Asymmetric Meckel Cave Enlargement: A Potential Marker of PHACES Syndrome

Wright, Jason N.; Wycoco, Victor

doi:10.3174/ajnr.a5140

Cited by 9 publications

(8 citation statements)

References 15 publications

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“…The MC is the dural space formed by the trigeminal nerve root, which is enclosed by the dural membrane of the posterior fossa in the middle fossa and crosses the rocky ridge; moreover, it contains the trigeminal ganglion and the trigeminal cistern [ 16 ]. Morphological changes in the MC are very important in the diagnosis [ 17 – 22 ] and treatment [ 23 , 24 ] of intracranial diseases, such as radiofrequency thermocoagulation and PBC of PTN [ 25 – 29 ]. Ipsilateral hypoplastic MC and crowded MC have been suggested to produce symptoms consistent with TN [ 8 , 9 ] At present, there is little research on the morphology of MC in patients with PTN and healthy people, and therefore a lack of statistical data comparing the MC morphology between these two groups.…”

Section: Discussionmentioning

confidence: 99%

“…In this study, we introduced the method of MC morphological features extraction based on radiomics. In the identification of MC anatomy by MRI, most studies [ 8 , 17 – 19 ] have only used one of the T1 or T2 sequences. T1 enhanced sequence is helpful to determine the wall of the MC [ 30 ], but lacks the sensitivity of cerebrospinal fluid and nerves in the MC.…”

Section: Discussionmentioning

confidence: 99%

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Flatness of the Meckel cave may cause primary trigeminal neuralgia: a radiomics-based study

Lin

Zhang

et al. 2021

J Headache Pain

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Background Neurovascular contact (NVC) is the main cause of primary trigeminal neuralgia (PTN); however, cases of PTN without NVC are still observed. In this study, the Meckel cave (MC) morphology in PTN were analyzed by radiomics and compared to healthy controls (HCs) to explore the cause of PTN. Methods We studied the 3.0T MRI data of 115 patients with PTN and 46 HCs. Bilateral MC was modeled using the 3D Slicer software, and the morphological characteristics of MC were analyzed using the radiomics method. Results The right side incidence rate in the PTN group was higher than the left side incidence. By analyzing the flatness feature of MC, we observed that the affected side of the PTN was lower than that of the unaffected side, the right MC of the PTN and HC was lower than that of the left MC, the MC of the affected side of the left and right PTN without bilateral NVC was lower than that of the unaffected side. Conclusions By providing a method to analyze the morphology of the MC, we found that there is an asymmetry in the morphology of bilateral MC in the PTN and HC groups. It can be inferred that the flatness of the MC may be a cause of PTN.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Flatness of the Meckel cave may cause primary trigeminal neuralgia: a radiomics-based study

Lin

Zhang

et al. 2021

J Headache Pain

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“…It is the dural space formed by the trigeminal nerve root, which is enclosed by the dural membrane of the posterior fossa in the middle fossa and crosses the rocky ridge; moreover, it contains the trigeminal ganglion and the trigeminal cistern 16 . Morphological changes in the MC are very important in the diagnosis [17][18][19][20][21][22] and treatment 23,24 of intracranial diseases, such as radiofrequency thermocoagulation and PBC of PTN [25][26][27][28][29] . Some cases 8,9 report that ipsilateral hypoplastic MC, which may lead to morphological atrophy, as well as crowded MC, may have produced symptoms consistent with TN.…”

Section: Discussionmentioning

confidence: 99%

“…In this study, we introduced the method of the MC morphological features extraction based on radiomics. In the identification of MC anatomy by MRI, most studies 8,[17][18][19] only use one of the T1 or T2 sequences. T1 enhanced sequence is helpful to determine the wall of the MC 30 , but lacks sensitivity of cerebrospinal fluid and nerves in the MC.…”

Section: Discussionmentioning

confidence: 99%

Flatness of the Meckel Cave May Cause Primary Trigeminal Neuralgia: a Radiomics-based Study

Lin

Zhang

et al. 2021

Preprint

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Background: Neurovascular contact (NVC) is the main cause of primary trigeminal neuralgia (PTN); however, cases of PTN without NVC are still observed. In this study, the Meckel cave (MC) morphology in PTN were analyzed by radiomics and compared to healthy controls (HCs) to explore the cause of PTN.Methods: We studied the 3.0T MRI data of 115 patients with PTN and 46 HCs. Bilateral MC was modeled using the 3Dslicer software, and the morphological characteristics of MC were analyzed using the radiomics method.Results: The right side incidence rate in the PTN group was higher than the left side incidence. By analyzing the flatness feature of MC, we observed that the affected side of the PTN was lower than that of the unaffected side, the right MC of the PTN and HC was lower than that of the left MC, the MC of the affected side of the left and right PTN without bilateral NVC was lower than that of the unaffected side.Conclusions: By providing a method to analyze the morphology of MC, we found that there is asymmetry in the morphology of bilateral MC in the PTN and HC groups. It can be inferred that the flatness of MC may be a cause of PTN.

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“…Sixteen of 41 (39%) patients had trigeminal cistern enlargement, which has been described in PHACE syndrome and is possibly related to aberrant migration of the cephalic neural crest in a metameric distribution. 15 Fourteen of 41 (34%) patients had hemangiomas identified on imaging, while the remaining hemangiomas had involuted by the time of scanning. The hemangiomas were ipsilateral to the arteriopathy in 16 of the 17 patients (94%) with unilateral arteriopathy, which is in keeping with findings in another publication.…”

Section: Conventional Mr Imaging Findingsmentioning

confidence: 98%

Arterial Spin-Labeling Perfusion for PHACE Syndrome

Mamlouk

Vossough

Caschera

et al. 2020

AJNR Am J Neuroradiol

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BACKGROUND AND PURPOSE: Arterial stroke is a rare-but-reported complication in patients with posterior fossa brain malformations, hemangiomas, arterial anomalies, coarctation of the aorta and cardiac defects, and eye abnormalities (PHACE) syndrome. Currently, stroke risk is inferred by the severity of arterial anomalies identified on MRA, though no evidenced-based data exist. The purpose of our study was to determine whether arterial spin-labeling MR imaging perfusion can detect alterations in CBF in patients with PHACE syndrome. MATERIALS AND METHODS: Records were reviewed from 3 institutions for all patients with PHACE syndrome who underwent arterial spin-labeling from 2000 to 2019. CBF was qualitatively investigated with arterial spin-labeling to determine whether there was decreased or normal perfusion. Arterial anomalies were characterized on MRA imaging, and parenchymal brain findings were evaluated on conventional MR imaging sequences. RESULTS: Forty-one patients with PHACE syndrome had arterial spin-labeling imaging. There were 30 females and 11 males (age range, 7 days to 15 years). Of the 41 patients, 10 (24%) had decreased CBF signal corresponding to a major arterial territory. Ten of 10 patients had decreased CBF signal in the anterior circulation, 2/10 had decreased anterior and posterior circulation CBF signal, 2/10 had decreased bilateral anterior circulation CBF signal, and 1/10 had globally decreased CBF signal. Forty of 41 (97.5%) patients had at least 1 arteriopathy, and in those with decreased CBF signal, the arteriopathy corresponded to the CBF signal alteration in 10/10 patients. CONCLUSIONS: Arterial spin-labeling can potentially characterize hemodynamic changes in patients with PHACE syndrome. ABBREVIATIONS: ASL ¼ arterial spin-labeling; PHACE ¼ posterior fossa brain malformations, hemangiomas, arterial anomalies, coarctation of the aorta and cardiac defects, and eye abnormalities A rterial ischemic stroke is a rare-but-devastating complication in a minority of patients with posterior fossa brain malformations, hemangiomas, arterial anomalies, coarctation of the aorta and cardiac defects, and eye abnormalities (PHACE) syndrome. Stroke has been reported in multiple patients with PHACE syndrome, 1-6 but the etiology of stroke is poorly understood. 1 Possible mechanisms for stroke with PHACE include the following: 1) artery-to-artery embolisms, 2) ischemia from reduced blood flow, or 3) cardioembolism. These etiologies are predicated on arteriopathies in the brain, neck, and aortic arch, which are the most common extracutaneous finding in patients with PHACE syndrome. 7 In 1 study, arteriopathies were observed in 91% of 33 patients 8 and ranged from an anomalous course to marked stenosis with a Moyamoya pattern. 3 Currently, stroke risk is only inferred by the severity of these arteriopathies identified on MRA imaging. 9 Despite knowledge of the types of arteriopathies in PHACE, 3 it is unclear why certain patients with arteriopathies experience a stroke and others do not, even if there are s...

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Asymmetric Meckel Cave Enlargement: A Potential Marker of PHACES Syndrome

Abstract: Asymmetric Meckel cave enlargement was a common feature of patients with PHACES in our cohort and may serve as a novel imaging marker. Increased awareness of this imaging feature has the potential to increase the diagnostic accuracy of PHACES.

Cited by 9 publications

References 15 publications

Flatness of the Meckel cave may cause primary trigeminal neuralgia: a radiomics-based study

Flatness of the Meckel cave may cause primary trigeminal neuralgia: a radiomics-based study

Flatness of the Meckel Cave May Cause Primary Trigeminal Neuralgia: a Radiomics-based Study

Arterial Spin-Labeling Perfusion for PHACE Syndrome

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