Odontogenic tumors and cysts, arising in the jawbones, grow by resorption and destruction of the jawbones. However, mechanisms underlying bone resorption by odontogenic tumors/cysts remain unclear. Odontogenic tumors/cysts comprise odontogenic epithelial cells and stromal fibroblasts, which originate from the developing tooth germ. It has been demonstrated that odontogenic epithelial cells of the developing tooth germ induce osteoclastogenesis to prevent the tooth germ from invading the developing bone to maintain its structure in developing bones. Thus, we hypothesized that odontogenic epithelial cells of odontogenic tumors/cysts induce osteoclast formation, which plays potential roles in tumor/cyst outgrowth into the jawbone. The purpose of this study was to examine osteoclastogenesis by cytokines, focusing on transforming growth factor-β (TGF-β), produced by odontogenic epithelial cells. We observed two pathways for receptor activator of NF-κB ligand (RANKL) induction by keratocystic odontogenic tumor fluid: the cyclooxygenase-2 (COX-2)/prostaglandin E2 (PGE2) pathway through interleukin-1α (IL-1α) signaling and non-COX-2/PGE2 pathway through TGF-β receptor signaling. TGF-β1 and IL-1α produced by odontogenic tumors/cysts induced osteoclastogenesis directly in the osteoclast precursor cells and indirectly via increased RANKL induction in the stroma.
Basal cell nevus syndrome (BCNS) is a rare, multisystem, autosomal dominant disorder that is characterized by various phenotypes, including multiple basal cell carcinomas of the skin, odontogenic keratocysts of the jaws, and occasionally cleft lip and/or palate. In this report, we describe a 6-year-old Japanese girl with a novel heterozygous nonsense mutation in
PTCH1
who exhibited rare craniofacial phenotypes, such as oligodontia and a short-tooth root.
Congenital midline upper lip fistula is a very rare congenital anomaly that can be accompanied by other congenital abnormalities such as cleft lip and palate, and may appear as a symptom of a syndrome such as Pirre-Robin' s syndrome. It has been reported that this disease can be transmitted and presents signs of infection such as swelling of the upper lip, abscess formation, and pus discharge from the fistula. However, a case of infection in infancy has never been reported before. Here, we report a case of a six-month-old baby with an infected congenital midline upper lip fistula. The patient visited our hospital with swelling of the upper lip and suckling disorder. With the midline upper lip fistula infected, signs of infection such as swelling of the upper lip, pus discharge from the fistula, and domeshaped swelling of the oral vestibule of the maxillary anterior tooth were observed. Due to these acute symptoms, the patient had a suckling disorder. After anti-inflammatory treatment, we resected the lip fistula under general anesthesia. Congenital midline upper lip fistula is likely to cause infection in infancy, leading to a suckling disorder.Early examination and treatment for a congenital midline upper lip fistula is suggested.
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