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CORRESPONDENCE |
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| Year : 2022 | Volume
: 67
| Issue : 6 | Page : 755-756 |
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| Multiple hypo- and hyper-pigmentation: An example of twin spotting? |
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Li-wen Zhang1, Juan Wu2
1 From the Department of Dermatovenereology, Chengdu Second People's Hospital, Chengdu, Sichuan, China
2 Sexually Transmitted Disease Institute, Shanghai Skin Disease Hospital, School of Medicine, Tongji University, Shanghai, China
| Date of Web Publication | 23-Feb-2023 |
Correspondence Address:
Li-wen Zhang
From the Department of Dermatovenereology, Chengdu Second People's Hospital, Chengdu, Sichuan
China
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/ijd.ijd_651_21
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How to cite this article:
Zhang Lw, Wu J. Multiple hypo- and hyper-pigmentation: An example of twin spotting?. Indian J Dermatol 2022;67:755-6 |
Sir,
A 17-year-old boy visited our hospital complaining of cutaneous hypo- and hyper-pigmentation. An irregular hypopigmented macula on the left neck and back, and brown maculae on the left back and right lower leg were found shortly after birth [Figure 1]a and [Figure 1]b. About 10 years later, a progressively enlarging asymptomatic brown macula with tiny papules occurred on his right neck and shoulder [Figure 1]a. The patient was otherwise healthy and had no correlative family history. The Wood lamp examination showed off-white accentuation without chalky-white fluorescence in the hypopigmented region. The reflectance confocal microscopy revealed pigment decrease in the epidermal basal layer of the hypopigmented region [Figure 2]a, pigment increase in the epidermal basal layer of the brown maculae on the left back and right lower leg [Figure 2]b, and slight acanthosis and elongation along with fusion of the epidermal ridges showing mild hyperpigmentation of the epidermal basal layer on the right neck and shoulder [Figure 2]c. Based on these results and clinical manifestations, the patient was diagnosed with Becker's nevus, naevus depigmentosus, and café-au-lait spots (CALs). | Figure 1: (a) Multiple hypo- and hyper-pigmented maculae on the neck, back, and shoulder. (b) A brown macula on the right lower leg
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 | Figure 2: reflectance confocal microscopy: (a) Pigment decreases in the epidermal basal layer of the hypopigmented region on the left back (500 × 500 μm). (b) Pigment increases in the epidermal basal layer of the brown maculae on the left back (500 × 500 μm). (c) Acanthosis with slight hyperpigmentation of the epidermal basal layer on the right neck and shoulder (500 × 500 μm)
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Becker's nevus is characterized by a unilateral, hairy, pigmented macule usually involving the chest or scapular region. The plausible explanations for Becker's nevus include an androgen-dependent lesion and postzygotic mutations in beta-actin.[1] Naevus depigmentosus is classically defined as congenital, nonprogressive hypopigmentation with a localized, segmental, or systemic distribution. Naevus depigmentosus is a hypomelanotic disorder through postzygotic somatic mutation, giving rise to a reduced melanogenic potential in a clone of cells during embryogenesis.[2] CALs, characterized by brown maculae with increased melanin and melanocytes in the epiderm, are common birthmarks seen sporadically and in association with several genetic syndromes such as neurofibromatosis, Legius syndrome, familial multiple CALs and so on.[3]
We hereby reported an unusual coexistence case of Becker's nevus, naevus depigmentosus, and CALs. Afsar et al.[4] described a case of Becker's naevus with segmental naevus depigmentosus, and considered an example of twin spotting. The twin spots have been suggested for the combination of leukoderma and CALS in Bloom's syndrome, lentiginosis and nevus depigmentosus, congenital melanocytic nevus and nevus depigmentosus, as well as nevus telangiectaticus and nevus anemicus.[5],[6] Both different lesions have different recessive mutations occurring on the same chromosome and manifest visibly only in the rare event of somatic recombination occurring at an early developmental stage. This hypothesis requires further genetic studies involving more patients.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
 References | |  |
| 1. | Cai ED, Sun BK, Chiang A, Rogers A, Bernet L, Cheng B, et al. Postzygotic mutations in Beta-Actin are associated with Becker's nevus and Becker's nevus syndrome. J Invest Dermatol 2017;137:1795-8.  |
| 2. | Bolognia JL, Orlow SJ, Glick SA. Lines of blaschko. J Am Acad Dermatol 1994;31:157-90; quiz 190-2. |
| 3. | Lalor L, Davies OM, Basel D, Siegel DH. Cafe au lait spots: When and how to pursue their genetic origins. Clin Dermatol 2020;38:421-31. |
| 4. | Afsar FS, Aktas S, Ortac R. Becker's naevus and segmental naevus depigmentosus: An example of twin spotting? Australas J Dermatol 2007;48:224-6. |
| 5. | Alkemade H, Juhlin L. Unilateral lentiginosis with nevus depigmentosus on the other side. J Am Acad Dermatol 2000;43:361-3. |
| 6. | Keisham C, Thokchom N, Sarkar R. Twin spotting − A rare association with congenital melanocytic nevus and nevus depigmentosus. Pigment Int 2018;5:59. [Full text] |
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