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Trigeminal hypoplasia due to vertebrobasilar dolichoectasia: A new entity.

Jha A, Gupta P, Haroon M, Shah G, Gupta G, Khalid M - J Pediatr Neurosci (2015 Apr-Jun)

Bottom Line: The etiology of the disease is unknown and is usually detected incidentally.The predominant clinical manifestations arise due to the mass effect of the dilated vessels and may include cranial nerve compression, extrinsic aqueductal compression, motor and sensory disturbances.Trigeminal hypoplasia is a very uncommon condition, usually described in association with Goldenhar-Gorlin syndrome and has not yet been attributed to vertebrobasilar dolichoectasia.

View Article: PubMed Central - PubMed

Affiliation: Department of Radiodiagnosis, Jawaharlal Nehru Medical College, Aligarh Muslim University, Aligarh, Uttar Pradesh, India.

ABSTRACT
The term "vertebrobasilar dolichoectasia" refers to anomalous dilatation of the intracranial arteries associated with elongation or tortuosity of the affected vessels. The etiology of the disease is unknown and is usually detected incidentally. The predominant clinical manifestations arise due to the mass effect of the dilated vessels and may include cranial nerve compression, extrinsic aqueductal compression, motor and sensory disturbances. Trigeminal hypoplasia is a very uncommon condition, usually described in association with Goldenhar-Gorlin syndrome and has not yet been attributed to vertebrobasilar dolichoectasia. The current case report highlights this rare association of trigeminal nerve hypoplasia and vertebrobasilar dolichoectasia, leading to hemifacial and corneal anesthesia.

No MeSH data available.


Related in: MedlinePlus

(a) Axial contrast-enhanced computed tomography scan of the head showing dilated left vertebral artery (black arrow), with an elongated aberrant course seen crossing toward the right side (white arrow). (b) At the next consecutive cranial scan, note the atrophy of the anterosuperior part of the left cerebellar hemisphere with widened cerebellopontine angle cistern (black arrow). The anomalous crossed left vertebral artery is now lying at the right side anterior to the midbrain (white arrow)
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Figure 1: (a) Axial contrast-enhanced computed tomography scan of the head showing dilated left vertebral artery (black arrow), with an elongated aberrant course seen crossing toward the right side (white arrow). (b) At the next consecutive cranial scan, note the atrophy of the anterosuperior part of the left cerebellar hemisphere with widened cerebellopontine angle cistern (black arrow). The anomalous crossed left vertebral artery is now lying at the right side anterior to the midbrain (white arrow)

Mentions: The patient also complained of occasional deviation of the tongue during speaking and chronic pulsatile headache especially in morning. On examination, the vitals of the patient were stable, and general examination of the patient was uneventful except for pallor. Neurological examination revealed hypoesthesia along the left side of the face corresponding to ophthalmic, maxillary and mandibular divisions of the trigeminal nerve. There was corneal anesthesia with corneal ulceration. Rest of neurological examination was normal. The patient was then referred for contrast-enhanced computed tomography scan of the head, which revealed abnormally dilated and tortuous left vertebral artery, with an aberrant course. The caliber of the vertebral artery was 7.2 mm and it was seen compressing the left lateral aspect of the brainstem, with relative atrophy of the anterosuperior part of the left cerebellar hemisphere leading to dilation of ipsilateral cerebellopontine angle cistern and crossing toward the right side [Figure 1]. The ectatic vessel was also seen compressing the fourth ventricle with mild dilatation of bilateral lateral ventricles.


Trigeminal hypoplasia due to vertebrobasilar dolichoectasia: A new entity.

Jha A, Gupta P, Haroon M, Shah G, Gupta G, Khalid M - J Pediatr Neurosci (2015 Apr-Jun)

(a) Axial contrast-enhanced computed tomography scan of the head showing dilated left vertebral artery (black arrow), with an elongated aberrant course seen crossing toward the right side (white arrow). (b) At the next consecutive cranial scan, note the atrophy of the anterosuperior part of the left cerebellar hemisphere with widened cerebellopontine angle cistern (black arrow). The anomalous crossed left vertebral artery is now lying at the right side anterior to the midbrain (white arrow)
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4489062&req=5

Figure 1: (a) Axial contrast-enhanced computed tomography scan of the head showing dilated left vertebral artery (black arrow), with an elongated aberrant course seen crossing toward the right side (white arrow). (b) At the next consecutive cranial scan, note the atrophy of the anterosuperior part of the left cerebellar hemisphere with widened cerebellopontine angle cistern (black arrow). The anomalous crossed left vertebral artery is now lying at the right side anterior to the midbrain (white arrow)
Mentions: The patient also complained of occasional deviation of the tongue during speaking and chronic pulsatile headache especially in morning. On examination, the vitals of the patient were stable, and general examination of the patient was uneventful except for pallor. Neurological examination revealed hypoesthesia along the left side of the face corresponding to ophthalmic, maxillary and mandibular divisions of the trigeminal nerve. There was corneal anesthesia with corneal ulceration. Rest of neurological examination was normal. The patient was then referred for contrast-enhanced computed tomography scan of the head, which revealed abnormally dilated and tortuous left vertebral artery, with an aberrant course. The caliber of the vertebral artery was 7.2 mm and it was seen compressing the left lateral aspect of the brainstem, with relative atrophy of the anterosuperior part of the left cerebellar hemisphere leading to dilation of ipsilateral cerebellopontine angle cistern and crossing toward the right side [Figure 1]. The ectatic vessel was also seen compressing the fourth ventricle with mild dilatation of bilateral lateral ventricles.

Bottom Line: The etiology of the disease is unknown and is usually detected incidentally.The predominant clinical manifestations arise due to the mass effect of the dilated vessels and may include cranial nerve compression, extrinsic aqueductal compression, motor and sensory disturbances.Trigeminal hypoplasia is a very uncommon condition, usually described in association with Goldenhar-Gorlin syndrome and has not yet been attributed to vertebrobasilar dolichoectasia.

View Article: PubMed Central - PubMed

Affiliation: Department of Radiodiagnosis, Jawaharlal Nehru Medical College, Aligarh Muslim University, Aligarh, Uttar Pradesh, India.

ABSTRACT
The term "vertebrobasilar dolichoectasia" refers to anomalous dilatation of the intracranial arteries associated with elongation or tortuosity of the affected vessels. The etiology of the disease is unknown and is usually detected incidentally. The predominant clinical manifestations arise due to the mass effect of the dilated vessels and may include cranial nerve compression, extrinsic aqueductal compression, motor and sensory disturbances. Trigeminal hypoplasia is a very uncommon condition, usually described in association with Goldenhar-Gorlin syndrome and has not yet been attributed to vertebrobasilar dolichoectasia. The current case report highlights this rare association of trigeminal nerve hypoplasia and vertebrobasilar dolichoectasia, leading to hemifacial and corneal anesthesia.

No MeSH data available.


Related in: MedlinePlus