给编辑的信:板层状鱼鳞病和关节挛缩在一个早产儿。

Chiara De Leonibus, Claudio Lembo, Alfredo Santantonio, Tiziana Fioretti, Silvana Rojo, Francesco Salvatore, Massimiliano De Vivo, Gabriella Esposito, Paolo Giliberti
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引用次数: 4

摘要

板层状鱼鳞病是一种罕见的先天性疾病,以出生时的胶膜和面部异常(外翻和外翻)为特征。主要的潜在遗传缺陷是TGM1,在50%的患者中发现该基因突变。由于疾病的严重程度,早期诊断对于确定具体治疗方法至关重要。我们报告一例严重板层状鱼鳞病和关节挛缩症,没有典型的面部表现,阴性TGM1突变。临床改善仅在口服类维生素a治疗后实现,突出了早期诊断和及时给予特定治疗的重要性。
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Photoletter to the editor: Lamellar ichthyosis and arthrogryposis in a premature neonate.

Lamellar ichthyosis is a rare congenital disorder characterized by collodion membrane at birth and facial anomalies (eclabium and ectropion). The major underlying genetic defect is in TGM1, with mutations of this gene found in 50% of patients. An early diagnosis is fundamental in view of establishing a specific treatment due to the severity of the disease. We report a case of severe lamellar ichthyosis and arthrogryposis, without the typical facial presentation, negative for TGM1 mutations. The clinical improvement was achieved only after treatment with oral retinoids, highlighting the importance of early diagnosis and prompt administration of a specific therapy.

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