Recurrent Painful Ophthalmoplegic Neuropathy: A Diagnostic Challenge in the Pediatric Age

Authors

  • Margarida Camacho Sampaio Centro de Desenvolvimento da Criança – Neuropediatria / Centro Hospilatar e Universitário de Coimbra, Coimbra, Portugal https://orcid.org/0009-0009-4576-5262
  • Henrique Coimbra Queirós Serviço de Imagem Médica – Unidade de Neurorradiologia / Centro Hospilatar e Universitário de Coimbra, Coimbra, Portugal https://orcid.org/0000-0002-6891-8403
  • Filipe Palavra Centro de Desenvolvimento da Criança – Neuropediatria / Centro Hospilatar e Universitário de Coimbra, Coimbra, Portugal; Laboratório de Farmacologia e Terapêutica Experimental, Instituto de Investigação Clínica e Biomédica de Coimbra (iCBR), Faculdade de Medicina, Universidade de Coimbra, Coimbra, Portugal; Centro Académico Clínico de Coimbra, Coimbra, Portugal https://orcid.org/0000-0002-2165-130X
  • Rui Pedro Pais Serviço de Imagem Médica – Unidade de Neurorradiologia / Centro Hospilatar e Universitário de Coimbra, Coimbra, Portugal
  • Catarina Paiva Serviço de Oftalmologia – Unidade de Oftalmologia Pediátrica / Centro Hospilatar e Universitário de Coimbra, Coimbra, Portugal
  • Carmen Costa Centro de Desenvolvimento da Criança – Neuropediatria / Centro Hospilatar e Universitário de Coimbra, Coimbra, Portugal

DOI:

https://doi.org/10.46531/sinapse/CC/230059/2024

Keywords:

Child, Ophthalmoplegia/diagnosis, Ophthalmoplegic Migraine/ diagnosis, Ophthalmoplegic Migraine/ drug therapy

Abstract

Recurrent painful ophthalmoplegic neuropathy (RPON) is a rare clinical entity, whose pathophysiology is not well understood. We present the case of a girl who, at 14 months of age, suddenly started right eyelid ptosis and incomplete paresis of the ipsilateral oculomotor nerve. Magnetic resonance imaging revealed a thickening of the emergence of that nerve. She was treated with corticosteroids and, over the years, presented multiple similar episodes, concluding with the diagnosis of RPON. The second case involves a 17-year-old girl with a history of episodic migraine and asymptomatic SARS-CoV-2 infection, also with right palpebral ptosis, with rapid progression to incomplete paresis of the ipsilateral oculomotor nerve. Ocular myasthenia gravis, a cranial neuralgia associated with COVID-19, and RPON were considered as hypotheses, with no recurrences to date. RPON recognition is difficult. This situation may contribute to its underestimation of prevalence, particularly in children.

Downloads

Download data is not yet available.

References

Furia A, Liguori R, Donadio V. Recurrent Painful Ophthalmoplegic Neuropathy: A case report with atypical features and a review of the literature. Cephalalgia. 2023;43:3331024221133386. doi: 10.1177/03331024221133386.

Aleksic DZ, Drakulic SM, Ljubisavljevic S. Recurrent Painful Ophthalmoplegic Neuropathy: Migraine, Neuralgia, or Something Else? J Oral Facial Pain Headache. 2020;34:3748. doi: 10.11607/ofph.2656.

Günay Ç, Edem P, Hız-Kurul AS, Yasar E, Yis U. Recurrent painful ophthalmoplegic neuropathy: a report of two new pediatric cases. Turk J Pediatr. 2022;64:592-8. doi: 10.24953/turkjped.2021.1124.

Liu Y, Wang M, Bian X, Qiu E, Han X, Dong Z, et al. Proposed modified diagnostic criteria for recurrent painful ophthalmoplegic neuropathy: Five case reports and literature review. Cephalalgia. 2020;40:1657-70. doi: 10.1177/0333102420944872.

Falsaperla R, Presti S, Lo Bianco M, Catanzaro S, Marino S, Ruggieri M. Diagnostic controversies in recurrent painful ophthalmoplegic neuropathy: single case report with a systematic review. Ital J Pediatr. 2022;48:82. doi: 10.1186/s13052-022-01274-x.

Headache Classification Committee of the International Headache Society (IHS) The International Classification of Headache Disorders, 3rd edition. Cephalalgia. 2018;38:1211. doi: 10.1177/0333102417738202.

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. doi: 10.1177/0883073811426502.

Choi JY, Jang SH, Park MH, Kim BJ, Lee DH. Ophthalmoplegic migraine with alternating unilateral and bilateral internal ophthalmoplegia. Headache. 2007;47:726-8. doi: 10.1111/j.1526-4610.2007.00795_2.x.

Bharucha DX, Campbell TB, Valencia I, Hardison HH, Kothare SV. MRI findings in pediatric ophthalmoplegic migraine: a case report and literature review. Pediatr Neurol. 2007;37:59-63. doi: 10.1016/j.pediatrneurol.2007.03.008.

Kekatpure MV, Panmand P, Nalubolu S. Neuroimaging of a Paediatric Ophthalmoplegic Migraine. Ann Indian Acad Neurol. 2022;25:131-2. doi: 10.4103/aian.AIAN_327_21.

Fortin E, Cestari DM, Weinberg DH. Ocular myasthenia gravis: an update on diagnosis and treatment. Curr Opin Ophthalmol. 2018;29:477-84. doi: 10.1097/ICU.0000000000000526. .

Al-Haidar M, Benatar M, Kaminski HJ. Ocular Myasthenia. Neurol Clin. 2018;36:241-51.

Díaz-Maroto I, García-García J, Sánchez-Ayaso PA, Alcahut-Rodríguez C, González-Villar E, Pardal-Fernández JM, et al. Ocular myasthenia gravis and risk factors for developing a secondary generalisation: description of a Spanish series. Neurologia. 2023;38:229-35. doi: 10.1016/j.nrleng.2020.09.004.

Bohania N, Ish P, Nune A, Iyengar KP. Cranial neuropathy in COVID-19: a case series and review of literature. Infez Med. 2021;29:609-13. doi: 10.53854/liim-2904-15.

Finsterer J, Scorza FA, Scorza C, Fiorini A. COVID-19 associated cranial nerve neuropathy: A systematic review. Bosn J Basic Med Sci. 2022;22:39-45. doi: 10.17305/bjbms.2021.6341.

Published

2024-04-04

How to Cite

1.
Camacho Sampaio M, Coimbra Queirós H, Palavra F, Pais RP, Paiva C, Costa C. Recurrent Painful Ophthalmoplegic Neuropathy: A Diagnostic Challenge in the Pediatric Age. Sinapse [Internet]. 2024 Apr. 4 [cited 2024 May 18];24(1):33-8. Available from: https://sinapse.pt/index.php/journal/article/view/47

Issue

Section

Case Reports

Most read articles by the same author(s)

1 2 > >>