Letter to the Editor
Cervical myelopathy as a form of presentation of Whipple disease
Mielopatía cervical como forma de presentación de la enfermedad de Whipple
Á.I. Pérez Álvarez
, G. Morís de la Tassa
Corresponding author
Servicio de Neurología, Hospital Universitario Central deAsturias, Oviedo, Asturias, Spain
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Signal alterations are observed in the left central and lateral splenium (white arrow), with restricted diffusion and no contrast enhancement. These findings are compatible with cytotoxic oedema. No alterations were observed in the rest of the corpus callosum (D) or in any other part of the brain parenchyma.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "P. Gómez Iglesias, E. López Valdés, M. Vega Bayoll, M.N. Gómez Ruíz" "autores" => array:4 [ 0 => array:2 [ "nombre" => "P." "apellidos" => "Gómez Iglesias" ] 1 => array:2 [ "nombre" => "E." "apellidos" => "López Valdés" ] 2 => array:2 [ "nombre" => "M." "apellidos" => "Vega Bayoll" ] 3 => array:2 [ "nombre" => "M.N." "apellidos" => "Gómez Ruíz" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0213485319300532" "doi" => "10.1016/j.nrl.2019.03.006" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0213485319300532?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173580820301644?idApp=UINPBA00004N" "url" => "/21735808/0000003500000008/v1_202010030617/S2173580820301644/v1_202010030617/en/main.assets" ] "en" => array:17 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Letter to the Editor</span>" "titulo" => "Cervical myelopathy as a form of presentation of Whipple disease" "tieneTextoCompleto" => true "saludo" => "Dear Editor:" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "583" "paginaFinal" => "585" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Á.I. Pérez Álvarez, G. Morís de la Tassa" "autores" => array:2 [ 0 => array:4 [ "nombre" => "Á.I." "apellidos" => "Pérez Álvarez" "email" => array:1 [ 0 => "angelperez@telecable.es" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "G." "apellidos" => "Morís de la Tassa" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Neurología, Hospital Universitario Central deAsturias, Oviedo, Asturias, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Mielopatía cervical como forma de presentación de la enfermedad de Whipple" ] ] "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" => 1062 "Ancho" => 905 "Tamanyo" => 78703 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">T2-weighted spinal MRI scan (sagittal plane) showing a hyperintense lesion in the posterior column between C2 and T1.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Whipple disease is a multisystem disease caused by bacterial infection with <span class="elsevierStyleItalic">Tropheryma whipplei</span>. It was first described by George H. Whipple in 1907 as an “intestinal lipodystrophy” in a patient with weight loss, polyarthritis, diarrhoea, malabsorption, and mesenteric lymphadenitis.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Presence of neurological symptoms in the absence of systemic manifestations is rare.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We present a case of Whipple disease manifesting with isolated symptoms of spinal cord involvement. Our patient was a 66-year-old woman who consulted due to a one-year history of gait alterations, with no associated symptoms. The neurological examination revealed decreased deep sensitivity in the lower limbs, with no pyramidal signs; results were normal for all other areas. A somatosensory evoked potentials study revealed injury to the somatosensory pathway at the level of the posterior column. A non-contrast, T2-weighted spinal cord MRI scan performed to rule out myelopathy revealed a hyperintense lesion in the posterior column between C2 and T1, with no associated oedema (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>); no brain lesions were identified. A blood analysis including a complete biochemistry study and immunological tests found no evidence of autoimmune, systemic, or metabolic diseases, nutritional deficiencies, etc. Given that differential diagnosis includes viral and bacterial infections, we requested a microbiological analysis of a blood sample; PCR findings were positive for <span class="elsevierStyleItalic">T. whipplei</span>. Biochemical, microbiological, immunological, and cytological analyses of the CSF yielded normal results, with the exception of positive PCR results for <span class="elsevierStyleItalic">T. whipplei</span>. The pathogen was not detected in the faeces or saliva. An additional PCR test performed 15 days later also yielded positive results for <span class="elsevierStyleItalic">T. whipplei</span>. The patient received intravenous ceftriaxone for 14 days, showing good clinical response. Long-term treatment with oral trimethoprim-sulfamethoxazole achieved marked improvements in gait. An MRI scan performed several months later revealed stability of the spinal cord lesion. Subsequent blood and CSF PCR tests yielded negative results.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Whipple disease is an infectious disease associated with multisystem manifestations, most frequently affecting the gastrointestinal system and joints. Although the precise incidence is unknown,<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> central nervous system involvement is frequent, appearing in 43% of cases<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a>; however, presence of neurological symptoms in isolation occurs in only 5% of patients.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Neurological symptoms are nonspecific and highly variable, ranging from impaired consciousness to cognitive impairment, epileptic seizures, myoclonus, cerebellar or hypothalamic involvement, cranial and peripheral neuropathies, and extrapyramidal symptoms.<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,6</span></a> Oculomasticatory myorhythmia and oculofacial-skeletal myorhythmia are pathognomonic of Whipple disease.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7,8</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Although some cases have been described of myelopathy associated with other inflammatory lesions in different parts of the central nervous system (eg, the brain, optic chiasm, posterior fossa), as in the patient described by Kremer et al.,<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> presentation in the form of isolated myelopathy is even rarer. The first case was reported by Clarke et al.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> in 1998 in a 62-year-old patient; Messori and Salvolini<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> and Messori et al.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a> subsequently described the case of a 65-year-old patient with relapsing-remitting cervico-thoracic myelopathy due to Whipple disease who subsequently developed brain lesions. In 2005, Schröter et al.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">13</span></a> described the case of a 50-year-old patient with a cervical spinal cord lesion extending from the cervicomedullary junction to the upper thoracic region, without cerebral involvement; the patient responded well to antibiotic therapy against <span class="elsevierStyleItalic">T. whipplei</span>, which was detected in a blood PCR test. To our knowledge, our patient is the fourth reported case (and the first in Spain) of Whipple disease manifesting as isolated myelopathy affecting the posterior column. The cases described bear certain similarities, including age of presentation (between the sixth and seventh decades of life), symptoms of chronic progressive myelitis, extensive cervical spinal cord involvement with MRI hyperintensity, and marked improvement with antibiotics.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Differential diagnosis should include other bacterial, viral, and parasitic infections; such demyelinating diseases as multiple sclerosis, neuromyelitis optica, and acute disseminated encephalomyelitis; vascular disease (ischaemia, haemorrhage); tumours or paraneoplastic syndromes; and systemic (eg, systemic lupus erythematosus, Behçet disease, Sjögren syndrome, sarcoidosis) or deficiency diseases (vitamin deficiency).</p><p id="par0035" class="elsevierStylePara elsevierViewall">The limited published evidence on the topic and the nonspecific nature of the symptoms may hinder diagnosis and delay treatment. Neurological symptoms may constitute the initial manifestation of Whipple disease, with other more characteristic symptoms appearing at later stages.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">14</span></a> In any case, the lack of microbiological confirmation should not delay treatment in cases of suspected Whipple disease. Targeted antibiotic therapy for bacterial infection is recommended before spinal cord biopsy is performed.</p><p id="par0040" class="elsevierStylePara elsevierViewall">In conclusion, symptoms of spinal cord involvement are infrequent in Whipple disease. Therefore, infection with <span class="elsevierStyleItalic">T. whipplei</span> should be considered in patients with isolated myelopathy of unknown cause, even in the absence of systemic symptoms; this requires a high level of clinical suspicion. Early diagnosis enables the provision of effective treatment able to resolve symptoms and even to prevent lesions from spreading to other organs or regions of the nervous system; Whipple disease is potentially fatal if the patient does not receive targeted treatment.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0045" class="elsevierStylePara elsevierViewall">This study has received no specific funding from any public, private, or non-profit organisation.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Funding" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2018-10-10" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Pérez Álvarez ÁI, Morís de la Tassa G. Mielopatía cervical como forma de presentación de la enfermedad de Whipple. Neurología. 2020;35:583–585.</p>" ] ] "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1062 "Ancho" => 905 "Tamanyo" => 78703 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">T2-weighted spinal MRI scan (sagittal plane) showing a hyperintense lesion in the posterior column between C2 and T1.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 594 "Ancho" => 905 "Tamanyo" => 38115 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">T2-weighted spinal MRI scan (axial plane) showing a hyperintense lesion in the posterior column at the cervical level.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:14 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "A hitherto undescribed disease characterized anatomically by deposits of fat and fatty acids in the intestinal and mesenteric lymphatic tissues" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "G.H. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 10 | 1 | 11 |
| 2024 October | 36 | 9 | 45 |
| 2024 September | 31 | 20 | 51 |
| 2024 August | 50 | 6 | 56 |
| 2024 July | 27 | 10 | 37 |
| 2024 June | 31 | 1 | 32 |
| 2024 May | 16 | 4 | 20 |
| 2024 April | 27 | 1 | 28 |
| 2024 March | 57 | 5 | 62 |
| 2024 February | 52 | 8 | 60 |
| 2024 January | 51 | 5 | 56 |
| 2023 December | 48 | 13 | 61 |
| 2023 November | 49 | 6 | 55 |
| 2023 October | 63 | 7 | 70 |
| 2023 September | 36 | 3 | 39 |
| 2023 August | 38 | 7 | 45 |
| 2023 July | 47 | 4 | 51 |
| 2023 June | 49 | 3 | 52 |
| 2023 May | 55 | 3 | 58 |
| 2023 April | 64 | 5 | 69 |
| 2023 March | 55 | 5 | 60 |
| 2023 February | 41 | 8 | 49 |
| 2023 January | 35 | 4 | 39 |
| 2022 December | 26 | 14 | 40 |
| 2022 November | 46 | 16 | 62 |
| 2022 October | 41 | 7 | 48 |
| 2022 September | 36 | 13 | 49 |
| 2022 August | 31 | 7 | 38 |
| 2022 July | 32 | 7 | 39 |
| 2022 June | 25 | 6 | 31 |
| 2022 May | 32 | 23 | 55 |
| 2022 April | 35 | 12 | 47 |
| 2022 March | 38 | 15 | 53 |
| 2022 February | 49 | 9 | 58 |
| 2022 January | 62 | 8 | 70 |
| 2021 December | 36 | 11 | 47 |
| 2021 November | 25 | 8 | 33 |
| 2021 October | 43 | 13 | 56 |
| 2021 September | 31 | 8 | 39 |
| 2021 August | 35 | 6 | 41 |
| 2021 July | 18 | 12 | 30 |
| 2021 June | 25 | 13 | 38 |
| 2021 May | 39 | 8 | 47 |
| 2021 April | 94 | 21 | 115 |
| 2021 March | 52 | 13 | 65 |
| 2021 February | 26 | 13 | 39 |
| 2021 January | 30 | 6 | 36 |
| 2020 December | 21 | 12 | 33 |
| 2020 November | 19 | 9 | 28 |
| 2020 October | 18 | 10 | 28 |
| 2020 September | 4 | 4 | 8 |
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