Letter - Clinical
Case for diagnosis. A transient unilateral face rash upon eating: Frey syndrome
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A transient unilateral face rash upon eating: Frey syndrome" "tieneTextoCompleto" => true "saludo" => "Dear Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "108" "paginaFinal" => "109" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Ana Gusmão Palmeiro, Laura Azurara, Bernardo Pimentel, Cristina Amaro" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Ana Gusmão" "apellidos" => "Palmeiro" "email" => array:1 [ 0 => "apgpalmeiro@gmail.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" => "Laura" "apellidos" => "Azurara" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "Bernardo" "apellidos" => "Pimentel" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "Cristina" "apellidos" => "Amaro" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Department of Dermatology, Centro Hospitalar Lisboa Ocidental, Hospital de Egas Moniz, Lisboa, Portugal" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Department of Pediatrics, Centro Hospitalar Lisboa Ocidental, Hospital de São Francisco Xavier, Lisboa, Portugal" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1508 "Ancho" => 1508 "Tamanyo" => 195008 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0175" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Flushing of the right cheek.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 9-months-old infant, born at full term and delivered with the assistance of forceps, presented to the Dermatology clinic with a history of flushing and warming on his right cheek that appeared when he ate (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The flushing first become apparent to his parents at 6-months of age, upon introduction of solid foods. It usually began within seconds of ingesting the first bite and resolved in 30 minutes. It was not associated with hyperhidrosis, pruritus, discomfort, lip swelling, wheezing, or dyspnea. The patient had not undergone surgery, nor had he had a personal history of atopy.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">At the initial observation in the clinic, there were no skin lesions, but within seconds after a provocation test by eating a strawberry, unilateral erythema and localized warmth appeared across his right zygomatic arch region, malar eminence, and submalar region (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). The remainder dermatologic exam was normal and there were no associated signs or symptoms, including sweating. The rash faded after around 15 minutes. The neurological exam was unremarkable.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">What is your diagnosis?</span><p id="par0095" class="elsevierStylePara elsevierViewall"><ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">a</span><p id="par0015" class="elsevierStylePara elsevierViewall">Frey syndrome,</p></li><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">b</span><p id="par0020" class="elsevierStylePara elsevierViewall">Fifth disease,</p></li><li class="elsevierStyleListItem" id="lsti0015"><span class="elsevierStyleLabel">c</span><p id="par0025" class="elsevierStylePara elsevierViewall">Oral allergy syndrome,</p></li><li class="elsevierStyleListItem" id="lsti0020"><span class="elsevierStyleLabel">d</span><p id="par0030" class="elsevierStylePara elsevierViewall">Mast cell activation syndrome.</p></li></ul></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">The clinical history and provocation test are compatible with Frey syndrome.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Frey syndrome is a parasympathetic auriculotemporal nerve dysfunction, characterized by a transient flushing, localized warmth, and sometimes sweating of the face in the territory of the auriculotemporal nerve, a branch of the mandibular nerve of the trigeminal nerve complex. Symptoms classically occur in response to olfactory or gustatory stimuli, particularly with acidic, spicy, and sour foods.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">In adults, it is a frequent complication of parotid surgery, but in children, it is very rare. As such, especially in bilateral cases, it is often misdiagnosed as a food allergy, prompting unnecessary diagnostic testing and treatment. A recent review showed that the diagnosis was made in only 20% of children at the first consultation and inappropriate dietary restriction was prescribed in 21%.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">In children, the majority (around 70%) of reported cases are unilateral, while associated sweating is rare (present in about 10%, in contrast to the adult forms, in which it is often present).<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The pathophysiology is incompletely understood. The prevailing hypothesis in unilateral forms is a traumatic lesion of the nerve fibers, followed by aberrant regeneration of the parasympathetic fibers into the skin, perhaps via sympathetic gains. This trauma could be neonatal, in instrumented delivery with forceps or ventouse, or postnatal.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Bilateral forms appear to be largely idiopathic, but a familial bilateral syndrome has already been described.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The outcome is overall favorable in unilateral cases, with regression in around 57% of cases, at a median age of 27 months.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> No further workup or treatment is recommended.</p><p id="par0060" class="elsevierStylePara elsevierViewall">The authors report a classical Frey syndrome in childhood, after a forceps-assisted delivery, and with the absence of hyperhidrosis. The mother was reassured, and no specific treatment was provided. In contrast to the adult postsurgical form, Frey syndrome in children is rare and often constitutes a diagnostic challenge. In this case, the authors aim to increase familiarity with this benign condition, to avoid misdiagnosis and unnecessary procedures.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Financial support</span><p id="par0065" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Authors' contributions</span><p id="par0070" class="elsevierStylePara elsevierViewall">Ana Gusmão Palmeiro: Conceptualization; Methodology; Investigation; Data curation; Resources; Original draft preparation; Supervision.</p><p id="par0075" class="elsevierStylePara elsevierViewall">Laura Azurara: Validation; Formal analysis; Review and editing.</p><p id="par0080" class="elsevierStylePara elsevierViewall">Bernardo Pimentel: Validation; Formal analysis; Review and editing.</p><p id="par0085" class="elsevierStylePara elsevierViewall">Cristina Amaro: Conceptualization; Validation; Formal analysis; Review and editing.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Conflicts of interest</span><p id="par0090" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:6 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "What is your diagnosis?" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Financial support" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Authors' contributions" ] 4 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflicts of interest" ] 5 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2021-09-05" "fechaAceptado" => "2021-10-03" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Study conducted at the Hospital de Egas Moniz, Lisboa, Portugal.</p>" ] ] "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1508 "Ancho" => 1508 "Tamanyo" => 195008 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0175" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Flushing of the right cheek.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1417 "Ancho" => 3008 "Tamanyo" => 339418 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0180" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Before (A) and after (B) the provocation test in the clinic.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Marathon of eponyms: 6 Frey syndrome (Gustatory sweating)" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "C. 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"fecha" => "2019" "volumen" => "37" "paginaInicial" => "414" "paginaFinal" => "416" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/31710019" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0025" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Familial auriculotemporal nerve (Frey) syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "G. Sethuraman" 1 => "A.J. Mancini" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1111/j.1525-1470.2009.00909.x" "Revista" => array:6 [ "tituloSerie" => "Pediatr Dermatol" "fecha" => "2009" "volumen" => "26" "paginaInicial" => "302" "paginaFinal" => "305" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/19706092" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/03650596/0000009800000001/v2_202304080751/S036505962200232X/v2_202304080751/en/main.assets" "Apartado" => array:4 [ "identificador" => "95153" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letter - Clinical" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/03650596/0000009800000001/v2_202304080751/S036505962200232X/v2_202304080751/en/main.pdf?idApp=UINPBA00008Z&text.app=https://clinics.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S036505962200232X?idApp=UINPBA00008Z" ]
| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 29 | 12 | 41 |
| 2024 October | 146 | 92 | 238 |
| 2024 September | 174 | 111 | 285 |
| 2024 August | 195 | 149 | 344 |
| 2024 July | 193 | 115 | 308 |
| 2024 June | 160 | 112 | 272 |
| 2024 May | 108 | 79 | 187 |
| 2024 April | 114 | 81 | 195 |
| 2024 March | 110 | 72 | 182 |
| 2024 February | 115 | 83 | 198 |
| 2024 January | 99 | 60 | 159 |
| 2023 December | 64 | 45 | 109 |
| 2023 November | 96 | 95 | 191 |
| 2023 October | 92 | 74 | 166 |
| 2023 September | 93 | 86 | 179 |
| 2023 August | 85 | 38 | 123 |
| 2023 July | 93 | 48 | 141 |
| 2023 June | 78 | 39 | 117 |
| 2023 May | 52 | 23 | 75 |
| 2023 April | 47 | 23 | 70 |
| 2023 March | 64 | 50 | 114 |
| 2023 February | 60 | 59 | 119 |
| 2023 January | 105 | 58 | 163 |
| 2022 December | 74 | 30 | 104 |
| 2022 November | 39 | 53 | 92 |
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