Intraoperative rupture of blood blister-like aneurysm: a case report and review of literature
© The Author(s) 2015
Received: 28 April 2015
Accepted: 7 September 2015
Published: 18 November 2015
Blood blister-like aneurysms (BBLAs) are aneurysms from the non-branching sites of the internal carotid artery (ICA). Though rare lesions, they pose a high risk of intraoperative aneurysmal rupture. Definite treatment of these types of aneurysms has been debatable, but surgical approach is the ultimate rescue treatment. Microsuture of the intraoperative ruptured BBLA has been reported scarcely in literature, but no review of these cases has ever been reported. We here present our experience of a case of BBLA intraoperative rupture requiring microsuture of the ICA and conduct meticulous review of all similar cases.
First described by Sundt and Murphey , blood blister-like aneurysms (BBLAs) or frog-eye aneurysms are frequently seen at the non-branching sites of the supraclinoid internal carotid artery. Higher incidences of BBLAs have been reported among women, on the right side, in younger patients with subarachnoid hemorrhage (SAH), associated hypertension and arteriosclerosis, or dissection of the internal carotid artery (ICA) [2, 3]. Their bulging appearance with a broad base ostium indicates a “sick” carotid wall segment differing from the overall group of saccular aneurysms . BBLAs of the ICA account for about 0.3–1 % of intracranial aneurysms or 0.9–6.5 % of ICA aneurysms .
BBLAs are thin and fragile hemispherical bulges and can progress into a saccular shape. They are at high risk of recurrent hemorrhage no matter their shape or the treatment modality chosen . These lesions account for 0.5–2.0 % of ruptured intracranial aneurysms and prelude unusually high morbidity and mortality rates compared with typical ruptured saccular aneurysms of the ICA . A high degree of suspicion for this lesion is recommended in SAH of unknown origin, particularly when the bleeding pattern is more typical for an ICA source . However, similar lesions can be found at lower frequencies in other areas of the basal cerebral circulation (e.g., in the basilar artery) [8, 9]. Because these lesions are often not true aneurysms but rather pseudoaneurysms with a high incidence of thin and fragile walls and lack a definable neck, they are extremely difficult to treat surgically. This difficulty is reflected by a high incidence of intraoperative rupture and postoperative hemorrhage, resulting in significant disability and mortality from surgical repair .
Although endovascular techniques have revolutionized the treatment of aneurysms in general, these blister-like aneurysms present a challenge because of their small size, wide necks, and fragile nature. Treating these aneurysms surgically is usually not a problem, but surgeons are often faced with a hole in the carotid when the aneurysm disintegrates during dissection . BBLAs are uncommon and their management is subsequently based on what little has been published. The angiographic diagnosis of BBLAs may be difficult because they often represent tiny lesions that can be overlooked, mistaken for artifacts or focal atheromatous irregularity, or missed completely due to overlap of vessel curvature . Repeat angiography often documents luminal changes or growth of these lesions.
In many patients with this particular type of aneurysm, soon after subarachnoid hemorrhage, the initial angiogram shows only a small bulge, which may progress to a saccular appearance within a few weeks . Exact pathogenesis of BBLA is unknown, but several autopsy studies suggest that BBLA is not a true saccular aneurysm but a specific type of pseudoaneurysm and that a small ulceration penetrating the internal elastic lamina and the resulting wall defect is crucial in BBLA development. Others suggest that BBLA is specifically a type 4 dissection with focal defect of the internal elastic lamina mimicking a penetrating ulceration without a coexisting intramural hematoma, a condition similar to BBLA .
Sundth and Murphey were the first to describe the lesions at non-branching sites on internal carotid artery (ICA). Preoperative diagnosis of BBLA is very challenging, and angiographic findings do not always correlate with the intraoperative features. BBLA might be one of the causes of angiogram-negative subarachnoid hemorrhage (SAH), particularly when the distribution of blood on computed tomography (CT) is non-perimesencephalic . So, in many cases of SAH, the first angiographic findings of CTA or even digital subtraction angiography (DSA) are negative or suspicious and repeated exams must be done . Short-term angiography follow-up (10 days) may show a dramatic change in the conformation of these lesions with reasons attributed to the profound weakness of the aneurysmal wall . Recently, the use of CTA as a first-line diagnostic tool has been advocated on the basis of comparable sensitivity and specificity to DSA even for small aneurysms . Blood blister-like aneurysms are most correctly diagnosed when proper visualization of the aneurysm is obtained intraoperatively.
Review of reported intraoperative aneurysm microsuture cases
Stenosis of artery
Vashu et al. 
WFNS Grade II
Right supraclinoid ICA aneurysm
8/0 Prolene 4 stitches—20-min clipping + barbiturate
No neurological deficit—no recurrence at 11 months follow-up
Vashu et al. 
WFNS Grade I
Right supraclinoid ICA aneurysm
7/0 Prolene 4 stitches—23-min clipping + another 28-min clipping + barbiturate
GOS-5 at 5 months follow-up, left side limb weakness 4/5
Yanaka et al. 
Right supraclinoid ICA aneurysm
8/0 nylon 2 stitches—Surgicel + fibrin glue + encircling clip
Returned to normal activities at 3 months follow-up
Lee et al. 
Left dorsolateral ICA
Suture—temporary clip time 36 min
Otani et al. 
Clip on wrapping with suture
Right superior to medial wall ICA aneurysm
mRs 3 at 3 months follow-up
Joo et al. 
Suture tear with 8/0 nylon + wrapping + clipping—20 min
Discrete hemiparesis (4/5)
Joo et al. 
Right supraclinoid ICA aneurysm
Suture with 8/0 nylon + wrapping + clipping
Right supraclinoid ICA aneurysm
8/0 Prolene 3 sutures—22-min clipping
mRS 3 at 8 months follow-up
Kojra et al.  concluded that stent-assisted coil embolization is not a treatment of choice for BBLA but only an option. Parker at al.  concluded that endovascular coiling of BBLA cannot be recommended as a treatment strategy due to high rates of procedural rupture (75 %), aneurysmal regrowth, and rebleeding. Maling et al.  further discouraged endovascular treatment as 11 out of 14 of their patients were judged unsuitable in BBLA treatment. There are no randomized controlled trials and meta-analyses to compare the endovascular and surgical treatments of BBLA, therefore making it difficult to encourage any particular treatment plan. Moreover, to date, all the present cases in literature used microsuturing as the rescue treatment of BBLA.
Various surgical strategies have been developed to fight against BBLA, including parallel clip placement along the ICA wall producing mild stenosis, clipping on wrapping methods, trapping with or without extracranial-intracranial bypass surgery, and suturing methods. Most surgical case series reported that BBLA had a worse outcome than usual ruptured saccular aneurysms. The BBLA wall is fragile; direct clipping of the aneurysm neck is dangerous and likely to cause premature rupture during surgical manipulation .
From our review of literature, as illustrated in Table 1, we noticed that suturing was used as a rescue plan when BBLAs ruptured intraoperatively. Postoperative angiography showed no vessel stenosis in four patients but two patients had vessel spasm. DSA was the preferred follow-up tool to re-evaluate patient after BBLA rupture and suture. Gonzalez  carried a systemic review of surgically treated blood blister-like aneurysms, and out of 165 patients with clipping, 39 (23 %) needed rescue treatment. Out of these 39 patients, 10 (25.6 %) patients received arterial microsuture. This shows that arterial microsuture is a fundamental technique for neurovascular surgeons.
Suturing has been infamous because of the lack of experience, fragile muscle wall, bleeding, prolonged ischemic time, and uncertain outcomes. Suturing methods and surgical handlings can be difficult because of the limited space and time. Yanaka et al.  agreed that aneurysmal rupture requiring arterial suture does not occur often, but a proper strategy to address such problems can make a difference in bringing a good neurological outcome out of an intraoperative catastrophe. They suggest that deep field suturing required long microsuture forceps and long microneedle holder, and these instruments must be prepared and available in the operating room.
However, there are certain limitations to this technique, such as excessive vessel laceration, making suturing cause too much stenosis and extensive circumferential carotid disease. Joo et al. attempted suturing in a patient after several failed attempts of failed clipping, but the follow-up DSA demonstrated internal carotid artery occlusion . In such cases, parent vessel occlusion and bypass can be considered.
In conclusion, BBLAs are rare but very unpredictable lesions. Proper teamwork between the neurosurgeon and interventional neuroradiologist can improve efficacy of treatment. Angiogram-negative subarachnoid hemorrhage patients must be suspected of BBLA and 3D CTA (in preference to 2D DSA) or 3D DSA (in preference to 3D CTA) can be repeated about 10 days after initial SAH. Intraoperative rupture of BBLA is very common, and we here report that arterial microsuture can be a good reliable rescue treatment. Also, in many medical centers as ours, there is no wide variety of stents and encircling clips are not available; suturing is the very practical in these situations as a lifesaving procedure. We therefore strongly suggest that any medical institution providing surgical treatment option for aneurysmal treatment should have team members well trained in arterial microsuturing techniques and should have readily available long microsuture forceps and long microsuture needle holders.
Written informed consent was obtained from the patient for the publication of this report and any accompanying images
We are grateful to Ms. Purvarshi Gowreesunkur for her help in language editing and Ms. Lu Gui Hua for her support during this research.
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