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Table of Contents    
CASE REPORT
Year : 2021  |  Volume : 69  |  Issue : 6  |  Page : 1805-1807

A case of adult-onset ophthalmoplegic migraine


1 Department of Neurology, Nalanda Medical College and Hospital, Patna, Bihar, India
2 Department of Medicine, Nalanda Medical College and Hospital, Patna, Bihar, India

Date of Web Publication23-Dec-2021

Correspondence Address:
Dr. Anwar Alam
Department of Neurology, Nalanda Medical College and Hospital, Patna, Bihar
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.333444

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 » Abstract 


Ophthalmoplegic migraine (OM) also called recurrent painful ophthalmoplegic neuropathy (RPON) is not a so common disorder. It is characterized by childhood onset, ophthalmoplegia and migraine type of headache. The most common involved nerve is third cranial nerve. Involvement of fourth and sixth cranial nerve is unlikely. Adult cases are not so common. This is a case report of a man who presented with left-sided severe headache and diplopia of left eye. He had left oculomotor nerve palsy. The patient responded to treatment and recovered.


Keywords: Adult, diplopia, ophthalmoplegic migraine
Key Message: Ophthalmoplegic migraine is rare in adults. Migraine associated with ophthalmoplegia is found in children. So, here we are reporting a case of adult onset ophthalmoplegic migraine which recovered completely.


How to cite this article:
Alam A, Iqubal MS, Kumar B, Azad ZR. A case of adult-onset ophthalmoplegic migraine. Neurol India 2021;69:1805-7

How to cite this URL:
Alam A, Iqubal MS, Kumar B, Azad ZR. A case of adult-onset ophthalmoplegic migraine. Neurol India [serial online] 2021 [cited 2022 Jan 19];69:1805-7. Available from: https://www.neurologyindia.com/text.asp?2021/69/6/1805/333444




According to International Classification Of Headache Disorders (ICHD-3) Ophthalmoplegic migraine (OM) is classified as cranial neuralgia and this is now termed as recurrent painful ophthalmoplegic neuropathy (RPON).[1] OM is an uncommon disorder characterized by recurrent attacks of ophthalmoplegia and severe migraine type of headache. It was first recognized by Charcot in 1890. The onset of OM is generally in childhood and occurs rarely in adults.[2] However, OM may be reported for the first time in adult life.[3] Incidence of OM is rare in the general population, more in young adults. The incidence in general population is 0.7 per 1,000,000.[4] Different pathogenesis of OM is postulated currently including, ischemia, compressive and demyelinating causes.[5]


 » Case History Top


A 40-year-old man came to neurology OPD with the complaint of severe left-sided headache. He had a history of occasional headache for long duration which was less severe and self-limiting for which he did not consult any doctor due to this self-limiting headache and he was not taking any prophylactic medication. In the first visit, he was diagnosed as migraine without aura according to ICHD3 and treatment with prophylaxis with propranolol and flunarizine (40 + 10 mg) daily and naproxen and domperidone s.o.s started. After 2 days, he came with left sided-drooping of upper eyelid and double vision (left-sided ophthalmoplegia). He denied any history of fluctuation of ptosis in day and night. The pain was pressure like and sometimes pulsating sensation with no radiation.

The headache was usually associated with nausea and photophobia, and it aggravated by physical activity and would last for 2 to 3 days and remitted spontaneously. He had a history of migraine without aura attacks in a frequency of 12–16 times per year for more than 10 years.

He had no history of vision loss, weakness, or numbness of limb during headache. He had no history of vomiting, loss of consciousness, or abnormal body movement. There was no relevant family history.

On examination, there was left third nerve palsy with pupil involved. He was unable to lower and adduct his left eye [Figure 1]. There was diplopia. His left pupil was larger than his right, but pupillary reaction was slow to light and accommodation. Higher mental function was intact. There was no other cranial nerve involvement.
Figure 1: Third cranial nerve palsy of left eye. Panel 1: ptosis of left eye, panel 2: patient is unable to adduct his left eye, panel 3: normal abducent nerve of both eye, panel 4: third cranial nerve palsy of left side, panel 5: normal down gaze

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Fundoscopic examination was normal. Motor and sensory functions were normal in all limbs. Coordination and gait were normal. He had no meningeal signs.

Complete blood counts, erythrocyte sedimentation rate, blood sugar, liver function, and renal function were within normal limits. Serology for HIV and VDRL was negative. CSF analysis was normal. TSH was normal. CT of the brain (plain and with contrast) and MRI of the brain with contrast were normal [Figure 2].
Figure 2: Imaging of brain. Panel 1: normal CT Brain (plain), panel 2: normal CT Brain with contrast, panel 3: normal MRI brain plain (after treatment), panel 4: normal MR angiography of brain (after treatment)

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 » Discussion Top


OM is now termed as RPON. This is a rare disorder with features of migrainous type of headache followed by ophthalmoplegia. Onset is generally in childhood and adult-onset is very rare. Here, we are reporting a case of adult-onset OM. He had migraine type of headache for long duration. He came to neurology OPD for severe left-sided headache. After 2 days, he came with left-sided ophthalmoplegia, and provisional diagnosis OM made. Eye movements and headache became normal after treatment with tapering dose of oral steroids and migraine prophylaxis [Figure 3]. The patient was in follow-up for 1 year with propranolol and flunarizine (40 + 10 mg). Frequency and severity of headache became less but occasional headache was present. In follow-up, there was no relapse of ocular symptoms and ophthalmoplegia.
Figure 3: Complete resolution of third cranial nerve palsy. Panel 1: normal forward gaze, panel 2: normal right gaze, panel 3: normal left gaze, panel 4: normal up gaze, panel 5: normal down gaze

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Different pathogenesis of OM is postulated currently of which ischemia, compressive and demyelinating causes are main.

In the past, it was postulated that during a severe migraine attack, the carotid artery of ipsilateral side becomes edematous and it compresses the oculomotor nerve inside the cavernous sinus. In ischemic theory, it was postulated that the ostia of the vasa nervosa that supplies the oculomotor nerve inside the cavernous sinus become narrow.[5] The course and recovery of our patient is likely to fit in this postulation.[5] Later it became possible to do magnetic resonance imaging of brain with MR angiogragraphy of the brain, which turned to be normal. It might be due to the resolution of acute ischemia of vasa nervosa supplying oculomotor nerve [Figure 2].

However, Gelfand and his colleagues found no evidence to suggest a systemic inflammatory process associated with OM in his analysis.[2]

There is also other possible cause of OM is viral infection. This was postulated by Mark and colleagues.[6] Although there is no significant data that can show an association between viral infection and OM, there is a single report which claims an association between OM and a positive cytomegalovirus IgG, but not IgM. However, due to the absence of the evidence of acute infection, this report shows doubtful significance.[7]

Lance and Zigami reported five patients of OM, in which a child developed recurrent attacks of OM after vaccination on three occasions.[8] Therefore, combining these observations and presence of nerve edema of oculomotor nerve, the authors suggested that a recurrent demyelinating cranial neuropathy may be the cause of this syndrome, in which it was supposed that inflammatory process affecting the oculomotor nerve might have irritated trigeminal sensory fibers and triggered migraine headache.

Familial cases of recurrent attacks of Bell's palsy and ophthalmoplegia that was preceded by headache have been reported.[9] There is also several case reports of OM in which patient had personal or family history of Bell's palsy.[10]

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 » References Top

1.
Headache Classification Subcommittee of the International Headache Society. The international classification of headache disorders. Cephalalgia 2004;24(Suppl 1):9-160.  Back to cited text no. 1
    
2.
Gelfand AA, Gelfand JM, Prabakhar P, Goadsby PJ. Ophthalmoplegic “migraine” or recurrent ophthalmoplegic cranial neuropathy: New cases and a systematic review. J Child Neurol 2012;27:759-66.  Back to cited text no. 2
    
3.
Celebisoy N, Sirin H, Gokcay F. Ophthalmoplegic migraine: Two patients, one at middle age with abducens palsy. Cephalalgia 2005;25:151-3.  Back to cited text no. 3
    
4.
Hansen SL, Borelli-Moller L, Strange P, Nielsen BM, Olesen J. Ophthalmoplegic migraine: Diagnostic criteria, incidence of hospitalization and possible etiology. Acta Neurol Scand 1990;81:54-60.  Back to cited text no. 4
    
5.
Walsh JP, O'Doherty DS. A possible explanation of the mechanism of ophthalmoplegic migraine. Neurology 1960;10:1079-84.  Back to cited text no. 5
    
6.
Mark AS, Casselman J, Brown D, Sanchez J, Kolsky M, Larsen TC 3rd, et al. Ophthalmoplegic migraine: Reversible enhancement and thickening of the cisternal segment of the oculomotor nerve on contrast-enhanced MR images. AJNR Am J Neuroradiol 1998;19:1887-91.  Back to cited text no. 6
    
7.
Ogose T, Manabe T, Abe T, Nakaya K. Ophthalmoplegic migraine with a rise in cytomegalovirus-specific IgG antibody. Pediatr Int 2009;51:143-5.  Back to cited text no. 7
    
8.
Lance JW, Zagami AS. Ophthalmoplegic migraine: A recurrent demyelinating neuropathy? Cephalalgia 2001;21:84-9.  Back to cited text no. 8
    
9.
Lee AG, Brazis PW, Eggenberger E. Recurrent idiopathic familial facial nerve palsy and ophthalmoplegia. Strabismus 2001;9:137-41.  Back to cited text no. 9
    
10.
Peck R. Ophthalmoplegic migraine presenting in infancy: A self-reported case. Cephalalgia 2006;26:1242-4.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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