Letter to the Editor
Transverse Myelitis and Bilateral Optic Neuritis in a Patient With Systemic Lupus Erythematosus
Mielitis transversa y neuritis óptica bilateral en una paciente con lupus eritematoso sistémico
Iñaki Hernando Rubio
, Joaquín Belzunegui Otano, Olga Máiz Alonso, Belén Álvarez Rodríguez
Corresponding author
Servicio de Reumatología, Hospital Donostia, San Sebastián, Spain
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During the course of the disease she presented malar rash, polyarthritis, serositis, positive antinuclear antibodies (ANA), anti-native DNA antibodies (anti-DNA) and several episodes of glomerulonephritis, for which she received pulse intravenous (iv) cyclophosphamide and maintenance therapy with azathioprine, suspended when she was 29 and remained stable.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Her initial chief complaint was dysphagia caused by esophageal diverticulum. At that time she received prednisone (7.5<span class="elsevierStyleHsp" style=""></span>mg daily) and hydroxychloroquine (400<span class="elsevierStyleHsp" style=""></span>mg daily). After 48<span class="elsevierStyleHsp" style=""></span>h, she had an episode of acute low back pain and paraparesis and a few hours later it became paraplegia with areflexia, tactile and painful hypesthesia, abolished sense of position and a distended bladder. The exploration of the upper extremities was normal. To this a total blindness of the left eye (LE) and a significant decrease in visual acuity of the right eye (RE) was added. We performed magnetic resonance imaging (MRI) of brain and orbit which showed findings consistent with bilateral optic neuritis. The thoracolumbar spine MRI disclosed a lesion compatible with transverse myelitis in the segment between the D7 vertebral body and the conus medullaris. The cerebrospinal fluid analysis revealed 576<span class="elsevierStyleHsp" style=""></span>leukocytes/mm<span class="elsevierStyleSup">3</span> with 95% polymorphonuclear and 0 erythrocytes/mm<span class="elsevierStyleSup">3</span>. Proteins were 347.79<span class="elsevierStyleHsp" style=""></span>mg/dl with normal glucose. Cultures, Gram stain, smear, Lowenstein culture and herpes simplex 1 and 2, varicella zoster and enterovirus PCR testing were negative. The ADA was 17.1<span class="elsevierStyleHsp" style=""></span>U/l. Ophthalmologic examination revealed a bilateral afferent pupillary defect and a deficit in bilateral visual acuity (perception of large forms and hard colors in total amaurosis of the LE and RE). The fundus was normal. ANA titer was 1/1.280 and anti-DNA Ab 54<span class="elsevierStyleHsp" style=""></span>U/ml. Extractable core antibodies were negative. Complement levels were low. Coagulation was normal and anticardiolipin, antiphospholipid antibodies and lupus anticoagulant were negative. We established a diagnosis of ONM and started treatment with methylprednisolone 1<span class="elsevierStyleHsp" style=""></span>g iv/day for 5 consecutive days, 1<span class="elsevierStyleHsp" style=""></span>g iv cyclophosphamide and 1<span class="elsevierStyleHsp" style=""></span>g/kg iv immunoglobulin for 2 consecutive days. This was repeated a month later. Five weeks later, the patient was able to distinguish large forms with her LE and to distinguish colors and count fingers with the RE. There was no improvement in mobility of the lower extremities. Cervical, thoracic and lumbar spine MRI was performed, where 2 multisegmental intramedullary lesions were observed: the first one located from D7 to the conus medullaris and showed no changes from the previous MRI, and the second in the left posterolateral region of the conus medullaris at C4–C5, both compatible with transverse myelitis.</p><p id="par0020" class="elsevierStylePara elsevierViewall">After 6 years with continued azathioprine treatment, the patient remained paraplegic and with a significant visual deficit (blurred RE vision and able to distinguish large forms with the LE). She has had no new episodes of ONM.</p><p id="par0025" class="elsevierStylePara elsevierViewall">ONM is a rare and severe CNS demyelinating inflammatory condition characterized by the coexistence of optic neuritis and transverse myelitis. The description in 2004 of the IgG-NMO1 allowed the establishment, in 2006, of a new diagnostic criteria to distinguish this disease from MS.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> It is estimated that the annual incidence of ONM is 0.4 cases per 100<span class="elsevierStyleHsp" style=""></span>000 persons/year and prevalence in Caucasians is 4.4 cases per 100<span class="elsevierStyleHsp" style=""></span>000 population.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The course can be a single episode or recurrent and, although it may be idiopathic, it is often associated with autoimmune diseases such as SLE or Sjögren's syndrome, presence of autoantibodies, infectious agents and exposure to drugs. The involvement of B lymphocytes with the participation of NMO-IgG in relapses<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> has been proposed as contributing to its pathogenesis. MRI reveals hyperintense T2 images in 3 or more spinal cord segments and optic nerve damage that is enhanced after gadolinium administration. These findings differ from those observed in MS.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The immediate start of aggressive treatment with high dose IV methylprednisolone and cyclophosphamide is essential to reduce the consequences of acute episodes.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Azathioprine has been shown to reduce recurrence,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> although the prognosis is generally unfavorable.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Hernando Rubio I, et al. Mielitis transversa y neuritis óptica bilateral en una paciente con lupus eritematoso sistémico. Reumatol Clin. 2012. <span class="elsevierStyleInterRef" href="http://dx.doi.org/10.1016/j.reuma.2012.01.005">http://dx.doi.org/10.1016/j.reuma.2012.01.005</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" => "A serum autoantibody marker of neuromyelitis optica: distinction from multiple sclerosis" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "V.A. Lennon" 1 => "D.M. Wingerchuk" 2 => "T.J. Kryzer" 3 => "S.J. Pittock" 4 => "C.F. Lucchinetti" 5 => "K. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 October | 69 | 36 | 105 |
| 2024 September | 60 | 17 | 77 |
| 2024 August | 91 | 33 | 124 |
| 2024 July | 108 | 45 | 153 |
| 2024 June | 82 | 33 | 115 |
| 2024 May | 99 | 32 | 131 |
| 2024 April | 102 | 29 | 131 |
| 2024 March | 67 | 48 | 115 |
| 2024 February | 38 | 51 | 89 |
| 2024 January | 59 | 40 | 99 |
| 2023 December | 45 | 52 | 97 |
| 2023 November | 53 | 36 | 89 |
| 2023 October | 46 | 48 | 94 |
| 2023 September | 82 | 42 | 124 |
| 2023 August | 64 | 23 | 87 |
| 2023 July | 46 | 39 | 85 |
| 2023 June | 41 | 28 | 69 |
| 2023 May | 46 | 24 | 70 |
| 2023 April | 40 | 25 | 65 |
| 2023 March | 49 | 44 | 93 |
| 2023 February | 51 | 45 | 96 |
| 2023 January | 33 | 35 | 68 |
| 2022 December | 54 | 44 | 98 |
| 2022 November | 63 | 40 | 103 |
| 2022 October | 54 | 41 | 95 |
| 2022 September | 55 | 48 | 103 |
| 2022 August | 52 | 37 | 89 |
| 2022 July | 49 | 63 | 112 |
| 2022 June | 68 | 52 | 120 |
| 2022 May | 100 | 48 | 148 |
| 2022 April | 65 | 65 | 130 |
| 2022 March | 97 | 77 | 174 |
| 2022 February | 133 | 47 | 180 |
| 2022 January | 96 | 65 | 161 |
| 2021 December | 73 | 60 | 133 |
| 2021 November | 58 | 71 | 129 |
| 2021 October | 80 | 62 | 142 |
| 2021 September | 54 | 54 | 108 |
| 2021 August | 39 | 41 | 80 |
| 2021 July | 47 | 30 | 77 |
| 2021 June | 38 | 34 | 72 |
| 2021 May | 66 | 50 | 116 |
| 2021 April | 113 | 81 | 194 |
| 2021 March | 100 | 44 | 144 |
| 2021 February | 51 | 20 | 71 |
| 2021 January | 51 | 18 | 69 |
| 2020 December | 54 | 28 | 82 |
| 2020 November | 35 | 19 | 54 |
| 2020 October | 36 | 15 | 51 |
| 2020 September | 60 | 25 | 85 |
| 2020 August | 41 | 18 | 59 |
| 2020 July | 32 | 23 | 55 |
| 2020 June | 47 | 27 | 74 |
| 2020 May | 46 | 21 | 67 |
| 2020 April | 40 | 27 | 67 |
| 2020 March | 19 | 13 | 32 |
| 2020 February | 1 | 0 | 1 |
| 2020 January | 4 | 0 | 4 |
| 2019 September | 4 | 0 | 4 |
| 2019 June | 1 | 0 | 1 |
| 2019 March | 1 | 0 | 1 |
| 2019 January | 1 | 0 | 1 |
| 2018 May | 6 | 1 | 7 |
| 2018 April | 63 | 7 | 70 |
| 2018 March | 70 | 17 | 87 |
| 2018 February | 58 | 3 | 61 |
| 2018 January | 41 | 3 | 44 |
| 2017 December | 63 | 6 | 69 |
| 2017 November | 40 | 8 | 48 |
| 2017 October | 35 | 4 | 39 |
| 2017 September | 45 | 6 | 51 |
| 2017 August | 58 | 5 | 63 |
| 2017 July | 65 | 9 | 74 |
| 2017 June | 80 | 15 | 95 |
| 2017 May | 80 | 11 | 91 |
| 2017 April | 64 | 8 | 72 |
| 2017 March | 48 | 8 | 56 |
| 2017 February | 38 | 10 | 48 |
| 2017 January | 29 | 13 | 42 |
| 2016 December | 73 | 14 | 87 |
| 2016 November | 71 | 6 | 77 |
| 2016 October | 77 | 15 | 92 |
| 2016 September | 79 | 7 | 86 |
| 2016 August | 67 | 7 | 74 |
| 2016 July | 46 | 11 | 57 |
| 2016 May | 0 | 15 | 15 |
| 2015 December | 3 | 0 | 3 |
| 2015 October | 1 | 0 | 1 |
| 2015 September | 6 | 13 | 19 |
| 2015 August | 2 | 0 | 2 |
| 2015 July | 20 | 7 | 27 |
| 2015 June | 46 | 8 | 54 |
| 2015 May | 46 | 17 | 63 |
| 2015 April | 48 | 10 | 58 |
| 2015 March | 38 | 8 | 46 |
| 2015 February | 51 | 7 | 58 |
| 2015 January | 46 | 8 | 54 |
| 2014 December | 59 | 7 | 66 |
| 2014 November | 49 | 10 | 59 |
| 2014 October | 47 | 11 | 58 |
| 2014 September | 43 | 10 | 53 |
| 2014 August | 51 | 13 | 64 |
| 2014 July | 51 | 12 | 63 |
| 2014 June | 55 | 8 | 63 |
| 2014 May | 44 | 14 | 58 |
| 2014 April | 48 | 7 | 55 |
| 2014 March | 54 | 16 | 70 |
| 2014 February | 33 | 15 | 48 |
| 2014 January | 48 | 12 | 60 |
| 2013 December | 37 | 9 | 46 |
| 2013 November | 39 | 8 | 47 |
| 2013 October | 42 | 14 | 56 |
| 2013 September | 43 | 11 | 54 |
| 2013 August | 48 | 20 | 68 |
| 2013 July | 40 | 10 | 50 |
| 2013 June | 45 | 8 | 53 |
| 2013 May | 42 | 10 | 52 |
| 2013 April | 43 | 6 | 49 |
| 2013 March | 50 | 13 | 63 |
| 2013 February | 40 | 11 | 51 |
| 2013 January | 32 | 9 | 41 |
| 2012 December | 33 | 12 | 45 |
| 2012 November | 25 | 23 | 48 |
| 2012 October | 22 | 21 | 43 |
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