Case and Research Letter
Intradermal Lipoblastoma-Like Tumor of the Lip in an Adult Woman
Lipoblastoma intradérmico tumor tipo en el labio de una mujer adulta
Department of Anatomical Pathology, Marqués de Valdecilla University Hospital, Medical Faculty, University of Cantabria and IDIVAL, Santander, Spain
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(B) Close-up view.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "H. Hakenoshita, T. Yamamoto" "autores" => array:2 [ 0 => array:2 [ "nombre" => "H." "apellidos" => "Hakenoshita" ] 1 => array:2 [ "nombre" => "T." 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A, At the first visit. B, 2 months later, with self-resolution. Histological appearance of the biopsies corresponding to the clinical images immediately above, with partial reduction of the tumor infiltrate in the second of them. C, Hematoxylin-eosin (H-E), x40. D, H-E, x400.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "M.R. García de la Fuente, F. Vilardell-Villellas, R.M. Martí-Laborda, J.M. Casanova-Seuma" "autores" => array:4 [ 0 => array:2 [ "nombre" => "M.R." "apellidos" => "García de la Fuente" ] 1 => array:2 [ "nombre" => "F." "apellidos" => "Vilardell-Villellas" ] 2 => array:2 [ "nombre" => "R.M." "apellidos" => "Martí-Laborda" ] 3 => array:2 [ "nombre" => "J.M." "apellidos" => "Casanova-Seuma" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0001731015003361" "doi" => "10.1016/j.ad.2015.04.018" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731015003361?idApp=UINPBA000044" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219015003273?idApp=UINPBA000044" "url" => "/15782190/0000010700000001/v2_201704190205/S1578219015003273/v2_201704190205/en/main.assets" ] "en" => array:14 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case and Research Letter</span>" "titulo" => "Intradermal Lipoblastoma-Like Tumor of the Lip in an Adult Woman" "tieneTextoCompleto" => true "saludo" => "Dear Editor:" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "88" "paginaFinal" => "90" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "J.F. Val-Bernal, S. Hermana, R. Sánchez" "autores" => array:3 [ 0 => array:4 [ "nombre" => "J.F." "apellidos" => "Val-Bernal" "email" => array:1 [ 0 => "apavbj@humv.es" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "S." "apellidos" => "Hermana" ] 2 => array:2 [ "nombre" => "R." "apellidos" => "Sánchez" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Department of Anatomical Pathology, Marqués de Valdecilla University Hospital, Medical Faculty, University of Cantabria and IDIVAL, Santander, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Lipoblastoma intradérmico tumor tipo en el labio de una mujer adulta" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1415 "Ancho" => 1050 "Tamanyo" => 364001 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Multivacuolated and univacuolated lipoblasts constitute most of the dermal fat tissue. Mature adipocytes are scant (hematoxylin and eosin, original magnification ×200).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Lipoblastoma is a benign adipocytic tumor consisting of lobulated, immature white fat cells with a prominent capillary pattern and, frequently, a focal myxoid matrix.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> The tumor can be localized or diffuse, occurs predominantly in infancy and early childhood, and affects patients aged 5 years or younger in 84% of cases.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Nevertheless, rare cases have been reported in adolescents and adults.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3,4</span></a> Most lipoblastomas are subcutaneous lesions affecting the arms and legs, and they are more common in male patients.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a distinctive mesenchymal neoplasm of the lip showing adipocytic differentiation in an adult woman. We consider that this tumor is akin to infantile lipoblastoma. As far as we are aware, only 1 such lesion has been previously reported.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">A 36-year-old woman presented with a history of a small asymptomatic, pigmented, raised nodule on the vermilion border of the lower lip that had been present for about 9 months. The patient had not undergone lip piercing, filler injection for lip augmentation, or any other interventions. Her past medical history was not significant for systemic disease. Physical examination revealed a soft, brown-colored, sessile nodule measuring 0.8<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>0.4<span class="elsevierStyleHsp" style=""></span>cm on the right half of the lower lip. The clinical diagnosis was melanocytic nevus. The lesion was completely removed.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The microscopic study showed a poorly demarcated lesion, with epidermal acanthosis, irregular papillomatosis, and hyperpigmentation of the basal layer. Scattered aggregates of immature fat cells replaced much of the submucosa (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), with a preferential perivascular location. These cells showed variation in size and were multivacuolated lipoblasts, which indented the nuclei and were interspersed with univacuolated lipoblasts that displaced the nuclei and looked like signet-ring fat cells. Mature adipocytes were scant (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Hibernoma-like cells were not seen. All the nuclei were small and monomorphic without atypia. Mitotic figures were absent. No giant cells, lymphocytes, mucin deposits, or plexiform vascularization were observed. Dermal aggregates were surrounded by numerous small vessels, and were incompletely separated by the thick collagenous bundles of dermis. In the depth of the lesion, lipoblast aggregates were seen to form part of the adventitia of small veins or to infiltrate between the fascicles of the orbicularis muscle (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). No birefringence was observed in the adipocytic cells. Alcian blue (pH 2.5) did not show any acid mucin in the fat cell groups or in the dermal connective tissue. The cells stained positively for vimentin and retinoblastoma antibody and negatively for AE1/AE3 cytokeratin, CD34, CD31, CD68, and estrogen receptor. Immature adipocytes showed moderate amounts of cytoplasmic mitochondria around fat vacuoles that were visible with the antimitochondrial antibody. The amount of mitochondria was not comparable to that observed in the brown adipose tissue cells. The neoplasm involved the surgical margins.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">There has been no local recurrence or metastasis in the 9 months since surgery.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The tumor in the case reported here shared histological features with lipoblastoma, namely prominent lobulation, immature fat cells consisting of multivacuolated and univacuolated lipoblasts admixed with mature fat cells, conspicuous capillaries, variably developed collagenous septae, and diffuse borders. The clinical manifestations suggested a diagnosis of melanocytic nevus due to the concomitant pigmented epidermal lesion. We interpret the epidermal lesion as a reactive hyperplasia that may accompany lesions of this kind.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Lipoblastoma occurs primarily in infancy and early childhood, but it can be observed in adolescents and young adults.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> Lae et al.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> described 2 cases in women aged 23 and 38 years, and Silverman et al.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> reported a recurring lipoblastoma in a 48-year-old man.</p><p id="par0040" class="elsevierStylePara elsevierViewall">To our knowledge, the present case is the second report of a lipoblastoma-like tumor of the lip. Jeong et al.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> reported a similar intradermal lesion involving the lower lip of a 61-year-old woman. The dermal tumor was erythematous, slightly elevated, and measured 2<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>1<span class="elsevierStyleHsp" style=""></span>cm. The clinical differential diagnosis included Fordyce spot, nevus lipomatosus superficialis, and foreign body granuloma. The excised specimen showed abundant dermal lobules composed of lipoblasts and mature cells. The epidermis above the dermal tumor showed acanthosis and irregular papillomatosis, which is a change similar to that observed in the case we are reporting. Neither local recurrence nor metastasis was observed 1 year after surgery.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Lipoblastoma is a benign tumor, but it can return in 9% of cases.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Recurrence may occur in the form of a mature lipomatous tumor. The differential diagnosis includes other dermal tumors with lipoblasts such as atypical lipomatous tumor/well-differentiated liposarcoma, spindle cell/pleomorphic lipoma, and myxoid liposarcoma.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The nature of lipoblastoma remains to be explained. The structure of the lesion is reminiscent of the development of white adipose tissue. White embryonal/fetal fat is characterized by a very close association between adipocyte development and angiogenesis. Both small univacuolated and multivacuolated adipocytes form lobules with abundant delicate branching capillaries. It has been proposed that the white adipocyte precursor is derived from undifferentiated mesenchymal cells that reside in the mural cell compartment of the adipose vasculature.<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">8–10</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The embryonal/fetal structure of the lipoblastoma and its occurrence primarily in the very young suggests a hamartomatous lesion.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1415 "Ancho" => 1050 "Tamanyo" => 488116 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Partial view of epidermal acanthosis, papillomatosis, and basal hyperpigmentation. Scattered aggregates of immature fat cells are seen in the submucosa (hematoxylin and eosin, original magnification ×100).</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" => 1415 "Ancho" => 1050 "Tamanyo" => 364001 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Multivacuolated and univacuolated lipoblasts constitute most of the dermal fat tissue. Mature adipocytes are scant (hematoxylin and eosin, original magnification ×200).</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1415 "Ancho" => 1050 "Tamanyo" => 345218 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Immature adipocytes can be seen infiltrating between the fascicles of the orbicularis muscle (hematoxylin and eosin, original magnification ×100).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Tumors of the soft tissues. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 6 | 7 | 13 |
| 2024 October | 65 | 39 | 104 |
| 2024 September | 76 | 25 | 101 |
| 2024 August | 96 | 59 | 155 |
| 2024 July | 73 | 47 | 120 |
| 2024 June | 70 | 32 | 102 |
| 2024 May | 64 | 34 | 98 |
| 2024 April | 71 | 25 | 96 |
| 2024 March | 77 | 30 | 107 |
| 2024 February | 67 | 35 | 102 |
| 2024 January | 45 | 38 | 83 |
| 2023 December | 59 | 19 | 78 |
| 2023 November | 65 | 41 | 106 |
| 2023 October | 70 | 25 | 95 |
| 2023 September | 67 | 25 | 92 |
| 2023 August | 44 | 21 | 65 |
| 2023 July | 74 | 30 | 104 |
| 2023 June | 65 | 23 | 88 |
| 2023 May | 59 | 25 | 84 |
| 2023 April | 57 | 22 | 79 |
| 2023 March | 65 | 24 | 89 |
| 2023 February | 56 | 26 | 82 |
| 2023 January | 40 | 56 | 96 |
| 2022 December | 44 | 38 | 82 |
| 2022 November | 33 | 28 | 61 |
| 2022 October | 24 | 24 | 48 |
| 2022 September | 24 | 30 | 54 |
| 2022 August | 30 | 46 | 76 |
| 2022 July | 17 | 36 | 53 |
| 2022 June | 20 | 30 | 50 |
| 2022 May | 50 | 37 | 87 |
| 2022 April | 59 | 33 | 92 |
| 2022 March | 54 | 45 | 99 |
| 2022 February | 39 | 29 | 68 |
| 2022 January | 63 | 42 | 105 |
| 2021 December | 54 | 29 | 83 |
| 2021 November | 42 | 52 | 94 |
| 2021 October | 51 | 45 | 96 |
| 2021 September | 54 | 43 | 97 |
| 2021 August | 69 | 22 | 91 |
| 2021 July | 69 | 33 | 102 |
| 2021 June | 57 | 24 | 81 |
| 2021 May | 36 | 34 | 70 |
| 2021 April | 91 | 30 | 121 |
| 2021 March | 48 | 22 | 70 |
| 2021 February | 77 | 25 | 102 |
| 2021 January | 39 | 15 | 54 |
| 2020 December | 28 | 8 | 36 |
| 2020 November | 29 | 20 | 49 |
| 2020 October | 22 | 18 | 40 |
| 2020 September | 32 | 10 | 42 |
| 2020 August | 31 | 14 | 45 |
| 2020 July | 17 | 15 | 32 |
| 2020 June | 22 | 24 | 46 |
| 2020 May | 15 | 19 | 34 |
| 2020 April | 26 | 18 | 44 |
| 2020 March | 17 | 19 | 36 |
| 2020 February | 4 | 0 | 4 |
| 2020 January | 4 | 1 | 5 |
| 2019 December | 4 | 0 | 4 |
| 2019 November | 4 | 0 | 4 |
| 2019 October | 0 | 3 | 3 |
| 2019 August | 4 | 0 | 4 |
| 2019 July | 4 | 0 | 4 |
| 2019 June | 4 | 0 | 4 |
| 2019 May | 5 | 2 | 7 |
| 2019 April | 3 | 0 | 3 |
| 2019 March | 2 | 5 | 7 |
| 2019 February | 5 | 0 | 5 |
| 2019 January | 3 | 0 | 3 |
| 2018 November | 17 | 0 | 17 |
| 2018 October | 10 | 0 | 10 |
| 2018 September | 6 | 0 | 6 |
| 2018 March | 2 | 1 | 3 |
| 2018 February | 24 | 2 | 26 |
| 2018 January | 33 | 5 | 38 |
| 2017 December | 32 | 3 | 35 |
| 2017 November | 12 | 7 | 19 |
| 2017 October | 23 | 4 | 27 |
| 2017 September | 10 | 5 | 15 |
| 2017 August | 21 | 7 | 28 |
| 2017 July | 22 | 9 | 31 |
| 2017 June | 26 | 10 | 36 |
| 2017 May | 25 | 3 | 28 |
| 2017 April | 30 | 8 | 38 |
| 2017 March | 5 | 5 | 10 |
| 2017 February | 13 | 4 | 17 |
| 2017 January | 19 | 7 | 26 |
| 2016 December | 14 | 11 | 25 |
| 2016 November | 25 | 10 | 35 |
| 2016 October | 13 | 11 | 24 |
| 2016 September | 0 | 3 | 3 |
| 2016 August | 0 | 3 | 3 |
| 2016 June | 0 | 4 | 4 |
| 2016 May | 0 | 6 | 6 |
| 2016 April | 0 | 1 | 1 |
| 2016 March | 0 | 7 | 7 |
| 2016 February | 0 | 4 | 4 |
| 2016 January | 0 | 2 | 2 |
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