Letter to the Editor
Orthostatic tremor secondary to recreational use of solvents
Temblor ortostático secundario al uso recreativo de disolventes
H. Cruz Tabuenca, J.L. Camacho Velásquez
, E. Rivero Sanz, S. Sánchez Valiente, J. López del Val.
Corresponding author
Servicio de Neurología, Hospital Clínico Universitario Lozano Blesa, Zaragoza, Spain
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Cruz Tabuenca, J.L. Camacho Velásquez, E. Rivero Sanz, S. Sánchez Valiente, J. López del Val." "autores" => array:5 [ 0 => array:2 [ "nombre" => "H." "apellidos" => "Cruz Tabuenca" ] 1 => array:4 [ "nombre" => "J.L." "apellidos" => "Camacho Velásquez" "email" => array:1 [ 0 => "jlcv2002@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 2 => array:2 [ "nombre" => "E." "apellidos" => "Rivero Sanz" ] 3 => array:2 [ "nombre" => "S." "apellidos" => "Sánchez Valiente" ] 4 => array:2 [ "nombre" => "J." "apellidos" => "López del Val." ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Neurología, Hospital Clínico Universitario Lozano Blesa, Zaragoza, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Temblor ortostático secundario al uso recreativo de disolventes" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 756 "Ancho" => 1400 "Tamanyo" => 131951 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">T2-weighted brain MRI shows hyperintense lesions on both pyramidal tracts at the pontine level and hypointense lesions at the mesencephalic level in the red nucleus and substantia nigra.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Organic solvents are volatile organic compounds used to dissolve raw materials or residual products. Their uses include cleaning agents, glues, paint components, plasticisers, and many other industrial purposes. Since these compounds are widely available, they may be used as psychostimulants; this tendency is more frequent in developing countries.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Our patient is a 30-year old former cocaine user. He had an 8-year history of bilateral optic neuritis with no known aetiology. He visited our emergency department due to frequent falls and gait disturbances which had evolved over 2 years to such an extent that he needed support to walk. Gait disturbances were initially attributed to probable epileptic myoclonic seizures (with normal EEG) and therefore treated with levetiracetam 500<span class="elsevierStyleHsp" style=""></span>mg/12<span class="elsevierStyleHsp" style=""></span>h. Treatment failed to control symptoms and provoked side effects which led the patient to stop taking the medication. Considering treatment failure in the context of the patient's age and degree of limitation, we decided to hospitalise him for further study.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Physical examination revealed that the patient was oriented in time and space, with no alterations in speech structures or content. Cranial nerves were unaffected. No motor or sensory alterations were observed in the limbs. He displayed upper limb tremor that was predominantly intention tremor with a postural component; it disappeared at rest and was compatible with tremor of cerebellar origin. Lower limbs showed orthostatic tremor in the proximal segment and a typical helicopter sign, which also provoked gait instability; the patient attempted to compensate with a wider stance. Tremor in the lower limbs was absent with the patient in the supine position or when seated. The patient also showed symmetrical hyperreflexia of the lower limbs.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Electroencephalography, electroneurography, and lumbar puncture procedures all yielded normal results. The electromyogram showed rhythmic muscle activity (tremor) at a frequency of 15<span class="elsevierStyleHsp" style=""></span>Hz.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Brain MRI revealed FLAIR sequence hyperintensity in both posterior limbs of the internal capsules, especially on the anterolateral pons. The T2-weighted sequence also showed hyperintense lesions on both pyramidal tracts at the brainstem level and a hypointense lesion at the mesencephalic level in the red nucleus and substantia nigra (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">A few days after the patient had been admitted, a relative informed us that the patient had been inhaling gasoline fumes for several years (8 approximately) and strongly suspected that he was still doing so without being able to speculate about the frequency. Considering findings from the physical examination and the neuroimaging study, and the patient's history of inhaling gasoline fumes, we established a diagnosis of orthostatic tremor secondary to solvent inhalation (gasoline vapour). Starting clonazepam as symptomatic treatment of orthostatic tremor achieved excellent results. The patient was discharged a few days later. In subsequent follow-up visits, the patient indicated having stopped inhaling solvents but still needed clonazepam treatment to lessen symptoms of tremor.</p><p id="par0035" class="elsevierStylePara elsevierViewall">We believe that the optic neuritis of unknown aetiology may also have been due to recreational use of organic solvents.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Discussion</span><p id="par0040" class="elsevierStylePara elsevierViewall">Literature on central nervous system lesions caused by exposure to volatile organic compounds is scarce. The most widely studied substances are aromatic hydrocarbons (such as toluene) and other hexacarbon solvents, since they are readily available and present in household chemicals.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">1</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The typical lesions to the nervous system caused by exposure to volatile substances usually include peripheral neuropathy, optic neuritis, and neurosensory hearing loss. Nevertheless, white matter lesions typically follow long-term exposure to hydrocarbons, as shown by the many case studies reporting these findings.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">2</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Lesions caused by long-term toluene exposure are visible in brain MRI, which may reveal white matter hyperintensities in T2-weighted sequences. These findings are more apparent in the posterior limbs of the internal capsules, cerebral peduncle, ventral pons, and middle cerebellar peduncles.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">3</span></a> Basal ganglia may appear hypointense.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">4</span></a> Cases of degeneration of the corpus callosum secondary to recreational use of solvents have been described in very recent studies.<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">5</span></a> Cocaine consumption can also cause white matter lesions that are observable with brain MRI; however, these lesions differ from those secondary to toluene in that they are predominantly located in the frontal lobe, with the brainstem and cerebellum usually remaining intact.<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">6</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Symptoms of white matter involvement may include orthostatic tremor<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">7</span></a> (seen in our patient), intention tremor, falling, ataxic gait, dysarthria, wide stance, instability, balance disturbances,<a class="elsevierStyleCrossRef" href="#bib0120"><span class="elsevierStyleSup">8</span></a> paraparesis, peripheral neuropathy,<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">9</span></a> and impaired concentration and abstract thought, as well as alterations in reading, writing, and speech content.<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">10</span></a> Such pyramidal signs as hyperreflexia and spasticity may manifest as symptoms.</p><p id="par0060" class="elsevierStylePara elsevierViewall">Studies have been conducted on neurological and neuropsychological impairment in adolescents who abuse volatile substances and in workers exposed to hydrocarbons on the job,<a class="elsevierStyleCrossRef" href="#bib0135"><span class="elsevierStyleSup">11</span></a> and they point to statistically significant differences between the exposed cohort and the unexposed group. Furthermore, damage to the dopaminergic pathway has been demonstrated in exposed subjects,<a class="elsevierStyleCrossRefs" href="#bib0140"><span class="elsevierStyleSup">12,13</span></a> and they have been shown to exacerbate secondary parkinsonism in some cases.<a class="elsevierStyleCrossRef" href="#bib0145"><span class="elsevierStyleSup">13</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Patients with isolated orthostatic tremor may take clonazepam or gabapentin as symptomatic treatment. Primidone, valproic acid, mistazapan, and acetazolamide may also be used as treatment alternatives.<a class="elsevierStyleCrossRef" href="#bib0150"><span class="elsevierStyleSup">14</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Clinical symptoms may subside provided that the patient stops consuming solvents completely and as early as possible.<a class="elsevierStyleCrossRef" href="#bib0155"><span class="elsevierStyleSup">15</span></a> If not, damages to the CNS become irreversible.<a class="elsevierStyleCrossRef" href="#bib0160"><span class="elsevierStyleSup">16</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0075" class="elsevierStylePara elsevierViewall">The authors have received no private or public funding for this case report.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Discussion" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Funding" ] 2 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Cruz Tabuenca H, Camacho Velásquez JL, Rivero Sanz E, Sánchez Valiente S, López del Val J. Temblor ortostático secundario al uso recreativo de disolventes. Neurología. 2017;32:401–403.</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" => 806 "Ancho" => 1400 "Tamanyo" => 129412 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">FLAIR sequence brain MRI shows mild hyperintense lesions on both posterior limbs of the internal capsule, with more apparent lesions on the anterolateral pons.</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" => 756 "Ancho" => 1400 "Tamanyo" => 131951 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">T2-weighted brain MRI shows hyperintense lesions on both pyramidal tracts at the pontine level and hypointense lesions at the mesencephalic level in the red nucleus and substantia nigra.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:16 [ 0 => array:3 [ "identificador" => "bib0085" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Treatment of the neurotoxic effects of organic solvents" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "M.H. 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| 2018 September | 28 | 0 | 28 |
| 2018 August | 8 | 1 | 9 |
| 2018 July | 8 | 2 | 10 |
| 2018 June | 14 | 0 | 14 |
| 2018 May | 21 | 4 | 25 |
| 2018 April | 3 | 1 | 4 |
| 2018 March | 4 | 0 | 4 |
| 2018 February | 3 | 1 | 4 |
| 2018 January | 11 | 4 | 15 |
| 2017 December | 19 | 1 | 20 |
| 2017 November | 11 | 3 | 14 |
| 2017 October | 7 | 1 | 8 |
| 2017 September | 15 | 10 | 25 |
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