Images of a patient with ocular vaccinia. A, Postgadodiamide spin-echo T1-weighted axial magnetic resonance image with fat saturation showing pronounced thickening and enhancement of the left preseptal soft tissue, left conjunctival thickening, inflammation extending into the postseptal extraconal fat around the left medial rectus, and left-sided retrobulbar fat stranding and enhancement. R indicates right; P, posterior. B, Ear lesion at the 1-week follow-up. C, Epithelial cells in a fibrin matrix with abundant eosinophilic cytoplasm and Guarnieri bodies (hematoxylin-eosin, original magnification ×400).
Periorbital edema and severe restriction of extraocular movements in the 9 cardinal positions of gaze.
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Ocular Vaccinia With Severe Restriction of Extraocular Motility. Arch Ophthalmol. 2009;127(12):1686–1693. doi:10.1001/archophthalmol.2009.319
Copyright 2009 American Medical Association. All Rights Reserved. Applicable FARS/DFARS Restrictions Apply to Government Use.2009
Ocular vaccinia reemerged amidst the Department of Defense Smallpox Vaccination Program launched in December 2002. Vaccinia is an Orthopoxvirus used in research and for immunization against smallpox virus. Blepharoconjunctivitis is a common manifestation usually appearing after autoinoculation from a weeping pustule, with keratitis occurring less frequently.1 We describe a patient with motility disturbances and orbital involvement suggestive of orbital cellulitis. Motility involvement in ocular vaccinia has been reported rarely, with the last known case report from 1927.2
A 24-year-old male laboratory technician visited the emergency department with marked swelling of his right ear and left eye, which he mistakenly attributed to a bee sting. A diagnosis of orbital cellulitis was made in the emergency department. An immediate computed tomographic scan demonstrated preseptal soft-tissue swelling, and a magnetic resonance image 2 days later showed a worsening preseptal inflammatory process with intraorbital extension (Figure 1A). On initial examination, visual acuity was 20/15 OD and 20/40 OS, auricular and periorbital edema was pronounced, and tender lymphadenopathy was noted. No eyelid vesicles or pustules were observed. Marked chemosis and a purulent discharge were present, but the cornea was clear. Motility examination revealed full ductions in the right eye with severe limitation in all fields in the left eye (Figure 2). A 1 × 1-cm weeping brown skin lesion surrounded by induration, erythema, and crusting was found on the posterior pinna (Figure 1B). Treatment with intravenous vancomycin hydrochloride, clindamycin phosphate, and piperacillin sodium with tazobactam sodium was started.
A conjunctival pseudomembrane obscuring the cornea was removed on the second hospital day and sent for pathologic analysis (Figure 1C), and a symblepharon discovered medially in the left eye was lysed. Neomycin sulfate, polymyxin B sulfate, and dexamethasone ophthalmic ointment was initiated. Four days after admission, MRC-5, A549, and primary rhesus monkey kidney cell cultures from the eye and ear discharge showed viral cytopathic effect consistent with vaccinia. All other bacterial and viral cultures were negative. The Centers for Disease Control and Prevention and the public health department were notified and the vaccinia strain was identified by polymerase chain reaction. After diagnosis, when specifically questioned about any possible exposure to vaccinia, the patient recalled the potential for autoinoculation approximately 14 days prior to the positive culture. He suggested that at that time he may have touched his ear with a gloved hand contaminated by infected blood while performing phlebotomy on experimentally inoculated research mice. The patient had not been previously vaccinated. Because of rapid improvement and the late stage at diagnosis, cidofovir and vaccinia immunoglobulin therapy were not started.
Ten days after admission, motility testing showed grossly full ductions in each eye and Maddox rod testing showed trace incomitant esodeviation and left hyperdeviation. Diffuse corneal haze in the left eye with an epithelial healing pattern and 1 small corneal defect were noted. No deep stromal changes or corneal thinning were observed, and topical therapy was changed to prednisolone acetate eyedrops. Continued follow-up over months revealed steady improvement, with a return to baseline motility and a small nummular area of slowly improving corneal haze. A functionally insignificant symblepharon and mild ptosis were noted sequelae. The final visual acuity was 20/15 OU.
We found this case to be interesting in its dramatic clinical appearance, mimicking orbital cellulitis, and the transient motility disturbances seen that were likely secondary to extensive edema and inflammation extending intraorbitally from the preseptal process. Hu et al3 reported a somewhat similar case with considerable preseptal inflammation and a small conjunctival membrane but without restricted motility. Long-term sequelae develop in 18% of ocular vaccinia cases with corneal involvement,1 and the symblepharon and mild ptosis were examples in this case. With the large-scale current vaccination program, clinicians should consider ocular vaccinia in unusual manifestations of blepharoconjunctivitis. Given that 5 cases of laboratory-acquired general vaccinia infection were reported to the Centers for Disease Control and Prevention between 2005 and 2007,4 proper personal protection and immunization guidelines should also be emphasized in laboratories using vaccinia.
Correspondence: Mr Baynham, Department of Ophthalmology, University of Virginia Health System, PO Box 800715, Charlottesville, VA 22908 (email@example.com).
Financial Disclosure: None reported.