Casavecchia Piero, Uzel M Ilhan, Kantarci Alpdogan, Hasturk Hatice, Dibart Serge, Hart Thomas C, Trackman Philip C, Van Dyke Thomas E
Department of Periodontology and Oral Biology, Boston University, Goldman School of Dental Medicine, Boston, MA 02118, USA.
J Periodontol. 2004 May;75(5):770-8. doi: 10.1902/jop.2004.75.5.770.
Hereditary gingival fibromatosis is a rare, genetically inherited overgrowth condition that is clinically characterized by a benign fibrous enlargement of maxillary and mandibular keratinized gingiva. A syndromic association between gingival fibromatosis and a wide variety of other genetically inherited disorders has been described. However, its coexistence with aggressive periodontitis has not been reported.
A 24-year-old African-American female, patient (proband X, [Px]) reported with a chief complaint of tooth mobility and gingival enlargement. Clinical examination revealed moderate to severe gingival overgrowth on both mandible and maxilla. Generalized attachment loss and mobility of the teeth were observed. Radiographic evaluation demonstrated severe alveolar bone loss. The patient was diagnosed with gingival fibromatosis and aggressive periodontitis based on the clinical and radiographic findings. Her brother (Bx) and her mother (Mx) were evaluated and diagnosed with gingival fibromatosis suggesting that this is a dominant trait in the family and gingival fibromatosis might be of hereditary origin. In addition, the brother also exhibited localized aggressive periodontitis. Medical history revealed no other systemic or local contributory factors associated with the oral findings in any of the subjects.
Surgical therapy included internal bevel gingivectomy combined with open flap debridement procedures for Px and Bx. Only internal bevel gingivectomy was performed for Mx since there was mild bone resorption and no intrabony defects. At the time of surgery, gingival biopsies were obtained and fixed in 4% paraformaldehyde. Multiple serial sections were stained with hematoxylin and eosin. Microscopic evaluation of the gingival specimens revealed large parallel collagen bundles associated with scarce fibroblasts in the connective tissue. The collagen bundles reached into the subepithelial connective tissue where elongated rete-pegs were also observed. Following the completion of the treatment, no signs of recurrence or bone resorption were observed over 2-year follow-up.
This is the first report of hereditary gingival fibromatosis associated with aggressive periodontitis. Combined treatment comprising removal of fibrotic gingival tissue and traditional flap surgery for the elimination of intrabony defects represents a unique treatment approach in periodontal therapy. Two-year follow-up revealed that both the gingival overgrowth and the destructive lesions were successfully treated.
遗传性牙龈纤维瘤病是一种罕见的、具有遗传倾向的过度生长疾病,临床特征为上颌和下颌角化牙龈的良性纤维性肿大。牙龈纤维瘤病与多种其他遗传性疾病之间存在综合征关联。然而,其与侵袭性牙周炎并存的情况尚未见报道。
一名24岁的非裔美国女性患者(先证者X,[Px])主诉牙齿松动和牙龈肿大。临床检查发现下颌和上颌均有中度至重度牙龈过度生长。观察到牙齿普遍存在附着丧失和松动。影像学评估显示严重的牙槽骨吸收。根据临床和影像学检查结果,该患者被诊断为牙龈纤维瘤病和侵袭性牙周炎。对其兄弟(Bx)和母亲(Mx)进行评估,诊断为牙龈纤维瘤病,提示这是该家族的显性性状,牙龈纤维瘤病可能具有遗传起源。此外,其兄弟还表现出局限性侵袭性牙周炎。病史显示,在任何受试者中均未发现与口腔检查结果相关的其他全身或局部促成因素。
手术治疗包括对Px和Bx进行内斜切口牙龈切除术联合开放瓣清创术。由于Mx仅有轻度骨吸收且无骨内缺损,仅对其进行了内斜切口牙龈切除术。手术时,获取牙龈组织活检标本并固定于4%多聚甲醛中。多个连续切片用苏木精和伊红染色。牙龈标本的显微镜评估显示结缔组织中有大量平行的胶原束,成纤维细胞稀少。胶原束延伸至上皮下结缔组织,在该处也观察到伸长的 rete 嵴。治疗完成后,在2年的随访中未观察到复发或骨吸收迹象。
这是遗传性牙龈纤维瘤病与侵袭性牙周炎相关的首例报道。联合治疗包括切除纤维化牙龈组织和传统瓣手术以消除骨内缺损,这是牙周治疗中的一种独特治疗方法。2年随访显示牙龈过度生长和破坏性病变均得到成功治疗。