Treatment of Recurrent Painful Ophthalmoplegic Neuropathy: A Case Where Pregabalin Was Successfully Employed

Zamproni, Laura N.; Ribeiro, Reinaldo Teixeira; Cardeal, Marina

doi:10.1155/2019/9185603

Cited by 13 publications

(14 citation statements)

References 19 publications

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“…As shown in Table 1, the demographic and medical characteristics of the cases in our clinic along with the reported cases from other studies that were diagnosed as OM or RPON from 2000 to 2020 were reviewed (4–6,9–15,19–59). From the database, most reported cases were from the USA, India, Japan, and European countries (e.g.…”

Section: Resultsmentioning

confidence: 99%

“…The other 67 patients got ophthalmoplegia concurrently or within 1 week after the headache attack (concurrently: Five cases; within one day: 20 cases; 1–4 days: 20 cases; 4–7 days: 22 cases). In one patient, there was a 15-day interval between the headache and the ophthalmoplegia (47). Sixty-one patients reported that they developed ophthalmoplegia during a headache attack (6,9).…”

Section: Resultsmentioning

confidence: 99%

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Proposed modified diagnostic criteria for recurrent painful ophthalmoplegic neuropathy: Five case reports and literature review

et al. 2020

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Background Recurrent painful ophthalmoplegic neuropathy (RPON) is an uncommon disorder characterized by recurrent unilateral headache attacks associated with ipsilateral ophthalmoplegia. We intend to study the clinical picture in our case series along with the published literature to discuss the pathogenesis and propose modified diagnostic criteria for recurrent painful ophthalmoplegic neuropathy. Methods We reported five cases diagnosed as ophthalmoplegic migraine/RPON in our medical centers and reviewed the published literature related to RPON from the Pubmed database between 2000 and 2020. In one of these cases, a multiplanar reformation was performed to look at the aberrant cranial nerve. Results The mean onset age for RPON was 22.1 years, and the oculomotor nerve was the most commonly involved cranial nerve (53.9%) in 165 reviewed patients. In most patients, ophthalmoplegia started within 1 week of the headache attack (95.7%, 67/70). Additionally, 27.6% (40/145) of patients presented enhancement of the involved nerve(s) from MRI tests. Finally, 78 patients received corticosteroids, out of which 96.2% benefited from them. Conclusion This is the first time multiplanar reformation has been performed to reveal the distortion of the oculomotor nerve. Modified diagnostic criteria are proposed. We hope to expand the current knowledge and increase the detection of recurrent painful ophthalmoplegic neuropathy in the future.

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Proposed modified diagnostic criteria for recurrent painful ophthalmoplegic neuropathy: Five case reports and literature review

et al. 2020

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“…16 In the literature, pregabalin was found to be useful in a patient whose headache and ocular paralysis regressed with steroid therapy but recurred with gradual dose reduction, and who was also unresponsive to prophylaxis with beta-blockers, calcium channel blockers, and topiramate. 18 Wang et al 19 reported an adult patient who did not experience an attack with the combination of valproate and flunarizine. Margari et al 7 reported that valproate was used in a pediatric patient with a history of focal seizures and epileptic abnormalities on electroencephalography, and both electroencephalography findings and the attack frequency decreased.…”

Section: Discussionmentioning

confidence: 99%

Recurrent painful ophthalmoplegic neuropathy: a report of two new pediatric cases

Günay¹,

Edem²,

Kurul³

et al. 2022

Turk J Pediatr

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Background. Recurrent painful ophthalmologic neuropathy (RPON), formerly known as ophthalmoplegic migraine, is characterized by repeated attacks of one or more ocular cranial nerve palsies with an ipsilateral headache. While steroid therapy has been reported to be beneficial for attacks, no clear consensus on prophylactic treatments exists. We present two cases emphasizing the diagnostic significance of the loss of enhancement during the symptom-free period and valproate as a beneficial option in prophylaxis. Case 1. A 4-year-old girl presented with a one-week right frontal headache, vomiting and photophobia. Neurological examination revealed ptosis, oculomotor nerve paresis, and delay in light reflex in the right eye. Brain magnetic resonance imaging (MRI) revealed a 5.5 mm nodular enhancement in the cisternal part of the 3rd cranial nerve in the right premesencephalic area. The enhancement regressed after a 6-month symptom-free period. While propranolol, topiramate and flunarizine were inefficacious in prophylaxis, the patient responded to valproate prophylaxis and benefited from the administration of steroids for one week during the attacks. Case 2. A 7-year-old girl presented with a ten-day right-sided, throbbing headache in the frontal region, oneday eye deviation and double vision. Neurological examination revealed inward gaze restriction and ptosis in the ipsilateral eye to the headache. Brain MRI revealed a 4.5 mm, enhancing, nodular lesion in the 3rd cranial nerve lodge in the right perimesencephalic area. Her symptoms regressed in one week with dexamethasone and she received prophylactic propranolol. Neuroimaging findings disappeared after a 3-month symptom-free period. After valproate was added because of a relapse, she did not experience any further attacks. Conclusions. RPON is an uncommon disease in childhood with unknown etiology. On brain MRI with contrast during the symptom-free period, regression of the enhancement or complete resolution of the lesion are guiding features in the diagnosis. Valproate may have beneficial effects on RPON treatment.

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“…Case reports for corticosteroid‐resistant cases, as defined by return of pain and cranial nerve palsy after withdrawal of corticosteroids, propose a few options. In one case, beta‐blocker eye drops were used with resolution of symptoms 8 . In another corticosteroid‐resistant case, which was also unresponsive to paracetamol and rizatriptan, pregabalin 150 mg daily resulted in resolution of pain within 7 days 9 …”

Section: Evaluation and Discussionmentioning

confidence: 99%

“…In one case, beta-blocker eye drops were used with resolution of symptoms. 8 In another corticosteroid-resistant case, which was also unresponsive to paracetamol and rizatriptan, pregabalin 150 mg daily resulted in resolution of pain within 7 days. 9 After acute management is successful, prevention must also be considered.…”

Section: Treatmentmentioning

confidence: 99%

Recurrent painful ophthalmoplegic neuropathy with headache and oculomotor palsy 18 years apart

Dhoot

Sengupta

2022

Headache

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Treatment of Recurrent Painful Ophthalmoplegic Neuropathy: A Case Where Pregabalin Was Successfully Employed

Cited by 13 publications

References 19 publications

Proposed modified diagnostic criteria for recurrent painful ophthalmoplegic neuropathy: Five case reports and literature review

Proposed modified diagnostic criteria for recurrent painful ophthalmoplegic neuropathy: Five case reports and literature review

Recurrent painful ophthalmoplegic neuropathy: a report of two new pediatric cases

Recurrent painful ophthalmoplegic neuropathy with headache and oculomotor palsy 18 years apart

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