Institute of Human Genetics, University Hospital Münster, Vesaliusweg 12, D-48149 Münster, Germany.
Research Unit Rare Diseases with Orofacial, Manifestations (RDOM), Department of Cranio-Maxillofacial Surgery, University Hospital Münster, Albert-Schweitzer-Campus 1, Building W 30, D-48149 Münster, Germany.
Biomolecules. 2021 Jan 24;11(2):149. doi: 10.3390/biom11020149.
We report an extremely rare case of combined classical and periodontal Ehlers-Danlos syndrome (EDS) with early severe periodontitis and a generalized lack of attached gingiva. A German family with classical EDS was investigated by physical and dental evaluation and exome and Sanger sequencing. Due to the specific periodontal phenotype in the affected child, an additional diagnosis of periodontal EDS was suspected. Physical and genetic examination of two affected and three unaffected family members revealed a family diagnosis of classical EDS with a heterozygous mutation in COL5A1 (c.1502del; p.Pro501Leufs*57). Additional to the major clinical criteria for classical EDS-generalized joint hypermobility, hyperelastic skin, and atrophic scarring -the child aged four years presented with generalized alveolar bone loss up to 80%, early loss of two lower incisors, severe gingival recession, and generalized lack of attached gingiva. Due to these clinical findings, an additional diagnosis of periodontal EDS was suspected. Further genetic analysis revealed the novel missense mutation c.658T>G (p.Cys220Gly) in C1R in a heterozygous state. Early severe periodontitis in association with generalized lack of attached gingiva is pathognomonic for periodontal EDS and led to the right clinical and genetic diagnosis in the present case.
我们报告了一例极为罕见的伴有早发性重度牙周炎和广泛附着龈缺失的经典型和牙周型埃勒斯-当洛斯综合征(EDS)病例。对一个有经典型 EDS 的德国家庭进行了体格检查和牙科评估,并进行了外显子组和 Sanger 测序。由于受影响儿童具有特定的牙周表型,因此怀疑其患有牙周型 EDS。对两名受影响和三名未受影响的家庭成员进行体格检查和基因检查,发现了经典型 EDS 的家族诊断,COL5A1 中存在杂合突变(c.1502del; p.Pro501Leufs*57)。除了经典 EDS 的主要临床标准(全身性关节过度活动、弹性皮肤和萎缩性瘢痕)外,这名四岁的孩子还表现为全身性牙槽骨丧失高达 80%、两颗下切牙早期丧失、严重的牙龈退缩和广泛的附着龈缺失。由于这些临床表现,怀疑其患有牙周型 EDS。进一步的基因分析显示,C1R 中存在杂合状态的新型错义突变 c.658T>G(p.Cys220Gly)。早发性重度牙周炎伴广泛附着龈缺失是牙周型 EDS 的特征性表现,在本病例中导致了正确的临床和基因诊断。