Case and Research Letters
Trigeminal Trophic Syndrome Secondary to Meningioma Recurrence
Síndrome trófico trigeminal secundario a recidiva demeningioma
A. Fernández-Bernáldez
, P. Rodríguez-Jiménez, M. Herrero-Moyano, J. Sánchez-Pérez
Corresponding author
Servicio de Dermatología, Hospital Universitario de La Princesa, Madrid, Spain
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Fernández-Bernáldez, P. Rodríguez-Jiménez, M. Herrero-Moyano, J. Sánchez-Pérez" "autores" => array:4 [ 0 => array:4 [ "nombre" => "A." "apellidos" => "Fernández-Bernáldez" "email" => array:1 [ 0 => "alberto_pswd@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "P." "apellidos" => "Rodríguez-Jiménez" ] 2 => array:2 [ "nombre" => "M." "apellidos" => "Herrero-Moyano" ] 3 => array:2 [ "nombre" => "J." "apellidos" => "Sánchez-Pérez" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Dermatología, Hospital Universitario de La Princesa, Madrid, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Síndrome trófico trigeminal secundario a recidiva demeningioma" ] ] "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" => 904 "Ancho" => 1674 "Tamanyo" => 214396 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A and B, Crusted erosive lesions on an atrophic background in the region innervated by the V1 branch of the trigeminal nerve.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Trigeminal trophic syndrome (TTS), also known as trigeminal neurotrophic ulceration, is a rare condition that occurs when the trigeminal nerve is damaged. It presents with anesthesia or unilateral dysesthesia that typically affects the ala nasi, and with ulcerations that are self-inflicted, usually unconsciously, by the patient.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">A 38-year-old woman was examined for pruriginous erosive and mildly painful lesions that had appeared 2 months earlier in the left frontoparietal region. The patient had undergone surgery for a meningioma measuring 46 × 40 × 38 mm in the left cavernous sinus a year and a half earlier by means of frontotemporal cutaneous incision and left pterional craniotomy. Since the surgery, the patient had presented sequelae including paresis of cranial nerves III and IV in the left eye, neurotrophic keratitis in the left eye, and panhypopituitarism with secondary hypogonadism and hypothyroidism. The last follow-up imaging study performed a month before visiting our department and approximately a month after onset of the skin condition, showed a recurrence of the meningioma in the left cavernous sinus. The patient had been examined a month earlier at the ophthalmology department for an episode of herpes zoster that involved the V1 branch of the trigeminal nerve, with no ophthalmologic involvement. Without microbiologic confirmation, she received treatment with valaciclovir and was sent to our department due to the persistence of the lesions when the treatment had been completed. When she visited our department, she presented several crusted erosive lesions of varying morphology on a background of hypersensitive atrophic skin compared to the contralateral region (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). We reached a diagnosis of TTS, with ulcers secondary to dysesthesia and anesthesia in the left V1 region, probably linked to the recurrence of meningioma. Treatment was instated at that time with tacrolimus cream, 0.01% and covered with a hydrocolloid dressing. The patient was also offered the option of starting treatment with oral gabapentin, which se rejected. After 2 months of treatment, the lesions improved considerably.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">TTS is characterized by the presence of unilateral facial ulcers caused by persistent scratching as a result of the dysesthesia (described as itching, or a burning or tingling sensation) and anesthesia in the sites innervated by the previously damaged trigeminal nerve or one of its branches. Patients often scratch to alleviate the feeling of discomfort and, due to the anesthesia, cause persistent ulceration. A systematic review of the literature in by Sawada et al. in 2014,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> which included 61 cases of TTS published to that date, describes the principal etiologies of this syndrome. The most commonly reported causes are ablation of the trigeminal nerve (30%), cerebrovascular accident (30%), and surgical complications, including a history of craniotomy (21%). Another, less frequently reported cause is herpes zoster, which is also mentioned by other authors, such as Dolohanty et al.,<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> who describe development of TTS in the V1 region 2 months after an episode of herpes zoster in the same dermatome, which was treated with valaciclovir. In our case, we believe that the initial clinical presentation, diagnosed as herpes zoster, was probably the onset of TTS.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Furthermore, damage to the trigeminal nerve may occur in up to 11% of patients who undergo surgery for meningioma.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The first case of TTS associated with meningioma was described in 1982.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Three further publications link TTS and meningioma surgery, although in one of them, the TTS appeared after surgery for a recurrence of the meningioma 9 years after the primary tumor.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Thus, although our patient potentially presents 3 of the etiologic factors most frequently associated with development of TTS (meningioma, craniotomy and a doubtful episode of herpes zoster), we believe that the sequence of appearance of the symptoms and the coincidence with the recurrence of the meningioma make the meningioma the most probable cause.</p><p id="par0025" class="elsevierStylePara elsevierViewall">In terms of treatment of TTS, improvement of cutaneous symptoms has been reported with tacrolimus cream, 0.01%, gabapentin, and hydrocolloid dressing.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Improvement of the lesions has also been observed after use of high doses of carbamazepine (200 mg/3 times daily).<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Other drugs, such as amitriptyline, pregabalin, and alprazolam have been used with little or no clinical benefit.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of interest</span><p id="par0030" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflicts of interest" ] 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: Fernández-Bernáldez A, et al. Síndrome trófico trigeminal secundario a recidiva de meningioma. Actas Dermosifiliogr. 2020;111:171–173.</p>" ] ] "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 904 "Ancho" => 1674 "Tamanyo" => 214396 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A and B, Crusted erosive lesions on an atrophic background in the region innervated by the V1 branch of the trigeminal nerve.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:8 [ 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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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 11 | 16 | 27 |
| 2024 October | 61 | 53 | 114 |
| 2024 September | 66 | 36 | 102 |
| 2024 August | 86 | 57 | 143 |
| 2024 July | 74 | 44 | 118 |
| 2024 June | 86 | 69 | 155 |
| 2024 May | 70 | 32 | 102 |
| 2024 April | 66 | 19 | 85 |
| 2024 March | 66 | 35 | 101 |
| 2024 February | 59 | 29 | 88 |
| 2024 January | 51 | 35 | 86 |
| 2023 December | 47 | 15 | 62 |
| 2023 November | 73 | 31 | 104 |
| 2023 October | 59 | 35 | 94 |
| 2023 September | 43 | 35 | 78 |
| 2023 August | 33 | 17 | 50 |
| 2023 July | 38 | 34 | 72 |
| 2023 June | 41 | 29 | 70 |
| 2023 May | 41 | 26 | 67 |
| 2023 April | 27 | 14 | 41 |
| 2023 March | 61 | 25 | 86 |
| 2023 February | 34 | 18 | 52 |
| 2023 January | 30 | 31 | 61 |
| 2022 December | 44 | 32 | 76 |
| 2022 November | 42 | 26 | 68 |
| 2022 October | 33 | 17 | 50 |
| 2022 September | 31 | 24 | 55 |
| 2022 August | 24 | 33 | 57 |
| 2022 July | 26 | 32 | 58 |
| 2022 June | 31 | 17 | 48 |
| 2022 May | 35 | 35 | 70 |
| 2022 April | 35 | 18 | 53 |
| 2022 March | 63 | 39 | 102 |
| 2022 February | 29 | 25 | 54 |
| 2022 January | 60 | 29 | 89 |
| 2021 December | 61 | 32 | 93 |
| 2021 November | 47 | 33 | 80 |
| 2021 October | 58 | 46 | 104 |
| 2021 September | 35 | 41 | 76 |
| 2021 August | 56 | 28 | 84 |
| 2021 July | 25 | 22 | 47 |
| 2021 June | 16 | 33 | 49 |
| 2021 May | 53 | 41 | 94 |
| 2021 April | 96 | 68 | 164 |
| 2021 March | 46 | 25 | 71 |
| 2021 February | 39 | 19 | 58 |
| 2021 January | 27 | 16 | 43 |
| 2020 December | 22 | 14 | 36 |
| 2020 November | 29 | 12 | 41 |
| 2020 October | 21 | 3 | 24 |
| 2020 September | 26 | 12 | 38 |
| 2020 August | 37 | 21 | 58 |
| 2020 July | 15 | 12 | 27 |
| 2020 June | 26 | 17 | 43 |
| 2020 May | 36 | 14 | 50 |
| 2020 April | 52 | 14 | 66 |
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