|Year : 2015 | Volume
| Issue : 2 | Page : 116-118
Sympathetic ophthalmia in a case of penetrating ocular injury associated with brain injury: A case report
Thokchom Gojendra Singh1, Keisham Sorojini Devi2, Laithangbam Pradipkumar3, Nongthombam Ratan Singh3
1 Department of Surgery, Neurosurgery Unit, Regional Institute of Medical Sciences (RIMS), Imphal, Manipur, India
2 Department of Ophthalmology, Jawaharlal Nehru Institute of Medical Sciences, Imphal, Manipur, India
3 Department of Anaesthesiology, Regional Institute of Medical Sciences (RIMS), Imphal, Manipur, India
|Date of Web Publication||20-Aug-2015|
Department of Anaesthesiology, Regional Institute of Medical Sciences (RIMS), Imphal, Manipur
Source of Support: None, Conflict of Interest: None
Sympathetic ophthalmia is a rare bilateral granulomatous inflammation that follows accidental or surgical insult to the uvea of one eye. The diagnosis may be confounded by the associated traumatic brain injury. Here, we report a case of sympathetic opthalmia in a patient who sustained perforating injury of the right eye and traumatic brain injury in the left frontal area following a road traffic accident. Timely diagnosis and surgical intervention was the key to a successful outcome.
Keywords: Eye injury, Sympathetic ophthalmia, Traumatic brain injury
|How to cite this article:|
Singh TG, Devi KS, Pradipkumar L, Singh NR. Sympathetic ophthalmia in a case of penetrating ocular injury associated with brain injury: A case report. J Med Soc 2015;29:116-8
|How to cite this URL:|
Singh TG, Devi KS, Pradipkumar L, Singh NR. Sympathetic ophthalmia in a case of penetrating ocular injury associated with brain injury: A case report. J Med Soc [serial online] 2015 [cited 2020 Oct 27];29:116-8. Available from: https://www.jmedsoc.org/text.asp?2015/29/2/116/163207
| Introduction|| |
Sympathetic ophthalmia is a rare, bilateral nonnecrotising granulomatous uveitis.  The injured eye (exciting eye) and the contralateral eye (sympathetic eye) demonstrate similar pathology suggesting an autonomous pathology.  It appears to have no predilection toward any particular age, race, or gender.  The incidence of sympathetic ophthalmia is 0.2% to 0.5% following injury and 0.1% following intraocular injury. ,
The onset of sympathetic ophthalmia is variable, appearing anytime between 1 week and 66 years after the inciting injury.  It has been reported as early as 5 days after the injury, and in 90% of the cases, it occurred within a span of 1 year. 
Other cases of granulomatous uveitis must be considered before a diagnosis of sympathetic ophthalmia can be made, particularly Vogt-Koyanagi-Harada syndrome More Details (VKH) and sarcoidosis, both of which have a systemic involvement and absence of ocular injury. 
Fundoscopic examination in the early stage of the disease may reveal posterior segment (of the eye) changes, optic nerve swelling, exudative retinal detachment, and subretinal pigment epithelium [subretinal pigment epithelium (sub-RPE)], nodular lesions that appear yellow-white, corresponding to histopathologic Dalen-Fusch nodules. However, these nodules are not pathognomonic of sympathetic ophthalmia as they occur only in 25% to 35% of the cases. 
Enucleation  of the injured eye (if primary repair is not possible) within 2 weeks and immunosuppressive therapy with corticosteroids are currently the mainstay of treatment. Controversy has long existed surrounding enucleation versus evisceration. As there is substantial evidence of the risk for sympathetic ophthalmia following any intraocular surgery, any residual uveal tissue following evisceration has the potential to be the causative factor for sympathetic ophthalmia. However, no study has as yet proved it.
| Case report|| |
A 30-year-old male patient was brought to the emergency department around 9 PM on August 19, 2014 following a road traffic accident. On examination, the patient was unconscious with a score of 6 (E1V1M4) on the Glasgow coma scale (GCS). His right eyeball was ruptured with the contents almost completely extruded. The left pupil was 4 mm in size with sluggish reaction to light. Noncontrast computed tomography (NCCT) of the skull showed skull fracture in the left frontoparietal area with left frontal extradural hematoma [Figure 1] and with multiple pneumoencephaloceles.
|Figure 1 : Noncontrast computed tomography (NCCT) of the skull showing skull fracture in the left frontoparietal area with left frontal extradural hematoma and multiple pneumoencephaloceles|
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Other associated injuries were fracture of the right zygoma (undisplaced) and fracture of the mandible (lower ramus) in the midline. His vitals and emergency laboratory investigations were within normal limits. The patient was taken to the operation theater and left frontal craniotomy was performed under balanced general anesthesia (duration: 2.5 h). At the end of the operative procedure, residual neuromuscular blockade was reversed but the endotracheal tube (ET) was not extubated to allow the oxygen supplementation via T-piece connected to the ET tube, and the patient was shifted to the intensive care (ICU).
The next morning at around 8 AM [postoperative day (PoD)1], the patient's left pupil was 3 mm in size, reacting to light, and the GCS score was E2VetM5. However, on the second postoperative day, his left pupil was again dilated to 5-6 mm with no reaction to light. Emergency repeat computed tomography (CT) scan (NCCT of the head) was taken and there was a formation of large extradural hematoma (that could be due to the collection of blood trickled from the left frontoparietal fracture line connected to the superior sagittal sinus) [Figure 2]. Emergency reoperation was done for evacuation of the hematoma.
|Figure 2 : Recollection of blood and formation of hematoma revealed by the repeat CT scan (head)|
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On the next day (PoD3, counted from the earlier first operation), the patient was fully conscious but the left pupil remained dilated (5-6 mm) and was not reacting to light. The possibilities of recollection of blood were considered and repeat NCCT was again done on PoD4. This time, there was no extradural or intracerebral hematoma [Figure 3]. This prompted us to consult an ophthalmologist to rule out sympathetic ophthalmia. The fundoscopic examination finding was nonspecific except for optic nerve swelling and exudative retinal detachment in the posterior segment (PoD5). At this stage, his vision was difficult - finger counting at 3 ft (0.9 m).
|Figure 3 : Repeat CT scans (head) on POD4, showing resolution of the clot|
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On PoD5, his vision (left eye) deteriorated to only the perception of light. Hence, in consultation with an ophthalmologist and a plastic surgeon, enucleation of the right eye was performed. On PoD6, his left pupil regained normal size (~3 mm) and was reacting to light, and the patient was shifted from the ICU to another high dependency area.
| Discussion|| |
To our knowledge, ours is the first case report where the diagnosis of sympathetic ophthalmia was confounded by associated traumatic brain injury. On PoD1, the left pupil became normal compared to the preoperative condition. It is quite natural to assume that redilation of the pupil on PoD2 was due to recollection of blood and/or formation of hematoma, as was revealed by the repeat CT scan (head).
On PoD3, the patient became conscious and responded to verbal commands but the left pupil remained dilated and did not react to light. It is here that we started suspecting sympathetic ophthalmia since it is quite absurd to maintain consciousness while the only functioning eye remained dilated (without mydriatic eyedrops) and nonreacting to light.
The most convincing evidence was the regaining of the left pupil to normal size and reaction to light after enucleation of the badly traumatized (irreparable) right eye on PoD5. We did not give emphasis on the diagnosis of sympathetic ophthalmia on histopathological finding of Dalen-Fusch nodules, that are found only in one-third  of the cases.
The original hypothesis proposed by Mackenzie and his contemporaries  in the nineteenth century suggested the bilateral impact of sympathetic ophthalmia to be the result of inflammation in the injured eye that propagated along the optic nerve and chiasma to the contralateral eye while some have proposed it to be a hypersensitivity reaction to melanin.  Others have pointed to the role of cell-mediated immune response to antigens from the retinal photo receptor layer-like retinal soluble antigen (S-antigen). 
Genetic predisposition to the development of sympathetic ophthalmia has also been proposed with particular focus on human leukocyte antigen (HLA).  Growing evidence points to the role of autoimmunity in sympathetic ophthalmia with elevated gelatinase B (matrix metalloproteinase 9), monocyte chemotactic protein 1(MCP 1)/CCL2) and stromal cell derived factor-1(SDF-1/CXCL12).
The loss of vision in the absence of inflammatory infiltrate in the retina and choriocapillaris in sympathetic ophthalmia has long been an unanswered question. Upregulation of tumor necrosis factor alpha (TNFα) and induction of oxidative stress induced by inducible nitric oxide synthase (iNOS) and peroxynitrite (ONOO - ) might lead to nitration of photoreceptor mitochondria-related proteins (such as cytochrome C) triggering the apoptosis cascade. 
Oxidative stress-induced products are found to immunolocalize to the inner segments of the photoreceptors in sympathetic ophthalmia. Whether regaining vision in the present case following enucleation and immunosuppression by corticosteroid therapy could be due to interference with this cascade is a matter of speculation.
| Conclusion|| |
In conclusion, timely diagnosis and intervention are required to avoid any fatal outcome and prevent any unwanted effect.
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Conflicts of interest
There are no conflicts of interest.
| References|| |
Duke-Elder S, Perkins ES. Sympathetic ophthalmitis. In: Duke-Elder S, editor. Diseases of the Uveal Tract. St. Louis: The CV Mosby Company; 1966. p. 558-93.
Abu El-Asrar AM, Struyt S, Van den Broek C, Van Damne J, Opdenakker G, Geboes K, et al
. Expression of chemokines and gelatinase B in sympathetic ophthalmia. Eye (Lond) 2007;21:649-57.
Albert DM, Diaz-Rohena R. A historical review of sympathetic ophthalmia and its epidemiology. Surv Ophthalmol 1989;34:1-14.
Makley Ta Jr, Azar A. Sympathetic ophthalmia. A long-term follow-up. Arch Ophthalmol 1978;96:257-62.
Mark Ge Jr. Recent advances in sympathetic ophthalmia. Surv Ophthalmol 1979;24:141-56.
Zaharia MA, Lamarche J, Laurin M. Sympathetic uveitis 66 years after injury. Can J Ophthalmol 1984;19:240-3.
Goto H, Rao NA. Sympathetic ophthalmia and Vogt-Koyanagi-Harada syndrome. Int Ophthalmol Clin 1990;30:279-85.
Damico Fm, Kiss S, Young LH. Sympathetic Ophthalmia. Semin Ophthalmol 2005;20:191-7.
Lubin JR, Albert DM, Weinstein M. Sixty-five years of sympathetic Ophthalmia. A clinicopathologic review of 105 cases (1913-1978). Ophthalmology 1980;87:109-21.
Bilyk JR. Enucleation, evisceration and sympathetic ophthalmia. Curr Opin Ophthalmol 2000;11:372-86.
Mackenzie W. A Practical Treatise on the Diseases of Eye. 3 rd
ed. London: Longmans; 1840. p. 523-34.
Elschnig A. Studies on sympathetic ophthalmia, II: The antigenic effect of eye pigments. Graef Arch Clin Exp 1910;76:365-456.
de Kozak Y, Sakai J, Thillaye B, Faure JP. S antigen-induced experimental autoimmune uveo-retinitis in rats. Curr Eye Res 1981;1:327-37.
Shindo Y, Ohno S, Usui M, Ideta H, Harada K, Masuda H, et al
. Immunogenetic study of sympathetic ophthalmia. Tissue Antigens 1997;49:111-5.
Parikh JG, Saraswathy S, Rao NA. Photoreceptor oxidative damage in sympathetic ophthalmia. Am J Ophthalmol 2008;146:866-75.e2.
[Figure 1], [Figure 2], [Figure 3]