Research 2018

Site presented by Bill Tillier.


(Because of the way articles come out, there is some overlap between years.)

Mariampillai, K., Granger, B., Amelin, D., Guiguet, M., Hachulla, E., Maurier, F., . . . Benveniste, O. (2018). Development of a New Classification System for Idiopathic Inflammatory Myopathies Based on Clinical Manifestations and Myositis-Specific Autoantibodies. JAMA Neurology. https://doi.org/10.1001/jamaneurol.2018.2598

Caughey, G. E., Gabb, G. M., Ronson, S., Ward, M., Beukelman, T., Hill, C. L., & Limaye, V. (2018). Association of Statin Exposure With Histologically Confirmed Idiopathic Inflammatory Myositis in an Australian Population. JAMA Internal Medicine, 2018. https://doi.org/10.1001/jamainternmed.2018.2859
See Fig. 1 below.


Naddaf, E., Barohn, R. J., & Dimachkie, M. M. (2018). Inclusion Body Myositis: Update on Pathogenesis and Treatment. Neurotherapeutics. https://doi.org/10.1007/s13311-018-0658-8

Selva-O'Callaghan, A., Pinal-Fernandez, I., Trallero-Araguás, E., Milisenda, J. C., Grau-Junyent, J. M., & Mammen, A. L. (2018). Classification and management of adult inflammatory myopathies. The Lancet Neurology, 17(9), 816-828. https://doi.org/10.1016/S1474-4422(18)30254-0

Fares, A., & Borrmann, D. (2018). Neurochemical Aspects of Alzheimer's Disease and Movement Disturbances: A Theory of beta-Amyloid and tau-Protein. American Journal of Alzheimer's Disease & Other Dementias, 153331751879063. https://doi.org/10.1177/1533317518790631

Herbelet, S., & De Bleecker, J. L. (2018). Immune checkpoint failures in inflammatory myopathies: An overview. Autoimmunity Reviews, 17(8), 746-754. https://doi.org/10.1016/j.autrev.2018.01.026

Lilleker, J. B. (2018). Advances in the early diagnosis and therapy of inclusion body myositis. Current Opinion in Rheumatology, 1. https://doi.org/10.1097/BOR.0000000000000537

Miller, F. W., Lamb, J. A., Schmidt, J., & Nagaraju, K. (2018). Risk factors and disease mechanisms in myositis. Nature Reviews Rheumatology, 14(5), 255-268. https://doi.org/10.1038/nrrheum.2018.48

Felice, K. J., Whitaker, C. H., Wu, Q., Larose, D. T., Shen, G., Metzger, A. L., & Barton, R. W. (2018). Sensitivity and clinical utility of the anti-cytosolic 5'-nucleotidase 1A (cN1A) antibody test in sporadic inclusion body myositis: report of 40 patients from a single neuromuscular center. Neuromuscular Disorders. https://doi.org/10.1016/j.nmd.2018.06.005
The anti-cN1A antibody test has limited diagnostic utility in IBM.

Gooch, C. L. (2017). How can we pay the piper? Inclusion body myositis and the high price of a longer life. Muscle & Nerve, 56(5), 837-838. https://doi.org/10.1002/mus.25749

Capkun, G., Callan, A., Tian, H., Wei, Z., Zhao, C., Agashivala, N., & Barghout, V. (2017). Burden of illness and healthcare resource use in United States patients with sporadic inclusion body myositis. Muscle & Nerve, 56(5), 861-867. https://doi.org/10.1002/mus.25686

Jabari, D., Vedanarayanan, V. V, Barohn, R. J., & Dimachkie, M. M. (2018). Update on Inclusion Body Myositis. Current Rheumatology Reports, 20(8), 52. https://doi.org/10.1007/s11926-018-0755-z
Summary: There has been growing evidence toward degeneration as the primary process in sIBM. This is consistent with the refractory nature of this disease. Improving our understanding of this disease pathogenesis will propel efforts to find an effective therapy.

Keshishian, A., Greenberg, S. A., Agashivala, N., Baser, O., & Johnson, K. (2018). Health care costs and comorbidities for patients with inclusion body myositis. Current Medical Research and Opinion, 204(2-3), 1-16. https://doi.org/10.1080/03007995.2018.1486294
Conclusions: IBM patients utilize more health care resources and incur higher health care costs than patients without IBM. Furthermore, IBM patients were more likely to have multiple comorbidities, including cardiovascular risk factors and events, muscle and joint pain, and pulmonary complications compared to those without IBM.

Schmidt, J. (2018). Current Classification and Management of Inflammatory Myopathies. Journal of Neuromuscular Diseases, 5(2), 109-129. https://doi.org/10.3233/JND-180308

Zhang, Y., Pu, C., Lu, X., Shi, Q., Wang, H., Ban, R., & Liu, H. (2018). Pathological findings in sporadic inclusion body myositis and GNE myopathy. International Journal of Clinical and Experimental Pathology, 11(5), 2907-2911. http://www.ijcep.com/files/ijcep0064777.pdf

Türk, M., Weber, I., Vogt-Ladner, G., Schröder, R., & Winterholler, M. (2018). Diaphragmatic dysfunction as the presenting symptom in neuromuscular disorders: A retrospective longitudinal study of etiology and outcome in 30 German patients. Neuromuscular Disorders, 28(6), 484-490. https://doi.org/10.1016/j.nmd.2018.04.001

Camargo, L. V. De, Carvalho, M. S. De, Shinjo, S. K., Oliveira, A. S. B. de, & Zanoteli, E. (2018). Clinical, Histological, and Immunohistochemical Findings in Inclusion Body Myositis. BioMed Research International, 2018, 1-15. https://doi.org/10.1155/2018/5069042

Britton, D., Karam, C., Schindler, J. S. (2018). Swallowing and Secretion Management in Neuromuscular Disease. Clinics in Chest Medicine, 39(2), 449-457. https://doi.org/10.1016/j.ccm.2018.01.007
Neuromuscular disease (NMD) frequently leads to difficulty swallowing (dysphagia) and managing secretions (sialorrhea and/or excessive thick mucus). . . . Swallowing function is also important for saliva management, because humans typically generate more than one-half liter of saliva per day and unconsciously swallow approximately every 1 to 3 minutes. . . . .For patients struggling with sialorrhea and/or excessive thick mucus, pharmacologic management may aid the balance between the extremes of excessive dryness or secretions. In addition to behavioral management techniques and/or the use of a suction machine, oral or transdermal medications may offer relief. For sialorrhea, medications with drying side effects are used, such as amitriptyline (Elavil), scopolamine (transdermal hyoscine), atropine, and/or glycopyrrolate (Robinul). For management of excessive thick mucus, medications that thin out saliva (eg, guaifenesin) without additional drying are helpful.

Oldroyd, A., Lilleker, J., & Chinoy, H. (2017). Idiopathic inflammatory myopathies - a guide to subtypes, diagnostic approach and treatment. Clinical Medicine, 17(4), 322-328. https://doi.org/10.7861/clinmedicine.17-4-322

Britson, K. A., Yang, S. Y., & Lloyd, T. E. (2018). New Developments in the Genetics of Inclusion Body Myositis. Current Rheumatology Reports, 20(5), 26. https://doi.org/10.1007/s11926-018-0738-0

Badrising, U. A., Tsonaka, R., Hiller, M., Niks, E. H., Evangelista, T., Lochmüller, H., . . . Spitali, P. (2017). Cytokine Profiling of Serum Allows Monitoring of Disease Progression in Inclusion Body Myositis. Journal of Neuromuscular Diseases, 4(4), 327-335.
Results: Ten cytokines (TRAIL, IL-8, MIF, MCP-1, LIF, IP-10, IFN-alpha2, MIG, bNGF and IL-3) were identified to be good to excellent markers to discern IBM patients from healthy controls. Three cytokines (IP-10, Eotaxin and SDF1A) changed significantly upon methotrexate treatment as compared with the natural clinical course. Muscle strength loss was associated with changes in IL-8 and SDF1A levels. IFN-gamma levels were only associated with survival of IBM patients before correction for multiple comparisons.
Discussion: Cytokine profiling can discriminate IBM patients from healthy controls and other neuromuscular disorders. Immunosuppression with methotrexate affects cytokine levels in IBM. IL-8 and SDF1A could serve as biomarkers for disease progression. https://doi.org/10.3233/JND-170234

Johari, M., Arumilli, M., Palmio, J., Savarese, M., Tasca, G., Mirabella, M., . . . Udd, B. (2017). Association study reveals novel risk loci for sporadic inclusion body myositis. European Journal of Neurology, 24(4), 572-577. https://doi.org/10.1111/ene.13244
Despite the challenges of sIBM being a complex disorder, genetic research is likely to result in important findings for understanding the etiology of the disease. Unusual combinations of different mutated genes in the same individual may result in a higher risk of developing a multifactorial sporadic disease like sIBM

Needham, M., & Mastaglia, F. (2017). Advances in inclusion body myositis: genetics, pathogenesis and clinical aspects. Expert Opinion on Orphan Drugs, 5(5), 431-443. https://doi.org/10.1080/21678707.2017.1318056

Dardis, C., Antezana, A., Tanji, K., & Maccabee, P. J. (2017). Inclusion Body Myositis: A Case Presenting with Respiratory Failure and Autopsy Findings Leading to the Hypothesis of a Paraneoplastic Cause. American Journal of Case Reports, 18, 700-706. https://doi.org/10.12659/AJCR.903566

Jorgensen, A. N., Aagaard, P., Nielsen, J. L., Christiansen, M., Hvid, L. G., Frandsen, U., & Diederichsen, L. P. (2017). Physical function and muscle strength in sporadic inclusion body myositis. Muscle & Nerve, 56(6), E50-E58. https://doi.org/10.1002/mus.25603

Alfano, L. N., Yin, H., Dvorchik, I., Maus, E. G., Flanigan, K. M., Mendell, J. R., & Lowes, L. P. (2017). Modeling functional decline over time in sporadic inclusion body myositis. Muscle & Nerve, 55(4), 526-531. https://doi.org/10.1002/mus.25373