The Management of Strabismus in Patients with Chronic Progressive External Ophthalmoplegia

Tinley, Christopher; Dawson, Emma; Lee, John

doi:10.3109/09273971003758388

Cited by 15 publications

(6 citation statements)

References 13 publications

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“…Should the eye become anchored in a grossly eccentric position, then muscle surgery to straighten it may be justified and beneficial but there is a risk of recurrence as the disease progresses (Tinley et al 2010). The eyes are relatively immobile in the later stages and prisms are then of little benefit.…”

Section: Managementmentioning

confidence: 83%

Myogenic Palsies

FRCOphth

2013

Diagnosis and Management of Ocular Motility Disorders

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Section: Managementmentioning

confidence: 83%

Myogenic Palsies

FRCOphth

2013

Diagnosis and Management of Ocular Motility Disorders

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“…Seven of 13 patients with a manifest deviation showed diplopia. Tinley et al (2010) reported 25 CPEO patients including exophoria or exotropia with/without vertical deviation in 22 patients (88%). The remaining three patients showed esotropia, esotropia with vertical deviation and hypertropia, respectively.…”

Section: Discussionmentioning

confidence: 99%

“…Chronic progressive external ophthalmoplegia (CPEO) features slowly progressive external ophthalmoplegia and ptosis (Hansen & Logan 1966; Sorkin et al 1997; Wallace et al 1997; Moon‐Woo et al 2005; Richardson et al 2005; Hwang et al 2009; Tinley et al 2010; Chatzistefanou et al 2018). Its diagnosis could be made by muscle biopsy showing ragged‐red fibres or molecular genetic tests confirming mitochondrial abnormalities (Hansen & Logan 1966; Sorkin et al 1997; Wallace et al 1997; Moon‐Woo et al 2005; Richardson et al 2005; Hwang et al 2009; Tinley et al 2010; Chatzistefanou et al 2018). Patterns of strabismus in CPEO as well as ophthalmoplegia have been scarcely reported because of its rarity (Sorkin et al 1997; Wallace et al 1997; Richardson et al 2005; Hwang et al 2009; Tinley et al 2010; Chatzistefanou et al 2018).…”

Section: Introductionmentioning

confidence: 99%

“…Its diagnosis could be made by muscle biopsy showing ragged‐red fibres or molecular genetic tests confirming mitochondrial abnormalities (Hansen & Logan 1966; Sorkin et al 1997; Wallace et al 1997; Moon‐Woo et al 2005; Richardson et al 2005; Hwang et al 2009; Tinley et al 2010; Chatzistefanou et al 2018). Patterns of strabismus in CPEO as well as ophthalmoplegia have been scarcely reported because of its rarity (Sorkin et al 1997; Wallace et al 1997; Richardson et al 2005; Hwang et al 2009; Tinley et al 2010; Chatzistefanou et al 2018). In addition, there is no description about strabismus in Asian CPEO patients.…”

Section: Introductionmentioning

confidence: 99%

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Strabismus in chronic progressive external ophthalmoplegia

Kim

Yang

Kim

et al. 2020

Acta Ophthalmologica

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Purpose To elucidate the patterns of strabismus and ophthalmoplegia associated with chronic progressive external ophthalmoplegia (CPEO) confirmed by mitochondrial DNA (mtDNA) deletions in Asians. Methods A total of 10 patients confirmed to have mtDNA deletion associated with CPEO were included. Long‐range PCR encompassing the entire mtDNA was carried out. In the cases with mtDNA deletion, the exact deletion ranges of mtDNA were identified by sequencing. A full ophthalmologic examination including prism and alternate cover test in the primary position, evaluation of ductions and versions, and binocularity was performed in 10 patients with confirmed mtDNA deletions associated with CPEO. Results All of the patients showed ophthalmoplegia as well as ptosis, even after eyelid surgeries. Ophthalmoplegia was symmetric between both eyes in nine patients (90%) while one patient (10%) showed asymmetric ophthalmoplegia with esotropia and left hypotropia. Among the nine patients with symmetric involvement, four patients (44%) showed exotropia, three (33%) had exotropia with vertical deviation, and the remaining two patients (22%) showed orthotropia. Five out of 10 patients (50%) complained of diplopia associated with strabismus, four of whom (80%) had vertical deviation. Three out of five patients (60%) without diplopia showed exotropia of 20 prism diopters (PD) to 50 PD. Conclusions Exotropia with/without vertical deviation is the most common form of strabismus in Asian patients with CPEO and only one of them showed a small angle of esotropia. Ophthalmoplegia could be asymmetric in 10% of CPEO patients.

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“…Prisms can also be helpful for small residual angles, particularly as there is suggestion that CPEO patients can have narrower fusional amplitudes [ 18 ]. Botulinum toxin alone, although insufficient for the larger exodeviations seen in CPEO, may be particularly useful for residual angles that may develop post-surgically [ 19 , 20 ].…”

Section: Extraocular Manifestationsmentioning

confidence: 99%

Mitochondria and the eye—manifestations of mitochondrial diseases and their management

Chen

Harvey

Gilhooley

et al. 2023

Eye

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Historically, distinct mitochondrial syndromes were recognised clinically by their ocular features. Due to their predilection for metabolically active tissue, mitochondrial diseases frequently involve the eye, resulting in a range of ophthalmic manifestations including progressive external ophthalmoplegia, retinopathy and optic neuropathy, as well as deficiencies of the retrochiasmal visual pathway. With the wider availability of genetic testing in clinical practice, it is now recognised that genotype-phenotype correlations in mitochondrial diseases can be imprecise: many classic syndromes can be associated with multiple genes and genetic variants, and the same genetic variant can have multiple clinical presentations, including subclinical ophthalmic manifestations in individuals who are otherwise asymptomatic. Previously considered rare diseases with no effective treatments, considerable progress has been made in our understanding of mitochondrial diseases with new therapies emerging, in particular, gene therapy for inherited optic neuropathies.

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The Management of Strabismus in Patients with Chronic Progressive External Ophthalmoplegia

Cited by 15 publications

References 13 publications

Myogenic Palsies

Myogenic Palsies

Strabismus in chronic progressive external ophthalmoplegia

Mitochondria and the eye—manifestations of mitochondrial diseases and their management

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