Monomelic Amyotrophy/Hirayama Disease: Surgical Outcome in a Large Cohort of Indian Patients

Hirayama disease (HD) is a cervical compressive myelopathy. Anterior cervical discectomy and fusion (ACDF) is identified as the best surgical approach. We evaluated surgical outcomes and factors influencing ACDF in HD. Between 2015 and 2019, 126 patients with HD underwent ACDF. Contrast magnetic res...

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Published in:	World neurosurgery Vol. 183; pp. e88 - e97
Main Authors:	Vengalil, Seena, Pruthi, Nupur, Bhat, Dhananjay, Uppar, Alok Mohan, Polavarapu, Kiran, Preethish-Kumar, Veeramani, Nashi, Saraswati, Rajesh, Srinithya, Aswini, Nangamangalam Srinivasan, Behera, Bidyut Prava, Vandhiyadevan, Govindan Dhanasekaran, Prasad, Chandrajit, Baskar, Dipti, Kulanthaivelu, Karthik, Saravanan, Akshaya, Kandavel, Thennarasu, Nishadham, Vikas, Huddar, Akshata, Unnikrishnan, Gopikrishnan, Thomas, Aneesha, Keerthipriya, Muddasu Suhasini, Sanka, Sai Bhargava, Manjunath, Nisha, Valasani, Ravi Kiran, Bardhan, Mainak, Nalini, Atchayaram
Format:	Journal Article
Language:	English
Published:	United States Elsevier Inc 01-03-2024
Subjects:	Cervical Vertebrae - diagnostic imaging Cervical Vertebrae - pathology Cervical Vertebrae - surgery Disabled Persons Flexion-induced myelopathy Hirayama disease Humans India Magnetic resonance imaging Monomelic amyotrophy Motor Disorders Spinal Muscular Atrophies of Childhood - diagnosis Spinal Muscular Atrophies of Childhood - surgery Surgery Treatment Outcome MMA Magnetic resonance imaging MRI Surgery UL T2W Monomelic amyotrophy ACDF Flexion-induced myelopathy HD Hirayama disease India
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Summary:	Hirayama disease (HD) is a cervical compressive myelopathy. Anterior cervical discectomy and fusion (ACDF) is identified as the best surgical approach. We evaluated surgical outcomes and factors influencing ACDF in HD. Between 2015 and 2019, 126 patients with HD underwent ACDF. Contrast magnetic resonance imaging of the cervical spine in full flexion was performed. Clinical examination and preoperative/postoperative assessment of hand function using Fugl-Meyer assessment, Jebsen-Taylor hand function test, and handheld dynamometry were performed at 3-monthly intervals for 1 year. Surgical outcomes were assessed as per the Odom criteria and Hirayama outcome questionnaire. Age at onset and duration of illness were 12–31 years (mean, 18 ± 2.7) and 1–96 months (32.7 ± 24.4), respectively. All patients had progressive weakness and wasting of the affected limb. Cord atrophy was seen in 97.1%, with epidural detachment and engorgement of the posterior epidural venous plexus in all. All patients underwent ACDF. Of these patients, 54% had an excellent/good outcome and 39% had a satisfactory outcome as per the Odom scale at last follow-up (mean, 44.9 ± 16.5 months) after surgery. Handheld dynamometry showed improvement from preoperative values to 1 year follow-up. Duration of illness and age at onset had a negative correlation and the preoperative Fugl-Meyer score had a positive correlation with improvement. ACDF resulted in remarkable improvement or stabilization in neurologic deficits in many patients with HD. Because motor disability ensues over time, early surgical intervention during the progressive phase is advocated.
Bibliography:	ObjectType-Article-1 SourceType-Scholarly Journals-1 ObjectType-Feature-2 content type line 23
ISSN:	1878-8750 1878-8769 1878-8769
DOI:	10.1016/j.wneu.2023.11.087