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06 February 2024: Articles  New Zealand

an Underrecognized Cause of Petrous Apicitis Presenting with Gradenigo Syndrome: A Case Report

Rare disease

Zaid Ibrahim ORCID logo1ABCDEF*, Shivani Fox-Lewis2E, Jason A. Correia13AE

DOI: 10.12659/AJCR.942652

Am J Case Rep 2024; 25:e942652

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Abstract

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BACKGROUND: With the advent of antibiotics, petrous apicitis (PA), inflammation of the petrous temporal bone, has become a rare complication of otitis media. Even more uncommon is Gradenigo syndrome (GS), a result of PA, characterized by a triad of otitis media or purulent otorrhea, pain within the regions innervated by the first and second division of the trigeminal nerve, and ipsilateral abducens nerve palsy. Recent literature has demonstrated increasing reports of Fusobacterium necrophorum isolated in cases of GS.

CASE REPORT: A 21-year-old man presented with otalgia, reduced hearing, and severe headache. Examination revealed right-sided purulent otorrhea, anesthesia within the trigeminal nerve distribution, and an ipsilateral abducens nerve palsy. F. necrophorum was isolated from an ear swab and a blood culture. Computed tomography and magnetic resonance imaging (MRI) demonstrated otomastoiditis, PA, cavernous sinus thrombosis, and severe stenosis of the petrous internal carotid artery. He was treated with intravenous benzylpenicillin, underwent a mastoidectomy and insertion of a ventilation tube, and was started on a 3-month course of dabigatran. Interval MRI showed improved internal carotid artery caliber, persistent petrous apex inflammation, and normal appearance of both cavernous sinuses. Follow-up clinical review noted persistent abducens and trigeminal nerve dysfunction.

CONCLUSIONS: We identified 190 cases of PA; of these, 80 presented with the classic Gradenigo triad. Fusobacterium sp. were cultured in 10% of GS cases, making them the most frequent isolates. Due to the fastidious nature of F. necrophorum, it may be underrepresented in the historical literature, and we recommend that empiric antibiotics cover anaerobic organisms.

Keywords: Fusobacteria, Fusobacteriaceae Infections, Fusobacterium necrophorum, Otitis Media, Otitis Media, Suppurative, Petrositis

Background

Otitis media (OM) is one of the most common infectious diseases. With the advent and widespread use of antibiotics, life-threatening complications of OM, such as petrous apicitis (PA), have become very rare. In 1937, it was estimated that PA occurred once in every 300 cases of OM [1]. Today, the incidence is likely less than 2 in every 100 000 cases of OM [2]. PA is caused by medial propagation of middle ear infection to the petrous apex of the temporal bone, where the trigeminal ganglion and abducens nerve, passing through the Dorello canal, reside [3]. Therefore, inflammation within the petrous apex can cause deep pain within the regions inner-vated by the first and second division of the trigeminal nerve and an ipsilateral abducens nerve palsy. When seen with OM/ purulent otorrhea, this is referred to as the Gradenigo triad, or Gradenigo syndrome (GS), first described by Giuseppe Gradenigo in 1904 [4]. Patients with PA rarely present with GS. In 1907, Gradenigo [5] suggested that only 42% of patients present with the classic triad.

Almost all individuals have pneumatized mastoid cells. Comparatively, one-third of adults have a pneumatized petrous apex. In these cases, there is a clear route for the middle ear infection to propagate medially to the petrous apex [6]. Therefore, these patients are at risk of developing PA and GS following OM. With a non-pneumatized petrous apex, the infection can occur directly through bony destruction, extension through fascial planes, or hematogenous spread [3,7]. These pathophysiologic processes offer insight into the 1-week to 3-month interval between the onset of OM and cranial nerve dysfunction [3]. If untreated, PA can lead to life-threatening complications, such as meningitis, empyema, cerebral abscess, or venous sinus thrombosis [3] with compression of the adjacent internal carotid artery (ICA); if severe, this can cause cerebral infarction. Immunocompromised patients are at significant risk of developing recurrent OM [8] and its life-threatening complications, including PA and GS. A literature review in 2018 suggested a mortality rate of 2.6% in GS cases [9].

F. necrophorum is an anaerobic, non-spore-forming, non-mo-tile, gram-negative bacillus [10]. It is a commensal of the upper respiratory tract with high virulence, as leukotoxin production leads to apoptosis of phagocytes [11]. It causes necrobacillosis, strictly defined as sepsis, due to F. necrophorum. However, the term is often synonymously used with Lemierre syndrome (septic thrombophlebitis of the internal jugular vein) [10]. F. necrophorum is known to cause platelet aggregation, possibly due to lipopolysaccharide release [12], increasing the risk of cerebral sinus thrombosis [13]. Interestingly, in recent reports of PA and GS, F. necrophorum appears to be an emerging pathogen [3,10,13–15]. Infrequent reports in the historical literature may be due to the fastidious nature of the organism, strictly growing under anaerobic conditions [16]. Here, we report a case of PA presenting with GS secondary to F. necrophorum and discuss the literature and treatment options.

Case Report

A 21-year-old immunocompetent man with a 2-year history of recurrent middle ear infections following a right tympanic membrane perforation that occurred after an accident while surfing presented to the Emergency Department with a 1-month history of otalgia, otorrhea, and reduced hearing in his right ear despite a 14-day course of topical ciprofloxacin and oral amoxicillin/clavulanic acid. He developed a severe headache 24 h before his presentation to the hospital. Examination revealed purulent, malodourous right-sided ear discharge, anesthesia within the ophthalmic and maxillary distributions of the ipsilateral trigeminal nerve, and an ipsilateral abducens nerve palsy. He did not have clinical signs of meningism. Laboratory test results revealed a white blood count of 46.51 E+9/L, a neutrophil count of 35.21 E+9/L, and a C-reactive protein level of 49 mg/L. Broad-spectrum antibiotics (ceftriaxone, vancomycin, and metronidazole) were started immediately. F. necrophorum was isolated from a right ear swab and 1 of 2 sets of blood cultures taken on the day of admission (Figure 1). The isolate was susceptible to penicillin (minimal inhibitory concentration [MIC]: 0.016 mg/L), amoxicillin/clavulanic acid (MIC: 0.016 mg/L), and metronidazole (MIC: 0.06 mg/L). The antibiotic treatment was rationalized to intravenous benzyl-penicillin. Computed tomography (CT) of his temporal bone, cerebral angiography, and venography demonstrated otomastoiditis extending into the petrous apex (Figure 2). A gas and fluid collection affecting the Meckel cave and cavernous sinus with associated thrombosis was noted. This process extended into the right carotid canal, causing severe stenosis of the petrous portion of the ICA (Figures 3–6). No distal stenosis was noted. There was no associated cerebral infarction. Magnetic resonance imaging (MRI) confirmed the presence of PA associated with otomastoiditis (Figures 5, 6). The day following his hospital admission, he underwent an examination under anesthesia of both ears and a right-sided cortical mastoidectomy via a post-auricular incision. Within the external acoustic meatus, a copious sum of purulent material was seen, in addition to a thickened tympanic membrane, with a small posterior perforation. A ventilation tube was inserted into the perfo-ration to encourage adequate drainage of the middle ear. The left-sided external acoustic meatus and tympanic membrane appeared normal. Mastoid granulation tissue was sent for culture and revealed no growth. To manage his cavernous sinus thrombosis, he was placed on a 3-month course of dabigatran 150 mg, orally, twice daily. He was discharged with a 28-day course of intravenous benzylpenicillin 7.2 g per 24 h. Interval 3-month MRI demonstrated improved but persistent PA, with improved ICA caliber, normal appearance of both cavernous sinuses, and resolution of the previously seen trigeminal nerve enhancement (Figures 4–6). His 3-month clinical review noted improved hearing and reduced headache, with persistent abducens and trigeminal nerve dysfunction.

Discussion

PA is a potentially life-threatening complication of middle ear infection propagating to the petrous apex of the temporal bone, causing inflammation of adjacent cranial nerves, which can lead to GS. With the advent of antibiotics, PA and GS have become rare. GS has a reported incidence of 1% to 3.6% among those who have experienced complications from OM [17] and a suggested mortality rate of 2.6% [9].

The scarcity of PA and GS, lack of familiarity among clinicians, and delay between the onset of initial OM symptoms and cranial nerve signs suggestive of a petrous apex infection can lead to a delay in diagnosis of PA [3], increasing the risk of morbidity, as propagation of the infection can establish the classic Gradenigo triad or lead to life-threatening complications, such meningitis, empyema, cerebral abscess, or, as seen in our case, venous sinus thrombosis with compression of the adjacent ICA. Urgent neuro-imaging must be obtained to outline the extent of the disease and aid with operative planning, if indicated. CT will highlight the bony anatomy, whereas MRI can demonstrate the involvement of neurovascular structures and detect intracranial complications [3,17].

The management of PA has evolved with time. In 2017, Gadre and Chole reviewed the management of 44 cases of PA over 40 years (1971–2011), noting a significant reduction in the number of major surgical interventions performed (excluding tympanostomy, ventilation tube placement, and simple mastoidectomy). During the following periods, the percentage of patients that underwent major surgical intervention were as follows: 1971–1980, 50%; 1981–1990, 28.6%; 1991–2010, 15.4%; and 2001–2011, 12.5% [2]. In their review, 34 of the 44 patients (77.3%) underwent successful treatment of PA with antibiotics alone, with or without tympanostomy and ventilation tube placement [2], contradicting a review by Parsons and Strauss, who assert that surgical debridement of devitalized tissue is of the utmost importance in the management of osteomyelitis [18]. Concordantly, Henke et al found that in foot and digit osteomyelitis, aggressive surgical debridement improves wound healing, and antibiotic therapy alone was associated with reduced wound healing, less chance of limb salvage, and poor outcomes [19]. Gadre and Chole postulate that in contrast to other forms of osteomyelitis, petrous apex debridement in PA should not be a first-line treatment, as antibiotics alone can be sufficient, possibly due to the rich vascularity of the petrous apex. They also suggested a treatment algorithm in which antibiotics are the first-line treatment. If a patient worsens clinically following 24 to 48 h of treatment, major surgical intervention combined with antibiotics should be considered [2].

If major surgical intervention is warranted, important factors in determining the surgical approach to the petrous apex include the extent and location of disease, the anatomy of adjacent structures, whether the patient has serviceable hearing, and what the surgical team deems to be the shortest and least morbid route [2,20]. If a patient has impaired hearing ipsilateral to the lesion, a translabyrinthine or transcochlear approach can be considered. In cases in which hearing is preserved, approaches that tend to spare hearing include endoscopic endonasal, open anterior petrosectomy, middle cranial fossa, or transcanal infracochlear [21]. Complications range from cerebrospinal fluid leak, postoperative meningitis, damage to vascular structures, which can lead to bleeding and stroke, and hearing loss, vertigo, or facial nerve damage [20]. Given the results by Gadre and Chole [2] and the potential for morbidity, following a multidisciplinary review of the case described, we chose to avoid surgical debridement of the petrous apex.

A systemic review by Loh, Phua, and Shaw found that in un-complicated cases of acute mastoiditis, conservative treatment with antibiotics alone or with myringotomy with or without ventilation tube insertion is as effective as mastoidectomy in producing favorable long-term outcomes [22]. In contrast, a review of 262 cases of acute mastoiditis by Gelbart et al found that 94.7% (18/19) of acute mastoiditis cases caused by F. necrophorum developed complications, the most common being a subperiosteal abscess, compared with 15.6% (38/243) of acute mastoiditis caused by an alternate organism. All cases caused by F. necrophorum required surgical intervention. Comparatively, only 15.6% of cases not caused by F. necrophorum required surgery. Additionally, cases caused by F. necrophorum required significantly longer periods of hospitalization [23]. Ulanovski et al reported 43 cases of F. necrophorum mastoiditis, with higher inflammatory markers, more severe clinical presentations, and a greater incidence of intracranial and extracranial complications, compared with cases of mastoiditis secondary to other organisms [24]. Similarly, Yarden-Bilavsky et al identified 7 cases of acute mastoiditis secondary to F. necrophorum. All cases were complicated by subperiosteal abscess formation, had significantly elevated inflammatory markers, and 4 were complicated by an extradural abscess. All cases required mastoidectomy with ventilatory tube insertion [25]. Bakhos et al reviewed the management of 31 patients with a subperiosteal abscess secondary to acute mastoiditis. They found that conservative management with postauricular puncture or tympanostomy tube placement with antibiotics is an effective alternative to cortical mastoidectomy. Of the 16 cases, only 1 failed conservative management, requiring cortical mastoidectomy. The organism isolated was F. necrophorum [26]. Gelbart et al, Ulanovski et al, and Bakhos et al, all conclude that mastoiditis caused by F. necrophorum appears to have a more aggressive and complicated clinical course, and mastoidectomy should be strongly considered [23,24,26]. The patient we described underwent neuro-imaging, demonstrating right-sided mastoiditis (Figures 2, 5) and cultures positive for F. necrophorum (Figure 1); in line with the reviewed literature, he was treated with antibiotics and underwent a right-sided cortical mastoidectomy, with ventilation tube insertion into a small tympanic membrane perforation. In the days following the cortical mastoidectomy, the patient showed no signs of clinical deterioration. Therefore, surgical debridement of the petrous apex was not undertaken.

As discussed, Gadre and Chole assert that the first-line treatment of PA should be empiric broad-spectrum antibiotics with effective central nervous system penetrance. They suggest using ceftriaxone, vancomycin, and metronidazole [2], the same empiric regime used in our case, to cover a broad range of microorganisms that may be causing PA or to account for the possibility of a polymicrobial infection, frequently seen in head and neck infections [27]. Among the studies we reviewed, highlighted in greater detail later in this discussion, we identified 19 polymicrobial PA infections (Table 1). Pseudomonas aeruginosa was the most common isolate in polymicrobial infections (37%), with Staphylococcus aureus being the second most common (32%). Consistently, P. aeruginosa was the most common isolate among the 190 cases of PA we identified in our literature review (14.21%) and the most common isolate in the study by Gadre and Chole (53.3%), who reviewed 44 cases of PA over 40 years [2]. This information suggests that antipseudomonal and anti-staphylococcal antibiotics with effective central nervous system penetrance are used when treating PA with empiric antibiotics. Despite having some activity against P. aeruginosa, ceftriaxone is not recommended for treating pseudomonal infections [28]. Therefore, clinicians should consider using an alternate empiric antibiotic with strong activity against P. aeruginosa.

Antimicrobial agents can then be adjusted depending on the organisms isolated. In the case we have described, F. necrophorum was isolated from a single blood culture and a swab from ear discharge and was susceptible to penicillin (MIC: 0.016 mg/L); a second blood culture and intraoperative specimens revealed no growth. With microbiological evidence suggesting our patient did not have a polymicrobial infection and the data regarding the isolate’s antibiotic sensitivities, the patient’s antibiotics were changed to intravenous benzylpenicillin, administered for 7 days as an inpatient, and a further 28 days after discharge. Consistently, a review by McLaren, Cohen, and El Saleeby [17] suggested a treatment duration of 4 to 6 weeks to be sufficient.

The most common pathogens associated with OM are Streptococcus pneumoniae, Moraxella catarrhalis, and Haemophilus influenzae [29]. This trend is not observed among reports of PA and GS [17]. Frequently, a pathogen is not isolated [30–70]. Interestingly, in recent reports of these conditions, F. necrophorum appears to be an emerging pathogen [3,10,13–15]. In 2020, McLaren, Cohen, and El Saleeby [17] identified 65 pediatric cases of PA, 45 presenting with GS, and 21 had positive cultures, 4 of which isolated F. necrophorum.

Similarly, recent studies have reported an increasing incidence of head and neck infections, including mastoiditis caused by F. necrophorum [23,24,71–73]. Gelbart et al reported a series of 149 cases of acute mastoiditis with an identified organism, 13% a result of F. necrophorum, showing a 7-fold increase in incidence from 2.8% in 2012 to 20.4% in 2015 [23]. Ulanovski et al reported 43 cases of F. necrophorum mastoiditis, 50% of which were culture-negative. The rest were identified through 16srRNA polymerase chain reaction (PCR) sequencing [24]. Over 11 years, Le Monnier et al identified 25 cases of F. necrophorum acute OM, 10 of which developed mastoiditis; 60% of the cases were diagnosed in the last 4 years of the study, and like the study by Ulanovski et al [24], PCR was used to identify F. necrophorum when cultures were negative [72]. The first report of F. necrophorum isolated in a case of GS in the English literature was in 2003 [10], in keeping with the organism’s fastidious nature. It appears that increased reports of F. necrophorum in head and neck infections are a consequence of improved anaerobic culture techniques and new molecular diagnostic methods, often used when cultures are negative and are therefore valuable when antibiotics have been started prior to cultures being sent to the laboratory [24,73]. Therefore, F. necrophorum is possibly underrecognized in the historical PA and GS literature.

Following a review of the English literature using PubMed from 1938 to 2023, we found 97 case reports [3,10,13–15,17, 30–70,74–121]. The papers were sorted on PubMed using the query “F. necrophorum” or “fusobacterium” and “Gradenigo” or “Petrositis” or “petrous apicitis”. A total of 173 papers were found, and 97 were included in our review. Including our findings, we identified 190 cases of PA due to infection, 92 of which cultured an organism. Among the 190 cases of PA, 80 presented with the classic Gradenigo triad. The most common microbe cultured was P. aeruginosa, seen in 27 patients (14.21%). Interestingly, it only featured in 4 (5%) cases presenting with GS. Of the PA cases, 7 cultured F. necrophorum (3.68%), all of which presented with GS (8.75%), making it the most frequent isolate in the 80 GS cases identified. In 1 case of GS, F. nucleatum was isolated, bringing the total cases caused by Fusobacterium sp. to 8 (10%; Table 2).

The higher incidence of the Gradenigo triad among cases of PA caused by F. necrophorum may be due to the highly virulent nature of the organism [11], inciting a greater inflammatory response at the petrous apex leading to the characteristic cranial nerve palsies. An intense inflammatory process would also explain the aggressive and complicated clinical course seen in cases of mastoiditis caused by F. necrophorum.

Conclusions

It appears that F. necrophorum may be an underrecognized cause of PA presenting with GS. We report a further case of PA presenting with GS secondary to F. necrophorum. This case highlights that although PA and GS are rare, prompt recognition, with early neuro-imaging, is essential to confirm the diagnosis. Recent evidence suggests that mastoidectomy is the first-line treatment for mastoiditis secondary to F. necrophorum, given its aggressive and complicated clinical course. In contrast, petrous apex debridement for PA should be reserved for cases in which antibiotic therapy has failed. Although antimicrobial resistance in F. necrophorum is uncommon [122], we suggest that empiric antibiotics used to treat PA and GS cover anaerobes, P. aeruginosa, and S. aureus. We also suggest that routine anaerobic culture of samples are obtained.

Figures

Gram stain of Fusobacterium necrophorum isolated from blood culture, demonstrating characteristic pleomorphic gram-negative bacilli. Scale bar is 50 µm.Figure 1.. Gram stain of Fusobacterium necrophorum isolated from blood culture, demonstrating characteristic pleomorphic gram-negative bacilli. Scale bar is 50 µm. Axial computed tomography scan demonstrating otomastoiditis (lateral arrow) extending into the petrous apex where there is expansion with a gas and fluid collection (medial arrow).Figure 2.. Axial computed tomography scan demonstrating otomastoiditis (lateral arrow) extending into the petrous apex where there is expansion with a gas and fluid collection (medial arrow). Axial time-of-flight magnetic resonance angiography demonstrating severe narrowing of the petrous segment of the right internal carotid artery (ICA; left-sided arrow) and normal caliber of the petrous segment of the left ICA (right-sided arrow).Figure 3.. Axial time-of-flight magnetic resonance angiography demonstrating severe narrowing of the petrous segment of the right internal carotid artery (ICA; left-sided arrow) and normal caliber of the petrous segment of the left ICA (right-sided arrow). (A) Three-dimensional (3D) time-of-flight magnetic resonance angiography (MRA) demonstrating severe narrowing of the petrous and cavernous segments of the right internal carotid artery (ICA; 2 left-sided arrows). (B) 3D time-of-flight MRA 3 months after treatment demonstrating significantly improved caliber of the petrous and cavernous segments of the right ICA (2 left-sided arrows).Figure 4.. (A) Three-dimensional (3D) time-of-flight magnetic resonance angiography (MRA) demonstrating severe narrowing of the petrous and cavernous segments of the right internal carotid artery (ICA; 2 left-sided arrows). (B) 3D time-of-flight MRA 3 months after treatment demonstrating significantly improved caliber of the petrous and cavernous segments of the right ICA (2 left-sided arrows). (A) Axial post-gadolinium T1-weighted magnetic resonance imaging (MRI) during hospital admission demonstrating right-sided otomastoiditis (lateral arrow) with petrous apicitis and minimal contrast enhancement of the ipsilateral petrous internal carotid artery (ICA; medial arrow). (B) Axial post-gadolinium T1-weighted MRI 3 months after treatment demonstrating resolution of otomastoiditis (lateral arrow) and significantly improved ipsilateral petrous ICA caliber (medial arrow).Figure 5.. (A) Axial post-gadolinium T1-weighted magnetic resonance imaging (MRI) during hospital admission demonstrating right-sided otomastoiditis (lateral arrow) with petrous apicitis and minimal contrast enhancement of the ipsilateral petrous internal carotid artery (ICA; medial arrow). (B) Axial post-gadolinium T1-weighted MRI 3 months after treatment demonstrating resolution of otomastoiditis (lateral arrow) and significantly improved ipsilateral petrous ICA caliber (medial arrow). (A) Axial post-gadolinium T1-weighted magnetic resonance imaging (MRI) during hospital admission demonstrating right-sided cavernous sinus thrombosis with reduced ipsilateral cavernous internal carotid artery (ICA) caliber (left-sided arrow). (B) Axial post-gadolinium T1-weighted MRI 3 months after treatment showing resolution of cavernous sinus thrombosis with significantly improved ipsilateral cavernous ICA caliber (left-sided arrow).Figure 6.. (A) Axial post-gadolinium T1-weighted magnetic resonance imaging (MRI) during hospital admission demonstrating right-sided cavernous sinus thrombosis with reduced ipsilateral cavernous internal carotid artery (ICA) caliber (left-sided arrow). (B) Axial post-gadolinium T1-weighted MRI 3 months after treatment showing resolution of cavernous sinus thrombosis with significantly improved ipsilateral cavernous ICA caliber (left-sided arrow).

Tables

Table 1.. A summary of the literature reviewed.A summary of the literature reviewed. Table 2.. The most common organisms isolated in the 190 case reports identified.The most common organisms isolated in the 190 case reports identified.

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Figures

Figure 1.. Gram stain of Fusobacterium necrophorum isolated from blood culture, demonstrating characteristic pleomorphic gram-negative bacilli. Scale bar is 50 µm.Figure 2.. Axial computed tomography scan demonstrating otomastoiditis (lateral arrow) extending into the petrous apex where there is expansion with a gas and fluid collection (medial arrow).Figure 3.. Axial time-of-flight magnetic resonance angiography demonstrating severe narrowing of the petrous segment of the right internal carotid artery (ICA; left-sided arrow) and normal caliber of the petrous segment of the left ICA (right-sided arrow).Figure 4.. (A) Three-dimensional (3D) time-of-flight magnetic resonance angiography (MRA) demonstrating severe narrowing of the petrous and cavernous segments of the right internal carotid artery (ICA; 2 left-sided arrows). (B) 3D time-of-flight MRA 3 months after treatment demonstrating significantly improved caliber of the petrous and cavernous segments of the right ICA (2 left-sided arrows).Figure 5.. (A) Axial post-gadolinium T1-weighted magnetic resonance imaging (MRI) during hospital admission demonstrating right-sided otomastoiditis (lateral arrow) with petrous apicitis and minimal contrast enhancement of the ipsilateral petrous internal carotid artery (ICA; medial arrow). (B) Axial post-gadolinium T1-weighted MRI 3 months after treatment demonstrating resolution of otomastoiditis (lateral arrow) and significantly improved ipsilateral petrous ICA caliber (medial arrow).Figure 6.. (A) Axial post-gadolinium T1-weighted magnetic resonance imaging (MRI) during hospital admission demonstrating right-sided cavernous sinus thrombosis with reduced ipsilateral cavernous internal carotid artery (ICA) caliber (left-sided arrow). (B) Axial post-gadolinium T1-weighted MRI 3 months after treatment showing resolution of cavernous sinus thrombosis with significantly improved ipsilateral cavernous ICA caliber (left-sided arrow).

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923