Abstract
IntroductionIsolated unilateral hypoglossal nerve palsy is an infrequent condition that usually appears in connection to an underlying disease. Rarely, the cause cannot be determined, and there is no symptom remission.Case descriptionWe report two cases of male patients who developed persistent idiopathic isolated unilateral hypoglossal nerve palsy. The younger patient complained of involuntary movements of the tongue, while the older patient developed dysarthria. In both cases, the symptoms lasted for several weeks before the initial clinical examination, and after comprehensive clinical, laboratory, and imaging investigations, no underlying cause was detected. Both patients did not show remission of symptoms on the follow-up examinations done after several months.DiscussionIdiopathic isolated unilateral hypoglossal nerve palsy is an underreported condition, most likely due to the expected reversible nature of the disease. In the case of persistence of symptoms, extensive medical assessment is needed before postulating a diagnosis of persistent idiopathic isolated unilateral hypoglossal nerve palsy. There are no treatment guidelines for this condition, but oral steroids were reported as a treatment option. Therefore, physicians should use a systematic approach to exclude serious underlying pathology or identify a treatable condition.
Highlights
Isolated unilateral hypoglossal nerve palsy is an infrequent condition that usually appears in connection to an underlying disease
In the case of persistence of symptoms, extensive medical assessment is needed before postulating a diagnosis of persistent idiopathic isolated unilateral hypoglossal nerve palsy
Nasopharyngeal cancer, Chiari malformation, and dural arteriovenous fistula are other known causes [1]. These quite different etiologies emphasize the necessity of extensive medical assessment when confronted with a case of hypoglossal nerve palsy (HNP), especially when postulating a diagnosis of idiopathic Isolated unilateral hypoglossal nerve palsy (IUHNP)
Summary
HNP in general appears together with other neurological abnormalities in a large variety of diseases. Nasopharyngeal cancer, Chiari malformation, and dural arteriovenous fistula are other known causes [1] These quite different etiologies emphasize the necessity of extensive medical assessment when confronted with a case of HNP, especially when postulating a diagnosis of idiopathic IUHNP. Two other cases of idiopathic IUHNP reported persistency of symptoms for a period of 36 months and 24 months respectively [4, 7]. To the best of our knowledge, our younger patient is the second reported having persistent idiopathic IUNHP at the age of 17 years. It has been suggested by Lee and colleagues that idiopathic IUHNP is a clinical entity similar to Bell’s palsy (BP) [6].
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