Caudill R, Saltzman D, Gaum S, Granite E
J Oral Surg. 1977 Jun;35(6):483-90.
This report has considered the possibility that the described patient had a maxillary and possibly a mandibular fibrous dysplasia concomitant with a parathyroid abnormality. It has attempted to arouse the diagnostician's suspicion of possible interrelations of fibrous dysplasia and endocrine abnormalities, and to strengthen the hypothesis of others that association of fibrous dysplasia and hyperparathyroidism is not coincidental. Clinical, radiographic, and laboratory examinations more than a year postoperatively further substantiate our hypothesis that the patient had a facial fibrous dysplasia concomitant with primary hyperparathyroidism. The maxilla and mandible continued to enlarge clinically. Systemic manifestations of hyperparathyroidism had resolved. Calcium phosphorus, and parathyroid hormone levels returned to normal limits. Radiographically, there had been remodeling and recalcification of all involved bones except the maxilla and mandible which continued to expand. A possible explanation is that the maxillofacial lesions represent the polyostotic fibrous dysplasia component of the Albright syndrome. The endocrine abnormality could be manifested by the hyperparathyroidism. The third component of Albright syndrome, café au lait lesions, has not been identified. The potential interrelation of fibrous dysplasia with endocrine abnormalities is obviously important from a diagnostic and therapeutic standpoint. If there is more than a coincidental relationship-hereditary, hormonal, neurological, developmental, or any as-yet-unknown factor-only further research and studies will confirm or disprove it.
本报告探讨了所描述的患者患有上颌骨纤维发育异常,可能还有下颌骨纤维发育异常并伴有甲状旁腺异常的可能性。本报告试图引起诊断医生对纤维发育异常与内分泌异常之间可能存在的相互关系的怀疑,并强化其他人提出的纤维发育异常与甲状旁腺功能亢进症的关联并非偶然的假说。术后一年多的临床、影像学和实验室检查进一步证实了我们的假说,即该患者患有面部纤维发育异常并伴有原发性甲状旁腺功能亢进症。上颌骨和下颌骨在临床上持续增大。甲状旁腺功能亢进症的全身表现已消退。钙、磷和甲状旁腺激素水平恢复到正常范围。影像学检查显示,除上颌骨和下颌骨持续扩大外,所有受累骨骼均有重塑和再钙化。一种可能的解释是,颌面部病变代表了奥尔布赖特综合征的多骨型纤维发育异常成分。内分泌异常可能表现为甲状旁腺功能亢进症。尚未发现奥尔布赖特综合征的第三个成分——牛奶咖啡斑。从诊断和治疗的角度来看,纤维发育异常与内分泌异常之间潜在的相互关系显然很重要。如果存在的不仅仅是偶然关系——遗传、激素、神经、发育或任何未知因素——只有进一步的研究才能证实或否定这一点。