Abstract
Background
Giant aneurysms of the intracranial vertebral artery are very rare cerebrovascular lesions. Due to the rarity of these aneurysms, we know little about them.
Methods
We performed a systematic review of the English literature by searching the PubMed database. The inclusion criteria were as follows: (a) the full text was available and (b) complete clinical data were available.
Results
A total of 45 articles were identified, containing 53 patients (53 aneurysms). The patients were aged from 5 to 77 years (48.8 ± 20.8 years). Four patients receiving conservative treatment died. The remaining 49 patients were divided into the aneurysm removal group (n = 17) and the aneurysm reserve group (n = 32). The outcomes of the 49 treated cases could be obtained in 45 cases, 31 of which (68.9%, 31/45) had a Glasgow outcome scale score of 4–5.
Conclusions
It is still difficult to treat intracranial giant vertebral artery aneurysms, regardless of the treatment selected. Because of the malignant natural history, aggressive treatment is still advocated.
Keywords: Medulla oblongata, giant aneurysm, vertebral artery, aneurysmectomy, endovascular treatment
Introduction
Giant aneurysms of the intracranial vertebral artery (VA) are very rare. If left untreated, the aneurysms will gradually grow and compress the medulla oblongata, resulting in a poor prognosis. 1 Especially for those ruptured, the prognosis is poor. Therefore, giant aneurysms of the intracranial VA need to be treated, and the treatments include open surgery and endovascular treatment (EVT).2,3
Theoretically, microsurgical clipping with the combination of aneurysmectomy could relieve the mass effect of the giant aneurysm on the medulla oblongata. However, the surgical risk is extremely high. Although EVT is easy to conduct, it cannot relieve the mass effect. Moreover, intra-aneurysmal thrombosis formed after EVT may enlarge the aneurysm and compress the medulla oblongata even further, leading to postoperative deterioration and even death. 4
To date, no studies have been conducted to compare the efficacy of microsurgical clipping with the combination of aneurysmectomy and EVT for giant aneurysms of the intracranial VA. In this study, we performed a systematic review of the relevant literature to address this issue.
Materials and methods
Search strategy
This systematic review was conducted in accordance with the Preferred Reporting Items for Systematic Review and Meta-Analyses (PRISMA) guidelines. Giant aneurysm of posterior circulation and giant aneurysm of vertebral artery were used as searching items. Eligible English language articles (case reports, case series, and studies of giant aneurysms of the VA) were identified by searching the PubMed database (the last searching date was 14 February 2021).
The reference lists of the identified articles were also manually searched for additional relevant studies. The inclusion criteria were as follows: (a) the full text was available; (b) complete clinical data were available; and (c) diameter greater than 2.5 cm or described as giant aneurysm. A flowchart of the selection process is shown in Figure 1.
Figure 1.
Flowchart of search strategy.
Statistical analysis
GraphPad software (LLC, San Diego, USA, 8.0) was used for statistical analysis. The continuous variables were expressed as mean ± standard deviation. If complying with Gaussian distribution, the t-test would be performed for continuous data. Otherwise, a non-parametric test would be performed. For count data, the chi-square test or Fisher’s exact test were used. P < 0.05 indicated a significant difference.
Results
A total of 45 articles containing 53 patients (53 aneurysms) were identified.3–47 In addition to the 53 patients identified in the literature, we also included two typical cases for illustration which were not included in the final analysis (Figures 2, 3 and 4).
Figure 2.
Rapid neurological deterioration in a patient with ruptured intracranial giant VA aneurysm. (a) Head CT shows subarachnoid haemorrhage concentrated at the ambient cistern. (b)–(d) Axial (b), sagittal (c) and coronal (d) CT show the occupying lesion with medulla oblongata compression. (e) CTA reveals a giant aneurysm in the VA. (f) CT shows the size of the aneurysm and the residual cavity (filled with contrast agent). (g) and (h) Several hours after admission, the patient rapidly deteriorates. Head CT shows further effacement of the brainstem (g) and acute hydrocephalus (h). The patient died 16 hours after admission. CT: computed tomography; CTA: computed tomography angiography; L: left; VA: vertebral artery.
Figure 3.
Pre-procedural images of a patient with giant right VA aneurysm. (a) and (b) Sagittal MRI on T2 (a) and T1 (b) sequences show a thrombosed giant aneurysm in front of the medulla oblongata (asterisk) with severe brainstem compression. (c) Axial MRI on T2 sequence shows a thrombosed aneurysm in front of the medulla oblongata with severe brainstem compression. (d) MRI on T1 sequence shows slight dilation of the lateral ventricles. (e) CTA shows that the aneurysm is located at the right VA. Single and double arrowheads denote the VA proximal and distal to the aneurysm, respectively. (f) MIP image shows that the residual cavity of the aneurysm is filled with contrast agent. The patient is a 54-year-old man admitted for right limb weakness and dysphagia. AN: aneurysm; BA: basilar artery; CT: computed tomography; CTA: computed tomography angiography; L: left; MIP: maximal intensity projection; MRI: magnetic resonance imaging; R: right; VA: vertebral artery.
Figure 4.
Intraoperative and postoperative images of the afore-mentioned patient. (a) Angiogram of the left VA shows the left VA is displaced by the aneurysm. (b) Angiogram of the right VA shows the aneurysm. Asterisk denotes the residual cavity and T denotes the thrombosed part. (c) Angiogram of the right VA after further advancing the guiding catheter, the aneurysm is more clearly shown. A jet flow (arrowhead) is noted. Some small arteries (asterisk) originate from the VA proximal to the aneurysm. (d) The VA is occluded with coils proximal to the aneurysm. (e) Unsubtracted angiogram of the right VA shows the right VA is coiled (arrow). And there is contrast agent retention in the aneurysm cavity. (f) Angiogram of the left VA shows no evident bloodflow to the contralateral aneurysm. (g) and (h) The patient deteriorates 8 hours after EVT. Head CT shows that the aneurysm is filled with contrast agent and that the bilateral ventricles are dilated. The patient died 20 hours after EVT. AN: aneurysm; BA: basilar artery; EVT: endovascular treatment; VA: vertebral artery.
The patients were aged from 5 to 77 years (mean 48.8 ± 20.8 years). Thirty (58.8%, 30/51) patients were boys or men. On admission, five patients (9.4%, 5/53) lived independently, while the remaining 48 (90.6%, 48/53) patients were dependent on others. Two patients (3.8%, 2/53) were admitted for aneurysmal subarachnoid haemorrhage due to rupture of the VA aneurysms, and the remaining 51 (96.2%, 51/53) patients had unruptured aneurysms. The diameters of 25 aneurysms were not reported and only described as giant, and the diameters of the other 28 aneurysms ranged from 2.5 to 5 cm (mean 3.8 ± 0.7 cm).
Morphology and proportion of residual cavity
There were two cases without information on the morphology and proportion of residual cavity. Morphological details of the remaining 51 cases were as follows. Ten patients (19.6%, 10/51) had serpentine aneurysms, with the proportion of residual cavity being less than one-third in nine cases and more than two-thirds in one case. Ten patients (19.6%, 10/51) had irregular dissections, with the proportion of residual cavity being less than one-third in nine cases and more than two-thirds in one case. Twenty-four patients (47.1%, 24/51) had spherical dissections, with the proportion of residual cavity between one-third and two-thirds in four cases and more than two-thirds in 20 cases. Seven patients (13.7%, 7/51) had fusiform dissections, with the proportion of residual cavity being less than one-third in six cases and between one-third and two-thirds in one case.
Relationship between the aneurysm and medulla oblongata
Positional information was not available for one aneurysm. Of the remaining 52 aneurysms, 41 (78.8%, 41/52) were located at the midline of the medulla oblongata, and 11 (21.2%, 11/52) were located at one side of the medulla oblongata. In terms of the degree of aneurysmal compression to the medulla oblongata (Figure 5), 15 aneurysms (28.8%, 15/52) posed moderate compression to the medulla oblongata and 37 cases (71.1%, 37/52) posed severe compression.
Figure 5.
Illustration of the severity of medulla oblongata compression. (a) and (b) The diagrams in sagittal (a) and axial (b) views show that the medulla oblongata is moderately compressed (less than 50% of its anteroposterior diameter). (c) and (d) The diagrams in sagittal (a) and axial (b) views show that the medulla oblongata is severely compressed (more than 50% of its anteroposterior diameter). AN: aneurysm.
Treatment options
Four patients received conservative treatment (Table 1), and the remaining 49 patients received invasive treatment. The treated aneurysms were divided into two groups: the aneurysm removal group and the aneurysm reserve group. In the aneurysm removal group, partial or complete aneurysmectomy was performed after the aneurysm was trapped, leading to complete or partial decompression of the medulla oblongata. A bypass would be performed if necessary. Details of the aneurysm removal are illustrated in Figure 6.
Table 1.
Clinical data of the patients with conservative treatment.
| No. | Author/year | Age/sex | Independence state at admission | Ruptured at admission | Size | Morphology and proportion of residual cavity | Positional relationship | Degree of brainstem compression | Cause of death |
|---|---|---|---|---|---|---|---|---|---|
| 1 | Nagahiro et al./1995 5 | 60/M | Dependent | No | 5 cm | Serpentine, <1/3 | Middle | Severe | Mass effect |
| 2 | Yasui et al./1998 6 | 76/M | Dependent | No | 5 cm | Serpentine, <1/3 | Lateral | Moderate | SAH |
| 3 | Inamasu et al./2000 7 | 54/M | Dependent | No | 4.5 cm | Irregular, <1/3 | Middle | Moderate | SAH |
| 4 | Ge et al./20168 | 65/F | Dependent | No | Giant | NM | NM | NM | SAH |
F: female; M: male; NM: not mentioned; SAH: subarachnoid haemorrhage.
Figure 6.
Schematic diagram of treatments with aneurysmal removal. (a) The VA aneurysm is trapped with clips and partially removed. (b) The aneurysm is completely resected after trapping. (c) A VA or PICA bypass is performed after trapping and partially removing the aneurysm. (d) A VA or PICA bypass is performed after trapping and completely resecting the aneurysm. (e) and (f) For an eccentric aneurysm (e), the VA wall is sutured after clipping and removing the aneurysm (f). BA: basilar artery; PICA: posterior inferior cerebellar artery; VA: vertebral artery.
In the aneurysm reserve group, blood flow to the aneurysm was diminished or occluded by surgical or endovascular methods, without removing the aneurysm. Thus, the space-occupying effect was not relieved. The details of aneurysm reserve treatments are illustrated in Figure 7.
Figure 7.
Schematic diagram of treatments with aneurysm reserve. (a) The VA is clipped at the proximal end to reduce blood flow to the aneurysm. (b) The aneurysm is trapped with clips. (c) The VA is proximally occluded with coils or a balloon to reduce blood flow to the aneurysm. (d) The VA is proximally occluded with a balloon or coils, and the distal end of the aneurysm is coiled through the contralateral VA. (e) Both the VA and aneurysm are coiled. (f) The aneurysm is coiled with stent assistance. (g) The VA is reconstructed with a FDS alone. (h) The aneurysm is loosely coiled and the VA is reconstructed with a FDS. BA: basilar artery; FDS: flow-diverting stent; VA: vertebral artery.
Aneurysm removal group
Of the 17 patients receiving aneurysmal removal (Table 2), four (28.6%, 4/17) underwent complete aneurysmectomy, five (29.4%, 5/17) underwent partial aneurysmectomy, two (11.8%, 2/17) underwent partial aneurysmectomy in combination with bypass, three (17.6%, 3/17) underwent complete aneurysmectomy in combination with bypass, one (5.9%, 1/17) underwent complete aneurysmectomy in combination with suturing reconstruction and two (11.8%, 2/17) underwent aneurysmal clipping.
Table 2.
Clinical data of the patients with aneurysm removal.
| No. | Author/year | Age/sex | Independence state at admission | Ruptured at admission | Size | Morphology and proportion of residual cavity | Positional relationship | Degree of brainstem compression | Treatment | Deterioration | Follow-up period | Outcome (GOS) |
|---|---|---|---|---|---|---|---|---|---|---|---|---|
| 1 | Sugita et al./19889 | 51/F | Dependent | No | 4.5 cm | Serpentine; <1/3 | Middle | Severe | Complete aneurysmectomy | Y | 3 Months | 4 |
| 2 | Sugita et al./19889 | 61/M | Dependent | No | 3.5 cm | Serpentine; <1/3 | Middle | Severe | Complete aneurysmectomy | N | 3 Months | 3 |
| 3 | Sugita et al./19889 | 65/M | Dependent | No | 3.5 cm | Spherical; >2/3 | Middle | Severe | Partial aneurysmectomy | N | 6 Months | 4 |
| 4 | Sugita et al./19889 | 68/F | Dependent | No | 2.5 cm | Spherical; >2/3 | Middle | Severe | Aneurysmal clipping and complete aneurysmectomy | N | 6 Months | 5 |
| 5 | Honda et al./199110 | 64/M | Dependent | No | Giant | Irregular dissection; <1/3 | Lateral | Moderate | Complete aneurysmectomy + suturing reconstruction | N | 3 Months | 4 |
| 6 | Wakui et al./199211 | 46/M | Dependent | No | 5 cm | Serpentine; <1/3 | Middle | Severe | Bypass + partial aneurysmectomy | Y | 3 Months | 4 |
| 7 | Nagasawa et al./1994 12 | 42/M | Dependent | No | 4 cm | Fusiform dissection; <1/3 | Lateral | Moderate | Bypass + complete aneurysmectomy | Y | NA | 1 |
| 8 | Okudera et al./1994 13 | 57/M | Dependent | No | 2.5 cm | NM | Lateral | Moderate | Partial aneurysmectomy | N | 2 Years | 4 |
| 9 | Greene et al./1994 14 | 9/M | Dependent | No | Giant | Serpentine; <1/3 | Middle | Severe | Partial aneurysmectomy | Y | 2 Years | 4 |
| 10 | Nagahiro et al./1995 5 | 60/M | Dependent | No | 4.5 cm | Serpentine; <1/3 | Middle | Severe | Bypass + partial aneurysmectomy | Y | 1 Year | 4 |
| 11 | Aoki et al./1997 15 | 65/M | Dependent | No | 3.6 cm | Irregular dissection; <1/3 | Middle | Severe | Complete aneurysmectomy | Y | 2 Weeks | 3 |
| 12 | Iihara et al./2003 16 | 58/F | Dependent | No | Giant | Spherical dissection; >2/3 | Middle | Moderate | Partial aneurysmectomy | Y | 5 Months | 4 |
| 13 | Benes et al./2006 17 | 65/F | Dependent | No | Giant | Fusiform dissection; <1/3 | Lateral | Moderate | Bypass + complete aneurysmectomy | N | 7 Years | 5 |
| 14 | Grigoryan et al./2016 18 | 48/F | Dependent | No | 4.3 cm | Spherical dissection; 1/3–2/3 | Middle | Severe | Complete aneurysmectomy | Y | NM | 3 |
| 15 | Suzuki et al./2018 19 | 52/F | Dependent | No | Giant | Fusiform dissection; <1/3 | Middle | Severe | Partial aneurysmectomy | N | 1 Year | 5 |
| 16 | Jaikin et al./2018 20 | 11/M | Dependent | No | 4 cm | Spherical dissection; >2/3 | Middle | Moderate | Aneurysmal clipping and complete aneurysmectomy | N | 13 Months | 5 |
| 17 | Tsunoda et al./2019 21 | 55/M | Dependent | No | Giant | Serpentine; <1/3 | Middle | Severe | Bypass + complete aneurysmectomy | N | NM | 5 |
F: female; GOS: Glasgow outcome scale; M: male; N: no; NA: not applicable; NM: not mentioned; Y: yes.
Aneurysm reserve group
Of the 32 cases (Table 3) receiving aneurysmal reserve, 10 (31.2%, 10/32) underwent proximal VA occlusion, 11 (34.4%, 11/32) underwent aneurysmal trapping, one (3.1%, 1/32) underwent coiling of the aneurysm, one (3.1%, 1/32) underwent coiling of the aneurysm and VA, one (3.1%, 1/32) underwent bypass and coiling of the aneurysm and VA, three (9.4%, 3/32) underwent stent-assisted coiling of the aneurysm, one (3.1%, 1/32) underwent VA reconstruction with flow-diverting stent (FDS), two (6.3%, 2/32) underwent partial coiling and VA reconstruction with FDS and two (6.3%, 2/32) underwent VA reconstruction with overlapping stents.
Table 3.
Clinical data of the patients with aneurysm reserve.
| No. | Author/year | Age/sex | Independence state at admission | Ruptured at admission | Size | Morphology and proportion of residual cavity | Positional relationship | Degree of brainstem compression | Treatment | Deterioration | Treatment | Follow-up period | Aneurysm change after treatment | Outcome (GOS) |
|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|
| 1 | Guglielmi et al./198922 | 38/F | Dependent | No | Giant | Spherical dissection, >2/3 | Lateral | Severe | Balloon occlusion of proximal VA | N | NA | 12 Months | NM | 4 |
| 2 | Abe et al./199423 | 57/M | Dependent | No | Giant | Fusiform dissection, <1/3 | Lateral | Moderate | Clipping of proximal VA | N | NA | 1 Month | NM | 5 |
| 3 | Khayata et al./199424 | 5/M | Dependent | Yes | Giant | Serpentine, <1/3 | Middle | Moderate | Bypass + trapping VA and aneurysm coiling | N | NA | 3 Months | Shrink | 5 |
| 4 | Gurian et al./199525 | 37/F | Independent | No | Giant | Spherical dissection, >2/3 | Middle | Severe | Balloon occlusion of proximal VA | Day 1; SAH | VA PAO and coiling | 5 Months | NM | 4 |
| 5 | Chang et al./199926 | 72/F | Dependent | No | 3 cm | Spherical dissection, >2/3 | Middle | Severe | Balloon and coil occlusion of proximal VA | Day 9; mass effect | Conservative treatment | NA | NA | 1 |
| 6 | Piepgras et al./200127 | 27/F | Dependent | No | 3 cm | Spherical dissection, >2/3 | Middle | Severe | Balloon and coil occlusion of proximal VA | Day 7; mass effect | Aneurysmectomy | NA | NA | NA |
| 7 | Otsuka et al./200128 | 10/M | Dependent | No | 3.3 cm | Serpentine, >2/3 | Lateral | Moderate | Trapping VA with coils | N | NA | 4 Months | Shrink | 5 |
| 8 | Leibowitz et al./200329 | 71/M | Dependent | No | Giant | Fusiform dissection, <1/3 | Lateral | Moderate | Trapping VA with coils | N | NA | 19 Months | Shrink | 5 |
| 9 | Morita et al./200330 | 65/F | Dependent | No | 3 cm | Spherical dissection, >2/3 | Middle | Severe | Occlusion of proximal VA with coils | Month 1; SAH | Aneurysm coiling | NA | NA | 1 |
| 10 | Lubicz et al./200431 | 52/M | Dependent | No | 4 cm | Spherical dissection, 1/3-2/3 | Middle | Severe | Trapping VA with coils | A few hours; mass effect | Conservative treatment | 36 Months | Shrink | 4 |
| 11 | Iwabuchi et al./20054 | 72/F | Dependent | No | Giant | Spherical dissection, >2/3 | Middle | Severe | Occlusion of proximal VA with coils | Day 9; SAH | Conservative treatment | NA | NA | 1 |
| 12 | Feddersen et al./200633 | 53/F | Dependent | No | Giant | Spherical dissection, >2/3 | Middle | Severe | Coiling aneurysm | Day 4; bradycardia | Conservative treatment | NM | NM | 5 |
| 13 | Kasliwal et al./200734 | 20/F | Dependent | No | 3.2 cm | Spherical dissection, >2/3 | Middle | Severe | Trapping VA with coils | Day 3; mass effect | Conservative treatment | NA | NM | 1 |
| 14 | Terakawa et al./200835 | 52/F | Dependent | Yes | 4.4 cm | Spherical dissection, 1/3–2/3 | Middle | Severe | Clipping and coiling trapping VA and coiling aneurysm | N | NA | 28 Months | Shrink | 4 |
| 15 | Limaye et al./201236 | 63/F | Independent | No | Giant | Spherical dissection, >2/3 | Middle | Severe | Stent-assisted coiling | N | NA | 6 Months | Unchanged | 5 |
| 16 | Saito et al./201337 | 70/F | Dependent | No | Giant | Fusiform dissection, <1/3 | Middle | Severe | Clipping and coiling the proximal VA | Several weeks; mass effect | NA | Several months | Shrink | 5 |
| 17 | Chen et al./201338 | 21/M | Dependent | No | 4.0 cm | Fusiform dissection, 1/3–2/3 | Middle | Severe | Trapping VA with coils | N | Conservative treatment | 3 Months | Shrink | 5 |
| 18 | Park et al./20143 | 7/F | Dependent | No | 3.4 cm | Spherical dissection, >2/3 | Middle | Severe | Occlusion of proximal VA with coils | Day 14; mass effect | Continuous coiling | 2 Years | Shrink | 5 |
| 19 | Zarzecka et al./201432 | 15/F | Independent | No | Giant | Irregular dissection, >2/3 | Lateral | Moderate | FDS | NO | Conservative treatment | 2 Years | Disappear | 5 |
| 20 | Zhang et al./201439 | 44/F | Dependent | No | Giant | Spherical dissection, >2/3 | Middle | Severe | Stent-assisted coiling | Immediately; SAH | Conservative treatment | NA | NA | 1 |
| 21 | Lee et al./201640 | 60/M | Dependent | No | 4 cm | Spherical dissection, >2/3 | Middle | Severe | Coiling trapping VA | Day 7; mass effect | NA | NA | NA | NA |
| 22 | Zhang et al./201641 | 55/M | Dependent | No | 3.1 cm | Irregular dissection, <1/3 | Middle | Severe | Trapping VA with coils | Immediately; mass effect | Conservative treatment | 26 Months | Enlarge | 3 |
| 23 | Zhang et al./201641 | 53/M | Dependent | No | Giant | Irregular dissection, <1/3 | Middle | Severe | Trapping VA with coils | N | NA | 4 Months | Unchanged | 3 |
| 24 | Zhang et al./201641 | 57/M | Dependent | No | Giant | Irregular dissection, <1/3 | Middle | Severe | Trapping VA with coils | Progressive deterioration after surgery; mass effect | Conservative treatment | 3 Months | Enlarge | 3 |
| 25 | Pahl et al./201642 | 54/M | Dependent | No | Giant | Irregular dissection, <1/3 | Middle | Severe | Trapping VA and coiling | Progressive deterioration after surgery; mass effect | Aneurysmectomy | NA | NA | NA |
| 26 | Ge et al./20168 | 77/M | Dependent | No | Giant | Spherical dissection, >2/3 | Middle | Severe | Stent-assisted coiling | Day 5; SAH | Conservative treatment | NA | NA | 1 |
| 27 | Murai et al./201743 | 71/M | Dependent | No | 4.2 cm | Irregular dissection, <1/3 | Middle | Severe | Trapping aneurysm with clips | Day 5; infarction | Conservative treatment | 12 Months | NM | 3 |
| 28 | Morishita et al./201744 | 56/M | Dependent | No | Giant | Irregular dissection, <1/3 | Middle | Moderate | PAO of proximal VA | N | NA | NM | NM | 5 |
| 29 | Wang et al./201945 | 12/M | Independent | No | Giant | Spherical dissection, >2/3 | Middle | Severe | FDS + partial coiling | N | NA | 6 Months | Shrink | 5 |
| 30 | Shirani et al./202046 | 12/M | Independent | No | Giant | Spherical dissection, >2/3 | Middle | Severe | FDS + coiling | N | NA | 6 Months | NM | 5 |
| 31 | Suzuki et al./202047 | NM/NM | Dependent | No | 3.8cm | Spherical dissection, >2/3 | Lateral | Moderate | Overlapping stents | N | NA | 6 Months | Shrink | 5 |
| 32 | Suzuki et al./202047 | NM/NM | Dependent | No | 3.5cm | Spherical dissection, 1/3–2/3 | Middle | Severe | Overlapping stents | N | NA | 12 Months | Enlarge | NA |
F: female; FDS: flow-diverting stent; GOS: Glasgow outcome scale; M: male; N: no; NM: not mentioned; NA: not applicable; PAO: parent artery occlusion; SAH: subarachnoid haemorrhage; VA: vertebral artery; Y: yes.
Outcome
Conservative treatment group
All the four patients died during conservative treatment.
Aneurysm removal group
Eight (47.1%, 8/17) of the 17 patients experienced neurological exacerbation after surgery. The follow-up period ranged from 3 to 84 months. The Glasgow outcome scale (GOS) scores of the patients were 1, 3, 4 and 5 in one (5.9%, 1/17), three (17.6%, 3/17), eight (47.1%, 8/17) and five (29.4%, 5/17) patients, respectively.
Aneurysm reserve group
Of the 32 patients, 17 (53.1%, 17/32) experienced neurological exacerbation postoperatively, of whom five exacerbated immediately after surgery, seven exacerbated in the first week, three between 1 and 2 weeks, and two beyond 2 weeks. The follow-up period ranged from one to 48 months. Sixteen patients underwent postoperative imaging follow-up, three aneurysms grew, 10 shrank, one disappeared and two remained unchanged. The GOS scores were available in 28 patients, that were 1, 3, 4 and 5 in six (21.4%, 6/28), four (14.3%, 4/28), four (14.3%, 4/28) and 14 (50%, 14/28) patients, respectively.
Statistical analysis
Baseline data
Age, sex, whether the patient was independent, mode of onset (ruptured or unruptured), aneurysm size, proportion of residual cavity greater than two-thirds, aneurysm position and severity of medulla oblongata compression did not differ significantly between the two groups, indicating that the baseline data were comparable (Table 4).
Table 4.
Comparison of the baseline data and treatment results between the aneurysm removal group and the aneurysm reserve group.
| Aneurysm removal group (n = 17) | Aneurysm reserve group (n = 32) | P value | |
|---|---|---|---|
| Age | 9–68 years (51.6 ± 17.3) (n = 17) | 5–77 years (45.3 ± 22.9) (n = 30) | 0.3287 |
| Sex | 11 M, 6 F (n = 17) | 16 M, 14 F (n = 30) | 0.5460 |
| Independent | 0 (n = 17) | 5 (n = 32) | 0.1488 |
| SAH | 0 (n = 17) | 2 (n = 32) | 0.5374 |
| Size | 2.5–5 (3.8±0.8) (n = 11) | 3–4.4 (3.6 ± 0.5) (n = 14) | 0.3538 |
| Proportion of residual cavity >2/3 on angiogram | 4 (n = 16) | 18 (n = 32) | 0.0650 |
| Middle position | 13 (n = 17) | 26 (n = 32) | 0.7215 |
| Severe brainstem compression | 11 (n = 17) | 25 (n = 32) | 0.3309 |
| Death | 1 (n = 17) | 6 (n = 28) | 0.2196 |
| GOS 4–5 | 13 (n = 17) | 18 (n = 28) | 0.5134 |
The t-test was used to compare the age and aneurysm size between two groups, and the remaining indexes were compared by the chi-square test.
F: female; GOS: Glasgow outcome scale; M: male; SAH: subarachnoid haemorrhage.
Treatment results
The occurrence of postoperative deterioration, the occurrence of death and the prognosis did not differ significantly between the two groups. The details are summarised in Table 4.
Discussion
Giant aneurysms of the intracranial VA are rare intracranial lesions, and only 53 reported cases were identified in this review.3–47 The intracranial segment of the VA runs along the anterolateral aspect of the medulla oblongata. Aneurysms originating from the intracranial segment of VA are rare and often dissecting aneurysms, 48 and most of them are stable and do not rupture. Some VA aneurysms may progressively grow into giant aneurysms. The growth of giant aneurysms is related to intra-aneurysm thrombosis, repeated bleeding and reconstruction of the aneurysm wall. 49
During the growth of intracranial VA aneurysms, the medulla oblongata would endure progressive compression, leading to the thinning and elongation of the medulla oblongata. In extreme circumstances, the normal medulla oblongata tissue could even not be observed on axial magnetic resonance scans (Figure 3(c)). As this process is slow, some patients may only have slight or even no symptoms due to the compensatory ability of the medulla oblongata. 41 In fact, detailed physical examination may reveal the subtle symptoms of medulla oblongata, cerebellum and cranial nerve compression. The clinical symptoms are not only related to the degree of compression on the medulla oblongata but are also related to their positional relationship. When the aneurysm is located at one side, part of the medulla oblongata may function normally, leading to mild clinical symptoms.
Medulla oblongata compression caused by a giant aneurysm of the intracranial VA may lead to a poor outcome. For example, all the patients treated with conservative management died (Table 1), indicating that the natural history of giant aneurysm in this region is aggressive. 50 Therefore, this finding is consistent with that of previous studies, suggesting that giant dissecting aneurysms of the intracranial VA should be aggressively treated either by open surgery or EVT. However, simple stratification of the results according to the treatments failed to reveal the treatment nature. The main factor that determines the treatment selection is whether it can relieve the mass effect on the medulla oblongata.
Therefore, in this study, the patients with aggressive treatment were divided into two groups: the aneurysm removal group and the aneurysm reserve group. The baseline data did not differ significantly between the two groups, and thus, comparison of the prognosis between the two groups may reflect the differences in treatment efficacy.
Theoretically, it is important to remove the aneurysm, thereby directly relieving the mass effect of the aneurysm on the medulla oblongata. However, it is very difficult to remove giant aneurysms in this region. They often adhere to the lower cranial nerves and medulla oblongata or obscure the surrounding important neurovascular structures. Consequently, exposing aneurysms can cause inadvertent injuries and even severe complications. In addition, the posterior inferior cerebellar artery is often involved, and a bypass is often needed. Hence, surgical procedures aimed at removing the giant aneurysm have a high risk.12,21
Previously, for treating giant VA aneurysms with aneurysm reserving, balloons or clips were usually used to occlude or reduce blood flow to the aneurysm.22,23 In recent years, many EVT materials have been developed, making it possible to trap aneurysms both at the proximal and distal ends. Of note, coils are not routinely used, because they might increase the aneurysm size. In addition to aneurysm trapping, conventional stent-assisted coiling and FDSs have also been used. 46 However, treatments with aneurysm reserving may lead to postprocedural enlargement of the aneurysm due to thrombus formation in the aneurysm cavity, and thus resulting in catastrophic consequences. Postprocedural deterioration after EVT may occur within the first month.
Conclusion
Despite decades of exploration, it is still difficult to manage giant intracranial VA aneurysms, regardless of the treatment selected. In this review, a satisfactory outcome was achieved in 68.9% of the cases. Because of the malignant natural history, aggressive treatment is still advocated.
Footnotes
Conflict of interest: The author(s) declare that they have no conflict of interest.
Ethical approval: Ethics approval is not required for review articles in our institution.
Funding: The author(s) received no financial support for the research, authorship, and/or publication of this article.
Informed consent: Written informed consent was obtained from the patient’s family for publication of the accompanying images.
ORCID iD: Jinlu Yu https://orcid.org/0000-0003-2329-7946
References
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