Chopra Ajay, Mitra Debdeep, Kandpal Renu, Agarwal Reetu
Department of Dermatology, Base Hospital Delhi Cantt, New Delhi, India.
Int J Trichology. 2018 Jan-Feb;10(1):17-20. doi: 10.4103/ijt.ijt_55_17.
Ankyloblepharon-ectodermal defect-cleft lip/cleft palate (AEC) syndrome is one of the variants of ectodermal dysplasia. It is an autosomal dominant disorder comprising of ankyloblepharon, ectodermal dysplasia, and cleft palate or cleft lip. In 1976, it wasfirst described by Hay and Wells, therefore also known as Hay-Wells syndrome. The characteristic feature of this syndrome is "ankyloblepharon filiforme adnatum", which refers to the partial thickness fusion of the eyelid margins. The "curly hair-ankyloblepharon-nail disease (CHAND) syndrome" is a clinical variant of AEC syndrome. We report a rare case of a 7-year-old girl child who presented with history of abnormal dentition, 20 nail dystrophy, and light-colored, sparse curly hairs since birth. Parents gave history that at the time of birth, her both eyelids were fused partially, which was surgically corrected by an ophthalmologist at 1 month of age. There was no history of hypohidrosis or anhidrosis, heat intolerance, cleft lip or cleft palate. Microscopy of the hair shaft found "bubbly hair" morphology. This case is unique as it is a rare presentation, and awareness should be there for this constellation of findings so that the systemic associations can be investigated. "Bubble hair" morphology on microscopy is a unique feature in this rare autosomal recessive condition.
睑缘粘连-外胚层缺陷-唇腭裂(AEC)综合征是外胚层发育异常的一种变体。它是一种常染色体显性疾病,包括睑缘粘连、外胚层发育异常以及腭裂或唇裂。1976年,它首次由海伊和韦尔斯描述,因此也被称为海伊-韦尔斯综合征。该综合征的特征性表现是“丝状粘连性睑缘粘连”,指的是睑缘的部分厚度融合。“卷发-睑缘粘连-甲病(CHAND)综合征”是AEC综合征的一种临床变体。我们报告了一例罕见病例,一名7岁女童自出生以来有牙列异常、20个指甲营养不良以及浅色、稀疏卷发的病史。父母称,出生时她的双眼睑部分融合,1个月大时由眼科医生进行了手术矫正。无少汗或无汗、不耐热、唇裂或腭裂病史。毛发镜检发现“气泡样毛发”形态。该病例很独特,因为其表现罕见,对于这一系列表现应提高认识,以便能对全身相关情况进行调查。镜检时的“气泡样毛发”形态是这种罕见常染色体隐性疾病的独特特征。