Letter to the Editor
Internal ophthalmoplegia as the initial symptom of Miller-Fisher syndrome
Oftalmoplejía interna como comienzo de un síndrome de Miller-Fisher
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Terry López, D. Sagarra Mur, A.M. Gutiérrez Álvarez, C. Jiménez Corral" "autores" => array:4 [ 0 => array:4 [ "nombre" => "O." "apellidos" => "Terry López" "email" => array:1 [ 0 => "oscarterry@hotmail.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" => "D." "apellidos" => "Sagarra Mur" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "A.M." "apellidos" => "Gutiérrez Álvarez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "C." "apellidos" => "Jiménez Corral" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Medicina Interna, Hospital Santa Bárbara, Soria, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Neurología, Hospital Santa Bárbara, Soria, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Oftalmoplejía interna como comienzo de un síndrome de Miller-Fisher" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Guillain-Barré syndrome (GBS) is an autoimmune polyradiculoneuropathy that may follow infection. The syndrome is divided into several subgroups: chronic inflammatory demyelinating polyneuropathy (the most frequent variant), acute pandysautonomia, acute motor axonal neuropathy, acute motor and sensory axonal neuropathy, and Miller-Fisher syndrome (MFS).<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> This last syndrome, a rare variant of GBS, is characterised by the classic symptom triad of ophthalmoplegia, ataxia, and areflexia. In addition to this triad, bulbar paralysis, weakness, and sensory loss may also appear.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We present the case of a patient with internal ophthalmoplegia as the initial symptom of MFS.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Our patient is a 74-year-old woman with a history of primary hypothyroidism and long-term treatment with levothyroxine. She was referred to the emergency department by her primary care doctor due to moderate fronto-orbital headache and non-reactive mid-size pupils without loss of visual acuity. She presented no photophobia or sonophobia and had not been using eye drops. During the preceding week, the patient had experienced itching, mild pharyngeal pain, and self-limiting fever. She was treated with amoxicillin/clavulanic acid and anti-inflammatory drugs.</p><p id="par0020" class="elsevierStylePara elsevierViewall">During the neurological examination, the patient was conscious and oriented but showed non-reactive mid-sized pupils. Doctors observed no light-near dissociation, diplopia, nystagmus, abnormal eye movements, visual field deficits, abnormal cranial nerves, dysphagia, language and speech disorders, dysmetria, or motor or sensory disorders. Gait was normal and muscle stretch reflexes were rated 1/5. Romberg test was negative and there were no meningeal signs.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The day after her admission, the patient presented mildly limited eye movement and mild gait instability. By the second day, she presented total external and internal ophthalmoplegia, with minimum ataxia and hyporeflexia.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Blood tests yielded normal results, and results of antineuronal antibody tests were negative. Serology studies for human immunodeficiency virus, syphilis, <span class="elsevierStyleItalic">Brucella</span>, Epstein-Barr virus, cytomegalovirus and <span class="elsevierStyleItalic">Borrelia</span> yielded negative results.</p><p id="par0035" class="elsevierStylePara elsevierViewall">According to the analysis, cerebrospinal fluid (CSF) was acellular and showed a high protein concentration (69<span class="elsevierStyleHsp" style=""></span>mg/dL); all other parameters were within normal ranges. CSF culture and serology were negative. Tests for anti-GQ1b IgG and the remaining anti-ganglioside antibodies (GM1-4, GD1a, GD1b, GD2-3, GT1a, GT1b) were negative.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Head CT scan showed discreet signs of parenchymatous atrophy. Head MRI scan showed a small image indicative of ischaemic lesion on the left frontal lobe. The rest of the examination yielded normal results.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Results of the ophthalmological study were also normal with the exception of the non-reactive mid-sized pupils.</p><p id="par0050" class="elsevierStylePara elsevierViewall">After receiving the results from the CSF analysis, doctors started treatment with intravenous immunoglobulins. On the fifth day of infusion, the patient's condition began to improve. Two months after discharge, the patient was asymptomatic and results from her neurological examination were normal.</p><p id="par0055" class="elsevierStylePara elsevierViewall">GBS and its variants (including MFS) are part of a series of autoimmune disorders that trigger an acute inflammatory response that may follow infection or vaccination. MFS was first described in 1956,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> and it manifests with the classic symptom triad of external ophthalmoplegia, ataxia, and areflexia. Its course is usually monophasic and it is one of the least frequent subtypes of GBS.</p><p id="par0060" class="elsevierStylePara elsevierViewall">Our patient presented symptoms of upper respiratory tract infection one week before syndrome onset, which is in line with descriptions found in the literature. The most relevant point in our case is the atypical initial manifestation as internal ophthalmoplegia with non-reactive mid-size pupils, as well as fronto-orbital headache. Internal ophthalmoplegia is much less frequent than external ophthalmoplegia.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a> The literature contains few references to presence of internal ophthalmoplegia in the absence of the external type, and we have only found one case resembling our own in that isolated internal ophthalmoplegia was the initial symptom.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Pupil disorders can be observed in up to 50% of MFS cases and they tend to progress regardless of whether or not external ophthalmoplegia is present. These disorders usually resolve in a significantly shorter period than is the case for ophthalmoplegia.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> These findings show that the involvement of pupillomotor fibres is independent of any lesions in other subdivisions of the oculomotor nerve.</p><p id="par0070" class="elsevierStylePara elsevierViewall">The patient will subsequently start to develop partial external ophthalmoplegia and mild gait ataxia with hyporeflexia.</p><p id="par0075" class="elsevierStylePara elsevierViewall">As a general rule, anti-GQ1b IgG antibodies are present in more than 90% of MFS cases.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7–9</span></a> This is because oculomotor nerves and the optic nerve contain large quantities of GQ1b gangliosides.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Evaluating these antibodies and other gangliosides is helpful but not essential to the diagnostic process. In our case, no anti-GQ1b IgG antibodies were detected despite the fact that the patient presented external and internal ophthalmoplegia, but this does not rule out MFS. In this case, the diagnosis was determined based on clinical signs, albuminocytological dissociation in CSF, and having ruled out other processes.</p><p id="par0080" class="elsevierStylePara elsevierViewall">Neurophysiological studies were not necessary in this case given that clinical progression and CSF analyses provided sufficient data. However, such studies can help determine early diagnosis in some specific cases, especially in the acute phase.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a></p><p id="par0085" class="elsevierStylePara elsevierViewall">The treatment of choice for MFS is IV immunoglobulins, or plasmapheresis in drug-resistant cases.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a> The patient started to improve on the fifth day after starting treatment with IV immunoglobulins, and her condition had resolved completely within 2 months.</p><p id="par0090" class="elsevierStylePara elsevierViewall">In conclusion, we present a rare case of MFS with an exceptional form of onset consisting of isolated internal ophthalmoplegia and fronto-orbital headache. Furthermore, anti-GQ1b IgG antibodies are not present even though the patient presented internal and external ophthalmoplegia. The incidence rate of this condition has probably been underestimated since it may be overlooked in the initial diagnostic process when symptoms are so mild that they may spontaneously resolve. In our case, lack of concomitant neurological signs meant that onset could have been overlooked, but the condition progressed rapidly in this patient, resulting in manifestation of the classic triad. We must be aware of this type of condition since it progresses quickly and consequences for patients may be severe.</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: Terry López O, Sagarra Mur D, Gutiérrez Álvarez AM, Jiménez Corral C. Oftalmoplejía interna como comienzo de un síndrome de Miller-Fisher. Neurología. 2014;29:504‐505.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:12 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Neuropatías disinmunes adquiridas. Sintomatología clínica y clasificación" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "A. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 7 | 0 | 7 |
| 2024 October | 55 | 4 | 59 |
| 2024 September | 57 | 9 | 66 |
| 2024 August | 44 | 1 | 45 |
| 2024 July | 61 | 6 | 67 |
| 2024 June | 53 | 5 | 58 |
| 2024 May | 48 | 4 | 52 |
| 2024 April | 89 | 5 | 94 |
| 2024 March | 91 | 8 | 99 |
| 2024 February | 86 | 10 | 96 |
| 2024 January | 108 | 7 | 115 |
| 2023 December | 95 | 11 | 106 |
| 2023 November | 70 | 3 | 73 |
| 2023 October | 118 | 3 | 121 |
| 2023 September | 66 | 9 | 75 |
| 2023 August | 51 | 9 | 60 |
| 2023 July | 72 | 9 | 81 |
| 2023 June | 73 | 6 | 79 |
| 2023 May | 123 | 9 | 132 |
| 2023 April | 84 | 5 | 89 |
| 2023 March | 70 | 7 | 77 |
| 2023 February | 97 | 4 | 101 |
| 2023 January | 87 | 8 | 95 |
| 2022 December | 52 | 7 | 59 |
| 2022 November | 61 | 10 | 71 |
| 2022 October | 68 | 9 | 77 |
| 2022 September | 75 | 18 | 93 |
| 2022 August | 74 | 11 | 85 |
| 2022 July | 63 | 11 | 74 |
| 2022 June | 54 | 8 | 62 |
| 2022 May | 62 | 6 | 68 |
| 2022 April | 69 | 19 | 88 |
| 2022 March | 72 | 18 | 90 |
| 2022 February | 86 | 12 | 98 |
| 2022 January | 106 | 9 | 115 |
| 2021 December | 74 | 19 | 93 |
| 2021 November | 90 | 7 | 97 |
| 2021 October | 140 | 11 | 151 |
| 2021 September | 93 | 13 | 106 |
| 2021 August | 118 | 8 | 126 |
| 2021 July | 93 | 10 | 103 |
| 2021 June | 96 | 12 | 108 |
| 2021 May | 130 | 11 | 141 |
| 2021 April | 321 | 22 | 343 |
| 2021 March | 201 | 9 | 210 |
| 2021 February | 144 | 10 | 154 |
| 2021 January | 131 | 15 | 146 |
| 2020 December | 91 | 7 | 98 |
| 2020 November | 109 | 7 | 116 |
| 2020 October | 74 | 10 | 84 |
| 2020 September | 98 | 10 | 108 |
| 2020 August | 112 | 3 | 115 |
| 2020 July | 76 | 8 | 84 |
| 2020 June | 85 | 9 | 94 |
| 2020 May | 105 | 12 | 117 |
| 2020 April | 128 | 5 | 133 |
| 2020 March | 148 | 10 | 158 |
| 2020 February | 155 | 5 | 160 |
| 2020 January | 122 | 16 | 138 |
| 2019 December | 146 | 14 | 160 |
| 2019 November | 138 | 21 | 159 |
| 2019 October | 176 | 5 | 181 |
| 2019 September | 228 | 13 | 241 |
| 2019 August | 98 | 14 | 112 |
| 2019 July | 137 | 12 | 149 |
| 2019 June | 265 | 27 | 292 |
| 2019 May | 344 | 54 | 398 |
| 2019 April | 223 | 33 | 256 |
| 2019 March | 89 | 8 | 97 |
| 2019 February | 62 | 11 | 73 |
| 2019 January | 70 | 17 | 87 |
| 2018 December | 46 | 5 | 51 |
| 2018 November | 57 | 13 | 70 |
| 2018 October | 51 | 10 | 61 |
| 2018 September | 25 | 6 | 31 |
| 2018 August | 27 | 1 | 28 |
| 2018 July | 12 | 2 | 14 |
| 2018 June | 14 | 3 | 17 |
| 2018 May | 22 | 6 | 28 |
| 2018 April | 29 | 2 | 31 |
| 2018 March | 23 | 0 | 23 |
| 2018 February | 6 | 3 | 9 |
| 2018 January | 23 | 1 | 24 |
| 2017 December | 16 | 0 | 16 |
| 2017 November | 42 | 6 | 48 |
| 2017 October | 33 | 2 | 35 |
| 2017 September | 29 | 1 | 30 |
| 2017 August | 24 | 4 | 28 |
| 2017 July | 48 | 2 | 50 |
| 2017 June | 44 | 1 | 45 |
| 2017 May | 18 | 5 | 23 |
| 2017 April | 11 | 23 | 34 |
| 2017 March | 13 | 43 | 56 |
| 2017 February | 9 | 3 | 12 |
| 2017 January | 7 | 8 | 15 |
| 2016 December | 21 | 5 | 26 |
| 2016 November | 18 | 2 | 20 |
| 2016 October | 28 | 8 | 36 |
| 2016 September | 37 | 7 | 44 |
| 2016 August | 25 | 7 | 32 |
| 2016 July | 22 | 6 | 28 |
| 2016 June | 24 | 9 | 33 |
| 2016 May | 16 | 10 | 26 |
| 2016 April | 14 | 12 | 26 |
| 2016 March | 24 | 24 | 48 |
| 2016 February | 24 | 19 | 43 |
| 2016 January | 5 | 8 | 13 |
| 2015 December | 10 | 9 | 19 |
| 2015 November | 14 | 10 | 24 |
| 2015 October | 19 | 12 | 31 |
| 2015 September | 21 | 12 | 33 |
| 2015 August | 23 | 7 | 30 |
| 2015 July | 16 | 7 | 23 |
| 2015 June | 8 | 3 | 11 |
| 2015 May | 25 | 9 | 34 |
| 2015 April | 13 | 12 | 25 |
| 2015 March | 25 | 12 | 37 |
| 2015 February | 42 | 6 | 48 |
| 2015 January | 29 | 10 | 39 |
| 2014 December | 69 | 22 | 91 |
| 2014 November | 49 | 18 | 67 |
| 2014 October | 101 | 39 | 140 |
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