Surgical debulking for idiopathic dacryoadenitis: a diagnosis and a cure

Ophthalmology. 2014 Feb;121(2):603-9. doi: 10.1016/j.ophtha.2013.09.010. Epub 2013 Oct 25.

Abstract

Purpose: Idiopathic inflammatory tumor of the lacrimal gland, also called idiopathic dacryoadenitis, generally is treated with high-dose, long-term systemic corticosteroids, despite their limited success, high recurrence rate, and incidence of drug-induced side effects. This study describes the outcome of patients with idiopathic dacryoadenitis who were managed with surgical debulking.

Design: Retrospective case series from 2 tertiary referral centers.

Participants: Forty-six patients (46 lacrimal glands).

Methods: Review of the clinical records, radiologic scans, and histopathologic specimens, with additional immunoglobulin G4 immunostaining.

Main outcome measures: Clinical signs and symptoms at 2 months after the surgery and off medications.

Results: Before referral, 41% (19 of 46) of the patients had received systemic high-dose corticosteroids, after which they all showed recurrence, of whom 26% (5 of 19) became dependent on corticosteroids. At referral, all patients underwent debulking surgery of the inflammatory lacrimal gland mass for diagnostic and therapeutic reasons. Additionally, intralesional or systemic low-dose corticosteroids were given during the operation or the first postoperative days in 54% (25 of 46) of the patients. At 2 months after the debulking surgery, a full clinical recovery was seen in 80% (37 of 46) of the patients. A recurrence occurred in 8% (3 of 37) of the patients 4 months and 2.2 and 4.6 years later. Surgical failure (20%; 9 of 46) was correlated with prior corticosteroid treatment (P = 0.002, Fisher exact test), but not with sclerosing inflammation present in 28% (13 of 46). The median follow-up time was 7.2 years (range, 0.7-18 years).

Conclusions: Debulking biopsy procedures for idiopathic dacryoadenitis, in addition to being diagnostic, may be therapeutic.

Publication types

  • Multicenter Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Child
  • Dacryocystitis / diagnosis*
  • Dacryocystitis / surgery*
  • Female
  • Follow-Up Studies
  • Humans
  • Lacrimal Apparatus / surgery*
  • Magnetic Resonance Imaging
  • Male
  • Middle Aged
  • Ophthalmologic Surgical Procedures*
  • Retrospective Studies
  • Tomography, X-Ray Computed
  • Treatment Outcome
  • Young Adult