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
J.F. Val-Bernal
, S. Hermana, R. Sánchez
Autor para correspondencia
Department of Anatomical Pathology, Marqués de Valdecilla University Hospital, Medical Faculty, University of Cantabria and IDIVAL, Santander, Spain
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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" => "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>" ] ] ] "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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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 2 | 2 | 4 |
| 2024 Octubre | 57 | 42 | 99 |
| 2024 Septiembre | 63 | 31 | 94 |
| 2024 Agosto | 75 | 66 | 141 |
| 2024 Julio | 70 | 44 | 114 |
| 2024 Junio | 77 | 64 | 141 |
| 2024 Mayo | 40 | 37 | 77 |
| 2024 Abril | 57 | 30 | 87 |
| 2024 Marzo | 71 | 43 | 114 |
| 2024 Febrero | 60 | 44 | 104 |
| 2024 Enero | 55 | 35 | 90 |
| 2023 Diciembre | 63 | 25 | 88 |
| 2023 Noviembre | 47 | 29 | 76 |
| 2023 Octubre | 67 | 30 | 97 |
| 2023 Septiembre | 64 | 32 | 96 |
| 2023 Agosto | 50 | 20 | 70 |
| 2023 Julio | 98 | 50 | 148 |
| 2023 Junio | 49 | 31 | 80 |
| 2023 Mayo | 62 | 34 | 96 |
| 2023 Abril | 47 | 28 | 75 |
| 2023 Marzo | 49 | 23 | 72 |
| 2023 Febrero | 48 | 26 | 74 |
| 2023 Enero | 36 | 34 | 70 |
| 2022 Diciembre | 45 | 39 | 84 |
| 2022 Noviembre | 50 | 48 | 98 |
| 2022 Octubre | 53 | 39 | 92 |
| 2022 Septiembre | 47 | 50 | 97 |
| 2022 Agosto | 47 | 66 | 113 |
| 2022 Julio | 45 | 36 | 81 |
| 2022 Junio | 39 | 39 | 78 |
| 2022 Mayo | 27 | 37 | 64 |
| 2022 Abril | 31 | 45 | 76 |
| 2022 Marzo | 40 | 66 | 106 |
| 2022 Febrero | 29 | 31 | 60 |
| 2022 Enero | 29 | 41 | 70 |
| 2021 Diciembre | 23 | 38 | 61 |
| 2021 Noviembre | 44 | 41 | 85 |
| 2021 Octubre | 33 | 79 | 112 |
| 2021 Septiembre | 21 | 41 | 62 |
| 2021 Agosto | 24 | 32 | 56 |
| 2021 Julio | 31 | 49 | 80 |
| 2021 Junio | 26 | 43 | 69 |
| 2021 Mayo | 29 | 28 | 57 |
| 2021 Abril | 44 | 47 | 91 |
| 2021 Marzo | 60 | 36 | 96 |
| 2021 Febrero | 81 | 43 | 124 |
| 2021 Enero | 39 | 21 | 60 |
| 2020 Diciembre | 42 | 23 | 65 |
| 2020 Noviembre | 25 | 41 | 66 |
| 2020 Octubre | 35 | 32 | 67 |
| 2020 Septiembre | 31 | 30 | 61 |
| 2020 Agosto | 21 | 34 | 55 |
| 2020 Julio | 41 | 26 | 67 |
| 2020 Junio | 43 | 37 | 80 |
| 2020 Mayo | 28 | 47 | 75 |
| 2020 Abril | 29 | 23 | 52 |
| 2020 Marzo | 31 | 24 | 55 |
| 2020 Febrero | 2 | 10 | 12 |
| 2020 Enero | 4 | 3 | 7 |
| 2019 Diciembre | 4 | 7 | 11 |
| 2019 Noviembre | 4 | 4 | 8 |
| 2019 Octubre | 0 | 6 | 6 |
| 2019 Septiembre | 0 | 8 | 8 |
| 2019 Agosto | 4 | 7 | 11 |
| 2019 Julio | 4 | 8 | 12 |
| 2019 Junio | 4 | 0 | 4 |
| 2019 Mayo | 4 | 8 | 12 |
| 2019 Abril | 2 | 1 | 3 |
| 2019 Marzo | 2 | 6 | 8 |
| 2019 Febrero | 0 | 1 | 1 |
| 2019 Enero | 2 | 0 | 2 |
| 2018 Diciembre | 1 | 1 | 2 |
| 2018 Noviembre | 1 | 2 | 3 |
| 2018 Octubre | 1 | 1 | 2 |
| 2018 Septiembre | 5 | 2 | 7 |
| 2018 Agosto | 0 | 4 | 4 |
| 2018 Julio | 0 | 1 | 1 |
| 2018 Junio | 0 | 3 | 3 |
| 2018 Mayo | 0 | 13 | 13 |
| 2018 Abril | 0 | 2 | 2 |
| 2018 Marzo | 3 | 2 | 5 |
| 2018 Febrero | 23 | 3 | 26 |
| 2018 Enero | 34 | 4 | 38 |
| 2017 Diciembre | 8 | 8 | 16 |
| 2017 Noviembre | 8 | 9 | 17 |
| 2017 Octubre | 5 | 3 | 8 |
| 2017 Septiembre | 10 | 13 | 23 |
| 2017 Agosto | 6 | 7 | 13 |
| 2017 Julio | 14 | 7 | 21 |
| 2017 Junio | 13 | 25 | 38 |
| 2017 Mayo | 5 | 21 | 26 |
| 2017 Abril | 13 | 18 | 31 |
| 2017 Marzo | 9 | 41 | 50 |
| 2017 Febrero | 7 | 15 | 22 |
| 2017 Enero | 3 | 7 | 10 |
| 2016 Diciembre | 7 | 15 | 22 |
| 2016 Noviembre | 10 | 17 | 27 |
| 2016 Octubre | 6 | 14 | 20 |
| 2016 Septiembre | 0 | 4 | 4 |
| 2016 Agosto | 1 | 1 | 2 |
| 2016 Julio | 4 | 3 | 7 |
| 2016 Junio | 1 | 0 | 1 |
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