Vascular Compression of the Medulla Oblongata by the Vertebral Artery: Report of Two Cases Kazuhiro Hongo, M.D., Hiroshi Nakagawa, M.D., Nobuhito Morota, M.D., Masanori Isobe, M.D. Department of N eu ro lo g ical Surgery, A ic h i M e d ic a l U n iv e rs ity , N ag akute, A ic h i, Japan OBJECTIVE AND IMPORTANCE: Compression of the medulla oblongata by a tortuous vertebral artery is rare. W e report two patients with this lesion who were treated with vascular decompression of the vertebral artery. CLINICAL PRESENTATION: A 36-year-old man developed right hemiparesis with lower cranial nerve deficits, and a 47-year-old man developed left lower cranial nerve deficits and left cerebellar dysfunction. In both patients, magnetic resonance imaging revealed a tortuous vertebral ar­ tery compressing the medulla oblongata. INTERVENTION: In both patients, the compressed medulla oblongata was treated by detaching the vertebral artery from the medulla oblongata, shifting it, and anchoring it to the nearby dura mater. Postoperatively, both patients are asymptomatic and have returned to their previous jobs. CONCLUSION: Although compression of the medulla oblongata by a tortu­ ous vertebral artery is rare, it can cause brainstem dysfunction. Magnetic resonance imaging clearly revealed the vascular compression in these patients. Surgical treatment was effective. The symptoms related to a tortuous vertebral artery and some techniques for surgical treatment are discussed. Awareness of this rare lesion is necessary to ensure appropriate treatment. (N eu ro surge ry 4 5 :9 0 7 - 9 1 0 , 1999) Key words: M edu lla oblongata, V a s c u la r co m p ressio n , V erteb ral artery T h e concept of neurovascular com­ pression as cranial nerve dysfunction resulting from the vascular com­ pression of cranial nerves is w idely accepted. However, there are very few rtP()fts of brainstem dysfunction caused } Oscular compression. Kim et al. (5) VUre the first to report such a patient, clnd there have been several subsequent reP°rts (2, 4, 6, 8-10). The authors re­ ported a similar case previously in w ich the elongated vertebral artery Caused a progressive pyram idal tract and the artery was eventually sec*0ned to relieve the neurological signs ^ )•In this report, we present two patients u 0 were treated successfully with mi­ crovascular decompression achieved by shifting the tortuous vertebral artery. Because a tortuous, thick vertebral artery usually causes the compression, careful and adequate decompression is needed to treat this type of lesion operatively. The routine placement of a prosthesis to achieve microvascular decompression in the treatment of hemifacial spasm or trigeminal neuralgia may not be adequate to achieve a definitive surgical result and to avoid recurrence in these patients. W e report two such patients, describe the symptoms related to the tortuous vertebral artery, and discuss some of the su rg ica l tech n iq u es used for treatment. Neurosurgery , Vol. 45, No. 4, O ctober 1999 Downloaded from https://academic.oup.com/neurosurgery/article-abstract/45/4/907/2832767 by University College of the Cariboo user on 28 January 2018 CASE REPORTS Patient 1 A 36-year-old man, who was a pedi­ atrician, was admitted to our institute with a 9-month history of right hemipa­ resis, hemisensory disturbance, and slight difficulty swallowing. These symptoms gradually worsened before admission. Exam ination A t admission, the neurological exam­ ination disclosed an onionskin-like hypalgesia, a diminished gag reflex on the right side, and slight atrophy of the right sternocleidomastoid and supraspinatus muscles. M ild right hemiparesis was also noted. Preoperatively, M R I re­ vealed significant compression of the right anterolateral side of the medulla oblongata, indicating a tortuous right vertebral artery that was causing severe indentation at the medulla (Fig. IA ). The right vertebral angiogram (Fig. 2) re­ vealed a tortuous, elongated right ver­ tebral artery and deviation of the verte­ bral union to the right. His symptoms and the M R I findings suggested that the vertebral artery was the cause of his symptoms. Surgery was performed af­ ter obtaining informed consent from the patient. O peration The patient was placed in the left lat­ eral decubitus position under general anesthesia. A right suboccipital craniot­ omy was performed. The dura mater was opened, and the inferolateral por­ tion of the posterior fossa was exposed. There was marked compression of the anterolateral portion of the medulla ob­ longata by a m edially bent and tortuous vertebral artery at the level of the hypo­ glossal nerve, anterior to the dentate lig­ ament. The vertebral artery was care­ fully separated from the medulla oblongata and from the small vessels. A 7- X 20-mm piece of Teflon felt was inserted beneath the vertebral artery and used to elevate the artery; 5-0 nylon sutures were used to anchor it to the dura mater near the foramen magnum. 907 908 Hongo et al F IG U R E 1. T2w eighted M R I artery. Surgery was performed after obtaining informed consent from the patient. scans of Patient 1. A , preoperative O p eration M R I scan revealing The patient was placed in the right decubitus position under general anes­ thesia, and a left suboccipital craniot­ omy down to the foramen magnum was performed. The lateral edge of the fora­ men magnum was widely drilled out near the occipital condyle, and the dura mater was opened. The vertebral artery was exposed by retracting the cerebel­ lum and evacuating the cerebrospinal fluid. The vertebral artery appeared to compress the anterolateral surface of the M pontom edullary junction, where the Hi posteroinferior cerebellar artery origi­ nates. W hen the vertebral artery was I IS detached from the medulla oblongata, an indentation caused by the artery was visible. The root entry zone of the VIHth cranial nerve was exposed, but no ab­ norm ality was found. W e suspected that vertebral artery compression of the me­ dulla oblongata might be responsible for the patient's symptoms, so the artery was shifted. A thin, 4- X 22-mm piece of Gore-Tex (W .L. Gore & Associates, Inc., Flagstaff, A Z ) was passed under the vertebral artery just p>roximal to the pos­ teroinferior cerebellar artery origin. A 6-0 nylon suture was passed through * led two slits made in the dura mater over the jugular tuberculum and tied to the Gore-Tex sheet to detach the vertebral artery from the medulla oblongata, IS: which achieved vascular decompression (Fig. 4). Throughout the procedure, the somatosensory evoked potential re­ mained unchanged. a flow void in the right side of the m edulla oblongata, w h ich indicates severe vascular com pression B, postoperative M R I scan dem onstrating that this void is no longer visible and indicating good decom pression of the m edulla oblongata. w Indentation, apparently caused by the vertebral artery, was noted on the me­ dulla where the right hypoglossal nerve originates, and the right vertebral artery was clearly indented by the hypoglossal nerve. The VUIth, IXth, Xth, and Xlth cranial nerves were all spared. Postoperative course The postoperative neurological exam­ ination revealed that hypalgesia had subsided, and the patient had a feeling of fatigue in his right arm and leg. The atrophy of the right sternocleidomas­ toid and supraspinatus muscles re­ mained. The postoperative M R I scan re­ vealed good decompression of the medulla oblongata (Fig. IB ). The patient was discharged 2 weeks after surgery. His symptoms gradually decreased, and he returned to his previous work 6 months after surgery. He has remained healthy in the almost 9 years since the operation. Patient 2 A 47-year-old man, the master of a sushi shop, first complained of vertigo 7 years before presentation. One year later, he noticed difficulty hearing on his left side. In March 1996, his gait was unsteady and he could not stand w ith ­ out holding onto a bar. At that time, he was referred to us by an otolaryngologist. Exam ination At admission, the patient was deaf in his left ear and had a decreased gag reflex on the left side. Standing on his left foot and maintaining a tandem gait were impossible because of left cerebel­ lar dysfunction. Preoperative M R I re­ vealed a tortuous left vertebral artery compressing the left anterolateral side of the medulla oblongata, resulting in deformity of the medulla oblongata (Fig. 5A). On the basis of the neurological and M R I findings, his symptoms were thought to be caused by the vertebral V F IG U R E 3. T2weighted MRI scans of Patient 2. A, preoperative M R I scan revealing the left-sided deformity of the medulla oblongata with the vertebral F IG U R E 2. Vertebral angiogram of artery attached to Patient 1, revealing tortuous vertebral the medulla, ft arteries bilaterally and the point of postoperative M R I scan dem onstrating space betw een the vertebral artery and the m edulla com pression (a rro w h e a d ). m edulla oblongata, w h ich indicates that vascular decom pression was achieved. Neurosurgery, Vol. 45, No. 4, O ctober 1999 Downloaded from https://academic.oup.com/neurosurgery/article-abstract/45/4/907/2832767 by University College of the Cariboo user on 28 January 2018 Jral th icle >rti ien !■/ iei ki icti, to C o m p r e s s io n o f th e M e d u lla O b lo n g a t a b y th e V e r t e b r a l A r t e r y FIGURE 4. Intraoperative photographs of Patient 2. A , the tortuous vertebral artery (VA) compressing the m edulla oblongata. B, the vertebral artery is shifted away from the m edulla oblongata w ith a Gore-Tex sheet and 6-0 nylon suture, which is anchored to the dura m ater near the jugular tuberculum . Postoperative course The patient's postoperative course was uneventful. The gag reflex seemed normal, and his cerebellar dysfunction decreased; he could stand and w alk without holding a bar. The postopera­ tive MRI scan revealed that the vertebral artery was detached from the medulla oblongata, although some deformity of the medulla remained (Fig. 3B). He was discharged 2 weeks after surgery and returned to his previous job 3 months later. d is c u s s io n Patient 1 presented with progressive pyramidal tract signs and lower cranial nerve deficits. The M R I scans and ver­ tebral angiograms revealed compres­ sion of the medulla oblongata by a tor­ tuous vertebral artery. The right vertebral artery was thought to be com­ pressing both the corticospinal tract just below the pyramidal decussation and the nucleus solitarius in the anterolatt rul portion of the medulla, causing the n£ht hemiparesis and difficulty swalowing. A surgical decompression was Performed with good results. Patient 2 presented with left hearing loss, de­ ceased gag reflex, and left cerebellar ysfunction. There were no pyram idal hact signs. The medulla oblongata was apparently compressed by a tortuous vertebral artery, especially at its antero­ medial side. The inferior olive and the olivocerebellar tract, as w ell as the nu­ cleus ambiguus, were thought to be dis­ turbed. Surgery was performed after ob­ taining informed consent from the patient regarding the benefits and pos­ sible risks of surgery. The reason for his hearing disturbance was not fully deter­ mined. Because the cerebellar signs dis­ appeared after surgery, the tortuous vertebral artery may have been com­ pressing the inferior olive and the olivo­ cerebellar tract at the level of the me­ dulla oblongata. In both patients, the symptoms disappeared almost com­ pletely after surgery, and both patients returned to their previous jobs. There are few reports describing a tor­ tuous vertebral artery compressing the medulla oblongata and causing neuro­ logical deficits, such as pyram idal tract signs or lower cranial nerve deficits. In 1985, Kim et al. (5) reported the first such case. Their patient responded well to vascular decompression. Kobayashi et al. (6) reported a patient with com­ pression of the medulla oblongata by the vertebral arteries bilaterally associ­ ated with a Chiari malformation, in which the symptoms improved after vascular decompression of the medulla oblongata. Except for the patient re­ ported by Tanaka et al. (10), in whom the involved artery was an excessively Neurosurgery, Vol. 45 , No. 4, O ctober 1999 Downloaded from https://academic.oup.com/neurosurgery/article-abstract/45/4/907/2832767 by University College of the Cariboo user on 28 January 2018 909 long basilar artery, the vessel responsi­ ble is usually a tortuous vertebral artery, but never one with a "m egadolicho" shape. It is not easy to determine when sur­ gery is indicated. For hemifacial spasm or trigeminal neuralgia, surgical micro­ vascular decompression is indicated by the clinical status, even when no appar­ ent offending vessels are observed on M R I scans or magnetic resonance angio­ grams. On the other hand, in symptom­ atic patients in whom compression of the medulla oblongata by the vertebral artery is suspected, a definite diagnosis should be made with the aid of M R I as well as with a precise neurological ex­ amination. In our two patients, the de­ formity of an apparently compressed medulla oblongata was visible on the M R I scans. There are several possible methods of surgical vascular decompression. Sim ­ ply inserting a prosthesis between the brainstem and the offending vessel can usually decompress the typical hemifa­ cial spasm or trigeminal neuralgia. However, with compression of the me­ dulla oblongata by the vertebral artery, the offending artery may be thick and quite difficult to decompress by this method. Fukushima (3) and Kondo (7) proposed lifting and anchoring the of­ fending vessel to the nearby dura mater with a variety of materials. In our cases, the tortuous vertebral artery was shifted and anchored to the dura mater with a Teflon sheet in Patient I and Gore-Tex in Patient 2, without inserting a prosthe­ sis. Good vascular decompression was achieved, which improved the neuro­ logical deficits. The vertebral artery should be shifted carefully, to avoid damaging small branches or perforating arteries from the vertebral artery enter­ ing the medulla oblongata. W e previ­ ously reported a patient treated by sec­ tioning the tortuous vertebral artery to produce vascular decompression of the medulla oblongata (4). This is the ulti­ mate w ay to achieve vascular decom­ pression, but indications for this ap­ proach may be quite limited. Shifting the vessel and anchoring it to the dura mater is a more appropriate choice. In conclusion, careful examination us­ ing M R I may help diagnose this rare lesion more frequently. A precise diag- 910 Hongo et al nosis on the basis of both M R I and a careful neurological examination is es­ sential to determine appropriate surgi­ cal interventions. R e c e iv e d , D e c e m b e r 17, 1998. A c c e p te d , Ju n e 7, 1999. R e p rin t req uests: K a z u h iro H o n g o , M .D ., D e ­ p a rtm e n t of N e u ro s u rg e ry , S h in sh u U n iv e rs ity Sch o o l o f M e d ic in e , A s a h i 3-1-1, M a ts u m o to 390-8621, Ja p a n . R EFER EN C ES 1. Deleted in proof. 2 . Dohi K, Kubota M , Ham ada H, Kuwabara K, Nishijim a Y, Kushihashi T, Matsum oto K: Com ­ pression of the medulla oblongata by a dissect­ ing aneurysm of the vertebral artery 7 years after its rupture: Case report [in Japanese]. No Sh in k ei G eka 2 2 :1 0 6 7 - 1 0 7 0 , 1 9 9 4 . 3 . Fukushima T: Posterior cranial fossa neurovas­ cular decompression (Jannetta method) for tri­ geminal neuralgia and facial spasm [in Japa­ nese]. No Sh in k ei G eka 1 0 :1 2 5 7 - 1 2 6 1 , 1 9 8 2 . 4 . Hongo K, Kobayashi S, Hokam a M, Sugita K: Vertebral artery section for treating arterial compression of the medulla oblongata: Case report. J Neurosurg 7 9 :1 1 6 - 1 1 8 , 1 9 9 3 . 5 . Kim P, Ishijima B, Takahashi H, Shim izu H, Yokochi M: Hem iparesis caused by vertebral artery compression of the medulla oblongata: Case report. J Neurosurg 6 2 :4 2 5 ^ 4 2 9 , 1 9 8 5 . 6 . Kobayashi T, Ogawa A, Kam eyam a M, Uenohara H, Yoshimoto T: Chiari malformation w ith compression of the medulla oblongata by the vertebral arteries: Case report. J Neurosurg 7 7 :3 0 7 - 3 0 9 , 1 9 9 2 . 7 . Kondo A: Follow-up results of m icrovascular decompression in trigeminal neuralgia and hemifacial spasm. Neurosurgery 4 0 :4 6 - 5 2 , 1 9 9 7 . 8 . M iyazaki M, Hashimoto T, Sakuram a N, Yoshimoto T, Tayama M , Kuroda Y: Central sleep apnea and arterial compression of the me­ dulla. A nn N eurol 2 9 :5 6 4 - 5 6 5 , 1 9 9 1 . 9 . M urata H, W aga S, Kojima T, Shim izu T, Shim izu S: M edulla oblongata compression by a tortuous vertebral artery: Case report [in Jap ­ anese], No Sh in k ei G eka 2 3 :3 4 9 - 3 5 3 , 1 9 9 5 . 1 0 . Tanaka Y, M iyazaki M , Tsuda M , M atsuyam a M , Kuzuhara S: Compression of the medulla oblongata by an elongated vertebral artery [in Japanese]. Sh in k ein aik a 3 4 :3 3 5 - 3 3 6 , 1 9 9 1 . C O M M EN TS The authors report the successful treatment of two patients who were rel­ atively young and who presented with symptoms suggestive of lower brain­ stem dysfunction. Imaging studies were reasonably correlated w ith neurological signs and suggested encroachment by the aberrant course of a vertebral artery. The authors' illustrations and intraoper­ ative photographs demonstrate a nice technique for displacing the vertebral artery from the brainstem. This unusual condition, as well as potential treatment alternatives, has been described previ­ ously, but the authors supplement the existing literature with useful surgical recommendations. M y typical experi­ ence has involved older patients with ectatic vertebral arteries that produced the mass effect. The expected perioper­ ative course in elderly patients can be expected to be more difficult than that reported in this article. In addition, in patients with more advanced and dis­ abling neurological symptoms, the dis­ section and displacement of the artery from the brainstem is not as well toler­ ated and has a higher morbidity. N e v­ ertheless, anecdotal evidence suggests that this condition can be progressive, and therefore treatment is warranted. I plan to use the Gore-Tex sling, as re­ ported by the authors, in my own practice. H. H unt Batjer Chicago, Illinois I read with interest these two unusual case reports by Hongo et al. from Aichi Medical University. Symptomatic vas­ cular compression of the medulla by a tortuous vertebral artery has been re­ ported previously. This rare condition presents several challenges to neurosur­ geons. As outlined by the authors, a careful neurological examination is im ­ perative to ensure that the patient's symptoms are attributable to vascular compression of the brainstem. In the era of magnetic resonance imaging, it is not uncommon to identify asymptomatic distortion of the brainstem caused by a tortuous vertebral or basilar artery. In Neurosurgery, Vol. 45, No. 4, O ctober 1999 Downloaded from https://academic.oup.com/neurosurgery/article-abstract/45/4/907/2832767 by University College of the Cariboo user on 28 January 2018 the clinical decision-making process, the neurosurgeon must determine whether the patient's symptoms are significant enough to warrant surgical interven­ tion. Each of the patients reported b\ Hongo et al. had significant and pro­ gressive neurological deficits that clearly justified surgical intervention. The surgical management of this partic­ ular disorder is much more complicated than microvascular decompression for cranial nerve dysfunction. These risks must be weighed when considering sur­ gical intervention. Finally, the technical aspects of vascular decompression of the medulla are formidable. As pointed out by the authors, simple placement of a prosthetic device between the artery and brainstem is inadequate treatment. More often, the artery must be displaced from the brainstem by creation of a sling to the adjacent dura. Care must be taken to avoid injury to small perforating branches off the vertebrobasilar system. These authors have presented an impor­ tant report and have achieved excellent surgical results in these two patients. D an ie l L. Barrow Atlanta, Georgia Tortuosity of the vertebral and basilar arteries is the rule rather than the excep­ tion. It is unusual, however, for such tortuosity to cause clinical symptoms. Patient 1 in this article is a compelling example of such a vascular compression syndrome. Patient 2 is less compelling, although the M R I scans are quite dra­ matic. The authors are correct in em­ phasizing the need for careful patient selection before any operative decom­ pression. M oving large, tortuous pos­ terior circulation vessels is more haz­ ardous than the typical superior cerebellar artery manipulation in tri­ gem inal neuralgia. The Gore-Tex sling is an innovative w ay of managing this problem. Ste ve n L. Giannotta Los Angeles, California