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Sanchis-Sánchez, A. Mateu-Puchades, F. García-Herreros" "autores" => array:3 [ 0 => array:4 [ "nombre" => "C." "apellidos" => "Sanchis-Sánchez" "email" => array:1 [ 0 => "celia_sanchis@hotmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "A." "apellidos" => "Mateu-Puchades" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "F." "apellidos" => "García-Herreros" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Universitario Doctor Peset, Valencia, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital Universitario Doctor Peset, Valencia, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Nódulos en las manos y anomalías músculo-esqueléticas" ] ] "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" => 616 "Ancho" => 1667 "Tamanyo" => 359343 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Hematoxylin-eosin, original magnification<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>40. B, Hematoxylin-eosin, original magnification<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>100.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Medical History</span><p id="par0005" class="elsevierStylePara elsevierViewall">A 28-year-old man with no family history of interest presented with asymptomatic nodules on the hands that had first appeared during adolescence. The patient had a history of panhypopituitarism secondary to the excision of a cerebral enchondroma and had undergone surgery to lengthen the right femur and tibia.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Physical Examination</span><p id="par0010" class="elsevierStylePara elsevierViewall">Physical examination revealed 2 types of subcutaneous nodules: some that were soft, mobile, and violaceous, with a vascular appearance (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A) and others that were indurated to the touch, nonmobile, and the color of normal skin, suggesting a bone origin (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). Marked scoliosis, dysmetria, and shortening and varus deformity of the right extremities were also present.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Histopathology</span><p id="par0015" class="elsevierStylePara elsevierViewall">Biopsy of a lesion with a vascular appearance revealed a well-defined nodule in the dermis composed of dilated vascular channels delimited by a thin endothelium. Also present were more solid areas composed of fascicles of spindle cells (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A), interspersed with a few round cells with vacuolated cytoplasm (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>B).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Additional Tests</span><p id="par0020" class="elsevierStylePara elsevierViewall">Radiography (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>A) and magnetic resonance imaging revealed multiple irregular thin-walled images in the phalanges and metacarpals. Bone scintigraphy with technetium-99m hydroxy diphosphonate (<span class="elsevierStyleSup">99m</span>Tc-HDP) revealed multiple foci of increased uptake of the radiotracer, predominantly in the long bones of the right side of the body (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>B).</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleBold">What Is Your Diagnosis?</span></p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Diagnosis</span><p id="par0030" class="elsevierStylePara elsevierViewall">Maffucci syndrome.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical Course and Treatment</span><p id="par0035" class="elsevierStylePara elsevierViewall">After 2 years of periodic follow-up visits the patient has not developed any malignancies.</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Comment</span><p id="par0040" class="elsevierStylePara elsevierViewall">Maffucci syndrome is a rare disorder. Only about 200 cases have been reported since it was first described in 1881. The syndrome is caused by a congenital, nonhereditary mesodermal dysplasia and is characterized by the triad of enchondromas, multiple vascular lesions, and musculoskeletal anomalies.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1–6</span></a> The etiology is unknown, although recent articles suggest that it could be caused by a postzygotic somatic mutation in the enzymes isocitrate dehydrogenase 1 and 2.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Patients tend to be asymptomatic at birth and develop lesions in childhood or adolescence. The lesions are distributed asymmetrically, predominantly on one side of the body and on distal parts of the extremities.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1–3</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Enchondromas are benign cartilaginous tumors clinically characterized as subcutaneous nodules the color of normal skin and with the consistency of bone. Although enchondromas are benign lesions, they can present complications such as deformities or fractures. The most important complication is the possibility of malignant transformation to chondrosarcoma, which occurs in up to 40% of cases. Radiologic studies show irregular images with thin walls that are insufflated but not destroyed and bone scintigraphy shows areas of increased uptake. In most cases, treatment is not necessary except in the event of complications, in which case surgery is the therapeutic option of choice.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1–6</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The vascular lesions present clinically as soft bluish or violaceous subcutaneous nodules. There are 3 types: venous malformations, lymphatic malformations, and spindle cell hemangioma, the last being the most characteristic type.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1–6</span></a> Spindle cell hemangioma is a benign vascular proliferation that is histologically composed of 3 components: dilated vascular channels, fascicles of spindle cells, and round cells with vacuolated cytoplasm.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">3,4</span></a> The risk of malignant transformation of the vascular lesions is not established, although it appears to be rare. In fact, only 7 cases of malignant transformation have been reported, specifically 6 angiosarcomas and 1 lymphangiosarcoma, although some of these cases developed at sites exposed to radiotherapy, so the role of radiotherapy in their development is not known.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> Treatment is only required in selected cases and, although surgery is the therapeutic option of choice, recurrences are frequent. In recent years, there have been reports of cases treated with rapamycin, with variable results.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">In addition to the risk of malignant transformation of the enchondromas and vascular lesions, Maffucci syndrome can also be associated with other malignancies such as astrocytomas or pituitary adenomas.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Therefore, although there are no established follow-up protocols, close monitoring of these patients is essential for early detection of possible neoplasms.</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of Interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Medical History" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Physical Examination" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Histopathology" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Additional Tests" ] 4 => array:2 [ "identificador" => "sec0025" "titulo" => "Diagnosis" ] 5 => array:2 [ "identificador" => "sec0030" "titulo" => "Clinical Course and Treatment" ] 6 => array:2 [ "identificador" => "sec0035" "titulo" => "Comment" ] 7 => array:2 [ "identificador" => "sec0040" "titulo" => "Conflicts of Interest" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Sanchis-Sánchez C, Mateu-Puchades A, García-Herreros F. Nódulos en las manos y anomalías músculo-esqueléticas. Actas Dermosifiliogr. 2017;108:861–862.</p>" ] ] "multimedia" => array:3 [ 0 => array:6 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 618 "Ancho" => 1667 "Tamanyo" => 106726 ] ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 616 "Ancho" => 1667 "Tamanyo" => 359343 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Hematoxylin-eosin, original magnification<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>40. B, Hematoxylin-eosin, original magnification<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>100.</p>" ] ] 2 => array:6 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1272 "Ancho" => 1667 "Tamanyo" => 181821 ] ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0035" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Vascular malformations. Part II: Associated syndromes" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "M.C. Garzon" 1 => "J.T. Huang" 2 => "O. Enjolras" 3 => "I.J. 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año/Mes | Html | Total | |
---|---|---|---|
2024 Noviembre | 16 | 8 | 24 |
2024 Octubre | 84 | 34 | 118 |
2024 Septiembre | 111 | 25 | 136 |
2024 Agosto | 127 | 74 | 201 |
2024 Julio | 96 | 30 | 126 |
2024 Junio | 92 | 33 | 125 |
2024 Mayo | 86 | 32 | 118 |
2024 Abril | 84 | 19 | 103 |
2024 Marzo | 77 | 32 | 109 |
2024 Febrero | 74 | 29 | 103 |
2024 Enero | 64 | 29 | 93 |
2023 Diciembre | 63 | 17 | 80 |
2023 Noviembre | 71 | 18 | 89 |
2023 Octubre | 104 | 20 | 124 |
2023 Septiembre | 67 | 30 | 97 |
2023 Agosto | 41 | 24 | 65 |
2023 Julio | 74 | 23 | 97 |
2023 Junio | 59 | 21 | 80 |
2023 Mayo | 65 | 18 | 83 |
2023 Abril | 55 | 16 | 71 |
2023 Marzo | 54 | 30 | 84 |
2023 Febrero | 68 | 15 | 83 |
2023 Enero | 38 | 24 | 62 |
2022 Diciembre | 52 | 29 | 81 |
2022 Noviembre | 29 | 29 | 58 |
2022 Octubre | 29 | 15 | 44 |
2022 Septiembre | 47 | 30 | 77 |
2022 Agosto | 29 | 33 | 62 |
2022 Julio | 40 | 42 | 82 |
2022 Junio | 35 | 18 | 53 |
2022 Mayo | 55 | 33 | 88 |
2022 Abril | 58 | 28 | 86 |
2022 Marzo | 57 | 55 | 112 |
2022 Febrero | 41 | 33 | 74 |
2022 Enero | 40 | 29 | 69 |
2021 Diciembre | 39 | 35 | 74 |
2021 Noviembre | 43 | 40 | 83 |
2021 Octubre | 41 | 53 | 94 |
2021 Septiembre | 31 | 33 | 64 |
2021 Agosto | 33 | 34 | 67 |
2021 Julio | 30 | 19 | 49 |
2021 Junio | 26 | 20 | 46 |
2021 Mayo | 37 | 45 | 82 |
2021 Abril | 47 | 60 | 107 |
2021 Marzo | 57 | 25 | 82 |
2021 Febrero | 47 | 24 | 71 |
2021 Enero | 23 | 20 | 43 |
2020 Diciembre | 39 | 16 | 55 |
2020 Noviembre | 32 | 18 | 50 |
2020 Octubre | 28 | 16 | 44 |
2020 Septiembre | 31 | 18 | 49 |
2020 Agosto | 29 | 15 | 44 |
2020 Julio | 26 | 17 | 43 |
2020 Junio | 27 | 22 | 49 |
2020 Mayo | 23 | 14 | 37 |
2020 Abril | 23 | 19 | 42 |
2020 Marzo | 20 | 11 | 31 |
2020 Febrero | 5 | 0 | 5 |
2020 Enero | 4 | 0 | 4 |
2019 Diciembre | 4 | 0 | 4 |
2019 Noviembre | 4 | 0 | 4 |
2019 Septiembre | 4 | 0 | 4 |
2019 Agosto | 4 | 0 | 4 |
2019 Julio | 4 | 0 | 4 |
2019 Junio | 4 | 0 | 4 |
2019 Mayo | 4 | 0 | 4 |
2019 Abril | 2 | 0 | 2 |
2019 Marzo | 2 | 0 | 2 |
2019 Febrero | 1 | 0 | 1 |
2019 Enero | 1 | 0 | 1 |
2018 Diciembre | 4 | 0 | 4 |
2018 Noviembre | 1 | 0 | 1 |
2018 Octubre | 7 | 0 | 7 |
2018 Septiembre | 8 | 0 | 8 |
2018 Febrero | 21 | 4 | 25 |
2018 Enero | 30 | 5 | 35 |
2017 Diciembre | 37 | 10 | 47 |
2017 Noviembre | 62 | 35 | 97 |
2017 Octubre | 3 | 6 | 9 |
2017 Septiembre | 4 | 6 | 10 |