Abstract: We present a case of r4etherton disease, where the hairs lacked the characteristic microscopic feature of Uichorrhexis invaginata. In its piace were certain hairs with a goif tee tnorphoiogy. These represent the proximal half of the invaginate node seen in typicai Netherton disease. Scanning eiectron microscopy demonstrated the three-dimensiona) quality of this abnormaiity, which is subtie when assessed by iight microscopy aione. We describe this sign so that it might be recognized when seen in isolation, as here, and aiiow tire diagnosis of Netherton disease to be confirmed.