title: Surgical debulking for idiopathic dacryoadenitis: a diagnosis and a cure.
abstract: OBJECTIVE
  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.
  
  
  METHODS
  Retrospective case series from 2 tertiary referral centers.
  
  
  METHODS
  Forty-six patients (46 lacrimal glands).
  
  
  METHODS
  Review of the clinical records, radiologic scans, and histopathologic specimens, with additional immunoglobulin G4 immunostaining.
  
  
  METHODS
  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.affiliation: Department of Ophthalmology, University Hospitals Leuven, Leuven, Belgium. Electronic address: Ilse.Mombaerts@uzleuven.be.
authors.forename: Ilse
authors.initials: I
authors.lastname: Mombaerts
authors.affiliation: Departments of Ophthalmology and Laboratory Medicine and Pathology, University of Minnesota School of Medicine, Minneapolis, Minnesota.
authors.forename: J Douglas
authors.initials: JD
authors.lastname: Cameron
authors.affiliation: HRH Princess Maha Chakri Sirindhorn Medical Center, Srinakharinwirot University, Bangkok, Thailand; Department of Ophthalmology, Mayo Clinic, Rochester, Minnesota.
authors.forename: Waruttaporn
authors.initials: W
authors.lastname: Chanlalit
authors.affiliation: Department of Ophthalmology, Mayo Clinic, Rochester, Minnesota.
authors.forename: James A
authors.initials: JA
authors.lastname: Garrity
mesh_terms: D000293:Adolescent; D000328:Adult; D000368:Aged; D002648:Child; D003607:Dacryocystitis / Q000175:diagnosis* / Q000601:surgery*; D005260:Female; D005500:Follow-Up Studies; D006801:Humans; D007765:Lacrimal Apparatus / Q000601:surgery*; D008279:Magnetic Resonance Imaging; D008297:Male; D008875:Middle Aged; D013508:Ophthalmologic Surgical Procedures; D012189:Retrospective Studies; D014057:Tomography, X-Ray Computed; D016896:Treatment Outcome; D055815:Young Adult