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Corticobasal syndrome: A case of discordance between the clinical findings and the imaging biomarkers
Síndrome córticobasal: un caso de discordancia entre la clínica y los biomarcadores de imagen
E. Carrillo-Villamizara,
, A. Cardozo-Saavedraa, C. Lorenzo-Bosqueta, S. Lucas del Pozob, E. Martínez-Sáezc, J. Hernández-Varad
Corresponding author
a Servicio de Medicina Nuclear, Hospital Universitari Vall d’Hebron, Barcelona, Spain
b Grupo de Investigación en Enfermedades Neurodegenerativas, Vall d’Hebron Institut de Recerca (VHIR), Barcelona, Spain
c Servicio de Anatomía Patológica, Sección Neuropatología, Hospital Universitari Vall d’Hebron, Banco de Tejidos Neurológicos, Barcelona, Spain
d Servicio de Neurología, Hospital Universitari Vall d’Hebron, Grupo de Investigación en Enfermedades Neurodegenerativas, Vall d’Hebron Institut de Recerca (VHIR), Banco de Tejidos Neurológicos, Barcelona, Spain
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Quantitative analysis using MI Neurology from Siemens® without statistically significant alterations.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We present a 59-year-old man who consulted for a clinical condition that had started in the previous 6 months, consisting of motor clumsiness and gait disorder. The physical examination highlighted a predominantly left akinetic-rigid syndrome, ipsilateral hemiapraxia, left hand pyramidalism with dystonic posture, and the "Alien Hand" phenomenon. General laboratory tests were normal. Brain MRI showed a moderate/severe degree of symmetric global brain atrophy and signs of leukoaraiosis. The presynaptic dopamine transporters SPECT (123I-FP-CIT SPECT) showed no alterations (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Brain PET with <span class="elsevierStyleSup">18</span>F-FDG showed no alterations in brain metabolism (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). The electromyogram showed a marked impairment of the first motor neuron in the left lower limb.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Clinically, the patient fulfilled the clinical criteria for a probable corticobasal syndrome (CBS). In the following months, he presented a progressive clinical worsening, dying due to a hematoma secondary to traumatic brain injury.</p><p id="par0020" class="elsevierStylePara elsevierViewall">In the anatomopathological study, a systematic sampling of different areas was carried out, finding abundant phosphorylated tau deposits in the form of neurofibrillary tangles and pretangles, balloonized neurons, tufted astrocytes and oligodendroglial inclusions. The distribution of this deposits was mainly brainstem, with little cortical involvement. It was compatible with progressive supranuclear palsy (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">The <span class="elsevierStyleSup">123</span>I-FP-CIT SPECT evaluates the presynaptic integrity of the nigrostriatal dopaminergic neurons of the pars compact of the substantia nigra, showing a high sensitivity for parkinsonisms; however, in some series, as many as 10% of patients with diagnosis of CBS did not present a pathological result.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">On the other hand, brain PET with <span class="elsevierStyleSup">18</span>F-FDG evaluates different patterns of brain hypometabolism with a sensitivity up to 93% and specificity up to 90% for differentiating the different atypical parkinsonisms (PSP,<a class="elsevierStyleCrossRef" href="#fn0005"><span class="elsevierStyleSup">1</span></a> MSA<a class="elsevierStyleCrossRef" href="#fn0010"><span class="elsevierStyleSup">2</span></a> and CBD<a class="elsevierStyleCrossRef" href="#fn0015"><span class="elsevierStyleSup">3</span></a>); likewise, some series have established different patterns of hypometabolism in relation to the underlying primary neurodegenerative pathology, correlating these findings with a post-mortem neuropathological examination and thus demonstrating that brain PET scan with <span class="elsevierStyleSup">18</span>F-FDG is an effective complementary tool in differentiation of atypical parkinsonisms.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">From the clinical point of view, the absence of alterations in the brain <span class="elsevierStyleSup">18</span>F- FDG PET could lead to reconsideration of the diagnosis of a CBS, regardless of the underlying pathological substrate (CBD, Alzheimer's Disease, PSP).<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Atypical parkinsonisms are complex pathologies whose phenotypic overlap and pathophysiological mechanisms, not yet fully clarified, make their "in vivo" diagnosis remain a clinical challenge, despite the currently available biomarkers that have improved the diagnostic approaches and characterization of the different entities. These should not be considered as definitive and excluding criteria, since, as we exemplify with the current case, despite the negativity of all of all these tests, finally, the histopathological study can confirm the presence of a taupathy that, in this patient, explained the neurological symptoms and his clinical evolution.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:4 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Carrillo-Villamizar E, Cardozo-Saavedra A, Lorenzo-Bosquet C, Lucas del Pozo S, Martínez-Sáez E, Hernández-Vara J. Síndrome Córticobasal: un caso de discordancia entre la clínica y los biomarcadores de imagen. Rev Esp Med Nucl Imagen Mol. 2022;41:51–53.</p>" ] 1 => array:3 [ "etiqueta" => "1" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Progressive supranuclear palsy.</p>" "identificador" => "fn0005" ] 2 => array:3 [ "etiqueta" => "2" "nota" => "<p class="elsevierStyleNotepara" id="npar0015">Multiple system atrophy.</p>" "identificador" => "fn0010" ] 3 => array:3 [ "etiqueta" => "3" "nota" => "<p class="elsevierStyleNotepara" id="npar0020">Corticobasal degeneration.</p>" "identificador" => "fn0015" ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1797 "Ancho" => 2500 "Tamanyo" => 329048 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleSup">123</span>I-FP-CIT SPECT without presynaptic dopaminergic involvement.</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" => 3199 "Ancho" => 2500 "Tamanyo" => 686615 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Brain PET with <span class="elsevierStyleSup">18</span>F-FDG. Quantitative analysis using MI Neurology from Siemens® without statistically significant alterations.</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1122 "Ancho" => 1500 "Tamanyo" => 318935 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Histologically, scarce deposits of phosphorylated tau were observed in the frontal cortex, mainly in the pretangles form (A, AT8, 100x), somewhat more abundant in the caudate nucleus (B, AT8, 100×) and very frequent in the midbrain (C, red nucleus, AT8, 100×) and the pons (D, base nuclei, AT8, 100×), with abundant pretangles, ovoid tangles, tufted astrocytes and oligodendroglial inclusions.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:3 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Dopamine transporter SPECT imaging in corticobasal syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "R. 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