Case Report
Chiari type I malformation discovered through a glossopharyngeal neuralgia
Malformación de Chiari tipo I descubierta a través de una neuralgia glosofaríngea
Alejandro Doval Rodríguez
, Ramón Serramito García, Beatriz Menéndez Cortezón, Ángel Prieto González
Corresponding author
Servicio de Neurocirugía, Complejo Hospitalario Universitario de Santiago de Compostela, Santiago de Compostela, La Coruña, Spain
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Is it feasible? A case report" "tienePdf" => "en" "tieneTextoCompleto" => "en" "tieneResumen" => array:2 [ 0 => "en" 1 => "es" ] "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "394" "paginaFinal" => "397" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Estimulación medular como tratamiento de la meralgia parestésica. ¿Es factible? Reporte de caso" ] ] "contieneResumen" => array:2 [ "en" => true "es" => true ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "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" => 1084 "Ancho" => 2501 "Tamanyo" => 182779 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Meralgia paraesthetica. A) Pain distribution, paraesthesias in the anterolateral aspect of the thigh (shaded area and black arrow). B) Radiological control: epidural stimulator at T10 (white arrow).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Fernando J. 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It can be idiopathic or secondary. There are many different origins for secondary neuralgia, including demyelinating disease, tumours of the posterior fossa, vascular compression and craniocervical malformations.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">There have been few reports of GN related to a Chiari malformation. In this article, we describe a case and discuss the surgical treatment of this association.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">This was a 26-year-old female patient who was referred to Neurosurgery for progressive dysphagia. The origin of the dysphagia was a sharp, stabbing pain in the left pharyngeal region triggered by swallowing or touching the area with a toothbrush. The pain and, consequently, the dysphagia progressively increased and the stimulus necessary to trigger the pain decreased. The examination did not identify any abnormalities.</p><p id="par0025" class="elsevierStylePara elsevierViewall">MRI revealed a Chiari type I malformation with caudal displacement of the right cerebellar tonsil by 8.7 mm and of the left by 15.2 mm; more noticeable with the left tonsil, compatible with the side of the neuralgia. No vascular compression was detected.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Initially, the pain was controlled by medical treatment (carbamazepine). However, eight months later the pain worsened, leading to significant weight loss due to intractable pain, and it was decided that surgical treatment was necessary.</p><p id="par0035" class="elsevierStylePara elsevierViewall">A suboccipital craniectomy was performed by opening the foramen magnum and dura mater (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), with exploration of the left cranial nerves (CN) <span class="elsevierStyleSmallCaps">IX</span> and <span class="elsevierStyleSmallCaps">X</span> (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>) from their entry zone and interposing several pieces of Teflon between the nerves and the left posterior inferior cerebellar artery (PICA) and the left vertebral artery (VA) (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">Postoperatively, the patient reported complete disappearance of her previous neuralgia-related symptoms. This meant the medical treatment could be reduced without the need for further increasing doses.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0045" class="elsevierStylePara elsevierViewall">Chiari type I malformation is an abnormality characterised by caudal displacement of the cerebellar tonsils through the foramen magnum (more than 5 mm), without descent of the vermis or fourth ventricle (unlike Chiari type II or Arnold-Chiari malformation).<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> It tends to be an abnormality typical of young adults, with a mean age at diagnosis of 41, and somewhat more prevalent in females.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The most common symptom is pain, typically headache and more specifically in the posterior region of the head and neck. This pain may be aggravated by certain manoeuvres, such as the Valsalva or hyperextension of the neck. Other signs and symptoms of Chiari type I are different patterns of pain (arm, leg, etc.), limb weakness, numbness or other sensory findings, vomiting, etc.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Lower cranial nerve dysfunction appears as a presenting sign at diagnosis in about a quarter of cases. These signs, such as paralysis of the vocal cords, weakness of the palate or tongue, tendency to aspiration or hoarseness, are related to loss of nerve function. The “hyperfunction” of a cranial nerve, in this case the glossopharyngeal nerve, is an extremely rare onset for a Chiari type I. In the PubMed database, performing a search with the keywords "Chiari" and "glossopharyngeal neuralgia", there are only six results from the last 50 years, mainly case reports. The lack of data is probably explained by the unusual presentation of this malformation in conjunction with this neuralgia, despite pain as an entity being the most common symptom in patients with Chiari type I.</p><p id="par0055" class="elsevierStylePara elsevierViewall">In the case we present here, the neuralgia may have had multiple origins: vascular compression of the root entry zone; compression secondary to caudal displacement of the cerebellar tonsil; or stretching of the nerve secondary to Chiari and spatial compromise in the posterior fossa. The pain was probably caused by these different coexisting mechanisms combined.</p><p id="par0060" class="elsevierStylePara elsevierViewall">In other articles, patients had symptoms other than neuralgia, such as central apnoea, weakness derived from the Chiari malformation<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a> or cardiac syncope in relation to the neuralgia itself.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> In some cases the only pathological finding in the physical examination or medical history was neuralgia.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6,7</span></a> Our patient only had neuralgia, with no other Chiari-related signs or symptoms, and no neck pain.</p><p id="par0065" class="elsevierStylePara elsevierViewall">We did not perform resection of the cerebellar tonsil on our patient. With the dura mater graft and the expansion of the posterior fossa (with opening of the foramen magnum and resection of the posterior arch of C1), the volume of the posterior fossa was significantly increased, solving the space problem. During surgery, a microvascular exploration of the nerves was performed to resolve any possible vascular compression.</p><p id="par0070" class="elsevierStylePara elsevierViewall">Previous authors have reported performing cerebellar tonsillectomy,<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,6–8</span></a> but in our case it was not considered necessary, as we assumed that the lesion caused in cranial nerve <span class="elsevierStyleSmallCaps">ix</span> due to compromised space and the different arteries could be resolved without resection. We verified that optimal decompression of the cranial nerves was being achieved and thus avoided symptoms and complications deriving from tonsillectomy.</p><p id="par0075" class="elsevierStylePara elsevierViewall">The disappearance of the pain in the immediate postoperative phase suggests that the origin of the neuralgia was a mechanical issue. However, it was impossible to differentiate between whether the main compression was due to the vascular component or derived from displacement of the cerebellar tonsil. The Chiari malformation probably caused rearrangement of the different structures found in the posterior cranial fossa, giving rise to pain with a multifactorial origin.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0080" class="elsevierStylePara elsevierViewall">The presence of GN can lead to the discovery of an underlying cause of the pain such as a Chiari malformation. Decompression of the posterior fossa combined with microvascular decompression of the affected cranial nerves may be sufficient to achieve optimal pain control in this case, avoiding cerebellar tonsil resection.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Informed consent</span><p id="par0085" class="elsevierStylePara elsevierViewall">Informed consent was obtained from the patient included in the study.</p><p id="par0090" class="elsevierStylePara elsevierViewall">The participant has given her consent for the presentation of the case report to the journal.</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Funding</span><p id="par0100" class="elsevierStylePara elsevierViewall">No funding was received for this article.</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Conflicts of interest</span><p id="par0105" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:12 [ 0 => array:3 [ "identificador" => "xres1814940" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1584585" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1814941" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1584584" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Case report" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Conclusion" ] 8 => array:2 [ "identificador" => "sec0025" "titulo" => "Informed consent" ] 9 => array:2 [ "identificador" => "sec0035" "titulo" => "Funding" ] 10 => array:2 [ "identificador" => "sec0040" "titulo" => "Conflicts of interest" ] 11 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2021-09-03" "fechaAceptado" => "2021-10-23" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1584585" "palabras" => array:4 [ 0 => "Chiari malformation" 1 => "Glossopharyngeal neuralgia" 2 => "Dysphagia" 3 => "Cranio-cervical junction" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1584584" "palabras" => array:4 [ 0 => "Malformación de Chiari" 1 => "Neuralgia glosofaríngea" 2 => "Disfagia" 3 => "Unión cráneo-cervical" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Glossopharyngeal neuralgia is a rare disease whose initial treatment is pharmacological. When medical therapy is not effective, different surgical options are available including stereotactic radiosurgery, microvascular decompression or nerve section.</p><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">It is reported a case of a 26-year-old female with glossopharyngeal neuralgia and Chiari malformation. This rare type of neuralgia sometimes is associated with an abnormality of the cranio-cervical junction. It was performed a posterior fossa expansion with duraplasty and microvascular decompression. The patient showed a complete disappearance of the pain, with no need of tonsil resection.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">La neuralgia glosofaríngea es una enfermedad rara cuyo tratamiento inicial es farmacológico. Cuando la terapia médica no es eficaz, se encuentran disponibles diferentes opciones quirúrgicas, incluida la radiocirugía estereotáctica, la descompresión microvascular o la sección de nervios.</p><p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Se presenta el caso de una mujer de 26 años con neuralgia glosofaríngea y malformación de Chiari. Este tipo raro de neuralgia a veces se asocia con una anomalía de la unión cráneo-cervical. Se realizó una expansión de fosa posterior con duraplastia y descompresión microvascular. La paciente mostró una completa desaparición del dolor, sin necesidad de llevar a cabo la resección de las amígdalas cerebelosas.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Doval Rodríguez A, Serramito García R, Menéndez Cortezón B, Prieto González A. Malformación de Chiari tipo I descubierta a través de una neuralgia glosofaríngea. Neurocirugia. 2022;33:398–401.</p>" ] ] "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" => 1406 "Ancho" => 2500 "Tamanyo" => 218763 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Initial exposure.</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" => 1875 "Ancho" => 2500 "Tamanyo" => 287508 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Cranial nerves after exposure.</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" => 1406 "Ancho" => 2500 "Tamanyo" => 275666 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Final situation after placement of pieces of Teflon.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Glossopharyngeal neuralgia. 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