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  IN THIS Article
 ::  Abstract
  ::  Introduction
  ::  Case Details
  ::  Conclusion
 ::  References
 ::  Article Figures

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  Table of Contents     
GRAND ROUND CASE
Year : 2021  |  Volume : 67  |  Issue : 2  |  Page : 96-99

Unraveling a cephalalgic quagmire from a cavern to a cave


1 Department of General Medicine, Sri Manakula Vinayagar Medical College and Hospital, Puducherry, India
2 Department of Neurosurgery, Chettinad Academy of Research and Education, Chennai, Tamil Nadu, India

Date of Submission11-Jul-2020
Date of Decision08-Oct-2020
Date of Acceptance25-Jan-2021
Date of Web Publication08-Apr-2021

Correspondence Address:
S Girija
Department of General Medicine, Sri Manakula Vinayagar Medical College and Hospital, Puducherry
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jpgm.JPGM_809_20

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 :: Abstract 


Headache in women in their late forties can be primary or secondary. We report a 48-year-old female with chronic slowly progressive left temporal headache for 1 year. She also had ipsilateral eye pain and facial numbness for 1 month, with restricted abduction in the left eye and diplopia. On neurological examination, she had isolated left abducent nerve palsy, with loss of corneal and conjunctival reflexes, localizing the pathology to the cavernous sinus or its adjacent structures. Anatomically, cranial nerves V and VI are in close proximity to each other in the region of Meckel's cave. In view of her age, insidious onset, progressive symptoms and clinical findings, the provisional diagnosis in this patient was a Meckel's cave tumor. Magnetic resonance (MR) imaging revealed a 2 cm × 2 cm × 1.7 cm enhancing dumb-bell-shaped mass lesion with mild restricted diffusion in the Meckel's cave projecting into cavernous sinus with alanine, myoinositol and glutamine peaks on MR spectroscopy. Intradural debulking was done; lesion was confirmed by histopathology and patient was cured of her symptoms. An algorithm for diagnosing this entity at the bedside is presented.


Keywords: Cavernous sinus, headache, Meckel's cave, meningioma, temporal, unilateral


How to cite this article:
Thenmozhi S, Girija S, Viswanathan K N, Karthikeyan K V. Unraveling a cephalalgic quagmire from a cavern to a cave. J Postgrad Med 2021;67:96-9

How to cite this URL:
Thenmozhi S, Girija S, Viswanathan K N, Karthikeyan K V. Unraveling a cephalalgic quagmire from a cavern to a cave. J Postgrad Med [serial online] 2021 [cited 2021 Aug 1];67:96-9. Available from: https://www.jpgmonline.com/text.asp?2021/67/2/96/313347





 :: Introduction Top


Meckel's cave is a dural recess located in the posteromedial part of the middle cranial fossa, acting as a passage for the trigeminal nerve. It is situated between the prepontine cistern and the cavernous sinus, housing the trigeminal ganglion, and the proximal rootlets of the trigeminal nerve.[1],[2],[3] Compression of structures around the Meckel's cave results in a classical picture which can be clinically localized.[3] We report a middle-aged lady who presented with unilateral temporal headache and was subsequently diagnosed to have Meckel's cave meningioma confirmed by imaging. We present an algorithmic approach to localize the lesion to the Meckel's cave at the bedside.


 :: Case Details Top


A 48-year-old lady presented with recurrent, progressive dull-aching, left-sided temporal headache for 1 year.

What are the common causes for the left temporal headache?

Chronic dull-aching left-sided temporal headache in women in their late forties can be primary (tension headache, cluster headache, chronic daily headache, and migraine) or secondary (glaucoma, brain tumor, raised intracranial pressure, trigeminal neuralgia, and temporal arteritis) [Figure 1].[4],[5]
Figure 1: Causes of unilateral temporal headache with ipsilateral eye pain, numbness, and facial pain in a 48-year-old lady

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Case details (continued)

She also had giddiness, left-sided eye pain, and left-sided facial numbness for 1 month. She had restriction of left-sided gaze in the left eye with double vision. The double vision became single on closing the left eye (the lateral false image disappeared, leaving the medial true image intact). She had no crossing of images, diurnal variation of diplopia, tinnitus, vertigo, deafness, nausea, or vomiting. There were no neuro-cutaneous markers and her higher mental functions were normal. She had a loss of left-sided corneal and conjunctival reflexes with restricted left eye abduction. There were no other neurological deficits.

What are the causes for left temporal headache with ipsilateral eye pain, facial numbness, and gaze-restriction?

Our patient had unilateral headache with isolated abducent nerve palsy, with loss of corneal and conjunctival reflexes localizing the lesion to the pons. As she did not have hemiplegia, facial nerve palsy, deafness, nausea, and vomiting, a lesion in the course of nerve distal to the pontine exit becomes a possibility, further localizing it to the region of the cavernous sinus or its adjacent structures.[3],[6],[7],[8] This is explained in [Figure 2].[4],[9]
Figure 2: Algorithm to localize the lesion to Meckel's cave

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What are the structures related to the cavernous sinus?

The cavernous sinus is related to the optic tract, optic chiasma, internal carotid artery superiorly, the pituitary gland inferiorly, temporal lobe with uncus laterally, superior orbital fissure, and the apex of the orbit anteriorly and apex of petrous temporal bone posteriorly. Meckel's cave is a narrow complex space measuring 4 mm × 7.5 mm, situated inferolateral to the posterior part of the cavernous sinus [Figure 3].[3],[6],[7] Structures within the lateral wall of the compartment of the cavernous sinus, from superior to inferior include the oculomotor nerve, trochlear nerve, ophthalmic, and maxillary branches of the trigeminal nerve. Structures passing through the medial wall are the abducent nerve, internal carotid artery and associated carotid plexus.[7]
Figure 3: (a) Magnetic resonance imaging of brain showing 2 cm × 2 cm × 1.7 cm enhancing dumbbell-shaped mass lesion with mild restricted diffusion in Meckel's cave (marked by asterisk) projecting into the cavernous sinus, cerebellopontine angle with dural tail with a waist at the foramen; (b) The anatomy and relations of the Meckel's cave (marked by asterisk) on MRI are compared with a schematic diagram through the middle cranial fossa

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What are the lesions causing cavernous sinus pathology?

Thrombosis, meningioma, schwannoma, aneurysms, cavernous hemangiomas, arteriovenous malformations, infections such as neurocysticercosis, inflammation, lymphomas, dermoids, pituitary adenomas, chordomas, chondrosarcomas, craniopharyngioma, pseudotumor cerebri, Meckel's cave schwannomas or meningiomas, and secondary malignancies are the various pathological lesions in the cavernous sinus.[7]

Why was this lesion localized to Meckel's cave instead of the cavernous sinus?

Progressive temporal headache with isolated restricted abduction of the left eye with loss of ipsilateral corneal and conjunctival reflexes suggested that the lesion was in close proximity to the cranial V and VI nerves.[3],[8] As cranial nerves II, III, and IV were uninvolved, the lesion could probably arise from the structures adjacent to the cavernous sinus only. Cranial nerve VI is closely related to the trigeminal ganglion in the Meckel's cave. In the postero-inferior aspect of the cavernous sinus, trigeminal ganglion and VI cranial nerve are only 1.87 mm apart.[2],[7] This further localized the lesion to the Meckel's cave.

Case details (continued)

Magnetic resonance (MR) imaging of the brain with contrast revealed a 2 cm × 2 cm × 1.7 cm enhancing dumb-bell shaped mass lesion with mild restricted diffusion in Meckel's cave projecting into the cavernous sinus, cerebellopontine angle with dural tail [Figure 3]a. MR spectroscopy showed alanine, myoinositol [Figure 4], and glutamine peaks. A provisional diagnosis of Meckel's cave meningioma was made with a differential diagnosis of trigeminal schwannoma considering the age, gender, clinical localization, and MR findings [Figure 3]b.[3],[7],[10],[11]
Figure 4: Magnetic resonance spectroscopy of the brain showed alanine, myoinositol (labeled) and glutamine peaks

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What are the possible lesions that could arise from the Meckel's cave?

  1. Meningioma
  2. Chordoma
  3. Schwannoma.


Meckel's cave is a dural cleft around the trigeminal nerve.[2] Meckel's cave meningiomas usually present with sensory symptoms and signs with very minimal motor involvement.[12] The presence of facial pain in the trigeminal territory with isolated sixth nerve palsy should arouse suspicion of pathology in Meckel's cave.[7] Tumors of the Meckel's cave contribute to only 0.5% of all intracranial tumors with meningiomas being the commonest.[3] Tumors with enhancing dural tail in the periphery, located along the margins of Meckel's cave are usually meningiomas.[1],[13]

Case details (continued)

The tumor was approached by left fronto-temporo-parietal craniotomy. Dura was incised medially close to the cavernous sinus and intracapsular debulking was done. Histopathology showed ovoid, monomorphic tumor cells arranged in whorls and syncytial pattern, with occasional intranuclear inclusions, consistent with meningothelial meningioma WHO grade 1 [Figure 5]. Her headache resolved after the surgery. The patient is currently undergoing external beam radiotherapy.
Figure 5: Biopsy showed ovoid, monomorphic tumor cells arranged in whorls and in syncytial pattern (1 and 2) with intranuclear inclusions in few cells, consistent with meningothelial Meckel's cave meningioma WHO grade 1 (H and E, ×400)

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 :: Conclusion Top


Systematically approaching this patient with unilateral temporal cephalalgia aided in clinical localization of the lesion, which was confirmed by MRI and surgical debulking resulted in cure.

Declaration of patient consent

The authors certify that appropriate patient consent was obtained.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 :: References Top

1.
Malhotra A, Tu L, Kalra VB, Wu X, Mian A, Mangla R, et al. Neuroimaging of Meckel's cave in normal and disease conditions. Insights Imaging 2018;9:499-510.  Back to cited text no. 1
    
2.
Arslan M, Deda H, Avci E, Elhan A, Tekdemir I, Tubbs RS, et al. Anatomy of Meckel's cave and the trigeminal ganglion: Anatomical landmarks for a safer approach to them. Turk Neurosurg 2012;22:317-23.  Back to cited text no. 2
    
3.
Busch CM, Prickett JT, Stein R, Cuoco JA, Marvin EA, Witcher MR. Meckel cave epidermoid cyst presenting as multiple cranial nerve deficits due to indirect tumoral compression of the cavernous sinus: A case report and literature review. World Neurosurg 2019;121:88-94.  Back to cited text no. 3
    
4.
Amato AA, Brown Jr. RH. Muscular dystrophies and other muscle diseases. In: Jameson JL, Kasper DL, Longo DL, Fauci AS, Hauser SL, Loscalzo J, editors. Harrison's Principles of Internal Medicine. 20th ed. New Delhi: McGraw-Hill Education; 2018. p. 3239-54.  Back to cited text no. 4
    
5.
Walker RW. Nervous system. In: Glynn M, Drake WM, editors. Hutchison's Clinical Methods: An Integrated Approach to Clinical Practice. 24th ed. China: Elsevier Health Sciences; 2018. p. 309-54.  Back to cited text no. 5
    
6.
The trigeminal nerve. In: Campbell WW, Barohn RJ. DeJong's the Neurologic Examination. 8th ed. China: Wolters Kluwer; 2020. p. 462-501.  Back to cited text no. 6
    
7.
Evans JJ, Kenning TJ, Farrell C, Kshettry VR, editors. Endoscopic and Keyhole Cranial Base Surgery. Switzerland: Springer; 2019.  Back to cited text no. 7
    
8.
Bigder MG, Helmi A, Kaufmann AM. Trigeminal neuropathy associated with an enlarging arachnoid cyst in Meckel's cave: Case report, management strategy and review of the literature. Acta Neurochir (Wien) 2017;159:2309-12.  Back to cited text no. 8
    
9.
Azarmina M, Azarmina H. The six syndromes of the sixth cranial nerve. J Ophthalmic Vis Res 2013;8:160-71.  Back to cited text no. 9
  [Full text]  
10.
Lyndon D, Lansley JA, Evanson J, Krishnan AS. Dural masses: Meningiomas and their mimics. Insights Imaging 2019;10:11.  Back to cited text no. 10
    
11.
Pescatori L, Tropeano MP, Ciappetta P. Meckel's cave meningiomas: New classification system focalized on dumbbell lesions deriving from the morphometric analysis of the posterior petrous apicectomy on fresh non-formalin-fixed specimens. A correlative anatomoclinical study. World Neurosurg 2018;118:e489-e499.  Back to cited text no. 11
    
12.
Soni CR, Kumar G, Sahota P, Miller DC, Litofsky NS. Metastases to Meckel's cave: Report of two cases and comparative analysis of malignant tumors with meningioma and schwannoma of Meckel's cave. Clin Neurol Neurosurg 2010;112:927-32.  Back to cited text no. 12
    
13.
Watts J, Box G, Galvin A, Brotchie P, Trost N, Sutherland T. Magnetic resonance imaging of meningiomas: A pictorial review. Insights Imaging 2014;5:113-22.  Back to cited text no. 13
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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