Introduction
Spontaneous intracerebral hemorrhage (ICH) accounts for 7 to 20 % of all strokes, which is more than twice as common as subarachnoid hemorrhage (SAH).1)3) Spontaneous ICH mainly results from hypertension, amyloid angiopathy, and vascular malformations, and aneurysmal rupture can be associated with ICH adjacent to the ruptured aneurysms in 4 to 34 % of the patients.13)19) There have been reports on aneurysmal SAH and ICH unrelated to aneurysmal rupture. Most of them were postoperative ICH related to neurosurgical procedures and perioperative arterial hypertension.4)8) Rarely cases of simultaneous SAH and remote ICH, which are not related to neurosurgical procedures, have been reported.7)10)11)14)16)17)22)
However, there has been no report of bleeding of previously treated aneurysm and simultaneous hypertensive ICH. We report on a case of concurrent hypertensive ICH and aneurysmal SAH with review of the literature.
Case Report
A 64-year-old lady presented with persistent right hemiparesis which suddenly occurred during cooking at home. She did not experience headache at all. She has been taking antihypertensive drug and aspirin. She had undergone clipping operation for an unruptured aneurysm at the distal
anterior cerebral artery (ACA) 15 months before. She had two intracranial aneurysms (Fig. 1), and a 9.8 × 7.2 mm sized wide-neck aneurysm at the bifurcation of unpaired A2 trunk had been clipped and the other one of 2.2 × 2.7 mm in size at the A3 segment of right distal ACA has been observed. At operation, the clipping had not been complete due to the atherosclerotic change at the broad neck of the aneurysm, with the remaining portion wrapped with muslin
gauze. After discharge, she was lost to follow-up. At this time, she was alert and showed right
hemihypesthesia, right hemiparesis (Grade 3/5 for upper extremity and Grade 1/5 for lower extremity), and extensor toe sign on the right foot. Initial blood pressure was 145/90 mmHg and all laboratory tests were within normal limits. Brain computed tomographic (CT) scan showed focal SAH around the aneurysm clip at the anterior interhemispheric fissure and acute ICH of 2 cm in diameter at the left thalamus, extending to the posterior limb of internal capsule laterally and corona radiata superiorly, with intraventricular blood at the left occipital horn of the lateral ventricle (Fig. 2). Cerebral angiography revealed the 4.5 × 2.8 mm sized residual aneurysm sac at the bifurcation of the single A2 trunk, in which the clip was displaced at the posterior aspect
of the enlarged aneurysm, as well as the other tiny aneurysm at the A3 segment with no interval change (Fig. 3). The large one was thought to have ruptured in light of the location of
bleeding. Subsequently, the aneurysm was occluded with stent-assisted coil embolization without complication. The left thalamic ICH was treated conservatively. As the hematoma resolved, her motor weakness gradually improved. She was transferred for rehabilitation a week later
and has become able to walk with a cane over three weeks.
Discussion
To our knowledge, this is the first report demonstrating rupture of previously clipped aneurysm with concurrent hypertensive ICH. Our patient showed SAH from the previously treated azygos ACA aneurysm and ICH in the left thalamus. Because the location of ICH was not related to the ACA aneurysm as shown in Fig. 2, the ICH and SAH in our case were thought to be separate lesions. During the previous surgery, the sclerotic aneurysmal neck hindered the complete
exclusion of the aneurysm from the circulation with clipping, and thus we performed wrapping around the aneurysm neck with muslin gauze at that time. Atherosclerosis is known as a cause of the formation and rupture of intracranial aneurysms because the hemorrhage within atheromatous plaque can lead to the wall dissection.6)15) Thus, atherosclerosis itself and increasing
hemodynamic stress on the wall after incomplete clipping on atherosclerotic neck in our case might have elicited the regrowth and rupture of the A2 aneurysm. On the other hand, wrapping has been used for the unclippable aneurysms, expecting the protective effect against growth
and rebleeding. The rebleeding rate of wrapped aneurysms after long-term follow-up is known to range from 0% to 17%.2) In our case, wrapping failed to prevent bleeding from the incompletely clipped aneurysm. However, the wrapping played a role in limiting the extent of hemorrhage, which enabled us to confirm that the aneurysm, not the other distal one, had ruptured.
There have been several reports on simultaneous aneurysmal SAH and remote ICH.7)10)11)14)16)17)22) The reported cases and our own case are summarized in Table 1. Notably, angiographic demonstration of a pseudoaneurysm of the lateral lenticulostriate artery as well as the left MCA
bifurcation aneurysm provided a definite evidence of simultaneous aneurysmal SAH and ICH in a case.16)
Which is the first event, ICH or SAH ?
The pathogenesis of simultaneous SAH and ICH is not clear. The two lesions might have occurred at the same time coincidentally, or one of them might have preceded the other
since most of the patients showed symptom development in a sequential fashion, such as non-localizing symptoms (headache or altered mentality) followed by localizing ones (hemiparesis or sensory change), or vice versa. It was hard to find a time sequence of SAH and ICH in our case who presented with neurological deficits related to ICH without typical headache. We supposed that ICH would be the initial event, which could be followed by rupture of the
incompletely treated aneurysm for the following reasons.
First, the incidence of ICH is usually higher than that of SAH.1)12) Second, atypical primary symptoms, such as dizziness, visual or speech disturbance, or personality change other than headache are rare in patients with SAH.5)
Embarrassing situation of sudden neurological deficits might have masked the symptoms related to SAH in our case.
In consideration of the sequences of symptom development, we presumed that SAH might have been be followed by ICH in five patients (group 1) and ICH might have provoked SAH in the other three, including our own case (group 2) (see Table 1). In group 1, headache and/or deteriorated mentality were initial presentation, and then focal neurologic deficits ensued; with a reverse order in group 2. Three of five cases in group 1 and all the three patients in group 2 had preexisting arterial hypertension.
We speculate that chronic hypertension has already influenced on the patients cerebral vasculature, and an instant increase of blood pressure could result in SAH or ICH initially.9)18) The hemorrhage would produce a high intracranial pressure, causing increase in systemic blood
pressure (Cushing phenomenon). Finally, the raised blood pressure would provoke another hemorrhage.18)20)21) Although the relevant pathogenesis does not seem to be different from the others, our patient is unique in that she developed rupture of previously clipped aneurysm in association with hypertensive thalamic ICH.
Which determines the prognosis, ICH or SAH ?
Subarachnoid hemorrhage is a life-threatening disease and ICH from aneurysm rupture has a negative influence on the patients clinical course and outcome.13)19) However, whether this can be applied in cases of aneurysmal SAH with simultaneous ICH is uncertain. We reviewed such cases, as summarized in Table 1. The estimated mean size of the concurrent ICHs was 1.8 cm × 2.6 cm (range, 0.2 cm × 0.2 cm to 4.7 cm × 6.1 cm), and only one patient with a large
ICH of 4.7 cm × 6.1 cm underwent hematoma evacuation surgery. The Hunt-Hess grades of the patients were I in 1 patient, II in 3, III in 3, and IV in 1. Aneurysm treatments (direct aneurysm neck clipping in 6 patients and endovascular coil embolization in 2; emergent acute
treatment in 5 and delayed treatment in 3) were performed in all patients. In terms of clinical outcome, ICH contributed to the patients morbidity such as hemiparesis (n=5) and diplopia (n=1). There was no sequela associated with the aneurysm treatment. As a result, ICH, rather than SAH, mainly affected the prognosis in this specific group of patients although number of the cases is small.
Conclusion
Simultaneous aneurysmal SAH and remote ICH is a very rare situation. To our knowledge, only 8 cases including our case have been reported and our case is the first one demonstrating the rupture of previously clipped aneurysm with concurrent hypertensive ICH. Hypertensive crisis
seems to be the most probable cause.
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