Idiopathic isolated unilateral hypoglossal nerve palsy: a report of two cases and review of the literature

Publication date: Available online 2 February 2018
Source:Journal of Oral and Maxillofacial Surgery
Author(s): Akio Shibata, Masashi Kimura, Kenichiro Ishiabashi, Masahiro Umemura
Hypoglossal nerve palsy (HNP) is a common finding in neurological diseases when associated with other cranial nerve palsies or further pathology and exhibits characteristic clinical manifestations, including unilateral atrophy of the musculature of the tongue. It occasionally appears as the initial or solitary sign of an intracranial or extracranial space-occupying lesion, head/neck injury, or vascular abnormality of the internal carotid artery. There are few cases of idiopathic isolated unilateral HNP, which should be diagnosed through exclusion. Here, we describe two patients who experienced different outcomes and present a literature review of idiopathic isolated unilateral HNP. Case 1 was a 71-year-old man who was referred with a 1-month history of dysphagia and speech impairment. Intraoral examination revealed marked left-sided hemiatrophy of the tongue and deviation toward the left on protrusion. On coronal Tl-weighted magnetic resonance imaging (MRI), left-sided hemiatrophy of the tongue was clearly visible through the deviation of the median septum to the left. The patient was diagnosed with idiopathic isolated unilateral HNP through exclusion, and was treated with steroids and mecobalamin, but he did not recover. Case 2 was a 32-year-old man complaining of tongue weakness since 2 days. On examination, left HNP was evident, with deviation of the tongue to the left on protrusion. He was diagnosed with idiopathic isolated unilateral HNP through exclusion, and was treated with steroids. After 3 weeks, the patient had completely recovered. To the best of our knowledge, this is the first detailed literature review on idiopathic isolated unilateral HNP. This condition is very rare but should be considered for diagnosis. It warrants a thorough and stepwise approach for etiological diagnosis.



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