Department of Dermatology, College of Medicine, Imam Abdulrahman Bin Faisal University (formerly University of Dammam), Dammam, Saudi Arabia.
Saudi Board of Dermatology, Arab Board of Dermatology and Venereology (ABDV), King Fahd Hospital, Hofuf, Saudi Arabia.
Am J Case Rep. 2022 Feb 24;23:e935393. doi: 10.12659/AJCR.935393.
BACKGROUND Annular epidermolytic ichthyosis is a rare form of epidermolytic ichthyosis caused by specific pathogenic variants of KRT1 and KRT10. Classically, it manifests at birth with variable degrees of erythroderma and superficial erosions, which subsequently improve with time. Later, it is characterized by a cyclic history of annular hyperkeratotic erythematous plaques over the trunk and proximal extremities, with or without palmoplantar keratoderma. Greither syndrome, another autosomal dominant disorder of KRT1 mutation, is demonstrated by the diffuse, thick, scaly yellow PPK with transgrediens and erythematous border extending up to the Achilles' tendon, patchy hyperkeratotic plaques over the knees, shins, thighs, elbows, knuckles, and axillary folds. We describe a patient with clinical findings consistent with annular epidermolytic ichthyosis mimicking Greither disease with a likely associated pathogenic variant of KRT1. CASE REPORT A 3-year-old Saudi girl presented with a diffuse palmoplantar keratoderma (PPK) extending to the dorsal aspects of the hands and feet up to the Achilles' tendon, first noticed at the age of 3 months, with a history of recurrent coin-shaped erythematous crusted erosions over the trunk, which were spontaneously healed over time, and an associated history of hyperhidrosis. Patchy hyperkeratotic plaques were noticed upon further examination over the bilateral elbows, axillary folds, and oral commissures. CONCLUSIONS The phenotype of our patient is consistent with the clinical features described for AEI, making the new K1 variant a likely pathogenic variant. When K1 mutation is the causative variant of the disease expression, phenotypically, it can present with Greither-like PPK.
环状表皮松解性鱼鳞病是一种罕见的表皮松解性鱼鳞病,由 KRT1 和 KRT10 的特定致病变体引起。经典地,它在出生时表现为不同程度的红皮病和浅表糜烂,随后随时间改善。后来,它的特征是躯干和近端肢体出现环状角化过度性红斑斑块的周期性病史,伴有或不伴有掌跖角化过度症。Greither 综合征是另一种由 KRT1 突变引起的常染色体显性疾病,其特征是弥漫性、厚的、片状黄色 PPK,具有 transgrediens 和红斑边界延伸至跟腱,膝盖、小腿、大腿、肘部、指关节和腋窝褶皱处有斑片状角化过度斑块。我们描述了一名患者,其临床表现与环状表皮松解性鱼鳞病一致,类似于 Greither 病,可能与 KRT1 的相关致病变体有关。
一名 3 岁沙特女孩因弥漫性掌跖角化过度症(PPK)就诊,PPK 延伸到手和脚的背侧,直至跟腱,在 3 个月大时首次发现,有反复硬币状红斑结痂糜烂的病史,随时间自行愈合,并有相关的多汗症病史。进一步检查发现双侧肘部、腋窝褶皱和口腔口角处有斑片状角化过度斑块。
我们患者的表型与描述的 AEI 临床特征一致,因此新的 K1 变体可能是致病变体。当 K1 突变是疾病表达的致病变体时,表型上,它可以表现为类似于 Greither 的 PPK。