Letter to the Editor
Levodopa-responsive parkinsonism-dystonia due to a traumatic injury of the substantia nigra
Parkinsonismo-distonía unilateral sensible a levodopa por lesión traumática de la sustancia negra
F. Pérez Errazquin
, M.J. Gomez Heredia
Corresponding author
Unidad de trastornos del movimiento, Servicio de Neurología, Hospital Universitario Virgen de la Victoria, Málaga, Spain
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Pérez Errazquin, M.J. Gomez Heredia" "autores" => array:2 [ 0 => array:4 [ "nombre" => "F." "apellidos" => "Pérez Errazquin" "email" => array:1 [ 0 => "pacoerrazquin@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "M.J." "apellidos" => "Gomez Heredia" ] ] "afiliaciones" => array:1 [ 0 => array:1 [ "entidad" => "Unidad de trastornos del movimiento, Servicio de Neurología, Hospital Universitario Virgen de la Victoria, Málaga, Spain" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Parkinsonismo-distonía unilateral sensible a levodopa por lesión traumática de la sustancia negra" ] ] "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" => 1887 "Ancho" => 1412 "Tamanyo" => 141569 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">DaTSCAN image. Marked, unilateral, low uptake of the right striatum.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">The relationship between traumatic brain injury (TBI) and parkinsonism has been established for a long time.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> However, in exceptionally rare cases there have been reports of parkinsonism secondary to traumatic lesions of the substantia nigra (SN).<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Case reports of parkinsonism due to SN lesions of vascular origin, either by lacunar stroke<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4–7</span></a> or by small mesencephalic haemorrhages,<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> are better known, but also exceptional. Bhatt published a series of 3 patients who developed parkinsonism several months after a severe TBI with SN lesions. As characteristic data, there was a delay between the trauma and the onset of parkinsonism. The latter developed quickly and aggressively, with all patients responding to therapy with levodopa.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> A possible pathophysiological mechanism which was postulated to justify the delay in the onset of parkinsonism with respect to trauma, was iron deposition from the degradation products of the haemorrhagic lesion. This deposition would trigger the cascade of events typical of dopaminergic degeneration observed in idiopathic Parkinson's disease (IPD), thus explaining the response to dopaminergic therapy in these cases. When the SN is affected, the resulting parkinsonism is strictly unilateral, unless the injury is more extensive and affects other structures. We found very few references in the literature regarding the usefulness of computed tomography or positron emission tomography in this entity, and they mainly referred to cases with a vascular aetiology.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9,10</span></a> Recently, a case of parkinsonism secondary to trauma with SN lesion was published in which the transcranial duplex study had not registered hyperechogenicity in the SN, unlike the characteristic pattern in IPD.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report a case of unilateral parkinsonism-dystonia secondary to traumatic injury of the SN and partially responsive to levodopa. We present the DaTSCAN study, which shows a notable decrease in radioisotope uptake in the striatum ipsilateral to the lesion.</p><p id="par0015" class="elsevierStylePara elsevierViewall">The patient was a 62-year-old male. At the age of 45 he suffered a TBI with loss of consciousness of 30–60<span class="elsevierStyleHsp" style=""></span>min duration due to a fall from a second storey, with no apparent immediate neurological sequelae. One year later, he started to suffer uncontrolled and involuntary movements of the left limbs, which were more pronounced in the foot. The examination revealed hemidystonia, without any other significant signs. Two months later, in addition to hemidystonia, he suffered akinetic-rigid syndrome characterised by resting tremor, significant cogwheel rigidity and bradykinesia in the affected side of the body. These symptoms had a relatively rapid onset, with severe worsening of parkinsonism within a few weeks. A cranial MRI scan (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>) conducted at that time showed a right mesencephalic lesion at the level of the SN, with a hyperintense signal on T2-weighted sequences and a hypointense signal on T1-weighted sequences, and without signal alterations in neighbouring structures (adjacent red nucleus or cerebral peduncle) or at other brain levels. The radiologist reported the lesion as suggestive of residual gliosis in the context of the previous TBI. We conducted an analytical study which included thyroid hormones, Ca, P, Mg, ceruloplasmin, copper, blood smear for evaluation of acanthocytes and serology for syphilis, human immunodeficiency virus and <span class="elsevierStyleItalic">Borrellia</span>, with no relevant results. The patient was treated with levodopa/carbidopa at 100/25<span class="elsevierStyleHsp" style=""></span>mg every 8<span class="elsevierStyleHsp" style=""></span>h and displayed a partial response, but with a clear improvement over the pretreatment condition. Since then, his condition has remained stable (approximately 15 years have elapsed since the onset of symptoms), without exacerbation of symptoms or significant worsening or extension to the contralateral side of the body. He has not developed motor complications or dyskinesias associated with levodopa treatment. An attempt to withdraw levodopa was followed by clinical worsening, so it was reintroduced. Hemidystonia persists, but it is mild. At present, the patient continues treatment with levodopa-carbidopa at 300<span class="elsevierStyleHsp" style=""></span>mg/day and extended-release ropinirole at 16<span class="elsevierStyleHsp" style=""></span>mg/day. A DaTSCAN was performed recently (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>), and it showed a notable decrease in uptake in the right striatum and preservation of uptake in the left striatum.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">It is well known that strategic lesions of the striatum or SN can cause symptomatic parkinsonism. The present case behaved like a strictly unilateral parkinsonism, developed months after a TBI. We should note that this parkinsonism occurred after a TBI which was not severe, since there were no immediate and persistent neurological sequelae, as has been previously reported.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> It started as hemidystonia prior to the development of parkinsonism. Dystonia has been associated with traumatic lesions of the basal ganglia, mostly thalamus and putamen, and also with a delay between trauma and the onset of symptoms.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a> However, there were no lesions in those structures in our case. Recently, a case of hemidystonia due to SN lesion which responded to treatment with levodopa has been published.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">13</span></a> The cranial MRI scan revealed a lesion with gliotic characteristics at the level of the contralateral SN, with no evidence to justify its presence other than the TBI. Although a vascular lesion cannot be ruled out definitively, the fact is that there were no stroke symptoms at any time, no ischaemic lesions were observed at other levels and the patient presented no cardiovascular risk factors since the event occurred at an early age. In addition, vascular parkinsonism has been associated with an immediate onset after stroke and lack of response to levodopa. These requirements were not fulfilled in our case. Our patient responded to treatment (by this we mean a clear, initial improvement and subsequent stabilisation of symptoms), as described in the few cases reported.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> No motor fluctuations or dyskinesias associated with prolonged use of levodopa have been detected. We consider unlikely the possibility of IPD precipitated by the TBI, since parkinsonism has remained strictly unilateral after many years of evolution and the DaTSCAN findings are not those of IPD, with complete denervation of one side and preservation of the other. However, some authors have raised the possibility that TBI could facilitate the development of IPD <span class="elsevierStyleItalic">a posteriori</span>.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> DaTSCAN images verified striatal denervation by Wallerian degeneration following the SN lesion. We emphasise the uniqueness of this case and the novelty of providing a DaTSCAN study.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Pérez Errazquin F, Gomez Heredia MJ. Parkinsonismo-distonía unilateral sensible a levodopa por lesión traumática de la sustancia negra. Neurología. 2012;27:181–3.</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" => 2308 "Ancho" => 1585 "Tamanyo" => 369243 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Cranial MRI scan: sagittal T1 section and axial T2 section. Mesencephalic lesion, hypointense on T1 (“black hole”) and hyperintense on T2, at the level of the right <span class="elsevierStyleItalic">substantia nigra</span>.</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" => 1887 "Ancho" => 1412 "Tamanyo" => 141569 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">DaTSCAN image. Marked, unilateral, low uptake of the right striatum.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:13 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Head injury and posttraumatic movement disorders" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "J.K. Krauss" 1 => "J. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 6 | 0 | 6 |
| 2024 October | 28 | 6 | 34 |
| 2024 September | 26 | 11 | 37 |
| 2024 August | 30 | 13 | 43 |
| 2024 July | 24 | 12 | 36 |
| 2024 June | 37 | 11 | 48 |
| 2024 May | 50 | 17 | 67 |
| 2024 April | 41 | 1 | 42 |
| 2024 March | 29 | 15 | 44 |
| 2024 February | 44 | 6 | 50 |
| 2024 January | 38 | 11 | 49 |
| 2023 December | 55 | 18 | 73 |
| 2023 November | 62 | 12 | 74 |
| 2023 October | 72 | 16 | 88 |
| 2023 September | 39 | 10 | 49 |
| 2023 August | 32 | 16 | 48 |
| 2023 July | 57 | 8 | 65 |
| 2023 June | 47 | 6 | 53 |
| 2023 May | 100 | 6 | 106 |
| 2023 April | 82 | 4 | 86 |
| 2023 March | 75 | 2 | 77 |
| 2023 February | 49 | 6 | 55 |
| 2023 January | 39 | 6 | 45 |
| 2022 December | 44 | 14 | 58 |
| 2022 November | 36 | 14 | 50 |
| 2022 October | 36 | 11 | 47 |
| 2022 September | 31 | 34 | 65 |
| 2022 August | 38 | 11 | 49 |
| 2022 July | 30 | 10 | 40 |
| 2022 June | 33 | 10 | 43 |
| 2022 May | 38 | 12 | 50 |
| 2022 April | 49 | 13 | 62 |
| 2022 March | 78 | 11 | 89 |
| 2022 February | 79 | 7 | 86 |
| 2022 January | 87 | 7 | 94 |
| 2021 December | 37 | 17 | 54 |
| 2021 November | 57 | 10 | 67 |
| 2021 October | 48 | 14 | 62 |
| 2021 September | 40 | 16 | 56 |
| 2021 August | 39 | 6 | 45 |
| 2021 July | 41 | 15 | 56 |
| 2021 June | 36 | 15 | 51 |
| 2021 May | 34 | 6 | 40 |
| 2021 April | 151 | 26 | 177 |
| 2021 March | 59 | 5 | 64 |
| 2021 February | 54 | 8 | 62 |
| 2021 January | 55 | 8 | 63 |
| 2020 December | 64 | 12 | 76 |
| 2020 November | 46 | 6 | 52 |
| 2020 October | 40 | 6 | 46 |
| 2020 September | 32 | 10 | 42 |
| 2020 August | 53 | 8 | 61 |
| 2020 July | 24 | 8 | 32 |
| 2020 June | 15 | 4 | 19 |
| 2020 May | 29 | 6 | 35 |
| 2020 April | 35 | 3 | 38 |
| 2020 March | 64 | 3 | 67 |
| 2020 February | 47 | 5 | 52 |
| 2020 January | 31 | 5 | 36 |
| 2019 December | 51 | 5 | 56 |
| 2019 November | 6 | 4 | 10 |
| 2019 October | 13 | 2 | 15 |
| 2019 September | 22 | 4 | 26 |
| 2019 August | 18 | 4 | 22 |
| 2019 July | 12 | 16 | 28 |
| 2019 June | 16 | 21 | 37 |
| 2019 May | 42 | 46 | 88 |
| 2019 April | 32 | 33 | 65 |
| 2019 March | 2 | 7 | 9 |
| 2019 February | 10 | 9 | 19 |
| 2019 January | 5 | 7 | 12 |
| 2018 December | 3 | 3 | 6 |
| 2018 November | 2 | 10 | 12 |
| 2018 October | 15 | 7 | 22 |
| 2018 September | 4 | 4 | 8 |
| 2018 August | 1 | 13 | 14 |
| 2018 July | 5 | 6 | 11 |
| 2018 June | 3 | 3 | 6 |
| 2018 May | 5 | 8 | 13 |
| 2018 April | 1 | 6 | 7 |
| 2018 March | 2 | 2 | 4 |
| 2018 February | 1 | 8 | 9 |
| 2018 January | 7 | 1 | 8 |
| 2017 December | 2 | 2 | 4 |
| 2017 November | 9 | 9 | 18 |
| 2017 October | 2 | 12 | 14 |
| 2017 September | 11 | 8 | 19 |
| 2017 August | 8 | 7 | 15 |
| 2017 July | 8 | 3 | 11 |
| 2017 June | 13 | 14 | 27 |
| 2017 May | 11 | 14 | 25 |
| 2017 April | 9 | 9 | 18 |
| 2017 March | 5 | 21 | 26 |
| 2017 February | 8 | 5 | 13 |
| 2017 January | 18 | 3 | 21 |
| 2016 December | 9 | 8 | 17 |
| 2016 November | 15 | 5 | 20 |
| 2016 October | 15 | 8 | 23 |
| 2016 September | 13 | 7 | 20 |
| 2016 August | 21 | 2 | 23 |
| 2016 July | 11 | 1 | 12 |
| 2016 June | 17 | 9 | 26 |
| 2016 May | 24 | 19 | 43 |
| 2016 April | 21 | 9 | 30 |
| 2016 March | 37 | 12 | 49 |
| 2016 February | 16 | 10 | 26 |
| 2016 January | 13 | 3 | 16 |
| 2015 December | 18 | 6 | 24 |
| 2015 November | 14 | 3 | 17 |
| 2015 October | 20 | 8 | 28 |
| 2015 September | 20 | 7 | 27 |
| 2015 August | 10 | 5 | 15 |
| 2015 July | 10 | 5 | 15 |
| 2015 June | 14 | 0 | 14 |
| 2015 May | 13 | 3 | 16 |
| 2015 April | 7 | 13 | 20 |
| 2015 March | 9 | 3 | 12 |
| 2015 February | 13 | 1 | 14 |
| 2015 January | 18 | 3 | 21 |
| 2014 December | 28 | 12 | 40 |
| 2014 November | 7 | 2 | 9 |
| 2014 October | 20 | 1 | 21 |
| 2014 September | 12 | 1 | 13 |
| 2014 August | 21 | 6 | 27 |
| 2014 July | 14 | 2 | 16 |
| 2014 June | 11 | 0 | 11 |
| 2014 May | 14 | 4 | 18 |
| 2014 April | 13 | 1 | 14 |
| 2014 March | 11 | 2 | 13 |
| 2014 February | 11 | 2 | 13 |
| 2014 January | 9 | 2 | 11 |
| 2013 December | 11 | 1 | 12 |
| 2013 November | 18 | 1 | 19 |
| 2013 October | 23 | 3 | 26 |
| 2013 September | 19 | 5 | 24 |
| 2013 August | 18 | 2 | 20 |
| 2013 July | 5 | 0 | 5 |
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