Unilateral orbital inflammation in a child after a jellyfish sting to the lower extremities.

A 3-year 10-month-old child initially developed locally recurrent cutaneous eruptions within the first 2 weeks after sustaining a jellyfish sting to her lower extremities. After 5 asymptomatic weeks, she developed unilateral orbital inflammation that did not respond to systemic antibiotics, antihistamines, or steroids. Imaging revealed a rapidly growing mass of the right lacrimal gland. Urgent anterior orbitotomy was performed and the lacrimal gland was biopsied. Histopathologic diagnosis revealed sclerosing dacryodenitis consistent with orbital inflammatory syndrome and/or an immune response to an antigen challenge.

Amelioration of lacrimal gland inflammation by oral administration of K-13182 in Sjögren's syndrome model mice.

Regulation of the adhesion of mononuclear cells to endothelial cells is considered to be a critical step for the treatment of inflammatory diseases, including autoimmune diseases. K-13182 was identified as a novel inhibitor for these adhesions. K-13182 inhibited the expression of vascular cell adhesion molecule-1 (VCAM-1, CD106) on human umbilical vein endothelial cells (HUVECs) and on mouse vascular endothelial cell line (MAECs) induced by tumour necrosis factor (TNF)-alpha. K-13182 also inhibited the adhesion of mononuclear cells to these HUVECs and MAECs, indicating that K-13182 suppressed these adhesions mediated by cellular adhesion molecules including VCAM-1. To evaluate the therapeutic effect in autoimmune disease model mice, K-13182 was orally administered to non-obese diabetic (NOD) mice as Sjögren's syndrome (SS) model mice. Severe destructive inflammatory lesions were observed in the lacrimal glands of vehicle-treated control mice; however, 8-week administration of K-13182 inhibited the mononuclear cell infiltration into the inflammatory lesions of the lacrimal glands. In K-13182-treated mice, the decrease in tear secretion was also prevented compared to the control mice. In addition, the apoptosis and the expression of FasL (CD178), perforin, and granzyme A was suppressed in the lacrimal glands of K-13182-treated mice. Therefore, K-13182 demonstrated the possibility of therapeutic efficacy for the inflammatory region of autoimmune disease model mice. These data reveal that VCAM-1 is a promising target molecule for the treatment of autoimmune diseases as a therapeutic strategy and that K-13182 has the potential as a new anti-inflammatory drug for SS.

Nasal endoscopy in the treatment of congenital lacrimal sac mucoceles.

PURPOSE: To determine the incidence of intranasal cysts associated with lacrimal sac mucoceles and the cure rate with nasal endoscopic cyst marsupialization. DESIGN: Interventional case series. SETTING: University-affiliated teaching hospital. PATIENT POPULATION: Twenty-five infants with non infected or infected lacrimal sac mucoceles or dacrocystitis without obvious mucocele were consecutively enrolled. INTERVENTION PROCEDURES: Management included local lacrimal massage, parenteral antibiotics, and when still symptomatic, nasolacrimal duct probing with concomitant nasal endoscopy. Intranasal cysts identified were marsupialized until the distal end of the nasolacrimal duct probe was visualized. MAIN OUTCOME MEASURES: Presence of intranasal cyst identification and cure rate. RESULTS: Infants were 4 days to 10 weeks old (mean 19 days). Forty-eight percent had a bluish cutaneous mass inferior and lateral to the lacrimal sac. Twenty percent were bilateral. At presentation, 76 percent had dacrocystitis. Fourteen percent had respiratory distress. Only one child responded to medical management. At endoscopy, 23 of 24 infants had ipsilateral intranasal cysts. The one child without nasal cyst had recurrent dacrocystitis and no mucocele. All children with mucocele were cured except one child with residual nasolacrimal duct obstruction. CONCLUSIONS: Lacrimal sac mucoceles were almost always associated with intranasal cysts. Nasal endoscopy is a valuable addition to the treatment plan for lacrimal sac mucoceles not responding to a brief trial of massage or infantile dacrocystitis. To avoid potential complications, we recommend against waiting until infection occurs before proceeding with surgery.