Isolated nevus unius lateralis in a patient from Uganda
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Austin B. Ambur
and
Timothy A. Nyckowski
A 5-year-old male presented to our global health outreach clinic in Uganda in May 2018 for discoloration affecting the entire left side of his body since infancy. Cutaneous examination revealed unilateral distributed linear bands of skin-colored to dark-brown verrucous papules coalescing into plaques following Blaschko lines on the face, trunk, and extremities (Figures 1 and 2). Further examination revealed that the patient did not suffer from seizures, developmental delay, or visual and auditory disturbances. The history and clinical findings were most consistent with nevus unius lateralis.
Linear bands of skin-colored to dark-brown verrucous papules coalescing into plaques following Blaschko lines on the face, trunk, and upper extremity.
Linear bands of skin-colored to dark-brown verrucous papules coalescing into plaques following Blaschko lines on the face, trunk, and upper extremity with sharp demarcation at midline.
Nevus unius lateralis is a rare clinical variant of systematized epidermal nevus with an estimated prevalence of one in one million [1]. Patients classically present during infancy with linear patches or thin skin-colored to brown verrucous papules coalescing into plaques and typically darken and thicken around puberty. The lesions are commonly distributed on only one-half of the trunk and extremities following lines of Blaschko. The etiology is unknown, however gene mutations in FGFR3, HRAS, or PIK3CA may be involved [2]. While the diagnosis is made clinically, a biopsy may be useful to rule out additional causes of verrucous lesions following lines of Blaschko. The diagnosis is important for clinicians to recognize as it is rarely an isolated finding and may be associated with developmental delay, central nervous system tumors, seizure disorders, and auditory and visual deficiencies [2]. Treatment of the cutaneous lesions is challenging. Destructive therapy with ablative lasers or systemic therapy with oral retinoids have been utilized with variable success [2, 3].
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Research funding: None reported.
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Author contributions: Both authors provided substantial contributions to conception and design, acquisition of data, or analysis and interpretation of data; both authors drafted the article or revised it critically for important intellectual content; both authors gave final approval of the version of the article to be published; and both authors agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
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Competing interests: None reported.
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Informed consent: A parent of the patient described in this report provided written informed consent.
References
1. Pack, G, Sunderland, D. Naevus unius lateris. Arch Surg 1941;43:341–75. https://doi.org/10.1001/archsurg.1941.01210150017002.Search in Google Scholar
2. Bolognia, JL, Schaffer, JV, Cerroni, L. Chapter 109: benign epidermal tumors and proliferations. In: Dermatology, 4th ed. Philadelphia, PA: Elsevier; 2018:1910–1 pp.Search in Google Scholar
3. Alkhalifah, A, Fransen, F, Le Duff, F, Lacour, JP, Wolkerstorfer, A, Passeron, T. Laser treatment of epidermal nevi: a multicenter retrospective study with long-term follow-up. J Am Acad Dermatol 2020;83:1606–15. https://doi.org/10.1016/j.jaad.2019.06.013.Search in Google Scholar PubMed
© 2022 the author(s), published by De Gruyter, Berlin/Boston
This work is licensed under the Creative Commons Attribution 4.0 International License.
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- Frontmatter
- Innovations
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- Validation of cognitive and neuromuscular latencies using an iPad-based tool to quantify simple reaction times in asymptomatic subjects
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- The assessment of point-of-care-ultrasound (POCUS) in acute care settings is benefitted by early medical school integration and fellowship training
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