Acute-onset dacryoadenitis following immunisation with mRNA COVID-19 vaccine.

A 14-year-old boy was referred to the ophthalmology department with a 4-day history of rapid-onset right upper lid pain, swelling and erythema starting 9 hours after his first dose of COVID-19 mRNA vaccination (BNT162b2/Comirnaty, Pfizer-BioNTech). On examination, he had significant right upper lid ptosis, oedema and erythema, with associated limitation of right eye abduction and elevation. He was found to have acute dacryoadenitis with orbital inflammatory disease on clinical and laboratory investigations. He was given tapering oral prednisone and had full resolution of symptoms within 2 weeks. This is the first known case of orbital inflammation after COVID-19 mRNA vaccination. Given the temporal association between the patient's vaccination and symptom onset, we believe it is likely that immunisation prompted the onset of disease. © BMJ Publishing Group Limited 2022. No commercial re-use. See rights and permissions. Published by BMJ.

Background

The lacrimal gland is an integral component of the mucosal immune system, which protects the ocular surface.1 Acute dacryoadenitis is characterised by rapid enlargement of the lacrimal gland. It is often caused by viral or autoimmune disease.2

Ocular manifestations of the novel coronavirus (SARS-CoV-2) include changes consistent with follicular conjunctivitis in up to 32% of patients,3 with reports of anterior segment and posterior segment inflammatory4 5 and vascular changes6 and rare cases of orbital inflammation.2 7 8

Given the recent introduction of COVID-19 mRNA vaccines, data on short-term and long-term side effects are still evolving. Several case reports have noted the development of intraocular immunological and inflammatory events after COVID-19 19mRNA vaccination,9 including cranial nerve palsies,10 11 uveitis,12 13 episcleritis,14 anterior scleritis,12 acute-onset central serous chorioretinopathy15 and acute macular neuropathy16 within 72 hours of receiving COVID-19 mRNA vaccinations.

To date, orbital inflammatory side effects of COVID-19 mRNA vaccines have not been reported. We present a healthy teenage boy who presented with unilateral acute-onset dacryoadenitis, 9 hours after receiving a first dose of COVID-19 mRNA vaccine.

Case report

A 14-year-old previously healthy boy, presented with a 4-day history of rapid-onset right upper lid pain, swelling and erythema starting 9 hours after his first dose of COVID-19 mRNA vaccination (BNT162b2/Comirnaty, Pfizer-BioNTech). There was no history of fever or respiratory symptoms. Systemic review was unremarkable. There was no recent travel history or history of COVID-19 infection.

On examination, visual acuities were 6/6 in each eye. The patient had right upper lid S-shaped ptosis, erythema and oedema (figure 1). There was conjunctival chemosis and significant conjunctival injection was noted over the right lateral rectus muscle. The right globe was mildly proptosed with inferomedial globe displacement. Abduction and elevation of the right eye were severely limited with associated diplopia. Anterior segment and dilated fundal examination were normal.

Figure 1

Macroscopic photograph of both eyes. Right upper lid ptosis, erythema and oedema, with associated conjunctival injection and chemosis.

Investigations

CT scan with intravenous contrast demonstrated an elliptical rim-enhancing collection at the right lacrimal gland that was enlarged compared with the left side. There was associated thickening of the lateral rectus and stranding of the overlying subcutaneous soft tissue anteriorly. There was no associated sinus disease (figure 2).

Figure 2

Coronal CT scan. Enlargement of the right lacrimal gland and lateral rectus with associated preseptal and eyelid oedema.

Laboratory testing showed white blood cell count 8.2×109 /L, and C reactive protein 10 mg/L. Electrolytes, creatinine, hepatic transaminases, gamma glutamyltransferase, bilirubin, uric acid, lactate dehydrogenase and thyroid stimulating hormone were normal. Blood cultures and conjunctival swabs were negative. Epstein-Barr virus IgG and IgM antibody titres were negative.

Based on clinical and laboratory investigations, this patient was diagnosed with acute dacryoadenitis with associated orbital inflammatory disease.

Treatment

The patient was admitted to hospital and started on empiric treatment with intravenous ceftriaxone and metronidazole two times daily. However, after noting no improvement after 1 day, he was started on oral prednisone 60 mg daily.

Outcome and follow-up

After 2 days, the pain had resolved, and the lid and conjunctival inflammation were improved. Eye movements were now full. Intravenous antibiotics were discontinued. The prednisone dose was tapered to zero over 2 weeks. The patient subsequently declined his second COVID-19 immunisation.

Discussion

We report acute dacryoadenitis with associated ophthalmoplegia in a 14-year-old boy occurring 9 hours after COVID-19 mRNA vaccination. To our knowledge, this is the first known case of orbital inflammation after COVID-19 mRNA vaccination. Given the temporal association between the patient’s vaccination and symptom onset, we believe it is likely that immunisation prompted the onset of disease. However, as noted in other case reports, it is unknown whether this is a coincidental or causal relationship.

Four cases of orbital inflammation secondary to COVID-19 infection have been reported.2 7 8 All patients were sero-positive for Sars-CoV-2 infection and were otherwise asymptomatic. One of these patients had dacryoadenitis,2 one had dacryoadenitis with associated orbital myositis8 and two had non-specific intraconal orbital inflammatory changes with associated sinusitis.7

Ng et al recently comprehensively reviewed the ocular side effects of COVID-19 vaccination.14 The most prevalent complications of mRNA vaccination were Bell’s/facial nerve palsy (n=30), followed by corneal graft rejection (n=6), panuveitis (n=2), acute abducens nerve palsy (n=1), reactivation of Vogt-Koyanagi-Harada disease (n=2), acute central serous retinopathy (n=1), activation of Graves’ disease (n=2), acute macular neuroretinopathy (n=1) and central retinal vein occlusion (n=2).

Furthermore, Lin et al9 recently described adverse ocular outcomes of acute abducens nerve palsy, uveitis, Vogt-Koyanagi-Harada disease activation, anterior ischaemic optic neuropathy and acute corneal graft rejection after Pfizer-BioNTech vaccination. Testi et al12 reported 70 patients who presented with ocular inflammatory events within 14 days of COVID-19 vaccination, including anterior and posterior uveitis, and scleritis. Over half of this cohort had a previous ocular inflammatory event.

The contribution of the lacrimal gland to the ocular mucosal immune system is well described; committed B and T cells in the gland produce surface IgA and IgG, respectively.1 IgA plays a central role in the body’s local mucosal immune response. A recent paper by Jalkanen et al17 noted a significant increase of serum anti-SARS-CoV-2 (anti-S1) IgG and moderate increase of anti-S1 IgA in BNT162b2/Comirnaty vaccine recipients 3 weeks after immunisation. Furthermore, recent research by Muyldermans et al18 showed the presence of SARS-CoV-2 IgA and IgG antibodies in the tear film of convalescent COVID-19 infected individuals compared with control patients. We speculate that this patient developed an acute-onset dacryoadenitis and orbital inflammatory syndrome secondary to lacrimal gland immunological response to the COVID-19 mRNA vaccination, although a causal relationship cannot be confirmed at this stage.

Nine hours after my first dose of vaccine, I noticed that my right eye was really swollen and sore. I didn’t really get a fright. We went to the hospital, and I just trusted the doctors and nurses to make things better. I’m doing fine now. I’m glad that other people get to hear about my case. My advice to anyone in a similar situation would be to stay calm. What happened to me is really rare, I’m not going to frighten my friends about the vaccine. I do feel a bit worried about getting my second dose and I hope this doesn’t happen again.

This is the first described case of orbital inflammation after COVID-19 mRNA vaccination.

This patient presented with classical dacryoadenitis clinical findings.

Treatment with a tapering dose of oral prednisone led to full resolution of this patient’s inflammation.

Acute dacryoadenitis and orbital inflammatory syndrome due to local immunological response may potentially occur after COVID-19 mRNA immunisation, although a causal relationship cannot be confirmed at this stage.