Surgical debulking for idiopathic dacryoadenitis: a diagnosis and a cure.

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.

• Authors: Ilse Mombaerts, J Douglas Cameron, Waruttaporn Chanlalit, James A Garrity
• Journal: Ophthalmology (Ophthalmology) Vol. 121 Issue 2 Pg. 603-9 (Feb 2014) ISSN: 1549-4713 [Electronic] United States
• PMID: 24572677 (Publication Type: Journal Article, Multicenter Study, Research Support, Non-U.S. Gov't)
• Copyright: Copyright © 2014 American Academy of Ophthalmology. Published by Elsevier Inc. All rights reserved.

Topics

• Adolescent
• Adult
• Aged
• Child
• Dacryocystitis (diagnosis, 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