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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 2  |  Issue : 1  |  Page : 204-206

A case of adult-onset cyclic esotropia combined with hypotropia


Department of Pediatric Ophthalmology and Strabismus, Tianjin Eye Hospital, Clinical College of Ophthalmology of Tianjin Medical University, Tianjin Key Laboratory of Ophthalmology and Vision Science, Tianjin, China

Date of Submission17-Apr-2021
Date of Acceptance07-May-2021
Date of Web Publication07-Jan-2022

Correspondence Address:
Dr. Yueping Li
Tianjin Eye Hospital, Tianjin Key Laboratory of Ophthalmology and Vision Science, Tianjin - 300 020
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_877_21

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  Abstract 


A 39-year-old woman presented with 48 hours periodic esotropia and hypotropia after vitreoretinal surgery for retinal detachment and then loss of vision in the left eye. The ocular motility revealed the limitation of supraduction and abduction of the left eye on strabismic days and full duction on good days. After strabismic surgery only for correcting esotropia, the patient changed to hypotropia on the previous strabismic days and exotropia on the previous good days alternatively in a 48-hour period. Its pathogenesis is still unknown. We supposed the patient had similar neurological mechanisms as ocular neuromyotonia and secondary cyclic oculomotor palsy.

Keywords: Cyclic esotropia, ocular motility, strabismic surgery


How to cite this article:
Li Y, Zhang W. A case of adult-onset cyclic esotropia combined with hypotropia. Indian J Ophthalmol Case Rep 2022;2:204-6

How to cite this URL:
Li Y, Zhang W. A case of adult-onset cyclic esotropia combined with hypotropia. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 Jan 28];2:204-6. Available from: https://www.ijoreports.in/text.asp?2022/2/1/204/334986



Cyclic strabismus is a rare form of strabismus, of which the prevalence is 1 in 3,000 to 1 in 5,000. The childhood-onset cyclic esotropia in a 48-hour cycle is the most common. The 72-hour and 96-hour cycles, cyclic exotropia, and cyclic vertical deviation have also been reported in the literature. Strabismic surgery is the effective treatment for cyclic deviation and aims to correct the maximum deviation presenting on a strabismic day.[1] The adult-onset cyclic strabismus is extremely rare. It could be associated with severe monocular vision impairment in the cycling eye,[2],[3],[4],[5],[6] underlying neurological lesion,[7] and autoimmune diseases.[8] Unlike childhood-onset form, the surgical outcomes in adult-onset cyclic deviation are variable in previous reports.


  Case Report Top


A 39-year-old woman presenting with periodic esotropia (ET) and hypotropia (HypoT) was referred to our department for consultation and further treatment. The patient underwent vitreoretinal surgery for retinal detachment on the left eye 6 years ago and then suffered a total loss of vision in the left eye. She stated that periodic misalignment on the left eye occurred suddenly 5 years ago and since then the condition had been stable. She did not have diabetes, hypertension, or thyroid disease. She had no history of strabismus, amblyopia, patching therapy, ocular trauma, or family history of strabismus.

Manifest refractions and visual acuity were +0.25 diopter (D), 20/20 of the right eye, and −5.00 D, light perception of the left eye. The anterior segments were normal in both eyes. The fundus examination revealed the pallor papillae, narrow and straight vasculars, and scattered retinal pigmentation in the left eye but unremarkable manifestation in the right eye. The pupil of the left eye had a relative afferent pupillary defect without episodic mydriasis or constriction. The deviation was measured on 14 consecutive days. The period of the cyclic ET combined with HypoT was 48 hours. On good days, the left eye was orthotropic with full ocular motility [Figure 1]a. On strabismic days, the left eye presented 60 (prism diopter) ET and 15 △HypoT on the Krimsky test accompanied with mild to moderate limitations of supraduction and abduction [Figure 1]b. Cranial and orbital lesions were ruled out by magnetic resonance imaging. Laboratory tests of thyroid function and thyroid-related antibodies were normal. We prescribed carbamazepine (200 mg, twice a day) for 7 days. However the patient did not respond to the medicine.
Figure 1: Preoperative nine-gaze photographs (a) on “ good days”, orthotropia with full ocular motility, (b) on “strabismic days,” 60Δ ET and 15Δ hypotropia accompanied with moderate limitation of supraduction and mild limitation of abduction on the left eye

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Considering the stable period and large ET, the strabismic operation was scheduled on a strabismic day. Forced duction test under sedation revealed unremarkable restrictions from the extraocular muscles. A 5.5 mm medial rectus recession and an 8 mm lateral rectus resection with adjustable sutures were performed on the left eye for correcting ET. At the time of adjustment 24 hours later, the patient acquired orthotropia in the primary position and then the sutures were tied without any adjustment. Within 2 weeks after surgery, the patient maintained alignment and full ocular motility. After that, the left eye manifested 10 △ HypoT accompanied by mild limitation of supraduction on previous strabismic days [Figure 2]a and 30 △ exotropia (XT) with full ocular movement on previous good days [Figure 2]b. XT alternating with HypoT on a 48-hour cycle persisted for 1.5 years till now.
Figure 2: Postoperative photographs (a) 10Δ hypotropia in the primary position on previous “strabismic days,” (b) 30Δ XT in the primary position on previous “good days”

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  Discussion Top


Reviewing the literature on cyclic strabismus with adult-onset, the etiology is different from that in the childhood-onset form. Its pathogenesis is still unknown. Severely monocular visual impairment in cycling eye is the most common, which could be due to aphakia without spectacle correction,[2] optic nerve atrophy,[3] retinal detachment,[4] retinitis pigmentosa,[5] and so on. Fusion potential and binocular vision might be unnecessary for adult-onset cyclic strabismus because of severe monocular vision impairment. However, adult-onset cyclic strabismus could also occur in patients with normal vision.[9],[10] Other underlying causes and diseases might be neurological disease, craniofacial surgery, Graves' disease, and radiation therapy,[9],[10] especially in cases with cyclic vertical strabismus and limitation of ocular movement.[7],[8]

Some authors supposed it could be related to ocular neuromyotonia (ONM) due to responding to the treatment of membrane-stabilizing medications.[7],[11] Another rare type of cyclic strabismus is cyclic oculomotor palsy, of which the feature is spasm phase spontaneously occurring and lasting for 10 to 30 seconds and then paretic phase lasting for 2 to 3 minutes. The patient with cyclic oculomotor palsy secondary to radiotherapy for cerebellar tumor had response to carbamazepine therapy in the previous reports.[12] Therefore, cyclic oculomotor palsy, ONM, and adult-onset cyclic deviation were supposed to have a similar neurological mechanism.[12],[13] But our patient did not have any underlying disease and had no response to the membrane-stabilizing medications.

The surgical outcomes in adult-onset cyclic deviation are variable and unpredictable. Most of the patients might obtain alignment after strabismic surgeries. However, persistent previous cycles,[3] cyclic exotropia,[6] or residual microdeviation without cycle[7],[8] could be presented postoperatively. The patient with poor vision might incline to secondary cyclic exotropia after strabismic surgery. It is noteworthy that the cyclic deviation could be ceased after correcting the high hyperopia in cases with aphakia[2] and after treatment for the underlying disease.[8]


  Conclusion Top


In conclusion, adult-onset cyclic strabismus is very rare, of which the etiology, underlying disease, management, and prognosis are different from the childhood-onset form. It is key to exclude neurological and autoimmune diseases through comprehensive examinations and to treat the underlying diseases. The surgical prognosis should be taken into consideration. To our knowledge, this is the first report that a patient with cyclic ET combined with HypoT converted into cyclic XT in the contrary period but still maintained the same period of cyclic vertical deviation after a strabismic operation. Furthermore, the patient needs to be followed up in the long term.

Acknowledgments

We would like to thank Dr. Xia Chen for advising management for the patient.

Ethics approval and consent to participate

The study was approved by the review board of Tianjin Eye Hospital. Written informed consent was obtained from the patient.

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

This study was supported by Science and Technology Found of Tianjin Health Commission (ZC20187)

Science and Technology Found of Tianjin Eye Hospital (YKZD2001).

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Von Noorden GK, Campos EC. Binocular Vision and Ocular Motility: Theory and Management of Strabismus. St. Louis: C.V. Mosby Company; 2002. p. 480-2.  Back to cited text no. 1
    
2.
Cole MD, Hay A, Eagling EM. Cyclic esotropia in a patient with unilateral traumatic aphakia: Case report. Br J Ophthalmol 1988;72:305-8.  Back to cited text no. 2
    
3.
Frenkel RE, Brodsky MC, Spoor TC. Adult-onset cyclic esotropia and optic atrophy. J Clin Neuroophthalmol 1986;6:27-30.  Back to cited text no. 3
    
4.
Troost BT, Abel L, Noreika J, Genovese FM. Acquired cyclic esotropia in an adult. Am J Ophthalmol 1981;91:8-13.  Back to cited text no. 4
    
5.
Hwang JM, Kim J. Cyclic exotropia associated with retinitis pigmentosa. Graefes Arch Clin Exp Ophthalmol 2006;244:1549-51.  Back to cited text no. 5
    
6.
Garg SJ, Archer SM. Consecutive cyclic exotropia after surgery for adult-onset cyclic esotropia. J AAPOS 2007;11:412-13.  Back to cited text no. 6
    
7.
Roper-Hall G, Cruz OA, Espinoza GM, Chung SM. Cyclic (alternate day) vertical deviation–possible forme fruste of ocular neuromyotonia. J AAPOS 2013;17:248-52.  Back to cited text no. 7
    
8.
Paik JS, Yan SW, Park SH. A cyclic vertical deviation with dysthyroid ophthalmopathy: A case report. BMC Ophthalmol 2016;16:119.  Back to cited text no. 8
    
9.
Ngo CS, Araya MP, Kraft SP. Cyclic strabismus in adults. J AAPOS 2015;19:279-81.  Back to cited text no. 9
    
10.
Di Meo A, Costagliola C, Della Corte M, Romano A, Foria C, Di Costanzo A. Adult-onset cyclic esotropia. Optom Vis Sci 2013;90:e95-8.  Back to cited text no. 10
    
11.
Chung SM, Lee AG, Holds JB, Roper-Hall G, Cruz OA. Ocular neuromyotoniain in Graves dysthyroid orbitopathy. Arch Ophthalmol 1997;115:365-70.  Back to cited text no. 11
    
12.
Gadoth A, Kipervasser S, Korczyn AD, Neufeld MY, Kesler A. Acquired oculomotor nerve paresis with cyclic spasms in a young woman, a rare subtype of neuromyotonia. J Neuroophthalmol 2013;33:247-8.  Back to cited text no. 12
    
13.
Miller NR, Lee AG. Adult-onset acquired oculomotor nerve paresis with cyclic spasms: Relationship to ocular neuromyotonia. Am J Ophthalmol 2004;137:70-6.  Back to cited text no. 13
    


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