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Biomedical subjects

C Stephen Foster

Publications and source records attributed to C Stephen Foster.

At least 19 recordsLinked to original sources
PubMed2007

Solitary myofibroma of the sclera.

PURPOSE: To report a novel case of solitary myofibroma involving the sclera. METHODS: Case report and review of the literature. RESULTS: A 19-year-old woman had an expanding and painful epibulbar mass clinically diagnosed as nodular scleritis that did not respond to anti-inflammatory therapy. A biopsy showed a proliferation of spindle-shaped cells that reacted strongly positively with immunoperoxidase stains for vimentin and smooth-muscle actin. The diagnosis of solitary myofibroma was made. Six months postoperatively, the patient was well, and the lesion had not recurred. CONCLUSIONS: Myofibroma should be considered in the differential diagnosis of an epibulbar mass, especially in the setting that would mimic nodular scleritis.

Adult↗
PubMed2006

Visual outcomes in children with juvenile idiopathic arthritis-associated uveitis.

PURPOSE: To analyze visual outcomes in children affected by juvenile idiopathic arthritis (JIA)-associated uveitis. DESIGN: Retrospective interventional case series. PARTICIPANTS: Eighty-nine children with JIA-associated uveitis. METHODS: Charts of children with JIA-associated uveitis were reviewed. MAIN OUTCOME MEASURE: Change in patients' visual acuities (VAs). RESULTS: Of 269 children with uveitic syndromes referred, 89 (33%) had JIA-associated uveitis. The process was bilateral in 76 children. Seventy-three patients were female, and 84% of patients were Caucasian. Mean age of onset of uveitis was 5.7 years. Mean follow-up was 2.96 years. Antinuclear antibody positivity was detected in 56 patients, 44 of them female. Patients with JIA-associated uveitis developed numerous complications in the course of their disease: of 165 affected eyes, 105 (64%) developed cataracts, 33 (20%) developed increased intraocular pressure, and 76 (46%) developed band keratopathy; posterior synechiae were present in 96 (58%). Of 89 children, 73% were treated with immunomodulators, 40% were treated with nonsteroidal antiinflammatory agents alone or in combination with immunomodulators, and 21% were treated with topical and/or systemic steroids. Of 65 children who required immunomodulation, only one chemotherapeutic agent was used in 30, two agents in 21, and > or =3 in 14. Visual acuities of 65 children (122 eyes) were documented and compared at standard intervals. By mixed-models linear regression, improvement in VA of 0.03 logarithm of the minimum angle of resolution units per year was not found to be statistically significant (standard error, 0.02, P = 0.089). CONCLUSIONS: Juvenile idiopathic arthritis-associated uveitis is a sight-threatening disease. However, much of the children's vision can be preserved if patients are treated appropriately.

Adolescent↗
PubMed2006

Chronic cicatrizing conjunctivitis in a patient with epidermolysis bullosa acquisita.

OBJECTIVE: To describe a nonconventional diagnostic technique used to diagnose a case of cicatrizing conjunctivitis associated with epidermolysis bullosa acquisita. METHODS: Direct immunofluorescence of a biopsy specimen of the patient's conjunctiva was performed using fluorescein-conjugated rabbit antihuman antibodies against IgA, IgG, and IgM; complement C3; and fibrinogen. Immunoblot assay using healthy human skin as substrate was performed to investigate for the presence of antibodies in the patient's serum. After the diagnosis of systemic autoimmune disease was established, intravenous immunoglobulin therapy was administered. RESULTS: Direct immunofluorescence of the conjunctiva revealed linear deposition of IgA and IgG, and C3 at the epithelial basement membrane zone. Immunoblot analysis demonstrated the presence of IgG antibodies in patient serum directed against a 290-kDa protein in human skin. A diagnosis of epidermolysis bullosa acquisita was established. All signs and symptoms improved dramatically 4 months after initiation of intravenous immunoglobulin therapy and remained stable during follow-up. CONCLUSIONS: Epidermolysis bullosa acquisita can manifest in the eye as chronic cicatrizing conjunctivitis indistinguishable from ocular cicatricial pemphigoid. A nonconventional diagnostic tool (immunoblot assay) might be helpful in establishing the diagnosis of an underlying systemic autoimmune disease in patients with chronic cicatrizing conjunctivitis. Intravenous immunoglobulin therapy was effective against chronic cicatrizing conjunctivitis associated with epidermolysis bullosa acquisita.

Adult↗
PubMed2006

Patterns of exacerbations of chronic non-infectious uveitis in pregnancy and puerperium.

PURPOSE: To determine patterns of exacerbations of recurrent non-infectious uveitis during pregnancy and puerperium. DESIGN: Retrospective cohort study. METHODS: The medical records of 32 women with a history of chronic non-infectious uveitis, who were pregnant during their follow-up at the Ocular Immunology and Uveitis Service of the Massachusetts Eye and Ear Infirmary, from 1983 through 2003, were reviewed. The uveitis relapse rate during pregnancy was compared to the relapse rate during pregnancy-free periods in these women and to the relapse rate in a control group of women of childbearing age with recurrent non-infectious uveitis. RESULTS: Among the 32 women who were pregnant during follow-up (40 pregnancies), the rate of flare-ups during pregnancy (1.0 recurrence per year) was lower than that observed during non-pregnant periods (2.4 per year; p<0.001) and lower than that observed in the non-pregnant control group (3.1 per year; p<0.001). Flare-ups were most frequent in the first trimester of pregnancy and decreased markedly in the second and third trimesters (2.3, 0.5, and 0.4 recurrences per year, respectively; p<0.001). CONCLUSIONS: Pregnancy is associated with lower numbers of flare-ups of non-infectious uveitis compared to the non-pregnant state. If flare-ups do occur during pregnancy, they happen predominantly in the first trimester.

Adolescent↗
PubMed2006

Conjunctival non-caseating granulomas in a human immunodeficiency virus (HIV) positive patient attributed to sarcoidosis.

PURPOSE: To report a rare case of isolated ocular sarcoidosis in an HIV positive patient. DESIGN: Case report. METHODS: A 47-year-old HIV+ Caucasian male was referred for chronic bilateral follicular conjunctivitis. RESULTS: A conjunctival biopsy that was performed on the right eye showed sarcoidosis. General medical evaluation including a spiral thin cut chest CT scan revealed no systemic involvement. The ocular signs and symptoms resolved completely with topical corticosteroid treatment. CONCLUSIONS: HIV infection and sarcoidosis rarely coexist, presumably because their immunopathogenesis mechanisms diverge. In the absence of systemic involvement, a definite diagnosis can only be made by biopsy of the ocular tissues.

Administration, Topical↗
PubMed2006

Efficacy of intravenous immunoglobulin treatment in refractory uveitis.

PURPOSE: To assess the efficacy of intravenous immunoglobulin (IVIg) therapy in patients with severe uveitis otherwise unresponsive to conventional immunomodulatory agents. METHODS: Data on five consecutive patients treated with IVIg and followed to the present time by one of the authors (CSF) were reviewed. All patients had severe and recalcitrant uveitis of diverse etiologies. Main outcome measures were control of intraocular inflammation, steroid-sparing effect, visual acuity, and side effects. RESULTS: The duration of IVIg therapy was 3 to 36 months (mean, 16.8 months). Treatment was effective in controlling the intraocular inflammation in 3 of 5 patients. One of those patients required maintenance of systemic steroids at a dose of 10 mg per day. Visual acuity has stabilized or improved in these three patients. No immediate or long-term side effect was observed in any of the patients. CONCLUSIONS: Intravenous immunoglobulin therapy was an effective therapeutic modality in the treatment of three of five patients with severe uveitis that was unresponsive to conventional immunomodulatory agent(s). No adverse events were observed.

Adolescent↗
PubMed2006

Mucous membrane pemphigoid with fatal bronchial involvement in a seventeen-year-old girl.

PURPOSE: This study was designed to report a case of biopsy-proven mucous membrane pemphigoid with severe bronchial involvement in a young woman. METHODS: Case report of a 17-year-old girl who presented with worsening dyspnea, skin rash, and bilateral ocular injection, symblepharon, and fornix foreshortening. Conjunctival, skin, and bronchial biopsies were performed along with imaging and serological tests in an effort to establish a diagnosis for this unusual constellation of findings. The surprising occurrence of a cerebrovascular accident during her hospitalization also prompted a search for a concurrent coagulation disorder. RESULTS: Immunofluorescence studies of conjunctival, skin, and bronchial tissue specimens revealed deposition of multiple antibody classes at the basement membrane zone. The patient also possessed circulating basement membrane zone antibodies in her serum and a significant titer of antiphospholipid antibodies. She underwent dilation and stent placement for subglottic tracheal and left bronchial stenosis and was treated with immunosuppressive agents. After a favorable initial response, the patient experienced progressive bronchial stenosis and respiratory compromise, culminating in her death from bronchospasm and cardiopulmonary arrest. CONCLUSION: To our knowledge, this is the first report of mucous membrane pemphigoid involving the lower airways that was confirmed by immunofluorescence analysis. It highlights the potentially lethal, systemic nature of mucous membrane pemphigoid and underscores the need to question patients about symptoms of respiratory dysfunction.

Adolescent↗
PubMed2006

Mooren ulcer: an immunopathologic study.

PURPOSE: To determine the pattern and distribution of mononuclear cells, adhesion, and co-stimulatory molecules in the conjunctiva of patients with Mooren ulcer. METHODS: Conjunctival biopsy specimens were obtained from 6 patients with Mooren ulcer and 6 healthy individuals. Immunohistochemistry was performed on frozen sections of the cryopreserved human conjunctivas using monoclonal antibodies directed against CD1alpha, CD3, CD4, CD8, CD20, CD25, CD57, and CD68 cells; the adhesion molecules E-selectin, vascular cell adhesion molecule-1 (VCAM-1), very late activation-4 (VLA-4), ICAM-1, and LFA-1; and the co-stimulatory molecules CD28, B7-1, B7-2, and CTLA-4. RESULTS: Differences in expression on the conjunctival epithelium from patients with Mooren ulcer and normal subjects were noted only for VCAM-1, VLA-4, ICAM-1, and LFA-1. The ratio of CD4+/CD8+ cells in Mooren ulcer specimens was significantly higher (3.5-fold). However, in the substantia propria, Mooren ulcer specimens revealed significantly increased numbers of CD1alpha+, CD3+, CD4+, CD20+, CD28+, B7-1+, B7-2+, and CD68+ cells. The ratios of CD4+/CD8+ cells and B7-2+/antigen-presenting cells in Mooren ulcer specimens were significantly higher (5-fold). All tested adhesion molecules showed significant up-regulation in the patients' conjunctivas. Mooren ulcer vascular endothelial cells prominently expressed E-selectin, VCAM-1, VLA-4, and ICAM-1 compared with normal conjunctiva. CONCLUSION: The simultaneous presence of multiple types of inflammatory cells, adhesion, and co-stimulatory molecules in Mooren ulcer conjunctiva suggests that their interaction may contribute to a sustained immune activation as at least part of the pathogenic mechanism of this disorder.

Antibodies, Monoclonal↗
PubMed2006

Obliterative microangiopathy presenting as chronic conjunctivitis in a patient with relapsing polychondritis.

PURPOSE: To report histopathologic findings and treatment of obliterative microangiopathy in a patient with chronic conjunctivitis due to relapsing polychondritis (RP). METHODS: This interventional case report describes a 50-year-old woman with relapsing polychondritis was referred to the Ocular Immunology and Uveitis Service for bilateral tearing due to refractory chronic conjunctivitis. Ocular examination revealed the presence of bilateral follicular conjunctivitis. Conjunctival biopsy of the inferior palpebral conjunctiva was performed, and the histopathologic findings guided the therapeutic intervention. RESULTS: The pathology of the conjunctival biopsy included granulomatous obliterative microangiopathy with numerous eosinophils, plasma cells, lymphocytes, and epithelioid cells in the substantia propia. The chronic conjunctivitis resolved with systemic methotrexate therapy. CONCLUSIONS: Microangiopathy may cause chronic conjunctivitis in patients with RP and may be a harbinger of evolving nonocular problems as a consequence of incomplete control of this autoimmune disorder. Immunomodulatory therapy should be considered in such cases to prevent possible cardiovascular, renal, respiratory, and neurologic complications of vasculitis. Although methotrexate has been used in treatment of RP-related necrotizing scleritis with poor results, it can be sufficient for the conjunctivitis with microangiopathy associated with RP.

Chronic Disease↗
PubMed2005

Analysis of clonal immunoglobulin heavy chain rearrangements in ocular lymphoma.

BACKGROUND: The morphologic diagnosis of primary and metastatic intraocular lymphoma (IOL) was made difficult by the paucicellular specimens with fragile populations of lymphocytes retrieved through pars plana vitrectomy (PPV). The analysis of immunoglobulin heavy chain (IgH) gene rearrangements (AIGHR) was used as an adjunct to cytopathology and flow cytometry in systemic lymphoma. In IOL, the sensitivity and specificity of AIGHR are unknown. METHODS: The authors reviewed the clinical records of patients who underwent PPV for suspicion of IOL at the Immunology and Uveitis Service of the Massachusetts Eye and Ear Infirmary (Boston, MA) between 2000 and 2002. AIGHR was performed as a routine diagnostic test on cell lysates isolated from < 0.5 mL of vitreous fluid. The authors used seminested polymerase chain reaction (PCR) with consensus primers for the VDJ-region of the IgH gene. PCR products were analyzed by polyacrylamide gel electrophoresis. RESULTS: Thirty patients (37 specimens) with chronic vitritis and 17 patients (23 specimens) with IOL were included. The specificity of vitreous fluid cytopathology, flow cytometry, and AIGHR was 1.0, and the sensitivity values were 0.24, 0.36, and 0.64, respectively. AIGHR was negative in two patients for whom cytopathology or flow cytometry revealed the diagnosis of lymphoma. Clonal IGHR was found in four specimens classified as negative for lymphoma based on cytopathology and flow cytometry. CONCLUSIONS: AIGHR supplemented cytopathology and flow cytometry to increase the diagnostic yield in IOL.

Adolescent↗
PubMed2005

Psoriatic uveitis: a distinct clinical entity?

PURPOSE: To describe the clinical characteristics of uveitis occurring in patients with psoriasis and to compare them with patients with idiopathic anterior uveitis and HLA-B27-associated anterior uveitis. DESIGN: Case-control study. METHODS: The charts of 36 patients with a diagnosis of uveitis and psoriasis visiting the Immunology and Uveitis Service at the Massachusetts Eye and Ear Infirmary between January 2000 and December 2001 were reviewed. Clinical characteristics, therapy, and outcomes of uveitis were compared with 30 randomly selected patients with either idiopathic anterior uveitis or with HLA-B27-associated anterior uveitis. RESULTS: The mean age at presentation for uveitis was significantly higher in patients with psoriasis compared with nonpsoriatic groups (44.6 years in HLA-B27-psoriatic patients vs 35.9 years in HLA-B27- nonpsoriatic patients, P = .034; 52.7 years in HLA-B27+ psoriatic vs 35.7 years in HLA-B27+ nonpsoriatic patients, P = .001). Uveitis in patients with psoriasis also tended to be bilateral (62%) and prolonged (11.2 weeks). HLA-B27+ patients with psoriasis required supplemental therapy with oral nonsteroidal anti-inflammatory drugs (95% confidence interval [CI] 1.41 to 5.36, P = .003) for anterior uveitis more often than did the HLA-B27+ nonpsoriatic group. Patients with psoriasis and uveitis who were HLA-B27+ required oral NSAIDs (95% CI 1.51 to 9.79, P = .001) for anterior uveitis more often than did HLA-B27- psoriatic patients. CONCLUSION: Uveitis in patients with psoriasis may have distinguishing clinical features. Further epidemiologic studies are required to determine the strength of association between psoriasis without arthritis but with uveitis.

Adult↗
PubMed2005

Successful treatment of idiopathic orbital inflammation with mycophenolate mofetil.

PURPOSE: To report our experience with the use of mycophenolate mofetil for treatment of idiopathic orbital inflammation (IOI). DESIGN: Noncomparative interventional case series. METHODS: We reviewed the records of five patients with IOI who were treated with mycophenolate mofetil. The indications for use included recurrent inflammation after other therapies (four patients) and to avoid corticosteroid-induced blood sugar elevation in poorly controlled diabetes mellitus (one patient). Treated patients were followed for a median of 8.5 months (range, 4-30 months) after mycophenolate mofetil therapy was begun. Main outcome measures were response to treatment, treatment-related side effects, drug dosage, previous and final treatment regimens, and discontinuation of systemic corticosteroids. RESULTS: Mycophenolate mofetil therapy was discontinued in one patient because of nausea. Each of the remaining four patients experienced complete resolution of orbital inflammation. Of these, only one patient remains on prednisone, but at a significantly reduced dose (1 mg/d). CONCLUSION: Mycophenolate mofetil therapy resulted in the resolution of inflammation in patients with refractory or corticosteroid-dependent IOI and was used successfully for a first episode of IOI in a patient for whom corticosteroids were contraindicated.

Adult↗
PubMed2005

Ocular pemphigus.

BACKGROUND: Ocular involvement in patients with pemphigus vulgaris (PV) has been rarely reported. We report ocular involvement in 11 patients with PV. METHODS: Medical records of 11 biopsy-proven patients with PV treated during the period between 1990 and 2003 were reviewed and clinical information was analyzed. RESULTS: Mean age at onset of PV was 52.3 years (range, 30-80 years). Ocular disease was preceded by involvement of the skin, other mucosae, or both in all patients. Ocular involvement was limited to the conjunctivae, the eyelids, or both. PV did not affect the visual acuity of any of the patients. Suprabasal acantholysis was observed on routine histologic examination of the conjunctiva and skin of the eyelid. Direct immunofluorescence of perilesional eyelid skin demonstrated deposition of IgG on epithelial cell surface. Mean duration of follow up was 48.9 months (range, 4-100 months). Recurrence of ocular disease occurred in 3 patients; recurrence at nonocular mucosae occurred in 4 patients. No sequelae were observed during detailed follow-up. Ocular pemphigus improved with systemic therapy. The mean remission period was 32 months (range, 0-92 months). CONCLUSIONS: Ocular involvement in PV is rare. Involvement is limited to the conjunctiva, the eyelids, or both. PV does not appear to affect visual acuity. Patients have full recovery without sequelae.

Acantholysis↗