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Vol. 27. Num. 6.November - December 2016Pages 263-316
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Vol. 27. Num. 6.November - December 2016Pages 263-316
Case report
DOI: 10.1016/j.neucir.2016.05.003
Microvascular decompression for trigeminal neuralgia secondary to vertebrobasilar dolichoectasia. Case report, literature review, and pooled case analysis
Descompresión microvascular para la neuralgia del trigémino secundaria a dolicoectasia vertebrobasilar. Caso clínico, revisión de la literatura, de casos y análisis de casos agrupados
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Ignacio Arrese
Corresponding author
iarrese14@yahoo.es

Corresponding author.
, Rosario Sarabia
Unit of Vascular Neurosurgery, Hospital Universitario Río Hortega, Valladolid, Spain
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Table 1. F/M, female/male ratio. BA/VA, basilar artery/vertebral artery ratio of offending vessel.
Abstract
Introduction

Vertebrobasilar dolichoectasia is a condition in which there is elongation and dilatation of the vertebral and basilar arteries. Few studies have been reported that focus on cases of trigeminal neuralgia (TN) secondary to vertebrobasilar dolichoectasia (VD) and treated by microvascular decompression (MD).

Patients and methods

A case is presented of trigeminal neuralgia caused by vertebral artery compression. An analysis of the microsurgical technique, as well as a systematic review of the literature about this uncommon nerve compression is performed, in order to investigate, by pooled case analysis, if MD is a good option for this type of patient.

Results

A total of 7 studies were included for analysis, to which the present case was added, making a total of 56 patents. There were excellent results in 53 cases, and partial recovery in 3, with a mean follow up of 54 months. No major complications were found.

Discussion

The good clinical results and absence of postoperative mortality or severe morbidity in our pooled case series lead us to recommend MD as the preferred treatment for TN caused by VD in patients in whom major surgery is not contraindicated.

Keywords:
Microvascular decompression
Trigeminal neuralgia
Vertebrobasilar dolichoectasia
Resumen
Introducción

La dolicoectasia vertebrobasilar se caracteriza por la elongación y dilatación de las arterias vertebral y basilar. Muy pocos estudios se han enfocado a casos de neuralgia del trigémino secundaria a dolicoectasia vertebrobasilar tratada mediante descompresión microvascular.

Pacientes y métodos

Presentamos un caso de neuralgia del trigémino causada por compresión de la arteria vertebral. Se realiza un análisis de la técnica quirúrgica así como una revisión sistemática de la literatura sobre este tipo de compresión poco común, con el fin de hacer un análisis de los casos para evaluar si la descompresión microvascular es una buena opción terapéutica.

Resultados

Seleccionamos 7 estudios que fueron incluidos en el análisis a los que añadimos nuestro caso, contando con un total de 56 pacientes. Se obtuvieron excelentes resultados en 53 casos y mejoría parcial en 3 con una media de seguimiento de 54 meses. No se encontraron complicaciones severas.

Discusión

Los buenos resultados clínicos y la ausencia de morbimortalidad severa postoperatoria encontrados en nuestro estudio nos conducen a recomendar la descompresión microvascular como tratamiento de elección para pacientes con neuralgia del trigémino secundaria a compresión por dolicoectasia vertebrobasilar en pacientes en los que la cirugía mayor no esté contraindicada.

Palabras clave:
Descompresión microvascular
Neuralgia del trigémino
Dolicoectasia vertebrobasilar
Full Text
Introduction

Vertebrobasilar dolichoectasia (VD) is characterized by elongation and dilatation of the vertebral and basilar arteries. This entity may become symptomatic by compression of the brainstem, hydrocephalus, stretching of intracranial nerves, emboli from luminal thrombi or hemorrhage. It is associated with high risk of ischemic stroke and dead.1 Dolichoectathic posterior circulation arteries can compress the root of the trigeminal nerve leading to trigeminal neuralgia (TN).2

Few cases of TN secondary to VD treated by microvascular decompression (MD) have been reported. In large series of microsurgically treated patients, only in around 2% of cases, the vertebral or basilar arteries are described as the offending artery.3–5 These big arteries carry higher arterial flow than smaller ones as the superior cerebellar artery (the most frequently artery implied). So, one can doubt if a simple maneuver of interposing a thin sponge between the trigeminal root and the offending artery may be able to buffer the pressure of such vessels.

We present a case of TN caused by vertebral artery compression, emphasizing the microsurgical technique by means of including the video recording of the vascular decompression procedure. We have reviewed the literature about this uncommon nerve compression in order to investigate by pooled case analysis if MD is a good option to these patients.

Patients and methods

We collected the clinical data and surgical video tape of a patient who underwent MD for TN caused by a dolichoectatic vertebral artery vascular compression.

We searched the medical literature up to January 2015, by using Pubmed database. No limits of languages were used. Key words and free text searches, used in combinations (by using the Boolean operators OR and AND), were the following: “trigeminal neuralgia”, “tic douloureux”, “microvascular decompression”, “Vertebrobasilar ectasia” and “Vertebrobasilar dolichoectasia”. The references of the publications obtained were checked for additional studies.

The inclusion criteria of the studies were: (a) studies reporting cases of MD for TN caused by dolichoectatic arteries’ compression (b) documentation of the surgical results and (c) reporting of death and complications after the procedure.

The study exclusion criterion was impossibility to exclude other techniques than MD as treatment in the series.

The data recorded were: (a) Epidemiologic data: Number, sex, age and clinical presentation (number of branches affected and side); (b) Surgical results: excellent (pain disappearance without medication), partial recovery (pain relief with medication) and surgical failure; (c) Surgical complications and follow up.

Pooled rates were calculated for demographic information, clinical characteristics and results of intervention. If the study did not specify the data per patient, we used the mean or median of the results of that study. SPSS 14 software was used.

Results

A 68-year-old man presented with left TN for 9 months. Initially, his pain was relieved with carbamazepine, but it recurred 2 months later. Pain persisted despite carbamazepine dose increase and addition of morphine derivates. On admission, he complained of left-sided trigeminal neuralgia mainly affecting the second and third branches of the trigeminal nerve. MRI revealed a mechanical compression at the left side of the brainstem due to vertebrobasilar dolichoectasia (Fig. 1). Angiography showed a great distortion of the vertebrovasilar system with an extremely high origin of the PICA (Fig. 2).

Fig. 1.
(0.23MB).

Axial T2 weighted (upper) and 3D-TOF (lower) MRI. An important compression and displacement of the left side of the brainstem at due to vertebrobasilar dolichoectasia can be observed.

Fig. 2.
(0.09MB).

Digital subtraction angiography showing great distortion of the vertebrovasilar system with lateralization to the left side. An extremely high origin of the PICA can be observed (white arrow).

Surgical intervention (see video)

A left suboccipital craniotomy was performed with the patient placed supine with the head rotated 90° to the right. The dura was opened and arachnoid dissection revealed a large vascular structure identified as the vertebral artery, dislocating and compressing the left trigeminal nerve at its root entry zone. Two pieces of dura substitute goretex were introduced in the conflicting neurovascular area between the artery and the TN after dissection, achieving a satisfactory decompression.

Outcome

The patient's lancinating facial pain resolved immediately after surgery and his postoperative course was uneventful. He remains free of pain without medical treatment 18 months after surgery.

Literature review

After selection 7 studies were included for analysis2,6–11 (Fig. 3). To these we added our own case, making a total of 56 patients from 8 studies (Table 1). One patient of the study Linskey et al. was removed because partial rizothomy was added to MD.

Fig. 3.
(0.07MB).

Flow of information through the different phases of the review.

Table 1.

F/M, female/male ratio. BA/VA, basilar artery/vertebral artery ratio of offending vessel.

Study  N° Cases  Mean age  F/M  BA/VA  Follow up 
Ma et al.2  60.7  2/7  4/5  22 
Kraemer et al.6  63  0/1  1/0 
Campos et al.7  63  0/1  1/0  24 
Noma et al.8  59  0/1  1/0  11 
García De Sola et al.9  65  1/2  15.6 
El-Ghandour et al.10  10  54  4/6  6/4  93 
Linskey.11  30  61  21/9  10/20  60 
Arrese et al.  68  0/1  0/1  18 

The pooled group was constituted by 28 males and 28 females with a mean age of 60 years (SD 4.6). The mean of affected branches was 1.75. The 2nd and 3rd combination is present in more than half of the cases with this data specified in the studies (15 of 26). The right-left ratio was 19/37.

Results of MD were excellent in 53 cases and partial recovery in 3 with a mean follow up of 54 months.

In the 54 cases from 7 studies in which the offending vessel was reported, this was the basilar artery in 24 cases and the vertebral artery in 30.

No severe complications or deaths were found. Only in 10 cases there were minor reported complications affecting cranial nerves.

Discussion

Both our case and the pooled review of the literature point out the excellent results of MD for cases of TN secondary to VD. Although the blood flow in vertebrobasilar arteries is obviously higher than in the thinner offending vessels more frequently involved as cause of TN, and complementary techniques have been advocated,12,13 the decompression of the trigeminal nerve by a patch appears to be sufficient to resolve the cause of the pain in most interventions. These complementary techniques should be reserved to difficult cases, as compressions caused by mega-VD or a calcified basilar artery, in those the offending vessel can be difficult to move off the nerve.

This pooled series of patients presented similar clinical characteristics compared to general series of MD for TN. Only the ratio male-female was different: whereas most TN series report a 2:3 male–female ratio, in our pooled population the ratio was 1:1. This finding may be explained by the higher prevalence of VD in males, since the development of VD appears to be associated to atherosclerotic risk factors.

With regards to the surgical technique, difficulty of the intervention is not substantially different than in typical superior cerebellar artery compression cases. As one can see on the video record, the difficulty of dissection was not increased by the size of the offending vessel and the introduction of the patch between the artery and nerve was easily performed.

Other techniques as percutaneous ones, radiosurgery or MD plus partial sensory rhizotomy have been advocated for TN secondary to VD.14,15 In our opinion, due to the excellent results obtained with MD, this is the technique is superior to destructive ones if the patient is in good condition.

VD is characterized by elongation and dilatation of the vertebrobasilar arteries. Although in our case the patient underwent a catheter angiography, MRI may be sufficient to show the arterial tortuousity and the compression of the nerve in order to plan the surgery noninvasively. Moreover, by performing Angio-MR other pathologic findings as vascular malformations can be excluded.

In a systematic review of the literature, Wolters et al.1 found that patients with vertebrobasilar dolichoectasia are at high risk of ischemic stroke, brainstem compression and death, being hemorrhagic complications less common. Estimated 5 year complication risks were 17.6% for ischemic stroke, 10.3% for brainstem compression, 10.1% for transient ischemic attack, 4.7% for hemorrhagic stroke, 3.3% for hydrocephalus, and 2.6% for subarachnoid hemorrhage. Therefore, patients affected by TN secondary to VD should not only be approached as solely facial pain patients, but also as patients at high vascular risk. In this regard, primary prevention recommendations for atherosclerosis should be emphasized.

We are aware that several limitations may have affected our results. First, due to our selection protocol many cases have been lost. It is obvious that, if VA or BA is the offending arteries in around 2% of cases, much more cases have been treated and included in other studies. However, we have preferred to be strict in the selection of similar and well reported cases, instead of increasing the number of cases. Second, there exist inherent limitations related to the nature of the studies analyzed. The excellent results and the lack of any associated major complication question the possibility of selection bias, which is likely more common in case reports and small published series.

Conclusion

The good clinical results and absence of postoperative mortality or severe morbidity in our pooled case series led us to recommend MD as the preferred treatment for TN caused by VD in patients in which major surgery is not contraindicated. VD is related to vascular risk factors as hypertension or smoking and implies high risk of ischemic stroke, brainstem compression and death. So, apart from the management of the pain, these patients should be suggested to observe primary prevention recommendations for vascular diseases.

All authors certify that they have no affiliations with or involvement in any organization or entity with any financial interest (such as honoraria; educational grants; participation in speakers’ bureaus; membership, employment, consultancies, stock ownership, or other equity interest; and expert testimony or patent-licensing arrangements), or non-financial interest (such as personal or professional relationships, affiliations, knowledge or beliefs) in the subject matter or materials discussed in this manuscript.

Patient consent

The patient has consented to the submission of the case report for submission to the journal.

Sources of funding

None.

Disclosures

None.

Conflict of interest

The authors declare no conflict of interest.

Appendix A
Supplementary data

The following are the supplementary data to this article:

(40.58MB)

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Copyright © 2016. Sociedad Española de Neurocirugía
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