Case for diagnosis
Tongue Ulcer in a 7-Month-Old Infant
Úlcera lingual en un lactante de 7 meses
E. del Alcázar–Viladomiu
, M.A. Vicente–Villa, M.A. González-Enseñat
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Servicio de Dermatología Pediátrica, Hospital Sant Joan de Déu, Barcelona, Spain
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"apellidos" => "del Alcázar–Viladomiu" "email" => array:1 [ 0 => "elenadelalcazarviladomiu@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "M.A." "apellidos" => "Vicente–Villa" ] 2 => array:2 [ "nombre" => "M.A." "apellidos" => "González-Enseñat" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Dermatología Pediátrica, Hospital Sant Joan de Déu, Barcelona, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding Author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Úlcera lingual en un lactante de 7 meses" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:6 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1137 "Ancho" => 1667 "Tamanyo" => 124808 ] ] ] ] "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 7-month-old boy with no past history of interest was referred to the dermatology department for evaluation of a tongue ulcer that had appeared 1 month earlier. The mother stated that the ulcer was painful and that it had appeared at the same time as eruption of the lower incisor teeth. Treatment with topical corticosteroids had produced no improvement.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Physical Examination</span><p id="par0010" class="elsevierStylePara elsevierViewall">On examination, the infant presented a round indurated ulcer that measured 1<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>1<span class="elsevierStyleHsp" style=""></span>cm and had well-defined borders. The ulcer was situated on the ventral surface of the tongue, coincident with the lower incisor teeth (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). A whitish exudate covered the base of the lesion, in which there were 2 deeper linear ulcers. There were no palpable cervical lymph nodes and no other skin or mucosal lesions. The child presented good height and weight gain and neurological examination was normal.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Additional Tests</span><p id="par0015" class="elsevierStylePara elsevierViewall">Bacterial culture and polymerase chain reaction analysis for herpes 1 and 2 viruses, Epstein-Barr virus, and cytomegalovirus were negative. Based on the initial clinical suspicion, and given the age of the patient, skin biopsy was not performed.</p><p id="par0020" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleBold">What Is Your Diagnosis?</span></p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Diagnosis</span><p id="par0025" class="elsevierStylePara elsevierViewall">Riga-Fede disease.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Clinical Course and Treatment</span><p id="par0030" class="elsevierStylePara elsevierViewall">The patient received dental treatment with smoothing of the edges of the incisor teeth. At 4 weeks the lesion had reduced in size (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>), and a month later it had completely disappeared.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Comment</span><p id="par0035" class="elsevierStylePara elsevierViewall">The first clinical description of Riga-Fede disease was made by Antonio Riga in 1881, and its histological features were described by Francesco Fede in 1890.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is an uncommon, benign condition characterized by an ulcer on the oral mucosa of children aged less than 2 years.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> In adults the disease is known as eosinophilic ulcer of the oral mucosa, traumatic granuloma of the tongue, or eosinophilic granuloma of the tongue, among other names.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The ulcer is situated on the ventral surface and tip of the tongue in the majority of cases,<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> and the incidence is similar in both sexes.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The etiology is believed to be recurrent trauma secondary to continual movements of protrusion and retraction of the tongue over the lower incisor teeth.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,5</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Riga-Fede disease is usually observed in children with premature eruption of the teeth, whether natal or neonatal.<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,6</span></a> It may also be observed in children with neurological diseases or cerebral palsy, and it can even be the initial manifestation of neurological diseases such as familial dysautonomia (Riley-Day syndrome), congenital autonomic dysfunction with universal pain loss, or Lesch-Nyhan syndrome.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2–6</span></a> Domingues-Cruz et al.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> proposed a classification based on age at onset and associated diseases. Early cases (before 6 months of age) are caused by natal or neonatal teeth, and there is no association with neurological disorders, whereas late cases (after 6 months of age) are caused by the primary dentition and may be associated with neurological or developmental diseases.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Infections (including congenital syphilis, tuberculosis, and candidiasis), tumors (lymphoma, sarcoma, myofibroma, granular cell tumor), and hematological diseases (agranulocytosis) must be considered in the differential diagnosis.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,4,6</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Biopsy reveals an ulcer with granulation tissue and a diffuse, mainly eosinophilic infiltrate that extends down to the muscle.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">The most common complications are bleeding and pain, which may interfere with feeding and lead to malnutrition, dehydration, and even delayed development.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Recommended treatments include planing or smoothing of the incisal edges of the lower incisor teeth or tooth protection with caps or composite increments to the teeth. Extraction of the incisor teeth is indicated as a last resort for persistent ulcers.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Riga-Fede disease should be included in the differential diagnosis of oral ulcers in children. Clinical recognition of this disease will prevent unnecessary biopsy or surgery. Children with Riga-Fede disease should undergo dental examination as well as neurological evaluation to exclude neurological disease.</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Conflicts of Interest</span><p id="par0075" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:8 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Medical History" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Physical Examination" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Additional Tests" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Diagnosis" ] 4 => array:2 [ "identificador" => "sec0025" "titulo" => "Clinical Course and Treatment" ] 5 => array:2 [ "identificador" => "sec0030" "titulo" => "Comment" ] 6 => array:2 [ "identificador" => "sec0035" "titulo" => "Conflicts of Interest" ] 7 => 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: del Alcázar–Viladomiu E, et al. 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Camacho" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1111/j.1525-1470.2007.00564.x" "Revista" => array:6 [ "tituloSerie" => "Pediatr Dermatol" "fecha" => "2007" "volumen" => "24" "paginaInicial" => "663" "paginaFinal" => "665" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/18035997" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15782190/0000010400000007/v1_201308270818/S1578219013001509/v1_201308270818/en/main.assets" "Apartado" => array:4 [ "identificador" => "6171" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Cases for Diagnosis" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15782190/0000010400000007/v1_201308270818/S1578219013001509/v1_201308270818/en/main.pdf?idApp=UINPBA000044&text.app=https://actasdermo.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219013001509?idApp=UINPBA000044" ]
| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 13 | 14 | 27 |
| 2024 Octubre | 133 | 53 | 186 |
| 2024 Septiembre | 181 | 46 | 227 |
| 2024 Agosto | 234 | 81 | 315 |
| 2024 Julio | 214 | 56 | 270 |
| 2024 Junio | 213 | 46 | 259 |
| 2024 Mayo | 171 | 54 | 225 |
| 2024 Abril | 206 | 39 | 245 |
| 2024 Marzo | 203 | 44 | 247 |
| 2024 Febrero | 276 | 39 | 315 |
| 2024 Enero | 296 | 37 | 333 |
| 2023 Diciembre | 366 | 28 | 394 |
| 2023 Noviembre | 360 | 43 | 403 |
| 2023 Octubre | 340 | 34 | 374 |
| 2023 Septiembre | 384 | 45 | 429 |
| 2023 Agosto | 232 | 29 | 261 |
| 2023 Julio | 238 | 59 | 297 |
| 2023 Junio | 203 | 25 | 228 |
| 2023 Mayo | 235 | 68 | 303 |
| 2023 Abril | 258 | 34 | 292 |
| 2023 Marzo | 315 | 48 | 363 |
| 2023 Febrero | 231 | 28 | 259 |
| 2023 Enero | 167 | 50 | 217 |
| 2022 Diciembre | 200 | 49 | 249 |
| 2022 Noviembre | 149 | 24 | 173 |
| 2022 Octubre | 133 | 24 | 157 |
| 2022 Septiembre | 156 | 48 | 204 |
| 2022 Agosto | 61 | 40 | 101 |
| 2022 Julio | 85 | 33 | 118 |
| 2022 Junio | 75 | 31 | 106 |
| 2022 Mayo | 181 | 45 | 226 |
| 2022 Abril | 265 | 44 | 309 |
| 2022 Marzo | 224 | 77 | 301 |
| 2022 Febrero | 214 | 62 | 276 |
| 2022 Enero | 201 | 63 | 264 |
| 2021 Diciembre | 104 | 56 | 160 |
| 2021 Noviembre | 69 | 45 | 114 |
| 2021 Octubre | 127 | 61 | 188 |
| 2021 Septiembre | 69 | 52 | 121 |
| 2021 Agosto | 52 | 85 | 137 |
| 2021 Julio | 40 | 53 | 93 |
| 2021 Junio | 72 | 47 | 119 |
| 2021 Mayo | 58 | 40 | 98 |
| 2021 Abril | 129 | 84 | 213 |
| 2021 Marzo | 101 | 47 | 148 |
| 2021 Febrero | 82 | 38 | 120 |
| 2021 Enero | 39 | 33 | 72 |
| 2020 Diciembre | 42 | 26 | 68 |
| 2020 Noviembre | 33 | 46 | 79 |
| 2020 Octubre | 29 | 13 | 42 |
| 2020 Septiembre | 44 | 29 | 73 |
| 2020 Agosto | 25 | 24 | 49 |
| 2020 Julio | 30 | 36 | 66 |
| 2020 Junio | 45 | 58 | 103 |
| 2020 Mayo | 27 | 47 | 74 |
| 2020 Abril | 29 | 29 | 58 |
| 2020 Marzo | 44 | 24 | 68 |
| 2020 Febrero | 4 | 11 | 15 |
| 2020 Enero | 4 | 7 | 11 |
| 2019 Diciembre | 8 | 8 | 16 |
| 2019 Noviembre | 4 | 4 | 8 |
| 2019 Octubre | 0 | 6 | 6 |
| 2019 Septiembre | 11 | 10 | 21 |
| 2019 Agosto | 7 | 10 | 17 |
| 2019 Julio | 2 | 11 | 13 |
| 2019 Junio | 4 | 12 | 16 |
| 2019 Mayo | 3 | 24 | 27 |
| 2019 Abril | 2 | 20 | 22 |
| 2019 Marzo | 0 | 13 | 13 |
| 2019 Febrero | 2 | 11 | 13 |
| 2019 Enero | 0 | 3 | 3 |
| 2018 Diciembre | 1 | 5 | 6 |
| 2018 Noviembre | 3 | 2 | 5 |
| 2018 Octubre | 1 | 0 | 1 |
| 2018 Septiembre | 5 | 0 | 5 |
| 2018 Junio | 0 | 1 | 1 |
| 2018 Mayo | 0 | 2 | 2 |
| 2018 Marzo | 3 | 2 | 5 |
| 2018 Febrero | 123 | 6 | 129 |
| 2018 Enero | 125 | 4 | 129 |
| 2017 Diciembre | 111 | 10 | 121 |
| 2017 Noviembre | 132 | 5 | 137 |
| 2017 Octubre | 132 | 9 | 141 |
| 2017 Septiembre | 107 | 5 | 112 |
| 2017 Agosto | 133 | 6 | 139 |
| 2017 Julio | 88 | 13 | 101 |
| 2017 Junio | 107 | 8 | 115 |
| 2017 Mayo | 91 | 8 | 99 |
| 2017 Abril | 65 | 12 | 77 |
| 2017 Marzo | 69 | 20 | 89 |
| 2017 Febrero | 87 | 12 | 99 |
| 2017 Enero | 121 | 14 | 135 |
| 2016 Diciembre | 89 | 9 | 98 |
| 2016 Noviembre | 151 | 13 | 164 |
| 2016 Octubre | 176 | 16 | 192 |
| 2016 Septiembre | 211 | 20 | 231 |
| 2016 Agosto | 189 | 10 | 199 |
| 2016 Julio | 96 | 13 | 109 |
| 2016 Junio | 7 | 16 | 23 |
| 2016 Mayo | 9 | 19 | 28 |
| 2016 Abril | 3 | 5 | 8 |
| 2016 Marzo | 3 | 12 | 15 |
| 2016 Febrero | 8 | 2 | 10 |
| 2016 Enero | 7 | 13 | 20 |
| 2015 Diciembre | 9 | 7 | 16 |
| 2015 Noviembre | 35 | 9 | 44 |
| 2015 Octubre | 19 | 14 | 33 |
| 2015 Septiembre | 4 | 5 | 9 |
| 2015 Agosto | 3 | 1 | 4 |
| 2015 Julio | 171 | 3 | 174 |
| 2015 Junio | 114 | 11 | 125 |
| 2015 Mayo | 102 | 15 | 117 |
| 2015 Abril | 105 | 4 | 109 |
| 2015 Marzo | 94 | 2 | 96 |
| 2015 Febrero | 126 | 8 | 134 |
| 2015 Enero | 57 | 6 | 63 |
| 2014 Diciembre | 86 | 7 | 93 |
| 2014 Noviembre | 80 | 5 | 85 |
| 2014 Octubre | 116 | 8 | 124 |
| 2014 Septiembre | 78 | 0 | 78 |
| 2014 Agosto | 73 | 6 | 79 |
| 2014 Julio | 70 | 4 | 74 |
| 2014 Junio | 64 | 3 | 67 |
| 2014 Mayo | 75 | 9 | 84 |
| 2014 Abril | 77 | 4 | 81 |
| 2014 Marzo | 73 | 6 | 79 |
| 2014 Febrero | 55 | 3 | 58 |
| 2014 Enero | 47 | 5 | 52 |
| 2013 Diciembre | 41 | 6 | 47 |
| 2013 Noviembre | 44 | 10 | 54 |
| 2013 Octubre | 27 | 2 | 29 |
| 2013 Septiembre | 20 | 5 | 25 |
| 2013 Agosto | 2 | 1 | 3 |
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