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BMJ Case Rep. 2010; 2010: bcr0220091619.
Published online 2010 August 3. doi:  10.1136/bcr.02.2009.1619
PMCID: PMC3027569
Unusual association of diseases/symptoms

Acute dacryoadenitis in Crohn's disease

Abstract

A 48-year-old Filipino female presented with unilateral acute onset painful red eye, blurred vision and yellow discharge. On examination she had a corrected visual acuity of 6/5 in the right eye and 6/18 in the left eye.

There was a left-sided periorbital swelling, with chemosis involving the bulbar conjunctiva on the temporal aspect. Ocular motility showed limitation of left-sided abduction with mild limitation of laevoelevation and laevodepression. She was afebrile and systemic examination was unremarkable. Medical history included diagnosis of Crohn's disease since the age of 20. She was on oral mesalamine 1 g for mildly active colitis. Full blood count was normal but erythrocyte sedimentation rate and C reactive protein were raised. Blood culture and conjunctival swab were negative. Contrast-enhanced CT scan demonstrated enlargement of the lacrimal gland. She was managed conservatively with acetaminophen and codeine for pain and swelling.

She recovered completely in 2 weeks with no sequelae.

Background

  1. Acute dacryoadenitis is a very rare association with Crohn's disease.
  2. Non-infective dacryoadenitis should be considered among the differential diagnosis in patients with established Crohn's, presenting with painful eye and ocular motility dysfunction.
  3. The eye condition may present even if the systemic disease is minimally active or under remission.
  4. The dacryoadenitis is self-limiting, and does not require immunosuppression for cure.

Case presentation

A 48-year-old Filipino female presented with a sudden onset of pain, photophobia, blurred vision and yellow discharge from her left eye. On examination corrected visual acuity was 6/5 in right eye and 6/18 in the left eye. There was left-sided periorbital swelling with tenderness along the superotemporal orbital margin. The bulbar conjunctiva was congested, with marked chemosis involving the temporal region. Ocular motility showed limitation of abduction in the left eye along with mild limitation of elevation and depression. Rest of the eye examination was normal. The right eye did not show any abnormality of note.

Medical history was significant with a diagnosis of Crohn's disease since the age of 20. At the time of presentation, she was on oral mesalamine 1 g twice daily for mildly active colitis. She was afebrile and her systemic examination was unremarkable.

There was significant improvement in her condition within 48 h. After 3 days her visual acuity had improved to 6/6 in the left eye. The preseptal swelling was showing signs of resolution and her eye movements returned to normal at the end of first week. At 2 weeks of diagnosis her visual acuity was 6/6 with a completely normal examination.

Investigations

Full blood count was normal. Globulin, C reactive protein (64) and ESR (50 mm/h) were raised. Culture of the conjunctival discharge and blood did not show any organism.

Contrast-enhanced computerised axial tomography of the orbit showed preseptal swelling on the left side along with enlargement of the lacrimal gland (figure 1). There was no ocular, retrobulbar or bony involvement.

Figure 1
Contrast-enhanced computerised axial tomography of the orbit showed preseptal swelling on the left side along with enlargement of the lacrimal gland.

Differential diagnosis

  1. Acute infective dacryoadenitis
  2. Pseudotumour of the orbit
  3. Intra orbital mass.

Treatment

She was treated initially with intravenous flucloxacillin 1 g twice daily with a presumed diagnosis of acute infective dacryoadenitis. This had to be stopped the following day due to intolerance to the antibiotic. In the absence of worsening of symptoms the antibiotic was not substituted. She was maintained on oral acetaminophen and codeine for symptomatic relief of pain and swelling.

Outcome and follow-up

At 2 weeks of diagnosis there was complete resolution with no sequelae.

Discussion

Orbital pseudotumour is an idiopathic orbital inflammatory disorder, which affects primarily adults.1 It can present as generalised inflammation of the orbit or localised inflammation as dacryoadenitis, myositis, periscleritis and perineuritis.2 There are few reports in the literature showing association of orbital inflammation and Crohn's disease.35 To the best of our knowledge there are only two case reports in Medline showing association of Crohn's disease with dacryoadenitis.6 7

Dutt et al6 reported a case of dacryoadenitis in a 14-year-old boy with Crohn's disease. His systemic disease was on remission and he was not on any medication at the time of presentation. The dacryoadenitis resolved with a short course of systemic steroids. Hwang et al7 reported dacryoadenitis in a 32-year-old woman, which responded well to a 6-week course of oral prednisolone. Crohn's disease was diagnosed 5 months after her ophthalmic presentation. Unlike previous report, Crohn's disease was mildly active in our case, which suggests concurrent systemic activity of the disease. Our patient did not receive any steroid therapy as her condition started to improve within a few days of presentation and recovered completely in 2 weeks without any sequelae. We suggest initial observation with supportive treatment of pain for isolated dacryoadenitis in patients with mildly active Crohn's disease. To the best of our knowledge this is the first case in the literature of spontaneous resolution of dacryoadenitis in a patient with mildly active Crohn's disease.

Learning points

  • Consider dacryoadenitis in the differential diagnosis of patients with Crohn's, presenting with periorbital swelling and ocular motility dysfunction.
  • In the absence of toxic symptoms, antibiotics or immunosuppression is unnecessary as the disease may resolve spontaneously.

Footnotes

Competing interests None.

Patient consent Obtained.

References

1. Rothfuss KS, Stange EF, Herrlinger KR. Extraintestinal manifestations and complications in inflammatory bowel diseases. World J Gastroenterol 2006;12:4819–31. [PubMed]
2. Weber AL, Romo LV, Sabates NR. Pseudotumor of the orbit. Clinical, pathologic, and radiologic evaluation. Radiol Clin North Am 1999;37:151–68, xi. [PubMed]
3. Weinstein JM, Koch K, Lane S. Orbital pseudotumor in Crohn's colitis. Ann Ophthalmol 1984;16:275–8. [PubMed]
4. Verbraeken H, Ryckaert S, Demets W. Pseudotumor of the orbit and Crohn's disease. Bull Soc Belge Ophtalmol 1984;210:65–72. [PubMed]
5. Smith JW. Orbital pseudotumor and Crohn's disease. Am J Gastroenterol 1992;87:405–6. [PubMed]
6. Dutt S, Cartwright MJ, Nelson CC. Acute dacryoadenitis and Crohn's disease: findings and management. Ophthal Plast Reconstr Surg 1992;8:295–9. [PubMed]
7. Hwang IP, Jordan DR, Acharya V. Lacrimal gland inflammation as the presenting sign of Crohn's disease. Can J Ophthalmol 2001;36:212–13. [PubMed]

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