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<p>Recurrent painful ophthalmoplegic neuropathy (RPON) is a rare cranial neuropathy characterized by recurrent unilateral headache followed by paresis of one or more ocular cranial nerves. Diagnosis remains clinical and requires exclusion of structural, vascular, infectious, and inflammatory causes. We report a 35-year-old man with recurrent episodes of unilateral headache followed by painful ophthalmoplegia. Neuroimaging, including CT angiography and brain MRI with dedicated cranial nerve sequences, as well as cerebrospinal fluid analysis, was unremarkable. The patient experienced complete recovery after corticosteroid therapy. During follow-up, additional attacks involved alternating cranial nerves, with faster resolution when oral corticosteroids were initiated early. Episodes were consistently associated with increased occupational emotional stress. This case highlights the diagnostic challenges of RPON, particularly when neuroimaging is normal, and underscores the importance of thorough exclusion of secondary causes and individualized trigger management.</p>

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Recurrent painful ophthalmoplegic neuropathy with alternating cranial nerve palsies: a diagnostic challenge

  • 期刊论文

  • 发表日期：

    2026年3月27日

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  • Miguel Saianda Duarte,
  • Joana Neiva Correia,
  • Margarida Palermo,
  • Vasco Fonseca,
  • Ana Cláudia Ribeiro

摘要

Recurrent painful ophthalmoplegic neuropathy (RPON) is a rare cranial neuropathy characterized by recurrent unilateral headache followed by paresis of one or more ocular cranial nerves. Diagnosis remains clinical and requires exclusion of structural, vascular, infectious, and inflammatory causes. We report a 35-year-old man with recurrent episodes of unilateral headache followed by painful ophthalmoplegia. Neuroimaging, including CT angiography and brain MRI with dedicated cranial nerve sequences, as well as cerebrospinal fluid analysis, was unremarkable. The patient experienced complete recovery after corticosteroid therapy. During follow-up, additional attacks involved alternating cranial nerves, with faster resolution when oral corticosteroids were initiated early. Episodes were consistently associated with increased occupational emotional stress. This case highlights the diagnostic challenges of RPON, particularly when neuroimaging is normal, and underscores the importance of thorough exclusion of secondary causes and individualized trigger management.