: A Case Report
Abstract
Pseudoaneurysms of the superficial temporal artery are an uncommon vascular lesion of the external carotid system and most often the result of blunt head trauma. The frequency of pseudoaneurysms of the superficial temporal artery developing after craniotomy is exceedingly low and only a few cases have been reported. We present a case of pseudoaneurysm of this type in a 45-year-old male who underwent craniotomy for excision of meningioma. One month postoperatively, the craniotomy flap exhibited an enormous diffuse pulsate swelling. The suspected diagnosis of pseudoaneurysm arising from superficial temporal artery was confirmed on angiography. Surgical excision was done and no recurrences of the tumor or aneurysm were noted on subsequent follow up.
A pseudoaneurysm is a dilation of an artery that includes a defect in one or more layers of arterial wall (1). Pseudoaneurysm of the superficial temporal artery is a rare (2, 3) clinical entity that typically occurs after a blunt trauma (1, 4–6) to the frontotemporal region (5). There are reports of such traumatic pseudoaneurysms of superficial temporal artery (1, 2, 6–9), but within the context of craniotomy as a cause of pseudoaneurysm only few cases (2) have been described throughout the world. We present a case of pseudoaneurysm of the superficial temporal artery emerging after craniotomy for excision of meningioma. To our knowledge, this is the first case of superficial temporal artery pseudoaneurysm developing after craniotomy (for meningioma excision) from Pakistan. We have discussed the etiology, clinical presentation, diagnosis, and treatment with reference to previously reported cases.
Case Report
A 45-year-old male presented with a complaint of a headache lasting 3 months. On fundoscopy, he had papilledema. A CT Scan Brain revealed a large space occupying lesion arising from the sphenoid ridge along with hyperostosis. Brain MRI with contrast showed a homogenously enhancing lesion with significant mass effect. Radiological findings were consistent with a meningioma.
After angioembolization, a frontotemporal craniotomy was done and the lesion was excised completely. The patient had a smooth recovery in post-operative phase and he was discharged 10 days after surgery.
During his follow up 1 month after surgery, he presented with a diffuse swelling of craniotomy flap, although the edges of the wound were healthy. The swelling was diffuse homogenous, normal in color, pulsatile in nature and indolent (Figure (Figure1).1). With the suspicion of pseudoaneurysm, carotid angiography was performed which revealed a large pseudoaneurysm arising from a branch of the superficial temporal artery. He had a redo surgery for excision of pseudoaneurysm (Figures (Figures22 and and3).3). At the base of pseudoaneurysm, there was a free end of superficial temporal artery, which was coagulated and ligated. Subsequent follow up did not reveal any recurrence of the tumor or pseudoaneurysm.
Discussion
The first case of superficial temporal artery pseudoaneurysm was described by Thomas Bartholin (1, 2, 4, 6) in 1740 and was the result of blunt trauma (2). The most common etiology of superficial temporal artery pseudoaneurysm is blunt trauma (1, 4–6, 10) accounting for 75% (4) to 95% (10) of cases; penetrating injury or iatrogenic cases were responsible for the rest of cases (10). STA pseudoaneurysms due to iatrogenic injury have been reported to occur after cyst removal, temporomandibular joint excision arthroplasty, punch hair grafting, and craniotomies (5). However, craniotomy as a cause of pseudoaneurysm of STA is extremely rare (6–8). Of the few craniotomies reported as a cause in literature, the majority were done for aneurysmal clipping (2). However, in this report we present a case of craniotomy for meningioma excision leading to STA pseudoaneurysms. Since 1985, when Rousseaux et al reported a case of an STA pseudoaneurysm that developed following a craniotomy for frontal lobe meningioma resection (9), no such cases have been reported in literature.
The anterior branch of the STA is most vulnerable to injury from blunt trauma due to its superficial course and close proximity to the underlying bony structures (1, 4, 10). However, it was affected as a result of craniotomy. Multiple aneurysms are possible but uncommon (1, 5). True aneurysms of the STA are also reported but are extremely rare (1, 2, 5). Regarding the patho physiologic mechanism of pseudoaneurysm formation, there is a consensus in the literature on occurrence of some kind of penetrating trauma during the surgery (2) by skin incision, a pin head-holder, thread removal and subcutaneous drains (4). In our case, we believe a needle injury to the STA during subcutaneous closure resulting in slow bleeding and pseudoaneurysm formation.
The time period from craniotomy to pseudoaneurysm in the literature varied between 4 days and 3 months (7), and in our case it was 1 month post-operatively. The typical history involves trauma or surgery to the temporal region (5, 7) and subsequent development of a pulsatile, indolent or expanding swelling which may be associated with headache (4, 7). Other neurologic symptoms are not always present but may include facial pain, dizziness, ear discomfort, or facial droop due to cranial nerve VII compression (1). In some cases, pulsation may be absent if there is complete thrombosis of the aneurysmal sac (10). On examination, a compressible, tender, pulsatile mass over the superior temporal line is usually apparent and occasionally bruits may be auscultated (1, 5, 7).
Differential diagnoses for STA pseudoaneurysm include vascular tumor, arteriovenous fistula, meningeal artery aneurysm with bony erosion, subcutaneous lipoma, abscess, and localized hematoma (1–5, 7).
Many authors believe that superficial temporal artery pseudoaneurysm should be diagnosed clinically from history and physical examinations (1, 2, 4). However, diagnostic modalities involve invasive and non-invasive tests (2). For most cases, duplex ultrasound is currently the imaging modality of choice (1, 10), since it can provide detailed information about the vascular anatomy without incurring the risks of invasive methods or radiation. CT Scans with contrast, CT Angiography and Digital Substraction Angiography have been reported in the literature (5).
Indications for surgery are cosmetic, treat headache, and avoid pain and hemorrhage (2, 4). Surgical management under local anesthetic is appropriate and may include ligation and excision of the aneurysmal arterial segment or primary repair when the arterial injury is easily amenable to closure with sutures (1). Other treatment options for STA aneurysm have been reported, including endovascular obliteration, percutaneous endo-obliteration using coils, glue, or ethylene vinyl alcohol copolymer (3). Percutaneous thrombin injection as a treatment has been described with controversies (6). Recurrence rate after surgery have been documented as rare (3). In our patient surgical excision of pseudoaneurysm was carried out with no recurrence post-surgery.
Conclusion
This case is intended to caution surgeons dealing with craniotomy of this rare complication. A post-craniotomy temporal mass should be considered a STA pseudoaneurysm until proven otherwise, and needle decompression should not be attempted. Surgical excision of the pseudoaneurysm of superficial temporal artery results in a good clinical outcome.
Informed Consent
The patient to which the case study refers provided oral and written consent to be featured in this article.
Conflict of Interest Statement
The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.
References
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Giant cell arteritis (GCA) also known as temporal arteritis is an autoimmune disease. Autoimmune diseases are conditions in which immune cells, that normally help fight infections, are misdirected to attack your own tissues. In the case of GCA, these immune cells are involved in an inflammatory reaction in large arteries of the body, mainly branches of the aorta and often, the aorta itself. The inflammation damages the affected blood vessels. It has been estimated that 228,000 Americans have GCA. At present the exact cause of this condition is unknown.
GCA is a form of vasculitis. Vasculitis is defined as a condition in which blood vessels, particular arteries, become inflamed. In GCA, the aorta and its branches are usually affected. The inflammation in GCA can cause swelling of the blood vessel wall and narrowing of the blood vessel lumen causing decreased blood supply to the neighboring tissues. The blood vessel may also become thrombosed causing severe ischemia or necrosis of tissues ordinarily supplied by the blood vessel.
How common is GCA?
GCA is the most common form of vasculitis in patients over the age of 50 years. Data from population-based studies estimate that 1 in 5,000 people over the age of 50 years are affected by GCA each year. The prevalence of GCA is estimated at 278 per 100,000 people over the age of 50 years.
Who gets GCA?
Age, gender, and ethnicity are all risk factors for GCA. GCA affects people over the age of 50 years and the risk of GCA increases the older one gets. For reasons that are not clear, GCA is 2 to 3 times more common in women than in men. This condition is most commonly seen in people of Northern European descent and is rare in other ethnic groups like Asians and African Americans.
Is GCA different from Polymyalgia Rheumatica (PMR)?
Yes, GCA is different from polymyalgia rheumatic. Polymyalgia rheumatic is a condition in which people experience severe stiffness and pain in the neck, shoulders, and hips. Symptoms are usually worse in the morning and improve later in the day. Blood tests show evidence of inflammation. There is a close association between polymyalgia rheumatic and GCA. While approximately half of all patients with GCA also have symptoms of polymyalgia rheumatic, only 16-21% of patients with polymyalgia rheumatic develop GCA.
Symptoms
Most patients with GCA will have one or more of the symptoms listed below. However, in a minority of patients, these symptoms are absent or develop late in the course of the disease making the diagnosis of underlying GCA a challenge.
Common symptoms
- New headache, usually in the temples
- Tenderness of the scalp
- Swelling of the arteries near the temples
- Vision changes like a curtain in the field of vision, sudden vision loss (temporary or permanent) or double vision.
- Pain in the muscles of the jaw while chewing food
- Shoulder or hip joint aching and stiffness
- Weight loss
- Poor appetite
- Low grade temperatures
Less common symptoms
- Cramping or aching in the arms with activity
- Audible pulsations (bruits) over the axillary areas
- Cramping or aching in the legs while walking which improves with resting
- Dry cough or sore throat
- Stroke-type symptoms
Diagnosis
The diagnosis of GCA is based on identifying one or more of the symptoms or physical findings listed above, laboratory studies with appropriate changes, and temporal artery biopsy findings showing arteritis. Physical examination may show swelling or decreased pulses in the temporal artery (artery that is superficial and supplies the scalp). Occasionally, your doctor may find decreased pulses in the arms or legs suggesting this diagnosis. While there is no blood test for diagnosing GCA, laboratory tests show evidence of inflammation. Your doctor may check a sedimentation rate and C-reactive protein, two blood tests that are usually high in patients with GCA. The gold standard to confirm the diagnosis of this condition is biopsy of the temporal artery. In most cases of GCA, this artery will show evidence of inflammation. In some cases, if this diagnosis is suspected, imaging of the arteries in the body with CT scan or MRI may be helpful.
Treatment
GCA is a treatable condition. All patients are started on prednisone (or other corticosteroid) which helps decrease inflammation. Addition of aspirin may be beneficial. Your doctor will follow blood tests with markers of inflammation every few months while the prednisone dose is gradually decreased. Most people require about 1 to 2 years of treatment. In some cases, longer courses of prednisone are needed. Occasionally, other medications to target the immune system may be added.
What are the complications of GCA?
The most common severe complication of GCA is blindness. This happens because of inflammation and blockage of the blood vessels that supply the main nerve of the eyes. Other complications include the formation of aneurysms (widening or ballooning of the aorta). Rarely, people can develop inflammation in the vessels supplying the arms or brain.
Prognosis
Overall, the prognosis of GCA, if treated promptly, is good. However, blindness can occur in up to a fifth of cases and may be irreversible. The risk of blindness after starting treatment is very low. GCA is usually controlled with treatment but prolonged treatment with prednisone or addition of other immune suppressing medications may be required. People with GCA have a normal life expectancy unless the disease causes an aortic aneurysm, which can occasionally rupture (tear).
What’s new in GCA?
At present, prednisone remains the standard of care for GCA. While it works very well, there are many side-effects of this medication. Other immune suppressing medications have been tried but do not work very well for this condition. Methotrexate, a medication commonly used for treatment of rheumatoid arthritis, is sometimes used to help reduce the risk of disease flares. Other treatments are currently being investigated. The types of inflammatory cells that are involved in the arteritis are being identified in current research. Understanding the nature of the inflammatory processes may lead to improved treatment.
Revision: September 2012
The Vasculitis Foundation gratefully acknowledges Dr. Rula Hajj-Ali of the Cleveland Center for Vasculitis Care and Research, for her expertise and contribution in compiling this information.