Case report: cystic hygroma.

BACKGROUND: Cystic hygroma (CH) is a congenital malformation of the lymphatic system. It most commonly presents in the neck, and aetiological factors include environmental and genetic factors. CASE REPORT: A 13-year-old female presented with spontaneous dental pain affecting the maxillary left first and second permanent molar teeth. Medical history revealed a history of left sided cervico-facial-thoracic CH. She was diagnosed with periapical peridontitis and required extraction of both teeth. Clinical management was compromised by the CH involving the left face, neck, ear, tongue, larynx, oropharynx and mediastinum and circling the trachea and great vessels. TREATMENT: Initial management included the placement of obtundant dressings for teeth number 26 and 27 with resolution of dental pain. Intensive prevention was instigated, and teeth number 16 and 17 were restored with composite resin under local analgesia (LA) without incident. Extraction of teeth number 26 and 27 was complicated by significant trismus and the unacceptably high risk associated with general anaesthesia, due to intubation difficulties. It proved impossible to achieve satisfactory local analgesia. Due to her difficult airway, it was decided to treat the patient with inhalational sedation, but administered in an operating theatre by a consultant anaesthetist, and teeth were extracted using articaine LA. FOLLOW-UP: The patient coped well with this treatment, and was discharged home on the same day. Two year follow-up with intensive prevention showed improved oral health, with no new carious lesions detected. CONCLUSION: This is the first report to our knowledge describing dental extractions in the immediate vicinity of a cystic hygroma. A potential management strategy and the difficulties of conventional methods in such patients are discussed.

Enhanced expression of TGF-betas and their receptors in human acute pancreatitis.

OBJECTIVES: To determine which mechanisms are involved in pancreatic remodeling, repair, and fibrosis after acute necrotizing pancreatitis (NP) in humans. SUMMARY BACKGROUND DATA: Transforming growth factor betas (TGF-betas) are multifunctional polypeptides that have been implicated in the regulation and formation of extracellular matrix and fibrosis. They exert their functions by binding to specific receptors. In this study, we analyze the expression of TGF-beta1, TGF-beta2, and TGF-beta3 and their receptors type I (Tbeta-RI [ALK5]), type II (Tbeta-RII), and type III (Tbeta-RIII) in NP. PATIENTS: Pancreatic tissue samples were obtained from 6 female and 8 male patients with a median age of 65 years (range, 37 to 77 years) undergoing surgery for NP. The median Ranson score of the patients was 6 (range, 2 to 9). The operation was performed a median 5.5 days (range, 4 to 17 days) after the onset of acute pancreatitis. Pancreatic tissue obtained from 12 previously healthy organ donors (6 male, 6 female; median age of 43 years) served as controls. METHODS: The expression of TGF-beta1, TGF-beta2, TGF-beta3, Tbeta-RI (ALK5), Tbeta-RII, Tbeta-RIII, and collagen type I mRNA was analyzed by Northern blot analysis. In addition, immunohistochemical analysis using polyclonal antibodies was performed to detect TGF-beta1, TGF-beta2, TGF-beta3, Tbeta-RI (ALK5), and Tbeta-RII. RESULTS: Northern blot analysis showed an increase in TGF-betas and their receptors in NP tissue samples compared with samples from normal controls. The increase was 3.5-fold for TGF-beta1 (p < 0.05), 2.7-fold for TGF-beta2 (p < 0.05), 3.5-fold for TGF-beta3 (p < 0.05), 10-fold for Tbeta-RI (ALK5) (p < 0.05), 5.7-fold for Tbeta-RII (p < 0.05), and 1.4-fold for Tbeta-RIII (not significant). Collagen type I mRNA was also markedly increased in NP samples and correlated with the level of TGF-betas. Immunohistochemical analysis demonstrated intense TGF-beta1, TGF-beta2, TGF-beta3, Tbeta-RI (ALK5), and Tbeta-RII immunoreactivity in the remaining acinar and ductal cells in most NP samples; in the normal control pancreas, there was weak to moderate immunoreactivity for these factors only in some acinar cells and a few ductal cells. CONCLUSION: The marked increase in expression of TGF-betas and their signaling receptors Tbeta-RI (ALK5) and Tbeta-RII suggests a role for TGF-betas in the repair process after the onset of NP in humans and raises the possibility that TGF-betas might be involved in tissue remodeling and the fibrotic reaction that occurs in the pancreas after necrosis.