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Congenital dermal sinuses are epithelium-lined tracts that extend from an opening in the skin through deeper tissues. These sinuses consist of incompletely separated cutaneous ectoderm from the neuroectoderm due to embryological maldevelopment. We present two cases of cervical dermal sinus that presented to our outpatient clinic with diminished hand muscle strength. These two cervical dermal sinus cases were followed-up with the diagnosis of polyneuropathy and motor neuron disease based on atrophy and weakness of the intrinsic hand muscles. The pathophysiology of the cases is discussed