Introduction
Epistaxis following transnasal transsphenoidal (TNTS) removal of pituitary adenoma
can be massive and life-threatening.[1 ] The intracranial source of bleeding is usually the intracavernous segment of the
internal carotid artery (ICA) or adjacent branches. Injury to the cavernous ICA can
lead to pseudoaneurysm (PA) or fistula formation.[2 ] Management of PA is different from saccular aneurysm. The timely diagnosis and adequate
management can restore vessel integrity and prevent associated morbidity.[3 ] We are reporting a case of growth hormone-secreting pituitary adenoma in a young
girl who underwent microscopic TNTS excision of the tumor and had presented with massive
epistaxis. Pseudoaneurysm of the cavernous ICA was not seen on computed tomography
(CT) angiography initially and later diagnosed on digital subtraction angiography
(DSA). The attempted management of PA with coils without stent could not stop aneurysm
growth. The management of such complicated PAs is discussed, and a literature review
is done regarding epistaxis in growth hormone (GH) secreting adenoma.
Case Report
Twenty-six-year old young unmarried female was diagnosed with growth hormone-secreting
pituitary macroadenoma for the last 3 years with features of acromegaly. Magnetic
resonance imaging revealed a sellar mass of size 1.5 × 1.6 × 1.7 cm. The GH level
was 27.8 ng/mL. The near-total tumor excision was done using a microscopic TNTS approach.
Tumor was greyish pink and soft with rubbery inconsistency. Intraoperatively, there
was moderate bleeding and could be controlled without much effort. Seller floor was
reconstructed with fat, cartilage, and bone. The next 48 hours were uneventful until
the removal of the nasal pack, which resulted in torrential hemorrhage. The nasal
cavity was repacked. The patient was stabilized hemodynamically, and CT angiography
brain was done, which did not reveal any vascular injury ([Fig. 1B ]). The patient was reoperated to remove the nasal pack, and the tumor bed was inspected
for any bleeding point. There was no active bleed. The patient was discharged after
2 days. After 2 weeks of surgery, she had repeated minor epistaxis at home, followed
by one episode of massive epistaxis. The patient was readmitted, and nasal gauze packing
was done. The GH level was 7.8 ng/mL. Digital subtraction cerebral angiography was
performed, which revealed a PA arising from the cavernous segment of the right ICA
measuring 0.75 × 0.45 cm with no cross flow between two ICAs ([Fig. 2A ]). Two ev3 (Covidien) three-dimensional coils 6 mm × 10 cm and 3 mm × 6 cm coils
were placed within the aneurysmal sac to occlude the neck. A postoperative angiogram
showed complete obliteration of the aneurysmal sac ([Fig. 2B ]). The patient was discharged with no neurological deficits. Her features of acromegaly
were resolving. Examining angiogram 6 weeks thereafter showed refilling of aneurysm
and coil mass compaction ([Fig. 3A ]). The aneurysm was recoiled, and since there was no residual filling of the aneurysm
([Fig. 3B ]), no stent across the aneurysm neck was placed. However, checking DSA 4 weeks thereafter
again revealed coil mass compaction with recurrence of the aneurysm. Ultimately stent-assisted
(Solitaire, Covidien) coiling of the aneurysm was performed ([Fig. 4A ]). At one-year follow-up the patient was asymptomatic with stable obliteration on
MRI and MRA ([Fig. 4B ]).
Fig. 1 (A ) Saggital view of Gadolinium-enhanced MRI showing contrast-enhanced pituitary macroadenoma.
(B ) Immediate Postoperative CT angiography of the brain after the first episode of epistaxis
revealed no anatomic abnormality.
Fig. 2 (A ) First digital subtraction angiography detected butterfly pseudoaneurysm of the cavernous
segment of ICA (0.75 × 0.45 cm). (B ) Post coiling DSA of aneurysm showed complete filling of sac.
Fig. 3 (A ) DSA after six weeks showed re-growth of pseudoaneurysm possible dissection of cavernous
ICA. (B ) Post-coiling DSA showing complete obliteration of aneurysm sac with reserved distal
flow.
Fig. 4 (A ) Digital subtraction angiogram showing stent-assisted coiling of ICA pseudoaneurysm
with completely obliterated sac. (B ) MRI (Axial T1 weighted) showing thrombosed stable aneurysm.
Discussion
The incidence of vascular injury following TNTS is 0.2 to 1.2%.[2 ]
[3 ] Massive epistaxis can be due to cavernous ICA injury, which may result in a carotid-cavernous
fistula or PA.[4 ]
[5 ]
[6 ] Injury to the ICA during transsphenoidal resection of pituitary tumors typically
occurs during aggressive dissection of macroadenomas especially invading the cavernous
sinus and encasing the ICA.[6 ]
[7 ] Carotid injuries are more likely to occur in patients with previous transsphenoidal
surgery or radiation therapy, invasive adenomas, endoscopic surgery, anatomical variations
such as bulging of ICA into the sphenoid sinus or covered only by the dura mater of
the cavernous sinus and the mucosa of the sphenoid sinus without bony covering.[8 ]
[9 ] In the present case, the tumor was not very large, and the probable cause of injury
would have been a deviation from midline reaching more toward the right side of the
sella.
The diagnosis could not be made on a CT angiogram, the howsoever good quality it is
as in the present case. Perhaps, the vessel at the focal site was in spasm or the
CT angiogram was done with nasal pack in situ, which must have covered the injury
site of ICA. However, the dissection kept growing, which led to aneurysm formation,
and was diagnosed with DSA. Therefore, even if the CT angiogram is negative, DSA should
be done, and if first DSA is negative, repeat DSA should be done after 2 to 4 weeks,
and DSA/CTA should be repeated following nasal pack removal.[1 ]
[3 ]
[10 ]
The size of iatrogenic intimal injury is most important to produce PA or dissecting
aneurysm. If the injury is less than 2 mm, then most of the time, it heals without
morphological changes. However, if the size of arterial injury is around 4 mm, then
it has a high probability to produce an aneurysm, and if the size is more than 6 mm,
then it would cause complete stenosis of the injured vessel.[11 ] In the present case as well the neck of the PA was approximately 4 mm, and the rent
in the intima is approximate of the same size would have resulted in the formation
of PA. There have been many reports of vascular injury in pituitary adenoma, but only
eight cases of iatrogenic carotid PA in GH secreting pituitary macroadenoma with acromegaly
following transsphenoidal surgery were identified. Acromegalic patients are at a higher
risk of sustaining this complication because of the distortion of the nasal-sinus
anatomy and the tendency to have tortuous and ectatic carotid arteries, sometimes
protruding into the sella.
Treatment options include carotid ligation to endovascular intervention. The endovascular
procedure includes parent artery occlusion, coil embolization, stent-assisted coiling,
covered stent placement, and onyx embolization.[11 ]
[12 ]
[13 ]
[14 ] Endovascular intervention is the most preferred approach. The management of PA differs
from saccular nondissecting aneurysm. Since the repair of the intima is essential
in dissecting aneurysms, only coiling may not work. The wall can heal by covering
it with a stent. The risk of long-term anticoagulation to prevent stroke is a major
concern. Our assumption of putting the coils and trying to achieve a good result was
wrong as the treatment of dissecting aneurysm is the reconstruction of vessel. The
treatment got stable only when a stent was placed across the neck of the aneurysm,
which helped in healing the wall and prevented the subsequent flow of blood in the
sac.
Conclusion
Delayed massive epistaxis is rare but is a serious complication of transsphenoidal
surgery. Vessel wall healing is as important as occluding the pseudoaneurysm.
Studies
Age/Sex
Diagnosis
Time of diagnosis
Treatment
Outcome
Wilson et al[15 ]
NR
Delayed epistaxis
NR
Carotid ligation
NR
Cabezudo et al[16 ]
41/F
Delayed epistaxis
1 mo
Gradual closure of carotid with Selverstone clamp over 7 d
Good
Reddy et all[17 ]
56/F
Angiogram
6 wk
The surgical clip of supraclinoid ICA and ligation of extracranial ICA
Good
Ahuja et al[18 ]
52/F
Follow-up angiogram
9 d
Endovascular occlusion of ICA
Temporary hemiparesis
Raymond et all[19 ]
28/F
Angiogram
10 d
Surgical packing
Good
Cappabianca et al[6 ]
22/F
Angiogram
NR
Coil embolization of the aneurysm
NR
De Souza et al[5 ]
38/F
Postoperative MRI
NR
Endovascular cover stent
Good
Cinar et al[9 ]
69/M
Angiogram
9 d
Endovascular Parent artery occlusion
Expired on 12th day
Current case (2015)
26/F
Postoperative DSA
2 wk
Endovascular stent-assisted coiling
Good
Abbreviations: DSA, digital subtraction angiography; MRI, magnetic resonance imaging;
NR, no response.
