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Severe focal sialadenitis and dacryoadenitis in NZM2328 mice induced by MCMV: a novel model for human Sjögren's syndrome.

The genetic and environmental factors that control the development of Sjögren's syndrome, an autoimmune disease mainly involving the salivary and lacrimal glands, are poorly understood. Viruses which infect the glands may act as a trigger for disease. The ability of sialotropic murine CMV (MCMV) to induce acute and chronic glandular disease was characterized in an autoimmune-prone mouse strain, NZM2328. MCMV levels were detectable in the salivary and lacrimal glands 14-28 days after i.p. infection and correlated with acute inflammation in the submandibular gland. After latency, virus was undetectable in the glands by PCR. At this stage, NZM2328 female mice developed severe chronic periductal inflammation in both submandibular and lacrimal glands in contrast to the much milder infiltrates found in female B6-lpr and male NZM2328. The focal infiltrates consisted of CD4+ and B220+ cells as opposed to diffuse CD4+, CD8+, and B220+ cells during acute infection. Salivary gland functional studies revealed a gender-specific progressive loss of secretory function between days 90 and 125 postinfection. Latent MCMV infection did not significantly affect the low incidence of autoantibodies to Ro/SSA and La/SSB Ags in NZM2328 mice. However, reactivities to other salivary and lacrimal gland proteins were readily detected. MCMV infection did not significantly alter the spontaneous onset of kidney disease in NZM2328. Thus, chronic inflammation induced by MCMV with decreased secretory function in NZM2328 mice resembles the disease manifestations of human Sjögren's syndrome.

Acute Disease↗

Lacrimal gland involvement in sarcoidosis. The clinical features of 9 patients.

PATIENTS AND METHODS: Ocular disease is relatively common in sarcoidosis and can be the initial clinical manifestation in some instances. In this study, we retrospectively evaluated the clinical and demographic features of 9 (1.74%) patients with lacrimal gland (LG) involvement out of 516 sarcoidosis patients who were followed up at our centre over the preceding 36-years. In addition, the characteristics of patients with LG involvement were compared to those of other cases with eye involvement and to other sarcoidosis patients. RESULTS: In 5 subjects with LG involvement, the chest X-ray was normal. The number of stage 0 subjects among other sarcoidosis patients was significantly lower than among patients with LG involvement (p<0.001). In 2 of these cases, the diagnosis of sarcoidosis was reached by LG biopsy. The mean age of patients with LG involvement was significantly lower than that of other sarcoidosis patients (p<0.001). Also, sarcoidosis-related organ involvement--other than of the LG--was more frequent than in other sarcoidosis patients (p<0.001). CONCLUSIONS: It should be borne in mind that LG involvement might be the initial manifestation of sarcoidosis and the chest X-ray in these patients might be completely normal.

Adolescent↗

[Recurrent unilateral inflammatory exophthalmos].

The issue discusses the diagnosis difficulties concerning the case of 58-years-old woman who was watched for a recurrent exophthalmos after a mild conjunctival trauma. The debut was orbital cellulite appearance, complicated by a nonaxial exophthalmos due to a dacryoadenitis. The exophthalmos is axial in the other two recurrences. All exophthalmic episodes progressed with inflammatory features associated with optic neuritis and oculomotor nerves implication without any neurological signs. The paraclinical tests (ECHO, orbital radiography, CAT) find inflammation of orbital elements: extrinsic muscles and lacrimal gland. Biochemical tests showed minimal inflammatory changes without be able to provide the positive diagnosis. Initial combined therapy (antibiotics and steroids), then steroid monotherapy had favorable response.

Cellulitis↗

Th1 versus Th2 immune responses in autoimmune lacrimal gland disease in MRL/Mp mice.

PURPOSE: In MRL/Mp-lpr/lpr (MRL/lpr) and MRL/Mp-+/+ (MRL/+) mice, a T-cell-driven lacrimal gland inflammation spontaneously develops that is a model for Sjögren's syndrome. The lacrimal gland lesions in these mice were evaluated by immunohistochemistry for the relative contributions of T-helper (Th)1 versus Th2 immune responses. METHODS: Frozen sections of lacrimal glands from MRL/lpr and MRL/+ mice ages 1 through 5 months were stained with monoclonal antibodies to the cytokines interferon (IFN)-gamma and interleukin (IL)4 and to the cell surface costimulatory molecules B7-1 and B7-2, which are associated with Th1 and Th2 responses, respectively. RESULTS: The median proportion of cells staining for IL-4 ranged from 30% to 67% over time for MRL/lpr mice and from 30% to 55% for MRL/+ mice. The median proportion of cells staining for IFN-gamma ranged from 1% to 5% for MRL/lpr mice and from 0% to 3% for MRL/+ mice. The proportion of cells staining positively for IL-4 was significantly greater than for IFN-gamma in both MRL/lpr (mean difference, 33%; P = 0.0001) and MRL/+ mice (mean difference, 42%; P = 0.0002). The median proportion of cells staining positively for B7-2 ranged from 20% to 38% for MRL/lpr mice and from 16% to 34% for MRL/+ mice. The median proportion of cells staining for B7-1 ranged from 2% to 10% for MRL/lpr mice and from 2% to 5% for MRL/+ mice. The proportion of cells staining positively for B7-2 was significantly greater than for B7-1 for both MRL/lpr mice (mean difference, 15%; P = 0.001) and for MRL/+ mice (mean difference, 19%; P = 0.006). CONCLUSIONS: On the basis of immunohistochemistry for cytokines and costimulatory molecules, inflammatory lacrimal gland lesions in MRL/lpr and MRL/+ mice appear to be a largely Th2 phenomenon.

Animals↗

Clinostomum trematode from human eye.

The first human case of Clinostomum lacramalitis in Thailand is reported. A 38-year-old man visited an ophthalmology clinic after having itchy pain in the right eye for two days. Ophthalmological examination revealed a living worm adhered to the lacramal opening and after removal, it was identified as Clinostomum sp. The patient admitted that he used to eat raw freshwater fish, he caught in Prachin Buri Province where he lives.

Adult↗

[Endonasal dacryocystorhinostomy (DCR) with protected drill].

PURPOSE: To analyze the interest of protected drills to perform endoscopic endonasal DCR. MATERIAL AND METHODS: [corrected] Fifty patients suffering from chronic epiphora were operated successively. None of those patients had had prior nasal surgery. The same surgeons (BF and ER) did the surgical procedure for all the cases. The technical steps were: endoscopic visualization of the middle meatus and evaluation of the anatomic variations of the nose; identification of the lacrymal bag with a 1 mm light in the canaliculus; unciformectomy; the maxillary bone was drilled with a 3.2 mm bit (straightshot Xomed), protected and irrigated; the lacrymal bag was opened by cissors and the flap was pushed in the ethmoid and coagulated. RESULTS: The suction-irrigation system was efficient to clean the surgical site, with a good endoscopic control. The protected bit didn't damage the 4-mm optics and the septal wall mucosa, even in narrow noses. The staightshot bit (Xomed) was tough enough to drill the maxillary bone in only 68 % of the surgical cases. In 28 % the drilling was unsatisfactory and in 4 % it was inefficient. The functional results at 6 month were good in 85 %. There was no difference between normal and narrow noses. CONCLUSION: The concept of protected bits perfectly matches the endoscopic endonasal DCR. The visual comfort and security of the surgical procedure are increased. Short term results are equivalent to external DCR, but are rather inefficient and expensive.

Chronic Disease↗

Leakage of aquaporin 5 in the tear of dacryoadenitis mice.

PURPOSE: The objective of this study was to investigate whether leakage of aquaporin 5 (AQP5) in tear is associated with damage of lacrimal glands (LGs) in dacryoadenitis models. METHODS: Female MRL/lpr (24-week-old), male NOD/Shi Jci (5-, 8-, and 10-week-old), female NFS/s-TX (10-week-old), and lipopolysaccharide (LPS)-induced dacryoadenitis model mice were used. Tear fluid was collected by a cotton thread. Tear proteins in the thread were dissolved in sodium dodecyl sulfate buffer, and AQP5 proteins were analyzed by the Western blot technique using anti-AQP5 antibody. LGs were prepared for hematoxylin and eosin staining or immunostaining of AQP5. RESULTS: In MRL/lpr, NFS/s-TX, 8- and 10-week-old NOD/Shi Jci mice, AQP5 protein was detected in the tear by Western blot analysis. Inflammatory lymphocyte infiltrations were observed in LGs of these dacryoadenitis model mice. In contrast, AQP5 leakage and damage of LG were not observed in normal mice. In 5-week-old NOD/Shi Jci mice, infiltration was not seen in LG, and AQP5 leakage was not detected in the tear. In LPS-induced dacryoadenitis model mice, either tissue destruction with inflammation in LG or AQP5 leakage in the tear was observed. AQP5 in the tear and tissue inflammation in LGs was not found in control mice. These results indicate that AQP5 is leaked in tears when LGs are damaged by dacryoadenitis. CONCLUSIONS: Leakage of AQP5 in the tear was found to be related to LG damage. This finding suggests that detection of AQP5 in tear is useful for specific diagnosis of LG disorders with tissue destruction.

Animals↗

[Analysis of the efficacy of early probing in the treatment of symptomatic congenital lacrimal duct obstruction in infants. Apropos of 92 cases].

INTRODUCTION: Nasolacrimal duct obstruction (NLDO) is a common clinical problem (5 to 6% of newborns). Dacryostenosis is a partial or complete block in the nasolacrimal duct. PATIENTS AND METHODS: The records of 68 patients (92 cases) of NLDO with/without mucocele were reviewed. The probing was performed early and according to the same technique. RESULTS: The age at the time of probing ranged from 0 to 7 months (mean 4.6 months). The cure rate was 91.3% and the failure rate was 8.70%. DISCUSSION: Our results are consistent with other reports in the literature. The cure rate was higher with a earlier probing and was good after a second tentative of probing. A multiple obstruction of nasolacrimal system was cause of a failure. This was not the case with mucocele. The optimal timing of the probing is discussed in this report and we preferred to perform the probing to 4 at 6 months. CONCLUSION: We showed a high cure rate with early probing in our series. These results should be confirmed by a prospective randomised study (in process in our institution).

Age Factors↗