ABSTRACT
Apoplexy of pituitary tumors is a common occurrence. In addition to commonly known presentations, cerebral infarcts and consequent focal neurologic deficits are a rare presentation. A rare case of pituitary apoplexy with associated subarachnoid bleed and bilateral anterior cerebral artery infarcts is described. Vasospasm leading to cerebral infarcts and consequent focal neurologic deficits as a presentation of pituitary apoplexy needs to be better appreciated.
Keywords: Pituitary apoplexy, cerebral infarcts
Owing to their unusual anatomy, the pituitary tumors are predisposed to autoinfarction or apoplexy.1 Even when one-fifth of all pituitary tumors have pathologic evidence of infarction, only 7% of these neoplasms become symptomatic.2 Apoplectic pituitary tumor may have varied presentations. Sudden onset ophthalmoplegia, with severe headache and altered sensorium, is the usual presentation.
In this report, we describe a rare case of pituitary apoplexy developing fatal bilateral anterior cerebral artery (ACA) territory infarcts, consequent to associated subarachnoid bleed and vasospasm.
CASE REPORT
A 22-year-old female presented in surgical emergency with altered sensorium, without any antecedent history suggestive of sellar pathology. On examination, she had a right-sided ophthalmoplegia, with no pupillary reflex. Left-sided pupil reacted normally to light and had normal left-sided ocular movements. Plain computed tomographic (CT) scan (after 2 hours of the onset of symptoms) showed hyperdense sellar mass, suggesting intralesional bleed, indicative of apoplexy of pituitary adenoma (Fig. 1). Magnetic resonance imaging (MRI) of the sellar region confirmed hemorrhage within the pituitary tumor, with a suprasellar extension (Figs. 2–4). There was no subarachnoid blood in MRI scans. Follow-up CT scan, 12 hours later, showed similar findings, but with the hemorrhagic tumor rupturing through the diaphragm into the subarachnoid space, cisternal spaces, sylvian fissures, and interhemispheric fissure (Fig. 5). As the patient was being investigated for hormonal status and prepared for surgery, she deteriorated to decerebrating status. While on hydrocortisone infusion, intensive resuscitation, and mechanical ventilation, the patient underwent CT scan, which showed symmetrical bilateral frontal hypodensities, indicating infarcts in relation to both the ACAs (Fig. 6). On the fourth postapoplectic day, the patient expired, defying all resuscitative efforts.
Figure 1.
Axial section of plain computed tomographic (CT) scan, showing hyperdense sellar mass.
Figure 2.
Axial section of contrast-enhanced magnetic resonance imaging (MRI) scan, showing varying contrast-enhancing sellar mass.
Figure 3.
Coronal section of contrast-enhanced magnetic resonance imaging (MRI) scan, showing massive suprasellar extension of sellar tumor.
Figure 4.
Sagittal section of T2-weighted scan, showing hemorrhagic sellar lesion, with suprasellar extension.
Figure 5.
Axial section of computed tomographic (CT) scan, showing interhemispheric fissure bleed.
Figure 6.
Axial section of computed tomographic (CT) scan, showing infarcts involving both the frontal lobes, pertaining to both the anterior cerebral arteries.
DISCUSSION
Histologically, 17% of all pituitary tumors have evidence of infarction, but only 7% present symptomatically.1 Various precipitating factors like bromocriptine therapy, estrogen replacement, radiation therapy, head trauma, and anticoagulant therapy have been described, but a majority have no obvious presentation. Pituitary tumors are prone to autoinfarction or apoplexy because of the local vascular anatomy. Asymptomatic vasospasm in pituitary apoplexy or sellar surgery remains an underestimated entity.2 A few of these cases eventually become symptomatic after developing focal cerebral deficits, which may be irreversible consequent to infarct formation. Pathophysiology of vasospasm following pituitary apoplexy remains unknown.3,4 Transdiaphragmatic rupture of the sellar tumor into the subarachnoid space is a probable cause behind vasospasm. Ten cases of infarcts in association with pituitary apoplexy have been described (Table 1). Even when focal cerebral dysfunctions are uncommon, pituitary apoplexy, hemiplegia, or hemiparesis remain the commonest described presentation.1,2,5,6 Such cases have remained on the records for nearly half of a century, but the pathophysiology remains far from understood. Documented cases of cerebral infarction in consequence to pituitary apoplexy are few (Table 1). Cerebral vasospasm and mechanical obstruction of vessels making the circle of willis are the two major causative factors behind vascular insults. Further, mechanical obstruction has been cited by many authors and described on the basis of angiograms. Intratumoral pressures in comparison with arterial pressures have not been documented. Total occlusion seen on angiograms has been presumed to be indicative of mechanical obstruction.7 We feel severe vasospasm will also depict similar vascular occlusion on angiogram.8
Table 1.
Pituitary Apoplexy Leading to Cerebral Infarcts
| Author (Year) | Presentation | Radiology | Infarct Site | Management | Outcome | Pathophysiology |
|---|---|---|---|---|---|---|
| ACA, anterior cerebral artery; CT, computed tomography; DSA, digital subtraction angiography; ICA, internal carotid artery; MCA, middle cerebral artery; MRI, magnetic resonance imaging; SAH, subarachnoid hemorrhage. | ||||||
| Schnitker and Lehnert (1952)1 | Focal hemispheric signs (R)MCA, (R)ACA | X-rays | Gliosis (autopsy) | Nil | Death | Compression (autopsy) |
| Rosenbaum et al (1977)7 | (L)Hemiplegia | Angio – (R)ICA block | Bleed into infarction | Surgery | Death | Compression |
| Cardoso and Peterson (1983)3 | Headache, drowsiness | X-rays – enlarged sella | Multiple cortical | Surgery | Death | Vasospasm |
| (R)III Nerve palsy | Angio – laterally displaced | (Autopsy – SAH) | ||||
| (R)Homo-hemianopia | Both ICAs | |||||
| Majchrzak et al (1983)4 | Headache, vision loss | CT scan – tumor bleed, SAH | (R)ACA | Surgery | Partial recovery | Compression |
| Comatose, disoriented | Angio – (R)ACA block | |||||
| (L)Hemiparesis | ||||||
| Clark et al (1987)9 | Headache, photophobia | CT scan – hemorrhagic mass | (L)ACA | Radiotherapy | Favorable | Compression |
| Dysphasia, (R)hemiplegia | Angio – (L)ICA block | (L)MCA | Hormonal | |||
| (R)Homo-hemianopia | (R)ICA narrow | Head of caudate | ||||
| Pozzati et al (1987)8 | Headache, drowsiness | CT scan – hemorrhagic mass | (R)Parietal | Hormonal | Partial recovery | Vasospasm |
| (L)III, VI Palsy | X-rays – enlarged sella | |||||
| Lath and Rajshekhar (2001)10 | Headache, vomiting | CT scan – hemorrhagic mass | (R)ICA | Surgery | Death | Compression |
| Vision loss, altered sensorium | ||||||
| (L)Hemiplegia | ||||||
| Catenoix et al (2002)6 | (L)III Nerve palsy | CT scan | (R)Lenticulostriate | |||
| Bhansali et al (2005)5 | Frontal lobe syndrome | CT scan – tumor bleed, SAH | (L)ACA territory, acute preexisting stenosis | Conservative elective surgery | Partial recovery | Vasospasm |
| Vision loss, headache | MRI – pituitary apoplexy | |||||
| DSA – B/L A1 elevation, spastic | ||||||
| Lomban et al (2006)2 | Confused, neck rigidity | CT scan – isodense sellar mass | (L)MCA | Nil | Partial recovery | Compression |
| (L)III Nerve palsy | MRI – pituitary apoplexy | Preexisting stenosis | ||||
| (R)Hemiplegia | DSA – (L)ICA compressed | |||||
| (L) Cervical ICA stenosis | ||||||
| Present case | Unconscious, (R)III nerve palsy | CT scan – tumor bleed, SAH | Bilateral ACA | Nil | Death | Vasospasm |
| Decerebrating posture | MRI – tumor bleed | |||||
It is either ACA or internal carotid artery (ICA) that gets compromised on account of vasospasm after pituitary apoplexy.2,5,6,9 We propose that it should always be ICA or ACA that gets mechanically obstructed on account of sellar tumor, whereas middle cerebral artery (MCA) not being a part of closed loop escapes mechanical brunt.10 However, partial occlusion of ICA mechanically may cause MCA territory ischemia, especially when MCA is the end artery and ACA has dual supply.
Bilateral ACA infarcts have not been reported earlier as a consequence to pituitary apoplexy. The present case remains unique as there was no focal or side preponderance of symptomatology, thereby delaying investigations and management. Repeat scans showed symmetrical bilateral ACA infarcts. In the present case, ongoing deterioration defied all resuscitative efforts. Surgical intervention for such a case remains debatable as hemorrhage into infarct always remains a possibility.
CONCLUSION
Spasm of vasculature of circle of willis remains a rare but possible complication after apoplexy of pituitary tumor. Such a vasospasm may prove fatal, if infarcts of large cerebral territories ensue. Awareness of such a catastrophe may bring forth more cases of pituitary apoplexy, which end fatally unnoticed.
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