Chiari type I malformation discovered through a glossopharyngeal neuralgia

Doval Rodríguez, Alejandro; Serramito García, Ramón; Menéndez Cortezón, Beatriz; Prieto González, Ángel

doi:10.1016/j.neucie.2022.02.008

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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">1</a>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.Case reportThis 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.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.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.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) IX and X (<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>).<elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia>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.DiscussionChiari 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">2</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">3</a>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">2</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.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.In other articles, patients had symptoms other than neuralgia, such as central apnoea, weakness derived from the Chiari malformation<a class="elsevierStyleCrossRefs" href="#bib0020">4,5</a> or cardiac syncope in relation to the neuralgia itself.<a class="elsevierStyleCrossRef" href="#bib0005">1</a> In some cases the only pathological finding in the physical examination or medical history was neuralgia.<a class="elsevierStyleCrossRefs" href="#bib0030">6,7</a> Our patient only had neuralgia, with no other Chiari-related signs or symptoms, and no neck pain.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.Previous authors have reported performing cerebellar tonsillectomy,<a class="elsevierStyleCrossRefs" href="#bib0020">4,6–8</a> but in our case it was not considered necessary, as we assumed that the lesion caused in cranial nerve ix 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.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.ConclusionThe 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.Informed consentInformed consent was obtained from the patient included in the study.The participant has given her consent for the presentation of the case report to the journal.FundingNo funding was received for this article.Conflicts of interestThe authors declare that they have no conflicts of interest." "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" => "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.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." ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "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.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." ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "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." ] ] "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" => "Initial exposure." ] ] 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" => "Cranial nerves after exposure." ] ] 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" => "Final situation after placement of pieces of Teflon." ] ] ] "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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Neurocirugía (English edition)

ISSN: 2529-8496

Neurocirugía is the official Journal of the Spanish Society of Neurosurgery (SENEC). It is published every 2 months (6 issues per year). Neurocirugía will consider for publication, original clinical and experimental scientific works associated with neurosurgery and other related neurological sciences.

All manuscripts are submitted for review by experts in the field (peer review) and are carried out anonymously (double blind). The Journal accepts works written in Spanish or English.

Indexed in:

Research Alert Institute, C.A.B., Excerpta Medica, JCR, Index Medicus/Medline e IBECS

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Impact factor

The Impact Factor measures the average number of citations received in a particular year by papers published in the journal during the two preceding years.

© Clarivate Analytics, Journal Citation Reports 2022

Impact factor 2023

0.7

Citescore

CiteScore measures average citations received per document published.

Citescore 2023

1.3

SJR

SRJ is a prestige metric based on the idea that not all citations are the same. SJR uses a similar algorithm as the Google page rank; it provides a quantitative and qualitative measure of the journal's impact.

SJR 2023

0.255

SNIP

SNIP measures contextual citation impact by wighting citations based on the total number of citations in a subject field.

SNIP 2023

0.302

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Vol. 33. Issue 6.

Pages 398-401 (November - December 2022)

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