Objective: Fibrous dysplasia(FD) is an unusual developmental abnormality of the skeleton. When facial and cranial bones are involved in FD, it is termed craniofacial fibrous dysplasia(CFD). Although several reports ha...Objective: Fibrous dysplasia(FD) is an unusual developmental abnormality of the skeleton. When facial and cranial bones are involved in FD, it is termed craniofacial fibrous dysplasia(CFD). Although several reports have reported that CFD has a tendency for spontaneous cerebrospinal fluid(CSF) leakage, there have been no related English-language case reports. We present the first case of post-traumatic CSF rhinorrhea associated with CFD. Methods: A 30-year-old man presented with CSF rhinorrhea after a mild head trauma. Computed tomography cisternogram located a defect in the posterior wall of the right frontal sinus. Imaging examination also showed the evident expansion of multiple skull bones, spinal scoliosis, and multiple local enlargements of ribs.Without café-au-lait cutaneous spots and endocrine abnormalities, polyostotic FD was diagnosed instead of Mc Cune-Albright syndrome(MAS). The patient underwent craniotomy fistula repair surgery. The excised bone was contoured to be thinner to increase the cranial cavity. The patient recovered well and CSF leakage did not recur.But during a nineteen-month follow up, sight in the patient's left eye was decreased.MAS was suspected. Unfortunately the patient refused to take the proposed decompression surgery and laboratory tests of serum hormones. Conclusions: CFD, if the wall of the paranasal sinus is involved and the cranial cavity is decreased, may increase the risk of CSF rhinorrhea after head trauma.Expectant management is recommended in asymptomatic CFD patients even in the presence of optic nerve compression. As MAS may cause more problems, it should be precluded before polyostotic FD is diagnosed.展开更多
BACKGROUND Fibrous dysplasia(FD)is a developmental hamartomatous bone disease characterized by a blend of fibrous and osseous entities.Though rarely malignant,the tumor can vary from being small and asymptomatic,to a ...BACKGROUND Fibrous dysplasia(FD)is a developmental hamartomatous bone disease characterized by a blend of fibrous and osseous entities.Though rarely malignant,the tumor can vary from being small and asymptomatic,to a fairly large sized lesion,progressing gradually,compromising occlusion and facial esthetics.Treatment approach depends on the stage of skeletal maturity.It primarily involves surgical management for stabilizing the disease process.Post-surgical comprehensive dental treatment is necessary for restoring form and function of the jaws and teeth.This article describes comprehensive orthodontic management of severe malocclusion in a surgically operated case of FD maxilla.CASE SUMMARY A 19-year female presented with a chief complaint of excessive gingival display when smiling.Dental history included swelling of gums around the upper right front teeth,diagnosed at the age of 15 as FD of the right anterior maxillary segment and treated with surgical recontouring of the dysplastic bone.The clinical and radiological examinations showed adequate post-surgical healing.The surgically treated dysplastic area presented with right canting of the maxillary anterior occlusal plane.The maxillary teeth were torqued palatally,with the root of the right maxillary canine exposed clinically.We discuss sequential management of the associated malocclusion with comprehensive fixed orthodontics,along with special precautions taken to prevent reactivation of the quiescent and healed lesion.CONCLUSION The adequate healing of fibro-dysplastic bone post-surgery must be allowed before initiating orthodontic tooth movement in the dysplastic bone.Periodic follow-ups are needed to monitor stability of occlusion and any relapse of the lesion.展开更多
基金the National Science and Technology Support Program of the 12th Five-Year of China(grant number:2012BAI12B03)Natural Science Foundation of Beijing(grant number:7112049)
文摘Objective: Fibrous dysplasia(FD) is an unusual developmental abnormality of the skeleton. When facial and cranial bones are involved in FD, it is termed craniofacial fibrous dysplasia(CFD). Although several reports have reported that CFD has a tendency for spontaneous cerebrospinal fluid(CSF) leakage, there have been no related English-language case reports. We present the first case of post-traumatic CSF rhinorrhea associated with CFD. Methods: A 30-year-old man presented with CSF rhinorrhea after a mild head trauma. Computed tomography cisternogram located a defect in the posterior wall of the right frontal sinus. Imaging examination also showed the evident expansion of multiple skull bones, spinal scoliosis, and multiple local enlargements of ribs.Without café-au-lait cutaneous spots and endocrine abnormalities, polyostotic FD was diagnosed instead of Mc Cune-Albright syndrome(MAS). The patient underwent craniotomy fistula repair surgery. The excised bone was contoured to be thinner to increase the cranial cavity. The patient recovered well and CSF leakage did not recur.But during a nineteen-month follow up, sight in the patient's left eye was decreased.MAS was suspected. Unfortunately the patient refused to take the proposed decompression surgery and laboratory tests of serum hormones. Conclusions: CFD, if the wall of the paranasal sinus is involved and the cranial cavity is decreased, may increase the risk of CSF rhinorrhea after head trauma.Expectant management is recommended in asymptomatic CFD patients even in the presence of optic nerve compression. As MAS may cause more problems, it should be precluded before polyostotic FD is diagnosed.
文摘BACKGROUND Fibrous dysplasia(FD)is a developmental hamartomatous bone disease characterized by a blend of fibrous and osseous entities.Though rarely malignant,the tumor can vary from being small and asymptomatic,to a fairly large sized lesion,progressing gradually,compromising occlusion and facial esthetics.Treatment approach depends on the stage of skeletal maturity.It primarily involves surgical management for stabilizing the disease process.Post-surgical comprehensive dental treatment is necessary for restoring form and function of the jaws and teeth.This article describes comprehensive orthodontic management of severe malocclusion in a surgically operated case of FD maxilla.CASE SUMMARY A 19-year female presented with a chief complaint of excessive gingival display when smiling.Dental history included swelling of gums around the upper right front teeth,diagnosed at the age of 15 as FD of the right anterior maxillary segment and treated with surgical recontouring of the dysplastic bone.The clinical and radiological examinations showed adequate post-surgical healing.The surgically treated dysplastic area presented with right canting of the maxillary anterior occlusal plane.The maxillary teeth were torqued palatally,with the root of the right maxillary canine exposed clinically.We discuss sequential management of the associated malocclusion with comprehensive fixed orthodontics,along with special precautions taken to prevent reactivation of the quiescent and healed lesion.CONCLUSION The adequate healing of fibro-dysplastic bone post-surgery must be allowed before initiating orthodontic tooth movement in the dysplastic bone.Periodic follow-ups are needed to monitor stability of occlusion and any relapse of the lesion.
