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   Table of Contents      
BRIEF COMMUNICATION
Year : 2017  |  Volume : 65  |  Issue : 11  |  Page : 1256-1261

Spontaneous anatomical and functional recovery of bilateral electric shock maculopathy


Department of Vitreo-Retina, Aravind Eye Hospital and Postgraduate Institute of Ophthalmology, Coimbatore, Tamil Nadu, India

Date of Submission25-Jun-2017
Date of Acceptance27-Sep-2017
Date of Web Publication13-Nov-2017

Correspondence Address:
Ratnesh Ranjan
Department of Vitreo-Retina, Aravind Eye Hospital and Postgraduate Institute of Ophthalmology, Avinashi Road, Coimbatore - 641 014, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_536_17

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  Abstract 

A 12-year-old boy presented with best-corrected visual acuity (BCVA) of 6/9 in both eyes following an episode of electric shock. Optical coherence tomography (OCT) showed disruption of the ellipsoid zone as well as retinal pigment epithelium (RPE) layer. Fundus autofluorescence (FAF) showed increased central hypoautofluorescence in both eyes. At 3-month follow-up, BCVA improved to 6/6 with OCT showing spontaneous resolution of maculopathy in both eyes with reorganized RPE layer and ellipsoid zone. To the best of our knowledge, this is the first case of bilateral electric shock maculopathy (ESM) with spontaneous anatomical as well as functional recovery. Ophthalmologists must be aware of various forms of ESM. OCT and FAF must be done in patients presenting with defective vision and history of electric shock for the diagnostic as well as prognostic evaluation.

Keywords: Electric shock, electric shock maculopathy, electrocution, maculopathy, outer lamellar hole


How to cite this article:
Ranjan R, Manayath GJ, Dsouza P, Narendran V. Spontaneous anatomical and functional recovery of bilateral electric shock maculopathy. Indian J Ophthalmol 2017;65:1256-61

How to cite this URL:
Ranjan R, Manayath GJ, Dsouza P, Narendran V. Spontaneous anatomical and functional recovery of bilateral electric shock maculopathy. Indian J Ophthalmol [serial online] 2017 [cited 2019 Oct 19];65:1256-61. Available from: http://www.ijo.in/text.asp?2017/65/11/1256/218070

Electrocution is a common mode of injury accounting for 5%–20% burns among all causes of burns.[1] Ocular complications resulting from electric shock injuries are well known but uncommon including eyelid skin burns, cataract, recurrent uveitis, optic neuropathy, and rarely maculopathies.[2] Cataract is the most common ocular manifestation of electrocution, but electric shock maculopathy (ESM) is very rarely reported in the literature. Herewith, we are reporting a case of ESM with spontaneous anatomical as well as visual recovery.


  Case Report Top


A 12-year-old boy visited our outpatient department with a complaint of blurring of vision in both eyes for 2 weeks following an episode of electric shock. Electrocution resulted from domestic accident with hand being the entry point and feet being the exit point. It also caused a brief period of unconsciousness. There was no history of ocular trauma, sun gazing, or exposure to a solar eclipse or welding arc. On examination, best-corrected visual acuity (BCVA) was 6/9 in both eyes. Anterior segments of both eyes were unremarkable. Both fundi showed a dark red spot at the central fovea and otherwise were within normal limits [Figure 1]a and [Figure 1]b. Spectral domain-optical coherence tomography (SD-OCT) [SPECTRALIS HRA + OCT, Heidelberg] showed disruption of the ellipsoid zone as well as retinal pigment epithelium (RPE) layer [Figure 2]a and [Figure 2]b. Fundus autofluorescence (FAF) [SPECTRALIS HRA + OCT, Heidelberg] showed increased central hypoautofluorescence in both eyes [Figure 3]a and [Figure 3]b. Based on the history, clinical findings, and imaging, a diagnosis of ESM was made. The patient was treated conservatively and reviewed after 3 months. At the 3-month follow-up visit, his BCVA improved to 6/6 in both eyes. OCT showed near-complete resolution of maculopathy with reorganized ellipsoid zone and minimal irregularities in RPE layer [Figure 2]c and [Figure 2]d. FAF showed a slight improvement in central hypoautofluorescence of both eyes [Figure 3]c and [Figure 3]d. To the best of our knowledge, this is the first case of ESM in the literature with bilateral spontaneous complete anatomical and functional recovery.
Figure 1: Color fundus photograph of right and left eye (a and b, respectively) at the time of first presentation showing dark red spot at fovea appearing as cherry red spot

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Figure 2: Optical coherence tomography image of right and left eye at the first visit showing disruption of ellipsoid zone and retinal pigment epithelium layer with intact external limiting membrane (a and b respectively). At 3-month follow-up, optical coherence tomography image showing intact external limiting membrane, spontaneously reorganized ellipsoid zone, and near normal retinal pigment epithelium layer (c and d, respectively

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Figure 3: Fundus autofluorescence image of right and left eye at the first visit showing increased central hypoautofluorescence (a and b, respectively) and relatively decreased central hypoautofluorescence at 3-month follow-up (c and d, respectively)

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  Discussion Top


ESM is a rare clinical entity with very few case reports available in the literature.[3],[4],[5],[6],[7],[8] Our case probably represents the mildest form of ESM characterized by disruption of RPE and photoreceptor layer and showing spontaneous resolution. However, the most commonly reported form of ESM is characterized by defect in RPE and outer retinal layers progressing to form outer lamellar hole with intact internal limiting membrane (ILM) over the bare RPE, with or without associated cystic changes noted on OCT. This form of ESM represents a more extensive or progressive damage from electric shock.[3],[4],[5] Full-thickness macular hole (FTMH) is a relatively rare form of ESM and represents the most severe injury from electric shock.[6],[7],[8]

Tissue damage from electric shock may occur through one or more mechanisms including direct transmission of electric current through the tissues causing cell membrane disruption, thermal burn of the tissues by absorbing thermal energy resulting from conversion of electrical energy, tissue ischemia caused by generalized vascular constriction, or mechanical injury resulting from high voltage shock wave.[6],[9] There are various factors which make the macula, particularly, sensitive for electrocution-related damages. Foveal avascularity increases the risk of ischemic insult of macula following electric shock-induced multifocal choroidal ischemia.[6],[10] Higher concentration of melanin pigments at macula due to thicker and more tightly packed submacular RPE cells causes more heat production and consequently more thermal damage.[4],[11] Macula being one of the sites of strongest vitreoretinal adhesion carries more risk of mechanical injury resulting from concussive forces of the electric shock and vitreomacular traction due to thermal shrinkage of the vitreous body.[6]

FAF imaging of our case showed mild central hypoautofluorescence without any parafoveolar ring of hyperautofluorescence and correlated well with good visual prognosis. However, the case reported by Arya et al. showed an increased central hypoautofluorescence surrounded by decreased parafoveolar hypoautofluorescence, which corresponded to a drop in visual acuity and progression of OCT changes.[12] Although FAF imaging was not reported in other cases of ESM, the importance of perifoveal ring of hyperautofluorescence as a predictive marker for the progression of other macular disorders such as macular dystrophy, cone dystrophy, retinitis pigmentosa, and hydroxychloroquine toxicity is well established.[13] Hence, it may be considered for ESM also to get significant prognostic input.

ESM may remain stationary, regress spontaneously, or progress over a period. Cases have shown an improvement in BCVA with corresponding tomographic improvement while fundus picture remaining similar to or appearing better than the initial presentation.[3],[4] Our case, probably being the mildest form of ESM, showed complete anatomical as well as visual recovery. Over a longer period, outer lamellar defect may undergo remodeling or gliosis and may be replaced by atrophic macular scar as reported by Bayar et al.[5] Arya et al. reported a decrease in BCVA with OCT showing ellipsoid zone defect progressing to OLH over a period of 3 months.[12] Case reported by Faustino et al. presented with outer lamellar hole with associated cystoid changes, which progressed to FTMH and rhegmatogenous retinal detachment in both eyes.[8]

The visual prognosis of ESM is generally good even with observation as seen in our as well as previously reported cases. Surgical intervention with ILM peeling and perfluoropropane (C3F8) gas tamponade has been described for one eye with outer lamellar defect with intact ILM.[11] Surgical intervention resulted in visual improvement from 6/30 to 6/19 with a decrease in lamellar hole size with persistent discontinuity of RPE and photoreceptor layers. The other eye of the same patient was kept under observation, and condition remained stable.[11] The presenting, as well as final visual outcome, depends on the severity of maculopathy and the presence of visually significant ocular comorbidities such as anterior segment and optic nerve involvement.[3],[4],[6] Hence, eyes presenting with outer retinal layer defect should be kept under observation with OCT and FAF imaging at every visit to monitor the progression or regression. For a better understanding of various aspects of this rare clinical entity, we have provided a comprehensive table of previously reported cases of ESM detailing mode of injury, presenting visual acuity, ocular comorbidities, features of maculopathy, management, and final visual acuity [Table 1].
Table 1: Summary of all previously reported cases of electric shock maculopathy

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  Conclusion Top


We describe a heretofore unreported case of ESM with documented bilateral spontaneous functional and anatomic recovery. This case report also highlights the possible role of FAF for prognostic evaluation of ESM. Ophthalmologists must be aware of the various forms of ESM. A history of electric shock exposure must be enquired in young patients presenting with such maculopathy without any history of sun gazing, exposure to solar eclipse, welding arc, or ocular trauma. Patients presenting with defective vision and history of electric shock must be examined on OCT, even in the absence of a clinically obvious maculopathy.[17]

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

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
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2.
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Miller BK, Goldstein MH, Monshizadeh R, Tabandeh H, Bhatti MT. Ocular manifestations of electrical injury: A case report and review of the literature. CLAO J 2002;28:224-7.  Back to cited text no. 3
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Sony P, Venkatesh P, Tewari HK, Garg SP. Bilateral macular cysts following electric burn. Clin Exp Ophthalmol 2005;33:78-80.  Back to cited text no. 4
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Bayar SA, Sarigul A, Yilmaz G, Pinarci EY. Development of optic neuropathy and foveal pseudocyst in a case of high-voltage electrical injury: A three-year follow-up. Turkish J Opthalmol 2014;44:410-2. Available from: http://www.oftalmoloji.org/article_7387/Development-Of-Optic-Neuropathy-And-FovealPseudocyst-In-A-Case-Of-High-voltage-Electrical-Injury-A-Three-year-Follow-up. [Last accessed on 2017 Apr 02].  Back to cited text no. 5
    
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Walkow T, Meyer-Rüsenberg HW. Macular hole, optic neuropathy, keratoconjunctivitis sicca and bilateral cataract after high-voltage injury. Klin Monbl Augenheilkd 2011;228:821-2.  Back to cited text no. 7
    
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Faustino LD, Oliveira RA, Oliveira AF, Rodrigues EB, Moraes NS, Ferreira LM, et al. Bilateral maculopathy following electrical burn: Case report. Sao Paulo Med J 2014;132:372-6.  Back to cited text no. 8
    
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Grover S, Goodwin J. Lightning and electrical injuries: Neuro-ophthalmologic aspects. Semin Neurol 1995;15:335-41.  Back to cited text no. 9
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Al Rabiah SM, Archer DB, Millar R, Collins AD, Shepherd WF. Electrical injury of the eye. Int Ophthalmol 1987;11:31-40.  Back to cited text no. 10
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Ouyang P, Karapetyan A, Cui J, Duan X. Bilateral impending macular holes after a high-voltage electrical shock injury and its surgical outcome: A case report. J Med Case Rep 2014;8:399.  Back to cited text no. 11
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Arya SA, Sarvanan VR, Narendran V. Outer lamellar hole with severe visual loss following high tension electric shock. Kerala J Ophthalmol 2013;25:385-7.  Back to cited text no. 12
    
13.
Fleckenstein M, Schmitz-Valckenberg S, Holz FG. Fundus autofluorescence imaging in clinical use. Rev Ophthalmol 2010. Available from: http://www.reviewofophthalmology.com/content/d/retinal_insider/i/1201/c/22655/. [Last accessed on 2017 Aug 26].  Back to cited text no. 13
    
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Manrique-Cerrillo M, Murillo-López S, Leizaola-Fernández C, Quiroz-Mercado H, Guerrero-Naranjo JL, Vargas-Castillo R, et al. Bilateral macular cysts secondary to electric current strike. A case report. Arch Soc Esp Oftalmol 2004;79:37-9.  Back to cited text no. 14
    
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Lakosha H, Tremblay F, De Becker I. High-voltage electrical trauma to the eye. Can J Ophthalmol 2009;44:605-6.  Back to cited text no. 15
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Tandon M, Agarwal A, Gupta V, Gupta A. Peripapillary retinal thermal coagulation following electrical injury. Indian J Ophthalmol 2013;61:240-2.  Back to cited text no. 16
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17.
Ravikumar M, Srinivas Y, Kiran PA. Bilateral macular hole after a high voltage electrical shock injury. Int J Sci Res 2015;4:2879-81. Available from: http://www.ijsr.net/archive/v4i4/SUB1537804.pdf. [Last accessed on 2017 Apr 02].  Back to cited text no. 17
    


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