Letter to the Editor
Central Nervous System Involvement on Eosynophillic Fascitis
Afectación del sistema nervioso central y periférico en la fascitis eosinofílica
Dolors Grados
, Melania Martinez-Morillo, Pilar Latorre, Alejandro Olivé
Corresponding author
Sección de Reumatología, Servicio de Neurología, Hospital Germans Trias i Pujol, Barcelona, Spain
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Various systemic manifestations associated with EF have been described, such as renal, cardiac, respiratory, joint and peripheral nervous system manifestations. Below we present the case of a patient with FE of the central and peripheral nervous system.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was a 71-year-old male with a medical history of hypertension and tuberculosis. He came to the hospital with induration of the arms, legs, chest and neck, which had started 5 months prior. He referred dysphagia to solids and loss of 10<span class="elsevierStyleHsp" style=""></span>kg in the past year. Physical examination revealed skin induration in the arms, legs, trunk and neck, respecting the hands and feet. He had a positive sulcus sign and orange skin. Neurological examination was normal. Laboratory tests showed peripheral eosinophilia (8.1<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/l leukocytes with 2% eosinophils) and an ESR of 40<span class="elsevierStyleHsp" style=""></span>mm in the first hour. Blood biochemistry was normal. Protein, tumor markers, chest X-ray, l capillaroscopy and immunological tests were normal. The upper GI series revealed esophageal motor incoordination with passage of contrast between the larynx and esophagus. An MRI showed changes consistent with eosinophilic fasciitis. A muscle biopsy was performed which revealed deep and reactive inflammatory changes in adipose tissue and fascia, all compatible with EF. Glucocorticoid therapy was initiated at doses of 1<span class="elsevierStyleHsp" style=""></span>mg/kg/day. At 2 months he showed a left facial central paralysis and hypoglossal nerve palsy. Cranial MRI was normal. A lumbar puncture was performed which was also normal. Subsequently, paralysis of the external popliteal nerve was observed. Physical examination showed persistent induration of the arms, legs and trunk. The neurological examination revealed a central left facial nerve palsy, left hypoglossal nerve palsy, paresis of the external cyatic popliteal nerve, but sensitivity and tendon reflexes were present and symmetrical as was the bilateral flexor plantar cutaneous reflex. The blood count was normal and ESR was 22<span class="elsevierStyleHsp" style=""></span>mm in the first hour. Blood biochemistry showed mild hypoproteinemia. Tumor markers and a CT scan were normal. An electromyogram was performed which revealed an asymmetric non sensitive polyneuropathy. Treatment was begun with azathioprine 50<span class="elsevierStyleHsp" style=""></span>mg/day and glucocorticoids at a dose of 1<span class="elsevierStyleHsp" style=""></span>mg/kg/day. Six months later azathioprine was withdrawn. Today, 10 years after diagnosis, he remains in treatment with 2<span class="elsevierStyleHsp" style=""></span>mg of methylprednisolone, without neurological or skin complications.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Since the first description of the syndrome, over 250 cases of eosinophilic fasciitis have been published. The most frequent neurological manifestation of peripheral neuropathy is due to carpal tunnel syndrome (both unilateral and bilateral), which occurs in approximately 25% of cases.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a> Lower limb neuropathy secondary to<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,6</span></a> mononeuritis multiplex and multifocal peripheral neuropathy have also been described.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> In the case reported, involvement was seen simultaneously in the central and peripheral nervous system: central left facial paralysis, left hypoglossal nerve palsy and paresis of the cyatic popliteal nerve, with good response to corticosteroids and azathioprine. In a literature review using PubMed (descriptors: eosinophilic fasciitis & nervous system; 1974–2012) we have not found more cases of eosinophilic fasciitis associated with this type of neurological involvement. It may be recalled that eosinophilic fasciitis is a disease with many extracutaneous manifestations, including the nervous system.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please, cite this article as: Grados D, et al. Afectación del sistema nervioso central y periférico en la fascitis eosinofílica. Reumatol Clin. 2013. <span class="elsevierStyleInterRef" href="doi:10.1016/j.reuma.2012.09.004">http://dx.doi.org/10.1016/j.reuma.2012.09.004</span>.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Diffuse fascitis with eosinophilia: a new syndrome?" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "L.E. 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| 2017 February | 22 | 7 | 29 |
| 2017 January | 24 | 5 | 29 |
| 2016 December | 65 | 19 | 84 |
| 2016 November | 45 | 2 | 47 |
| 2016 October | 70 | 15 | 85 |
| 2016 September | 59 | 12 | 71 |
| 2016 August | 59 | 6 | 65 |
| 2016 July | 43 | 9 | 52 |
| 2016 May | 0 | 8 | 8 |
| 2016 February | 1 | 0 | 1 |
| 2016 January | 0 | 15 | 15 |
| 2015 December | 2 | 0 | 2 |
| 2015 October | 1 | 12 | 13 |
| 2015 September | 1 | 0 | 1 |
| 2015 August | 1 | 0 | 1 |
| 2015 July | 31 | 4 | 35 |
| 2015 June | 26 | 7 | 33 |
| 2015 May | 46 | 18 | 64 |
| 2015 April | 47 | 15 | 62 |
| 2015 March | 29 | 9 | 38 |
| 2015 February | 25 | 6 | 31 |
| 2015 January | 24 | 12 | 36 |
| 2014 December | 31 | 12 | 43 |
| 2014 November | 30 | 11 | 41 |
| 2014 October | 32 | 17 | 49 |
| 2014 September | 18 | 7 | 25 |
| 2014 August | 33 | 14 | 47 |
| 2014 July | 40 | 17 | 57 |
| 2014 June | 51 | 11 | 62 |
| 2014 May | 47 | 16 | 63 |
| 2014 April | 54 | 14 | 68 |
| 2014 March | 65 | 13 | 78 |
| 2014 February | 46 | 17 | 63 |
| 2014 January | 37 | 13 | 50 |
| 2013 December | 37 | 9 | 46 |
| 2013 November | 29 | 8 | 37 |
| 2013 October | 49 | 8 | 57 |
| 2013 September | 38 | 12 | 50 |
| 2013 August | 48 | 10 | 58 |
| 2013 July | 37 | 8 | 45 |
| 2013 June | 44 | 12 | 56 |
| 2013 May | 58 | 17 | 75 |
| 2013 April | 22 | 13 | 35 |
Show all
