment With in Vivo Reflectance Confocal
         Microscopy: an Easy Option to Consider
                     Giulio Gualdi1, Cesare Ariasi2, Paolo Amerio1, Giovanna Galdo3,
                             Piergiacomo Calzavara-Pinton2, Gaetano Licata2


1 Dermatologic Clinic, Department of Medicine and Aging Science, University G D’Annunzio Chieti-Pescara, Chieti, Italy
2 Department of Dermatology, University of Brescia, ASST Spedali Civili Brescia, Brescia, Italy
3 Department of Dermatology, AORN San Giuseppe Moscati, Avellino, Italy



Key words: inflammatory linear verrucous epidermal nevus, confocal microcopy, child, non-invasive technology
Citation: Gualdi G, Ariasi C, Amerio P, Galdo G, Calzavara-Pinton P, Licata G. Inflammatory Linear Verrucous Epidermal Nevus
Assessment With in Vivo Reflectance Confocal Microscopy: An Easy Option to Consider. Dermatol Pract Concept. 2023;13(2):e2023107.
DOI: https://doi.org/10.5826/dpc.1302a107
Accepted: October 21, 2023; Published: April 2023
Copyright: ©2023 Gualdi et al. This is an open-access article distributed under the terms of the Creative Commons Attribution-
NonCommercial License (BY-NC-4.0), https://creativecommons.org/licenses/by-nc/4.0/, which permits unrestricted noncommercial use,
distribution, and reproduction in any medium, provided the original authors and source are credited.
Funding: None.
Competing Interests: None.
Authorship: All authors have contributed significantly to this publication.
Corresponding Author: Gaetano Licata, MD, Department of Dermatology, University of Brescia, ASST Spedali Civili di Brescia, P.le Spedali
Civili 1, 25123, Brescia, Italy. Phone: +393276215976 E-mail gaetano.licata89@gmail.com




Introduction                                                             Case Presentation
Inflammatory linear verrucous epidermal nevus (ILVEN)                    A 6-year-old boy was seen for a linear, mild itchy lesion in-
is a rare disorder characterized by an early age onset [1]. It           volving the left part of the nose. Diagnosed one year earlier
consists of pruritic linear, or whorled array of inflamma-               as impetigo, after multiple ineffective topical and systemic
tory, following Blaschko lines [2], papules and/or plaques               antibiotic treatments, referred to our clinic for redness and
and histologic features resembling psoriasis or lichenoid                worsening of the lesion. Clinical examination showed a lin-
dermatitis [3]. The disease is a distinct variety of kerat-              ear dyskeratotic erythematous plaque with a slightly pig-
inocytic epidermal nevus that is clinically inflammatory                 mented halo and fine superficial desquamation located on
and reflects a phenomenon of mosaicism caused by so-                     the nose (Figure 1A). Because of the anatomic site and age
matic mutations. We report the case of a 6-year-old boy                  of the patient, to avoid biopsy he was subjected assessment
in which in vivo reflectance confocal microscopy (RCM)                   with hand-held RCM (Vivascope 3000). It proved minimal
allowed the diagnosis of ILVEN avoiding an invasive                      hyperkeratosis, spongiosis (Figure 2A, white circle) with
skin biopsy.                                                             a pattern closer to the honeycombed one with thickened,




Research Letter | Dermatol Pract Concept. 2023;13(2):e2023107                                                                         1
           Figure 1. (A) Linear dyskeratotic erythematous plaque with a slightly pigmented halo and
           fine superficial desquamation involving the columella, tip and bridge of the left part of the
           nose. (B) Clinical picture one months after treatment with hydrocortisone butyrate oint-
           ment 0.1% twice a day; almost complete resolution of lesion.




    Figure 2. (A-D) Summary of the main features imaged by in vivo reflectance confocal microscopy
    (RCM) (Vivascope 3000) in RCM in Inflammatory Linear Verrucous Epidermal Nevus.
    (A) Spongiosis (withe circle) with a pattern closer to the honeycombed one with thickened, highly re-
    fractive intercellular space between keratinocytes.
    (B) Presence of a single roundish refractive Demodex folliculorum (arrow) within the hair follicles and
    a pattern closer to the honeycombed.
    (C) At level of the epithelial-connective tissue junction, non-rimed connective tissue papillae were observed.
    (D) Below the basal layer, connective tissue papillae appeared disrupted and confluent in large dark
    areas (white circle), peculiar signs of the interface dermatitis.


2                                                                 Research Letter | Dermatol Pract Concept. 2023;13(2):e2023107
highly refractive intercellular space between keratinocytes          characteristics and the thinner skin of the pediatric patient
(Figure 2B). Non-rimed connective tissue papillae were ob-           make the use of this non-invasive technology particularly
served at the level of the epithelial-connective tissue junction     suitable in this case. Finally, it is important to underline how
(Figure 2C). Below the basal layer, connective tissue papil-         the current contact devices can be best used even on a non-
lae appeared disrupted and confluent in large dark areas             flat surface; fast and handy device make it much more prac-
­(Figure 2D, white circle), peculiar signs of interface derma-       tical also for pediatric patients.
titis. Based on these and on the clinical features patient was
diagnosed with ILVEN and treatment started at first with             References
hydrocortisone butyrate ointment 0.1% twice a day for one
                                                                     1. Wollina U, Tchernev G. ILVEN - COMPLETE REMISSION
month with good improvement (Figure 1B). Then was pre-
                                                                        ­AFTER ADMINISTRATION O TOPICAL CORTICOSTEROID
scribed tacrolimus ointment 0.03% twice a day for another
                                                                         (CASE REVIEW). Georgian Med News. 2017;(263):10-13.
month leading to complete resolution of the lesion.                      PMID: 28452721.
                                                                     2. Barney E, Prose NS, Ramirez M. Inflammatory linear verrucous

Conclusions                                                              epidermal nevus treated successfully with crisaborole ointment
                                                                         in a 5-year-old boy. Pediatr Dermatol. 2019;36(3):404-405.
                                                                         DOI: 10.1111/pde.13793. PMID: 30838675.
ILVEN is a relatively rare disease and remains a clini-
                                                                     3. Tanita K, Fujimura T, Sato Y, Lyu C, Aiba S. Widely Spread
cal-pathological diagnosis. There are numerous differen-
                                                                         Unilateral Inflammatory Linear Verrucous Epidermal N    ­ evus
tial diagnoses to be taken into consideration such as: other             (ILVEN). Case Rep Dermatol. 2018;10(2):170-175. DOI:
epidermal nevi, Darier disease, linear porokeratosis, linear li-         10.1159/000489876. PMID: 30022937. PMCID: PMC6047550.
chen planus, linear psoriasis and lichen striatus. Our diagno-       4. Csuka EA, Ward SC, Ekelem C, Csuka DA, Ardigò M,
sis of ILVEN was based on pathognomonic clinical signs and               ­ esinkovska NA. Reflectance Confocal Microscopy, Optical
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                                                                         Coherence Tomography, and Multiphoton Microscopy in Inflam-
RCM-assessed pictures of inflammatory lichenoid dermatitis
                                                                         matory Skin Disease Diagnosis. Lasers Surg Med. 2021;53(6):
thus excluding both psoriasiform dermatitis and spongiotic
                                                                         776-797. DOI: 10.1002/lsm.23386. PMID: 33527483.
dermatitis [4]. In vivo RCM is a non-invasive imaging and             5. Scharf C, Argenziano G, Brancaccio G, Licata G, Ronchi A,
appears interesting for assessment of lesion in special areas            Moscarella E. Melanocytic or Not? Dermoscopy and Reflec-
and in pediatric patients [5,6]. The main limitation of RCM              tance Confocal Microscopy for Lesions Difficult to Diagnose:
is its penetration in dermis, reaching nowadays 350–400                  A Cross-Sectional Diagnostic Accuracy Study. Dermatol Pract
                                                                         Concept. 2021;11(4):e2021127. DOI: 10.5826/dpc.1104a127.
μm. This prevents imaging of structures located in the deep
                                                                         PMID: 34631270. PMCID: PMC8480465.
dermis, especially in cases of hyperpigmented or hyperkera-
                                                                      6. Ko RF, Smidt AC, Durkin JR. Reflectance confocal microscopy in
totic lesions, as, in these cases, there is strong contrast atten-       pediatric dermatology: A state-of-the-art review. Pediatr Derma-
uation because of absorption and scattering of light going               tol. 2021;38(6):1488-1499. DOI: 10.1111/pde.14837. PMID:
through those structures. However, the ILVEN histological                34651341.




Research Letter | Dermatol Pract Concept. 2023;13(2):e2023107                                                                          3
