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Published on: October 13, 2017
Bilateral Idiopathic Dacryoadenitis in an Infant: A Case Report and Review of the Literature
Jai Paris1, Abdullah I Almater1,2,3, Khizar Rana1,4
1Department of Ophthalmology & Visual Sciences, University of Adelaide, North Terrace, Adelaide, South Australia, 5000, Australia, adelaide.edu.au.
Insights
Idiopathic dacryoadenitis, a rare condition in children, presents diagnostic challenges. Early imaging and biopsy are crucial for timely diagnosis and effective corticosteroid treatment in pediatric cases.
Area of Science:
- Ophthalmology
- Pediatric Medicine
- Pathology
Background:
- Dacryoadenitis, inflammation of the lacrimal gland, is typically caused by infections, inflammation, or tumors.
- Idiopathic dacryoadenitis is exceedingly rare in the pediatric population.
- This case involves a previously healthy 22-month-old boy with progressive bilateral eyelid swelling and ocular symptoms.
Purpose of the Study:
- To present a rare case of idiopathic dacryoadenitis in a young child.
- To emphasize the diagnostic difficulties and management strategies for pediatric dacryoadenitis.
- To highlight the importance of advanced imaging and histopathological examination.
Main Methods:
- Clinical presentation of bilateral eyelid swelling, conjunctival chemosis, and ocular discharge.
- Exclusion of infectious, autoimmune, and neoplastic etiologies through comprehensive investigations.
- Diagnostic confirmation via magnetic resonance imaging (MRI) showing lacrimal gland enlargement and subsequent lacrimal gland biopsy.
Main Results:
- Initial treatment for atypical conjunctivitis failed, with clinical deterioration.
- Bacterial, viral, and autoimmune tests were negative.
- Lacrimal gland biopsy revealed chronic lymphoplasmacytic inflammation, leading to a diagnosis of idiopathic dacryoadenitis.
- Rapid and complete resolution of symptoms within 3 weeks following systemic corticosteroid therapy.
Conclusions:
- Idiopathic dacryoadenitis is a rare but treatable cause of pediatric dacryoadenitis.
- Early diagnosis through imaging (MRI) and biopsy is essential for atypical or progressive cases.
- Systemic corticosteroids are an effective treatment for idiopathic dacryoadenitis in children.
Abstract:
Dacryoadenitis is most commonly caused by infectious, inflammatory or neoplastic processes. Idiopathic dacryoadenitis is exceptionally rare in children. We present a case of a 22-month-old previously healthy boy with progressive bilateral eyelid swelling, conjunctival chemosis and ocular discharge without systemic features. He was initially treated for atypical conjunctivitis but failed antimicrobial therapy and continued to deteriorate clinically. Investigations including bacterial swabs, viral serology and autoimmune testing were negative. Magnetic resonance imaging demonstrated bilateral lacrimal gland enlargement. Lacrimal gland biopsy revealed chronic lymphoplasmacytic inflammation without vasculitis or increased IgG4-positive plasma cells. The patient was diagnosed with idiopathic dacryoadenitis and responded rapidly to systemic corticosteroids, with complete resolution within 3 weeks. This case highlights the diagnostic challenge of paediatric dacryoadenitis and the importance of early imaging and tissue diagnosis in atypical or progressive cases.

