Primary Periocular Orofacial Granulomatosis: Case Series and Systematic Review.

PURPOSE: There is no known optimal treatment for primary periocular orofacial granulomatosis (PPOFG), a disorder that results in periocular edema. This case series and systematic review identifies management strategies and their reported improvement. METHODS: Systematic review and case series. PubMed and MEDLINE databases were searched following the Preferred Reporting Items for Systematic Reviews and Meta-Analysis guidelines to identify published cases of PPOFG. Cases were included when edema involved the periocular tissues and when a biopsy was interpreted to be PPOFG. Cases were excluded when edema did not involve the periocular tissues or when the edema was secondary to another process. The electronic medical records of the pathology department were studied to identify cases that were in keeping with PPOFG. The clinical charts were examined to confirm the diagnosis and provide a local case series. RESULTS: There are 38 published cases of PPOFG. An additional 9 cases were identified locally. These cases were combined and analyzed. Most PPOFG has eyelid swelling in isolation, without other facial swelling (36/47; 76.6%). It is most commonly a bilateral disease (30/47; 63.8%). Fissured tongue and facial nerve palsy occur, just as they do in other cases of orofacial granulomatosis. Treatment with surgical debulking or intralesional steroids resulted in high rates of symptomatic improvement of eyelid swelling, but recurrences were common. CONCLUSIONS: In light of no curative or highly successful treatment currently available, intralesional steroids and/or surgical debulking are therapies in the treatment of eyelid swelling associated with PPOFG that demonstrate reasonable short- and medium-term results. There is no established therapy that can offer disease remission or long-term symptom improvement.

Bacterial pyomyositis of the extraocular muscles: case report and systematic review.

OBJECTIVE: To describe the manifestations and treatment of extraocular muscle (EOM) bacterial pyomyositis. DESIGN: A systematic review following PRISMA guidelines and a case report. METHODS: PubMed and MEDLINE databases were searched for case reports and case series of EOM pyomyositis using the term "extraocular muscle" combined "pyomyositis" and "abscess". Patients were included as bacterial pyomyositis of the EOMs when there was a response to antibiotics alone or if a biopsy was consistent with the diagnosis. Patients were excluded when pyomyositis did not involve the EOMs or when diagnostic tests or treatment were not in keeping with the diagnosis of bacterial pyomyositis. An additional patient with bacterial myositis of the EOMs, treated locally, was added to the cases identified in the systematic review. Cases were grouped for analysis. RESULTS: There are 15 published cases of EOM bacterial pyomyositis including the one reported in this paper. Bacterial pyomyositis of the EOMs typically affects young males and is caused by Staphylococcus species. Most patients present with ophthalmoplegia (12/15; 80%), periocular edema (11/15; 73.3%), decreased vision (9/15; 60%) and proptosis (7/15; 46.7%). Treatment involves antibiotics alone or in combination with surgical drainage. CONCLUSIONS: Bacterial pyomyositis of the EOM presents with the same signs as orbital cellulitis. Radiographic imaging identifies a hypodense lesion with peripheral ring enhancement within the EOM. An approach to cystoid lesions of the EOMs is helpful in reaching the diagnosis. Cases can be resolved with antibiotics aimed at treating Staphylococcus, and surgical drainage may be required.

Restrictive strabismus following Jones tube insertion: a case series of 8 patients.

BACKGROUND: Restrictive strabismus and diplopia are an uncommon complication of conjunctivodacryocystorhinostomy, with insertion of the Lester-Jones tube. A literature review revealed only 4 published reports of this complication with a total of 4 patients affected. METHODS: We report on a series of 8 patients who presented with restrictive strabismus and diplopia following Jones tube insertion. RESULTS: Time to presentation was variable and was found to occur from several months to as long as 6 years after insertion. Treatment included topical steroid therapy initially. Surgery was done to release adhesions and scarring if topical treatment failed. Mitomycin C was used in 2 patients. Only 4 of the 8 patients had successful resolution of their diplopia with either therapy. INTERPRETATION: Diplopia following Jones tube insertion is an infrequent complication of surgery. Medical or surgical therapy can be help resolve symptoms but is often unsuccessful.