Letter to the Editor
Adult diagnosis of temporo-occipital leptomeningeal angiomatosis
Angiomatosis leptomeníngea temporo-occipital de diagnóstico en edad adulta
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Gómez-Moreno, C. Murrieta-Urruticoechea, E. Martinez-Acebes, R. Gordo-Mañas" "autores" => array:4 [ 0 => array:4 [ "nombre" => "M." "apellidos" => "Gómez-Moreno" "email" => array:1 [ 0 => "mayragomezmoreno@yahoo.es" ] "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" => "C." "apellidos" => "Murrieta-Urruticoechea" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "E." "apellidos" => "Martinez-Acebes" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "R." "apellidos" => "Gordo-Mañas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Sección de Neurología, Hospital Universitario Infanta Leonor, Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Neurología, Hôpital Gui de Chauliac, CHU Montpellier, Montpellier, France" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Angiomatosis leptomeníngea temporo-occipital de diagnóstico en edad adulta" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1171 "Ancho" => 2918 "Tamanyo" => 241292 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Cranial CT without contrast: cortical calcification and discrete atrophy of the right occipital lobe, and calcification and enlargement of the choroid plexus of the occipital horn of the right ventricle.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Sturge-Weber syndrome (SWS) or encephalotrigeminal angiomatosis is a sporadically presenting neuroectodermatosis characterised by a facial port-wine stain in the trigeminal distribution, ipsilateral leptomeningeal angioma, and ipsilateral choroidal angioma. Studies have described clinical variants with different combinations within this triad. SWS is primarily diagnosed in children, but it has also been diagnosed in adults in exceptional cases. We present a case of isolated leptomeningeal angiomatosis and symptom onset in an adult patient.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 44-year-old woman with no relevant medical history came to the emergency department due to sudden-onset right hemicranial headache that was intense, oppressive, and responding poorly to analgesics. Twenty-four hours later, symptoms also included disorientation, psychomotor agitation, and visual hallucinations. Physical examination revealed a herpes labialis sore on her upper lip. Laboratory analyses revealed leukocytosis with left shift and elevated C-reactive protein (CRP). Cranial axial computed tomography with and without contrast (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>) showed a right parietal-occipital gyriform hyperdensity and focal meningeal enhancement that was initially reported as being compatible with chronic meningitis. Lumbar puncture yielded acellular CSF with elevated protein levels, so the emergency department started provisional treatment with IV vancomycin, ceftriaxone, and acyclovir for possible meningoencephalitis. The patient was admitted to the neurology department and remained afebrile with an intense headache accompanied by nausea, vomiting, and fluctuating episodes of visual and auditory hallucinations. Neurological examination detected homonymous hemianopsia. Brain MRI scans performed with and without gadolinium contrast (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>) showed leptomeningeal angiomatosis in the right hemisphere (parietal-occipital-temporal area). Basal amplitude on the electroencephalogram was attenuated in the right temporal-occipital area. There were no data indicating infection (results from 2 blood and CSF cultures and CRP tests were all negative); since a second lumbar puncture yielded acellular CSF with elevated opening pressure and protein levels, antibiotics were discontinued. A consultation with the ophthalmology department ruled out ocular vascular malformation. The patient was diagnosed with SWS without facial angioma and exhibiting temporal-occipital partial seizures and possible venous thrombosis/stasis at the angiomatous lesion. She was treated with acetazolamide (for intracranial hypertension), levetiracetam, and acetylsalicylic acid. Both the headache and the confusional episodes/hallucinations resolved; the latter were thought to be caused by seizure or vascular migraine-like phenomena. The patient subsequently attended follow-up visits as a neurology outpatient. After an additional lumbar puncture yielded acellular CSF with normal opening pressure and protein levels, the dose of acetazolamide was gradually reduced until it could be discontinued. The patient remained asymptomatic at a year of follow-up.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Encephalotrigeminal angiomatosis or SWS is an uncommon neurocutaneous syndrome affecting brain microvasculature that presents sporadically and is usually diagnosed in childhood. Clinical characteristics of its typical form include a flat facial angioma (port-wine stain) in the distribution of the ophthalmic branch of the trigeminal nerve, ipsilateral leptomeningeal angiomatosis at the level of the occipital and parietal lobes, and ocular vascular malformation. Incomplete variants of the syndrome<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> include the following: (a) facial and leptomeningeal angioma, without choroid angioma; (b) leptomeningeal and choroid angioma without facial nevus; (c) facial nevus and choroid angioma with no clinical or imaging signs of cerebral angiomatosis; and <span class="elsevierStyleItalic">d)</span> isolated cerebral and pial angiomatosis.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2–4</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Although descriptions of SWS date back to 1879 (Sturge), Van Bogaert described a form of corticomeningeal telangiectasia without facial angioma as a variant of SWS in 1935. In Lund's 1949 review of the 144 reported patients affected by the syndrome, he identifies 7 cases with typical radiography images and no facial angiomas.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The medical literature describes few cases of SWS without facial angioma,<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6–9</span></a> and a diagnosis in an adult patient, as in our case, is very rare.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Neurological manifestations of SWS are related to the presence of leptomeningeal angioma.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> The most consistent sign is the presence of epileptic seizures, which affect 75% to 90% of all patients<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a>; only 7% experience the initial seizure after the age of 5 years. Neurological deficits may appear slowly over time, or as stroke-like episodes associated with seizures and/or headaches resembling migraines as in the clinical case described here. The most frequently observed signs of neurological deficit are motor hemiparesis, hemianopsia, and hemiatrophy, which present in 65% of all patients. Cognitive changes are frequent and they are related to early onset of refractory seizures and bihemispheric dysfunction.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a> Depressive symptoms are also common. Migraines and other headaches appear frequently, and this type is included in the second edition of the International Classification of the Headache Society (ICHD-II) among the secondary headaches in section 6.3.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">13</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">If there is a clinical suspicion of cerebral impairment, the condition is diagnosed using neuroimaging techniques, which provide very useful information in the absence of typical cutaneous or ocular signs. The characteristic radiography findings are gyriform cortical calcifications appearing adjacent to the leptomeningeal angioma in head CT images. Cranial MRI displays the typical vascular malformation; IV contrast provokes enhancement in T1-weighted images. In addition to these findings, we may also observe dilation and enhancement in the ipsilateral choroid plexus and enlargement of deep venous drainage underlying the affected cortical region.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Long-term treatment with antiepileptic drugs is fundamental for those patients with seizures. Since venous stasis and microvascular thrombosis probably contribute to the neurological decline occurring in SWS, the empirical recommendation calls for daily antiplatelet treatment with low doses of acetylsalicylic acid,<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">14</span></a> which has been shown to decrease the frequency of seizures and stroke-like events. Headaches and migraines are treated with conventional abortive drugs (including triptans<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">15</span></a>); preventive drugs may also be used if required due to episode frequency or severity. Surgery (resection of the malformation or even hemispherectomy) provides effective treatment, but it should not be considered at time of diagnosis because it is currently indicated only in cases of seizures refractory to drug treatment.</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: Gómez-Moreno M, Murrieta-Urruticoechea C, Martinez-Acebes E, Gordo-Mañas R. Angiomatosis leptomeníngea temporo-occipital de diagnóstico en edad adulta. Neurología. 2015;30:64–66.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1171 "Ancho" => 2918 "Tamanyo" => 241292 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Cranial CT without contrast: cortical calcification and discrete atrophy of the right occipital lobe, and calcification and enlargement of the choroid plexus of the occipital horn of the right ventricle.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1100 "Ancho" => 2920 "Tamanyo" => 248842 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Cranial MRI: axial T1-weighted contrast-enhanced images with striking meningeal enhancement mainly observed in the parietal-occipital-temporal regions. Enhancement in cerebral sulci was both dural and leptomeningeal (pial), but especially leptomeningeal. Size difference between choroid plexi of the temporal horns of the lateral ventricles; choroid plexi are larger and show more enhancement in the right lateral ventricle.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:15 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Encephalofacial angiomatosis sparing the occipital lobe and without facial nevus: on the spectrum of Sturge–Weber syndrome variants?" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "A.M. Comi" 1 => "R. Fisher" 2 => "E.H. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 11 | 0 | 11 |
| 2024 October | 63 | 18 | 81 |
| 2024 September | 99 | 19 | 118 |
| 2024 August | 67 | 4 | 71 |
| 2024 July | 58 | 3 | 61 |
| 2024 June | 57 | 5 | 62 |
| 2024 May | 43 | 4 | 47 |
| 2024 April | 53 | 4 | 57 |
| 2024 March | 67 | 8 | 75 |
| 2024 February | 71 | 16 | 87 |
| 2024 January | 112 | 19 | 131 |
| 2023 December | 98 | 29 | 127 |
| 2023 November | 116 | 20 | 136 |
| 2023 October | 161 | 15 | 176 |
| 2023 September | 95 | 10 | 105 |
| 2023 August | 87 | 8 | 95 |
| 2023 July | 156 | 9 | 165 |
| 2023 June | 131 | 9 | 140 |
| 2023 May | 210 | 9 | 219 |
| 2023 April | 213 | 16 | 229 |
| 2023 March | 208 | 9 | 217 |
| 2023 February | 164 | 14 | 178 |
| 2023 January | 160 | 16 | 176 |
| 2022 December | 128 | 17 | 145 |
| 2022 November | 120 | 7 | 127 |
| 2022 October | 132 | 15 | 147 |
| 2022 September | 136 | 20 | 156 |
| 2022 August | 131 | 20 | 151 |
| 2022 July | 82 | 17 | 99 |
| 2022 June | 80 | 11 | 91 |
| 2022 May | 110 | 20 | 130 |
| 2022 April | 101 | 13 | 114 |
| 2022 March | 139 | 17 | 156 |
| 2022 February | 185 | 13 | 198 |
| 2022 January | 207 | 23 | 230 |
| 2021 December | 159 | 20 | 179 |
| 2021 November | 120 | 20 | 140 |
| 2021 October | 115 | 20 | 135 |
| 2021 September | 119 | 23 | 142 |
| 2021 August | 99 | 21 | 120 |
| 2021 July | 97 | 15 | 112 |
| 2021 June | 70 | 11 | 81 |
| 2021 May | 91 | 26 | 117 |
| 2021 April | 214 | 31 | 245 |
| 2021 March | 138 | 24 | 162 |
| 2021 February | 110 | 26 | 136 |
| 2021 January | 168 | 17 | 185 |
| 2020 December | 113 | 27 | 140 |
| 2020 November | 128 | 17 | 145 |
| 2020 October | 110 | 17 | 127 |
| 2020 September | 131 | 21 | 152 |
| 2020 August | 136 | 30 | 166 |
| 2020 July | 98 | 20 | 118 |
| 2020 June | 125 | 22 | 147 |
| 2020 May | 98 | 24 | 122 |
| 2020 April | 100 | 11 | 111 |
| 2020 March | 103 | 14 | 117 |
| 2020 February | 107 | 13 | 120 |
| 2020 January | 102 | 12 | 114 |
| 2019 December | 99 | 16 | 115 |
| 2019 November | 83 | 19 | 102 |
| 2019 October | 110 | 12 | 122 |
| 2019 September | 117 | 16 | 133 |
| 2019 August | 71 | 16 | 87 |
| 2019 July | 81 | 19 | 100 |
| 2019 June | 136 | 31 | 167 |
| 2019 May | 206 | 62 | 268 |
| 2019 April | 165 | 88 | 253 |
| 2019 March | 68 | 12 | 80 |
| 2019 February | 58 | 23 | 81 |
| 2019 January | 39 | 18 | 57 |
| 2018 December | 40 | 12 | 52 |
| 2018 November | 51 | 28 | 79 |
| 2018 October | 69 | 12 | 81 |
| 2018 September | 110 | 31 | 141 |
| 2018 August | 78 | 18 | 96 |
| 2018 July | 68 | 13 | 81 |
| 2018 June | 87 | 8 | 95 |
| 2018 May | 147 | 8 | 155 |
| 2018 April | 73 | 6 | 79 |
| 2018 March | 70 | 5 | 75 |
| 2018 February | 39 | 4 | 43 |
| 2018 January | 48 | 7 | 55 |
| 2017 December | 34 | 3 | 37 |
| 2017 November | 51 | 4 | 55 |
| 2017 October | 53 | 13 | 66 |
| 2017 September | 56 | 23 | 79 |
| 2017 August | 83 | 8 | 91 |
| 2017 July | 64 | 9 | 73 |
| 2017 June | 86 | 8 | 94 |
| 2017 May | 118 | 18 | 136 |
| 2017 April | 53 | 13 | 66 |
| 2017 March | 105 | 12 | 117 |
| 2017 February | 152 | 17 | 169 |
| 2017 January | 76 | 16 | 92 |
| 2016 December | 74 | 14 | 88 |
| 2016 November | 89 | 20 | 109 |
| 2016 October | 102 | 18 | 120 |
| 2016 September | 177 | 14 | 191 |
| 2016 August | 155 | 17 | 172 |
| 2016 July | 84 | 13 | 97 |
| 2016 June | 81 | 35 | 116 |
| 2016 May | 75 | 25 | 100 |
| 2016 April | 66 | 40 | 106 |
| 2016 March | 109 | 44 | 153 |
| 2016 February | 56 | 24 | 80 |
| 2016 January | 64 | 25 | 89 |
| 2015 December | 53 | 10 | 63 |
| 2015 November | 55 | 22 | 77 |
| 2015 October | 64 | 18 | 82 |
| 2015 September | 84 | 47 | 131 |
| 2015 August | 72 | 6 | 78 |
| 2015 July | 106 | 46 | 152 |
| 2015 June | 56 | 4 | 60 |
| 2015 May | 60 | 7 | 67 |
| 2015 April | 72 | 30 | 102 |
| 2015 March | 97 | 19 | 116 |
| 2015 February | 113 | 31 | 144 |
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