Case Letter
A case of linear atrophoderma of Moulin
Li-Wen Zhang, Meng-Sha Ma, Tao Chen
, Li-Xin Fu
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Department of Dermatovenereology, Chengdu Second People's Hospital, Sichuan, China
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The lesions were asymptomatic. There were no prior skin lesions or inflammation. There was no significant medical or family history. Physical examination found linear hyperpigmented atrophic patches on the right arm and right trunk following Blaschko's lines, involving both the anterior and posterior aspects. The skin was slightly atrophic on palpation. No signs of induration or inflammation were noted (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A and B). Laboratory investigations – including full blood count, erythrocyte sedimentation rate, liver function test, renal profile, and antinuclear antibodies – were all negative or within the normal range. Biopsy of a lesion showed a normal epidermis with increased pigmentation of the basal layer, with more compact dermal collagen and mild upper dermal perivascular lymphocytic infiltration (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Dermoscopy found multiple light brown networks with unclear margins. The patient was diagnosed with linear atrophoderma of Moulin (LAM) and started treatment with topical halometasone 0.5% cream and hydroquinone 2% cream for two months, with no improvement.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">LAM is a rare and distinct clinical entity characterized by acquired unilateral, hyperpigmented, and atrophic bandlike skin lesions following the lines of Blaschko, without prior inflammation or sclerotic appearance. It is named after Moulin, who, in 1992, reported on five patients with pigmented and more-or-less atrophic bands along Blaschko's lines.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> LAM usually progresses as a linear atrophic lesion in the first few months; then the lesion ceases to progress and persists. The etiology of LAM remains unclear. All reported cases were so far sporadic. It may be connected with gene mosaicism or autoimmunity. A study of the atrophic component of LAM by ultrasonography revealed that subcutaneous volume reduction was the cause of the atrophic appearance, not dermal atrophy.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Even though the clinical manifestation of LAM is rather unique, the histopathology of LAM is quite inconspicuous. Hematoxylin and eosin staining usually shows hyperpigmentation only in basal epidermal layers, without abnormal collagen or elastic fibers in the dermis or any obvious inflammation.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> There may be some perivascular lymphocytic infiltration, acanthosis, epidermal atrophy, altered collagen in the dermis, and decreased or fragmented elastic tissue.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Lopez et al.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> proposed the following diagnostic criteria for -LAM, including: (1) Onset during childhood or adolescence; (2) Development of hyperpigmented, slightly atrophic, unilateral lesions following Blaschko lines on the trunk or limbs; (3) Absence of prior inflammation or subsequent scleroderma; (4) A stable, non-progressive clinical course without a pattern of remission; (5) Histologic findings showing hyperpigmentation of the basal epidermis and a normal dermis with unaltered connective tissue and elastic fibers. Up to now, more than 30 cases of LAM have been reported in the literature. However, the condition may be overestimated. If the diagnostic criteria are strictly adhered to, the diagnosis of LAM cannot be reached in some cases, as these authors reported histologic findings that are compatible with other clinical entities.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">LAM must be differentiated from atrophoderma of Pasini and Pierini (APP), which also presents with similar configuration, atrophy, and hyperpigmentation, but does not follow Blaschko's lines. In addition, LAM is different from linear morphea, which usually presents preceding inflammation, induration, or scleroderma.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Histopathologically, morphea shows collagen bundles that are closely packed and oriented horizontally, and dermal appendages and subcutaneous fat are progressively lost. However, it is still debated whether LAM is a distinct entity. There are many clinical and histologic similarities between LAM, APP, and morphea, thus some of the literature suggests that these diseases represent part of a disease spectrum, and that LAM may not be a distinct entity.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> LAM may be a Blaschko-linear variant of APP, and APP may be considered to be an abortive form of morphea.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">There is no effective treatment for LAM. Topical corticosteroids and heparin were not helpful. Some trial treatments showed partial response to LAM, including the following: topical calcipotriol, systemic methotrexate or aminobenzoate, and intralesional platelet-rich plasma therapy.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> The current report presented a case of LAM with classic clinical and histopathological features.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0035" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors’ contribution</span><p id="par0040" class="elsevierStylePara elsevierViewall">Li-Wen Zhang and Meng-Sha Ma contributed equally to this work. Li-Wen Zhang: Approval of the final version of the manuscript; conception and planning of the study; composition of the manuscript; critical review of the literature; critical review of the manuscript.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Meng-Sha Ma: Collection, analysis, and interpretation of data; critical review of the manuscript.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Tao Chen: Approval of the final version of the manuscript; conception and planning of the study.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Li-Xin Fu: Critical review of the literature.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0060" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Financial support" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Authors’ contribution" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2019-01-18" "fechaAceptado" => "2019-03-18" "NotaPie" => array:2 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">How to cite this article: Zhang L-W, Ma M-S, Chen T, Fu L-X. A case of linear atrophoderma of Moulin. An Bras Dermatol. 2020;95:119–21.</p>" ] 1 => array:2 [ "etiqueta" => "☆☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Study conducted at the Chengdu Second People's Hospital, Sichuan, China.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2648 "Ancho" => 1011 "Tamanyo" => 332513 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Linear hyperpigmented patches on the right arm and right trunk following Blaschko's lines. (B) Linear hyperpigmented patches on the right arm and right trunk following Blaschko's lines.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 941 "Ancho" => 1007 "Tamanyo" => 173673 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A normal epidermis with increased pigmentation of the basal layer, with more compact dermal collagen and mild upper dermal perivascular lymphocytic infiltration (Hematoxylin & eosin, ×40).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Acquired atrophic pigmented band-like lesions following Blaschko's lines" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "G. 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Herrera" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Actas Dermosifiliogr" "fecha" => "2008" "volumen" => "99" "paginaInicial" => "165" "paginaFinal" => "167" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/18346448" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Linear atrophoderma of Moulin: a distinct entity?" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "E. de Golian" 1 => "K. Echols" 2 => "H. Pearl" 3 => "L. Davis" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1111/pde.12003" "Revista" => array:6 [ "tituloSerie" => "Pediatr Dermatol" "fecha" => "2014" "volumen" => "31" "paginaInicial" => "373" "paginaFinal" => "377" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/23046463" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Linear atrophoderma of Moulin: a rare entity" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "V.D. Kharkar" 1 => "B.A. Abak" 2 => "S.A. 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| Ano/Mês | Html | Total | |
|---|---|---|---|
| 2024 Novembro | 19 | 15 | 34 |
| 2024 Outubro | 109 | 89 | 198 |
| 2024 Setembro | 138 | 96 | 234 |
| 2024 Agosto | 146 | 112 | 258 |
| 2024 Julho | 136 | 131 | 267 |
| 2024 Junho | 101 | 76 | 177 |
| 2024 Maio | 104 | 62 | 166 |
| 2024 Abril | 116 | 89 | 205 |
| 2024 Março | 108 | 74 | 182 |
| 2024 Fevereiro | 83 | 67 | 150 |
| 2024 Janeiro | 66 | 38 | 104 |
| 2023 Dezembro | 62 | 61 | 123 |
| 2023 Novembro | 95 | 91 | 186 |
| 2023 Outubro | 66 | 83 | 149 |
| 2023 Setembro | 74 | 73 | 147 |
| 2023 Agosto | 51 | 46 | 97 |
| 2023 Julho | 57 | 40 | 97 |
| 2023 Junho | 68 | 44 | 112 |
| 2023 Maio | 55 | 24 | 79 |
| 2023 Abril | 51 | 23 | 74 |
| 2023 Março | 57 | 46 | 103 |
| 2023 Fevereiro | 48 | 31 | 79 |
| 2023 Janeiro | 42 | 44 | 86 |
| 2022 Dezembro | 45 | 32 | 77 |
| 2022 Novembro | 58 | 52 | 110 |
| 2022 Outubro | 112 | 48 | 160 |
| 2022 Setembro | 49 | 55 | 104 |
| 2022 Agosto | 48 | 59 | 107 |
| 2022 Julho | 48 | 53 | 101 |
| 2022 Junho | 79 | 54 | 133 |
| 2022 Maio | 38 | 48 | 86 |
| 2022 Abril | 47 | 53 | 100 |
| 2022 Março | 47 | 68 | 115 |
| 2022 Fevereiro | 26 | 31 | 57 |
| 2022 Janeiro | 47 | 75 | 122 |
| 2021 Dezembro | 37 | 52 | 89 |
| 2021 Novembro | 33 | 61 | 94 |
| 2021 Outubro | 58 | 81 | 139 |
| 2021 Setembro | 33 | 50 | 83 |
| 2021 Agosto | 43 | 55 | 98 |
| 2021 Julho | 37 | 49 | 86 |
| 2021 Junho | 37 | 49 | 86 |
| 2021 Maio | 66 | 68 | 134 |
| 2021 Abril | 106 | 207 | 313 |
| 2021 Março | 85 | 60 | 145 |
| 2021 Fevereiro | 32 | 14 | 46 |
| 2021 Janeiro | 23 | 20 | 43 |
| 2020 Dezembro | 49 | 18 | 67 |
| 2020 Novembro | 97 | 12 | 109 |
| 2020 Outubro | 26 | 11 | 37 |
| 2020 Setembro | 17 | 19 | 36 |
| 2020 Agosto | 17 | 7 | 24 |
| 2020 Julho | 19 | 9 | 28 |
| 2020 Junho | 14 | 13 | 27 |
| 2020 Maio | 22 | 13 | 35 |
| 2020 Abril | 1 | 1 | 2 |
| 2020 Março | 6 | 2 | 8 |
| 2020 Fevereiro | 0 | 1 | 1 |
| 2020 Janeiro | 0 | 8 | 8 |
| 2019 Dezembro | 0 | 1 | 1 |
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