Letter to the Editor
Recurrent Miller Fisher syndrome: Case report
Recurrencia del síndrome de Miller Fisher: descripción de un caso
A. Peral Quirós
, F. Acebrón, M. del Carmen Blanco Valero, F. Labella Álvarez
Corresponding author
Servicio de Neurología, Hospital Universitario Reina Sofía, Córdoba, Spain
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Peral Quirós, F. Acebrón, M. del Carmen Blanco Valero, F. Labella Álvarez" "autores" => array:4 [ 0 => array:4 [ "nombre" => "A." "apellidos" => "Peral Quirós" "email" => array:1 [ 0 => "alepq994@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "F." "apellidos" => "Acebrón" ] 2 => array:2 [ "nombre" => "M." "apellidos" => "del Carmen Blanco Valero" ] 3 => array:2 [ "nombre" => "F." "apellidos" => "Labella Álvarez" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Neurología, Hospital Universitario Reina Sofía, Córdoba, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Recurrencia del síndrome de Miller Fisher: descripción de un caso" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">The term Miller Fisher syndrome (MFS) refers to the clinical spectrum described in 1932 by Cullier, characterised by ataxia, ophthalmoparesis, and areflexia; it is considered an infrequent variant of Guillain-Barré syndrome.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–5</span></a> Its aetiology is considered to be autoimmune. MFS is caused by a cross-reaction, after history of a respiratory or gastrointestinal infection or vaccination, to the GQ1b antigens present in the axons of oculomotor nerves, neurons of the posterior spinal root ganglia, and neuromuscular spindles.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> Anti-GQ1b antiganglioside antibodies are detected in 80% of patients.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–4</span></a> Annual incidence is between 1% and 5% of all cases of GBS spectrum.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,6</span></a> Treatment includes intravenous (IV) immunoglobulins (Ig) or plasmapheresis, and outcomes are frequently favourable. In most cases, the clinical course is monophasic, with recurrences being very rare (approximately 5% of cases).<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,4,5</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We present the case of a 21-year-old man with recurrent MFS and history of seborrheic dermatitis and hyperhomocysteinaemia. In 2018, one week after manifesting tonsillitis and diarrhoea, treated with amoxicillin/clavulanic acid, he was admitted due to gait instability and binocular diplopia in the horizontal plane, progressing for less than 24 hours, with no dysautonomic signs. The patient presented right palpebral ptosis, bilateral limited upgaze, paralysis of the right medial and lateral rectus muscles, hypoactive deep tendon reflexes in the upper limbs and abolished deep tendon reflexes in the lower limbs, preserved positional and arthrokinetic sensitivity, and reduced distal vibration sensitivity in the left lower limb, with ataxic gait. Analysis of a cerebrospinal fluid (CSF) sample collected on the day of admission only revealed an increased protein level (34 mg/dL; normal range: 8–32 mg/dL). Serology results for HIV, <span class="elsevierStyleItalic">Rickettsia</span>, <span class="elsevierStyleItalic">Brucella</span>, <span class="elsevierStyleItalic">Coxiella</span>, and hepatitis C virus yielded negative results. We detected IgG antibodies against the hepatitis B virus surface antigen, with a PCR test yielding negative results. A blood sample collected on day 2 after admission was negative for antiganglioside antibodies (anti-GM1, GM2, GM3, GD1a, GD1b, GT1b, and GQ1b). We detected vitamin B<span class="elsevierStyleInf">12</span> deficiency (83 pg/mL; normal range: 211–911 pg/dL), which was treated with supplementation. Non-contrast magnetic resonance imaging (MRI) yielded normal results. No spinal MRI scan was performed. A motor nerve conduction study revealed generalised motor sensory axonal polyradiculoneuritis. After diagnosis of MFS, the patient started treatment with IV Ig, which led to pronounced worsening, with ophthalmoplegia and truncal ataxia, at 3 days. He then started 12 cycles of plasmapheresis, which improved ophthalmoparesis and ataxia. The patient was asymptomatic at 6 months, without recovery of stretch reflexes (with the exception of hypoactive right brachioradialis reflex).</p><p id="par0015" class="elsevierStylePara elsevierViewall">At 2 years, a week after presenting symptoms of cold without fever, he was admitted due to similar symptoms of gait instability and diplopia, generalised tingling sensation, and difficulty speaking. The examination revealed mild dysarthria, complete ophthalmoplegia, universal areflexia, ataxic gait, left-sided dysmetria on the finger-to-nose test, and preserved deep sensitivity, with no dysautonomic signs. Complementary testing yielded normal or negative results, including PCR testing for SARS-CoV-2 in nasopharyngeal aspirate, motor nerve conduction study (performed at 48 hours after symptom onset and repeated after 2 weeks), non-contrast brain MRI scan, and antiganglioside antibody determination, with no changes with respect to previous serology tests. He received treatment with Ig for 5 days, which improved symptoms, and was discharged with mild ophthalmoparesis.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Recurrence of MFS is infrequent.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,4,5</span></a> We present the case of a patient whose symptoms resolved after a first episode, before experiencing a relapse 2 years later, with similar symptoms and an infectious trigger in both episodes. We should highlight the negative results for antiganglioside antibodies during both admissions, as well as the normal findings in the motor nerve conduction study performed during the second admission. This lack of pathological findings has previously been described in other cases of recurrence,<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,7</span></a> and may be explained by the early performance of the study.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7,8</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">In other reported cases of recurrent GBS spectrum disorders, symptoms are usually more severe in the second episode,<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> which was not the case with our patient. He also presented an excellent response to Ig during the recurrence, unlike in the first episode. We do not know the reason for the lesser severity and better response to Ig during the relapse, although they may be related to immunological factors, as well as aetiological factors of the precipitating agent.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The causes predisposing to recurrent GBS are unclear. Patients younger than 30 years, with moderate impairment and MFS phenotype, are reported to present a higher risk of recurrence.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> While the reasons for this are not clear, some human leukocyte antigens (HLA-Cw3 and HLA-DR2), as well as some immunological factors, are reported to be involved.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,6,9,10</span></a></p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Peral Quirós A, Acebrón F, del Carmen Blanco Valero M, Labella Álvarez F. Recurrencia del síndrome de Miller Fisher: descripción de un caso. Neurología. 2021;36:561–562.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Update: the Miller Fisher variants of Guillain-Barré syndrome [published correction appears in Curr Opin Ophthalmol. 2020 Jan;31(1):80]" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "B. Al Othman" 1 => "J. Raabe" 2 => "A. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 24 | 0 | 24 |
| 2024 October | 84 | 13 | 97 |
| 2024 September | 75 | 21 | 96 |
| 2024 August | 74 | 10 | 84 |
| 2024 July | 59 | 8 | 67 |
| 2024 June | 69 | 6 | 75 |
| 2024 May | 69 | 11 | 80 |
| 2024 April | 53 | 6 | 59 |
| 2024 March | 69 | 10 | 79 |
| 2024 February | 95 | 16 | 111 |
| 2024 January | 97 | 8 | 105 |
| 2023 December | 98 | 15 | 113 |
| 2023 November | 67 | 13 | 80 |
| 2023 October | 85 | 23 | 108 |
| 2023 September | 42 | 12 | 54 |
| 2023 August | 47 | 9 | 56 |
| 2023 July | 46 | 3 | 49 |
| 2023 June | 45 | 12 | 57 |
| 2023 May | 58 | 20 | 78 |
| 2023 April | 58 | 5 | 63 |
| 2023 March | 51 | 5 | 56 |
| 2023 February | 33 | 5 | 38 |
| 2023 January | 29 | 10 | 39 |
| 2022 December | 30 | 6 | 36 |
| 2022 November | 27 | 9 | 36 |
| 2022 October | 52 | 9 | 61 |
| 2022 September | 54 | 9 | 63 |
| 2022 August | 35 | 12 | 47 |
| 2022 July | 28 | 11 | 39 |
| 2022 June | 53 | 9 | 62 |
| 2022 May | 46 | 23 | 69 |
| 2022 April | 50 | 8 | 58 |
| 2022 March | 62 | 16 | 78 |
| 2022 February | 76 | 10 | 86 |
| 2022 January | 76 | 17 | 93 |
| 2021 December | 39 | 9 | 48 |
| 2021 November | 27 | 15 | 42 |
| 2021 October | 30 | 11 | 41 |
| 2021 September | 13 | 10 | 23 |
| 2021 August | 0 | 3 | 3 |
| 2021 July | 0 | 8 | 8 |
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