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In June 2020, she was referred to dermatology due to a 5-month-old lesion on the nasal ala. The patient complained of mild loss of sensation and pinching, and openly acknowledged scratching the area. The examination revealed a rounded ulcer with regular edges, which caused an almost complete loss of the right nasal ala, with early involvement of the ipsilateral nasolabial fold proximal portion. The nasal tip was intact (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). There were no skin lesions in other sites. The patient denied epistaxis or other systemic symptoms. A biopsy revealed areas of ulceration and focal necrosis, with no signs of malignancy. No granulomas or vasculitis-related findings were identified.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Complete blood count, renal function, liver enzymes, protein profile and complement levels were normal. Antinuclear and antineutrophil cytoplasmic antibodies were negative, as well as microbiological cultures and serologies for hepatitis B, C and human immunodeficiency viruses. Given these findings, the diagnosis of TTS after ischemic stroke was established. Hygiene measures were prescribed, emphasising the avoidance of lesion manipulation, in addition to treatment with pregabalin. After 2 months, re-epithelialization of a large part of the nasal ala was observed.</p><p id="par0020" class="elsevierStylePara elsevierViewall">TTS is a rare disorder described in the early 20th century following the development of facial ulcers following ablative treatment of trigeminal neuralgia.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Central or peripheral damage to the V cranial nerve causes paraesthesia, dysesthesia or anaesthesia in the trigeminal territory and these sensory alterations seem to be the origin of the repeated self-manipulation leading to persistent ulcers.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Multiple associated causes have been described, especially ablative procedures for the treatment of trigeminal neuralgia and strokes affecting the posterior vascular territory.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–3</span></a> Other less common include tumours (meningioma, astrocytoma), infections, or trauma.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–4</span></a> The latency time from the occurrence of nerve damage to the development of clinical symptoms ranges from weeks to years. Generally, the lesions settle in the territory corresponding to the maxillary branch of the V cranial nerve, with the nasal ala being the most common location. The nasal tip is not involved because its innervation comes from the anterior ethmoidal nerve.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,5</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The differential diagnosis includes skin neoplasms, infections, autoimmune diseases, and psychiatric disorders.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Basal cell carcinoma, squamous cell carcinoma, and nasal NK lymphoma are the main tumours to rule out. Various viral, fungal, or bacterial infections, including mycobacteria, can cause similar lesions, so serial cultures are important. In addition, other disorders such as systemic vasculitis, pyoderma gangrenosum, and granulomatous diseases must be ruled out. Finally, factitial dermatitis is a psychiatric skin disorder that appears in individuals without a compatible neurological history or sensory disturbances, in the form of lesions with atypical distribution and whose patients deny their manipulation.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2,5</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Treatment of TTS requires a multidisciplinary approach, the cornerstone of which is patient education to achieve self-mutilation control. In addition to hygiene measures such as keeping nails short, the use of protective devices (gloves, dressings or face masks) can be helpful.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Educational measures are often complemented by oral (pregabalin, gabapentin, amitriptyline, carbamazepine) and/or topical (tacrolimus) drug therapy to control sensory symptomatology.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–5</span></a> Surgical reconstruction may be necessary in certain cases.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In conclusion, TTS should be considered in a patient with a history of neurological pathology and a persistent unilateral facial ulcer. Efforts to make the patient aware of the self-inflicted nature of the disease are essential in order to avoid manipulation and to achieve early epithelialisation of the lesion.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0045" class="elsevierStylePara elsevierViewall">The authors declare that they have not received any funding to carry out this work.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Funding" ] 1 => 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: Moreiras Arias N, Sopeña B, Sainz Gaspar L. Síndrome trófico del trigémino tras ictus isquémico. Med Clin (Barc). 2022;158:38–39.</p>" ] ] "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 605 "Ancho" => 905 "Tamanyo" => 71051 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Clinical presentation: round ulcer with regular borders affecting the right nasal ala with early involvement of the proximal portion of the nasolabial fold.</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" => "Trigeminal trophic syndrome: an updated review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "A.U. 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Chiota-McCollum" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1212/CPJ.0000000000000711" "Revista" => array:5 [ "tituloSerie" => "Neurol Clin Pract" "fecha" => "2020" "volumen" => "10" "paginaInicial" => "27" "paginaFinal" => "29" ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0020" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Trigeminal trophic syndrome secondary to meningioma recurrence" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "A. Fernández-Bernáldez" 1 => "P. Rodríguez-Jiménez" 2 => "M. Herrero-Moyano" 3 => "J. 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Vol. 158. Issue 1.
Pages 38-39 (January 2022)
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Vol. 158. Issue 1.
Pages 38-39 (January 2022)
Letter to the Editor
Trigeminal trophic syndrome after ischaemic stroke
Síndrome trófico del trigémino tras ictus isquémico
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