10.5826/dpc.1401a14
text/xml
2160-9381
3359
en
Camila Downey
https://creativecommons.org/licenses/by-nc/4.0
Dermatology Practical & Conceptual
pigmentary disorders
Lingual Epidermal Choristoma: Ultrasonography as a Diagnostic Tool
Text.Serial.Journal
https://dpcj.org/index.php/dpc
Research Letter
2024-01-31
2023-03-22
2024-01-31
2024-02-21
e2024014
Dermatol Pract Concept
Camila Downey
2024/01/31
10.5826/dpc.1401a14
e2024014
Department of Dermatology, Clínica Alemana de Santiago, Department of Dermatology, Hospital Luis Calvo Mackenna, Santiago, Chile
2160-9381
pigmentary disorders
en
e2024014
Dong SZ, Zhu M, Bulas D. Techniques for minimizing sedation in pediatric MRI. J Magn Reson Imaging. 2019;50(4):1047-1054. DOI: 10.1002/jmri.26703.
Lingual Epidermal Choristoma: Ultrasonography as a Diagnostic Tool
https://dpcj.org/index.php/dpc/article/download/3359/2554
     Lingual Epidermal Choristoma: Ultrasonography
                   as a Diagnostic Tool
                      Constanza del Puerto1, Ximena Wortsman2,3, Camila Downey1,4


1 Department of Dermatology, Clínica Alemana de Santiago, Santiago, Chile
2 Institute for Diagnostic Imaging and Research of the Skin and Soft Tissues Clinic, Santiago, Chile
3 Department of Dermatology, School of Medicine, Pontificia Universidad Católica de Chile, Santiago, Chile
4 Department of Dermatology, Hospital Luis Calvo Mackenna, Santiago, Chile



Key words: choristoma, ultrasonography, mucous membrane disorders, pigmentary disorders
Citation: del Puerto C, Wortsman X, Downey C. Lingual Epidermal Choristoma: Ultrasonography as a Diagnostic Tool. Dermatol Pract
Concept. 2024;14(1):e2024014. DOI: https://doi.org/10.5826/dpc.1401a14
Accepted: May 31, 2023; Published: January 2024
Copyright: ©2024 del Puerto 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: Constanza del Puerto, Department of Dermatology, Clínica Alemana de Santiago, Santiago, Chile. 5951
Vitacura Avenue, Santiago, Chile. Email address: conidelpuerto@gmail.com




Introduction                                                             patient family, clinical observation was chosen. The macules
                                                                         grew proportionally to the child’s development, becoming
Choristoma is a rare benign congenital malformation                      palpable and with surface changes at age of five (Figure 1B).
­consisting of normal growth of cells or tissue that is foreign          High frequency and ultra-high frequency color Doppler ul-
to that body site [1]. Epidermal choristoma (EC) is ectopic              trasound demonstrated submucosal thickening with multi-
epidermal tissue. It is a rare benign lesion on the tongue that          ple hypoechogenic oval structures, consistent with sebaceous
presents as solitary or multiple brown macules or papules [2].           glands, without hypervascularity (Figure 2, A-C). The find-
Recent publications suggest clinical follow-up ­considering              ings were consistent with lingual EC.
its benign course [2]. We report a patient with an EC with
a 5-year follow-up and whose diagnosis was supported by
                                                                         Conclusions
high-definition ultrasonography.
                                                                         The presence of sebaceous glands in the oral mucosa is not

Case Presentation                                                        uncommon, but on the tongue is exceptional and is consid-
                                                                         ered an EC [3]. Most cases present on the tongue in males
A healthy 2-month-old boy consulted for three light-brown                before the age of 2 [2]. It is clinically characterized by one
homogeneous macules on the right dorsal aspect of the                    or multiple macules or papules unilaterally or in the mid-
tongue (Figure 1A). Congenital oral melanotic macules and                line dorsal tongue. Color varies from light to dark brown;
epidermal choristoma were included in the differential di-               therefore, it is usually misdiagnosed as a congenital mela-
agnosis. After discussing management alternatives with the               notic macule [1,2]. The histopathological study shows the



Research Letter | Dermatol Pract Concept. 2024;14(1):e2024014                                                                        1
                       Figure 1. (A,B) (A) Three light-brown homogeneous macules on the dorsum of the tongue
                       when the patient was 2 months old. (B) At the age of 5, 3 light-brown homogeneous now
                       palpable lesions on the right dorsum of the tongue.




Figure 2. (A-C) Ultrasound at 70 MHz (Vevo MD; Visualsonics Fujifilm) of the tongue (transverse views) demonstrates multiple s­ ebaceous
glands as hyperechoic oval-shaped submucosal structures on the right side of the tongue (A) that were delineated with a white color (B).
Notice the difference with the left side of the tongue (C) that does not present sebaceous glands.


replacement of normal oral mucosal epithelium by strat-               sedation, with all the risks it implies, including 0.4%-17% [6]
ified squamous epithelium, hyperkeratosis, pigmentation               of acute complications and yet unknown long-term neuro-
of the basal layer, abortive hair follicles, and sebaceous            logical side effects. For this reason, we propose that ultra-
glands [1].                                                           sound and particularly ultra-high frequency ultrasound, may
    In the case of congenital exclusively lingual pigmenta-           be an alternative for the diagnosis and monitoring of EC in
tion, most relevant differential diagnoses are congenital lin-        cases where families and physicians decide to perform active
gual melanotic macule (CLMM) and EC. Clinical clues to                clinical observation, especially considering that oral congeni-
differentiate EC from CLMM are that EC usually becomes a              tal melanoma has not been reported yet, the difficulty of oral
palpable lesion over time, whereas CLMM usually presents              biopsy in a neonate and that sedation under three years of
as highly pigmented macules in dark skinned patients [4].             age may carry risks.
    High and ultra-high frequency color Doppler ultrasound
have gained relevance in skin and mucosal conditions and
can be useful in children where an oral cavity biopsy proce-
                                                                      References
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2                                                                         Research Letter | Dermatol Pract Concept. 2024;14(1):e2024014
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Research Letter | Dermatol Pract Concept. 2024;14(1):e2024014                                                                          3
