Currently there is no cure for ALS, so the focus of medical care is to provide symptomatic management for all patients. Disease-modifying pharmacologic therapies to treat ALS include riluzole, edaravone, and sodium phenylbutyrate/taurursodiol.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[56]Paganoni S, Macklin EA, Hendrix S, et al. Trial of sodium phenylbutyrate-taurursodiol for amyotrophic lateral sclerosis. N Engl J Med. 2020 Sep 3;383(10):919-30.
https://www.nejm.org/doi/10.1056/NEJMoa1916945
http://www.ncbi.nlm.nih.gov/pubmed/32877582?tool=bestpractice.com
There is no evidence for a differential response to treatment between patients with familial versus sporadic ALS.[57]Benatar M, Kurent J, Moore DH. Treatment for familial amyotrophic lateral sclerosis/motor neuron disease. Cochrane Database Syst Rev. 2009 Jan 21;(1):CD006153.
https://www.cochranelibrary.com/cdsr/doi/10.1002/14651858.CD006153.pub2/full
http://www.ncbi.nlm.nih.gov/pubmed/19160266?tool=bestpractice.com
Care of patients with ALS is best provided in a multidisciplinary ALS clinic, with a team comprising a respiratory therapist, physical and occupational therapists, a dietitian, a speech and swallowing specialist, a social worker, and other specialists as needed. This approach allows for optimization of care, and has been shown to be associated with improved survival, fewer hospital admissions, increased use of adaptive equipment, and better quality of life.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[58]Lee CN. Reviewing evidences on the management of patients with motor neuron disease. Hong Kong Med J. 2012 Feb;18(1):48-55.
https://www.hkmj.org/system/files/hkm1202p48.pdf
http://www.ncbi.nlm.nih.gov/pubmed/22302912?tool=bestpractice.com
[59]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: multidisciplinary care, symptom management, and cognitive/behavioral impairment (an evidence-based review). Neurology. 2009 Oct 13;73(15):1227-33.
https://n.neurology.org/content/73/15/1227
http://www.ncbi.nlm.nih.gov/pubmed/19822873?tool=bestpractice.com
[60]Van den Berg JP, Kalmijn S, Lindeman E, et al. Multidisciplinary ALS care improves quality of life in patients with ALS. Neurology. 2005 Oct 25;65(8):1264-7.
http://www.ncbi.nlm.nih.gov/pubmed/16247055?tool=bestpractice.com
[61]Lazovic M, Nikolic D, Boyer FC, et al. Evidence-based position paper on Physical and Rehabilitation Medicine practice for people with amyotrophic lateral sclerosis. Eur J Phys Rehabil Med. 2022 Apr;58(2):271-9.
https://www.minervamedica.it/en/journals/europa-medicophysica/article.php?cod=R33Y2022N02A0271
http://www.ncbi.nlm.nih.gov/pubmed/34786907?tool=bestpractice.com
Immunizations, including pneumococcal vaccination, COVID-19 vaccination, and annual seasonal influenza vaccination, are recommended in patients with ALS, given the underlying chronic pulmonary dysfunction.
Informing patients and their families about the diagnosis, and ongoing counseling regarding prognosis, treatments, and end-of-life issues, with special emphasis on advance directives, are extremely important aspects of the patient-physician relationship.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[35]Andersen PM, Abrahams S, Borasio GD, et al. EFNS guidelines on the clinical management of amyotrophic lateral sclerosis (MALS) - revised report of an EFNS task force. Eur J Neurol. 2012 Mar;19(3):360-75.
https://onlinelibrary.wiley.com/doi/10.1111/j.1468-1331.2011.03501.x
http://www.ncbi.nlm.nih.gov/pubmed/21914052?tool=bestpractice.com
[62]Taylor LP, Besbris JM, Graf WD, et al. Clinical guidance in neuropalliative care: an AAN position statement. Neurology. 2022 Mar 8;98(10):409-16.
https://n.neurology.org/content/98/10/409
http://www.ncbi.nlm.nih.gov/pubmed/35256519?tool=bestpractice.com
[63]van Eenennaam RM, Kruithof WJ, van Es MA, et al. Discussing personalized prognosis in amyotrophic lateral sclerosis: development of a communication guide. BMC Neurol. 2020 Dec 14;20(1):446.
https://bmcneurol.biomedcentral.com/articles/10.1186/s12883-020-02004-8
http://www.ncbi.nlm.nih.gov/pubmed/33308184?tool=bestpractice.com
Pharmacologic therapy
Riluzole
All patients are started on riluzole at the time of diagnosis.[35]Andersen PM, Abrahams S, Borasio GD, et al. EFNS guidelines on the clinical management of amyotrophic lateral sclerosis (MALS) - revised report of an EFNS task force. Eur J Neurol. 2012 Mar;19(3):360-75.
https://onlinelibrary.wiley.com/doi/10.1111/j.1468-1331.2011.03501.x
http://www.ncbi.nlm.nih.gov/pubmed/21914052?tool=bestpractice.com
[64]Miller RG, Mitchell JD, Moore DH. Riluzole for amyotrophic lateral sclerosis (ALS)/motor neuron disease (MND). Cochrane Database Syst Rev. 2012 Mar 14;(3):CD001447.
https://www.cochranelibrary.com/cdsr/doi/10.1002/14651858.CD001447.pub3/full
http://www.ncbi.nlm.nih.gov/pubmed/22419278?tool=bestpractice.com
[65]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: drug, nutritional, and respiratory therapies (an evidence-based review). Neurology. 2009 Oct 13;73(15):1218-26. [Errata in: Neurology. 2009 Dec 15;73(24):2134; Neurology. 2010 Mar 2;74(9):781.]
https://n.neurology.org/content/73/15/1218
http://www.ncbi.nlm.nih.gov/pubmed/19822872?tool=bestpractice.com
Riluzole prolongs survival in patients with ALS, with potential advantage in patients with bulbar onset.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[64]Miller RG, Mitchell JD, Moore DH. Riluzole for amyotrophic lateral sclerosis (ALS)/motor neuron disease (MND). Cochrane Database Syst Rev. 2012 Mar 14;(3):CD001447.
https://www.cochranelibrary.com/cdsr/doi/10.1002/14651858.CD001447.pub3/full
http://www.ncbi.nlm.nih.gov/pubmed/22419278?tool=bestpractice.com
[66]Bensimon G, Lacomblez L, Meininger V; ALS/Riluzole Study Group. A controlled trial of riluzole in amyotrophic lateral sclerosis. N Engl J Med. 1994 Mar 3;330(9):585-91.
https://www.nejm.org/doi/full/10.1056/NEJM199403033300901
http://www.ncbi.nlm.nih.gov/pubmed/8302340?tool=bestpractice.com
Annual survival benefit with riluzole is approximately 9%.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
Significant hepatic toxicity and neutropenia are associated with riluzole use, but are rare.[67]Weber G, Bitterman H. Riluzole-induced neutropenia. Neurology. 2004 May 11;62(9):1648.
http://www.ncbi.nlm.nih.gov/pubmed/15136708?tool=bestpractice.com
Liver function tests and complete blood count should be monitored monthly for the first 3 months, then every 3 months thereafter.[64]Miller RG, Mitchell JD, Moore DH. Riluzole for amyotrophic lateral sclerosis (ALS)/motor neuron disease (MND). Cochrane Database Syst Rev. 2012 Mar 14;(3):CD001447.
https://www.cochranelibrary.com/cdsr/doi/10.1002/14651858.CD001447.pub3/full
http://www.ncbi.nlm.nih.gov/pubmed/22419278?tool=bestpractice.com
Nausea and lethargy are possible adverse effects of riluzole.[59]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: multidisciplinary care, symptom management, and cognitive/behavioral impairment (an evidence-based review). Neurology. 2009 Oct 13;73(15):1227-33.
https://n.neurology.org/content/73/15/1227
http://www.ncbi.nlm.nih.gov/pubmed/19822873?tool=bestpractice.com
Edaravone
Edaravone is a free radical scavenger that exerts neuroprotective effects by reducing oxidative stress in motor neurons. It is approved in the US and some other countries (but not in Europe) for treating ALS. Although one phase 3 randomized controlled trial did not show an overall benefit of intravenous edaravone in slowing ALS progression, a benefit has been demonstrated in a subgroup of patients.[68]Abe K, Itoyama Y, Sobue G, et al. Confirmatory double-blind, parallel-group, placebo-controlled study of efficacy and safety of edaravone (MCI-186) in amyotrophic lateral sclerosis patients. Amyotroph Lateral Scler Frontotemporal Degener. 2014 Dec;15(7-8):610-7.
https://www.tandfonline.com/doi/full/10.3109/21678421.2014.959024
http://www.ncbi.nlm.nih.gov/pubmed/25286015?tool=bestpractice.com
[69]Abe K, Aoki M, Tsuji S, et al; Writing Group; Edaravone (MCI-186) ALS 19 Study Group. Safety and efficacy of edaravone in well defined patients with amyotrophic lateral sclerosis: a randomised, double-blind, placebo-controlled trial. Lancet Neurol. 2017 Jul;16(7):505-12.
http://www.ncbi.nlm.nih.gov/pubmed/28522181?tool=bestpractice.com
The effect of edaravone on survival was not evaluated in the study; one real-world analysis reported that intravenous edaravone was associated with prolonged overall survival in a large predominantly riluzole-treated US cohort.[70]Brooks BR, Berry JD, Ciepielewska M, et al. Intravenous edaravone treatment in ALS and survival: an exploratory, retrospective, administrative claims analysis. EClinicalMedicine. 2022 Oct;52:101590.
https://www.thelancet.com/journals/eclinm/article/PIIS2589-5370(22)00320-0/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/35958519?tool=bestpractice.com
However, one cohort study reported that although intravenous edaravone therapy for approximately 1 year was feasible and mainly well tolerated, no disease-modifying benefit was observed compared with standard therapy alone (riluzole).[71]Witzel S, Maier A, Steinbach R, et al; German Motor Neuron Disease Network (MND-NET). Safety and effectiveness of long-term intravenous administration of edaravone for treatment of patients with amyotrophic lateral sclerosis. JAMA Neurol. 2022 Feb 1;79(2):121-30.
https://jamanetwork.com/journals/jamaneurology/fullarticle/2787473
http://www.ncbi.nlm.nih.gov/pubmed/35006266?tool=bestpractice.com
The treatment regimen for edaravone is very intensive, with treatment needed for 14 days initially, followed by treatment for a further 10 consecutive days a month as maintenance therapy. Edaravone is available in intravenous and oral formulations.
Sodium phenylbutyrate/taurursodiol
Sodium phenylbutyrate/taurursodiol is a combination of the salt of an aromatic fatty acid and a bile acid. The mechanism of action in patients with ALS is unclear. This combination therapy was demonstrated in one clinical trial to modestly slow progression of ALS over a 6-month period in patients with definite ALS (as defined by the revised El Escorial criteria) who had onset of symptoms within the previous 18 months. The magnitude of effect was similar to the effect size of edaravone over a 6-month period.[56]Paganoni S, Macklin EA, Hendrix S, et al. Trial of sodium phenylbutyrate-taurursodiol for amyotrophic lateral sclerosis. N Engl J Med. 2020 Sep 3;383(10):919-30.
https://www.nejm.org/doi/10.1056/NEJMoa1916945
http://www.ncbi.nlm.nih.gov/pubmed/32877582?tool=bestpractice.com
A larger confirmatory trial with expanded inclusion criteria is ongoing, with results expected in 2024. The combination drug received Food and Drug Administration (FDA) approval in 2022 and has conditional approval in Canada, but is not yet approved in Europe.
Symptomatic management
ALS is a progressive disease of the motor system and, as such, produces symptoms primarily relating to the location and degree of weakness. The exact treatment needs vary between patients, and will change with time for individual patients.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[35]Andersen PM, Abrahams S, Borasio GD, et al. EFNS guidelines on the clinical management of amyotrophic lateral sclerosis (MALS) - revised report of an EFNS task force. Eur J Neurol. 2012 Mar;19(3):360-75.
https://onlinelibrary.wiley.com/doi/10.1111/j.1468-1331.2011.03501.x
http://www.ncbi.nlm.nih.gov/pubmed/21914052?tool=bestpractice.com
[36]National Institute for Health and Care Excellence. Motor neurone disease: assessment and management. Jul 2019 [internet publication].
https://www.nice.org.uk/guidance/ng42
[59]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: multidisciplinary care, symptom management, and cognitive/behavioral impairment (an evidence-based review). Neurology. 2009 Oct 13;73(15):1227-33.
https://n.neurology.org/content/73/15/1227
http://www.ncbi.nlm.nih.gov/pubmed/19822873?tool=bestpractice.com
[65]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: drug, nutritional, and respiratory therapies (an evidence-based review). Neurology. 2009 Oct 13;73(15):1218-26. [Errata in: Neurology. 2009 Dec 15;73(24):2134; Neurology. 2010 Mar 2;74(9):781.]
https://n.neurology.org/content/73/15/1218
http://www.ncbi.nlm.nih.gov/pubmed/19822872?tool=bestpractice.com
[72]Khan A, Frazer-Green L, Amin R, et al. Respiratory management of patients with neuromuscular weakness: an American College of Chest Physicians clinical practice guideline and expert panel report. Chest. 2023 Mar 13 [Epub ahead of print].
https://journal.chestnet.org/article/S0012-3692(23)00353-7/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/36921894?tool=bestpractice.com
Respiratory and nutritional support is critical, with the actual intervention required dependent on the status of the patient.
Patients with significant weakness of bulbar musculature may require interventions for dysphagia, sialorrhea, and communication difficulties.
Patients with weakness in the extremities may require support aimed at maintaining mobility, therapy for spasticity, or devices aimed at maintaining upper extremity function.
Mental status changes that may require treatment include depression, anxiety, and pseudobulbar affect. Cognitive and behavioral changes associated with frontotemporal impairment may also require interventions.
General principles of respiratory dysfunction management
Patients with mild or moderate respiratory dysfunction may be entirely asymptomatic, or may have symptoms that are either nocturnal or exercise-related. Monitoring of respiratory function every 3 months is recommended by screening for subjective symptoms of respiratory insufficiency and objective signs of respiratory insufficiency.
Symptoms of respiratory insufficiency may be shortness of breath on exertion or at rest, orthopnea, frequent awakenings at night, early morning headaches or confusion (due to nocturnal hypercapnia), poor vocal projection, and poor cough efficacy.
Options for objectively monitoring respiratory function include forced vital capacity (FVC), slow vital capacity (SVC), and sniff nasal inspiratory pressure (SNIP).[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[72]Khan A, Frazer-Green L, Amin R, et al. Respiratory management of patients with neuromuscular weakness: an American College of Chest Physicians clinical practice guideline and expert panel report. Chest. 2023 Mar 13 [Epub ahead of print].
https://journal.chestnet.org/article/S0012-3692(23)00353-7/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/36921894?tool=bestpractice.com
As supine FVC correlates well with symptoms of nocturnal hypoventilation, monitoring may be considered, along with erect FVC.[65]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: drug, nutritional, and respiratory therapies (an evidence-based review). Neurology. 2009 Oct 13;73(15):1218-26. [Errata in: Neurology. 2009 Dec 15;73(24):2134; Neurology. 2010 Mar 2;74(9):781.]
https://n.neurology.org/content/73/15/1218
http://www.ncbi.nlm.nih.gov/pubmed/19822872?tool=bestpractice.com
SNIP may be particularly useful in patients with bulbar weakness who might not be able to form a complete seal around the mouthpiece for obtaining FVC.[73]Morgan RK, McNally S, Alexander M, et al. Use of Sniff nasal-inspiratory force to predict survival in amyotrophic lateral sclerosis. Am J Respir Crit Care Med. 2005 Feb 1;171(3):269-74.
https://www.atsjournals.org/doi/10.1164/rccm.200403-314OC
http://www.ncbi.nlm.nih.gov/pubmed/15516537?tool=bestpractice.com
SNIP <40 cm H₂O has been found to be related to nocturnal hypoxia and has demonstrated a higher sensitivity for predicting 6-month mortality when compared with FVC <50%. In addition, nocturnal oximetry and/or sleep study can provide data regarding nocturnal hypoxia.[65]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: drug, nutritional, and respiratory therapies (an evidence-based review). Neurology. 2009 Oct 13;73(15):1218-26. [Errata in: Neurology. 2009 Dec 15;73(24):2134; Neurology. 2010 Mar 2;74(9):781.]
https://n.neurology.org/content/73/15/1218
http://www.ncbi.nlm.nih.gov/pubmed/19822872?tool=bestpractice.com
[72]Khan A, Frazer-Green L, Amin R, et al. Respiratory management of patients with neuromuscular weakness: an American College of Chest Physicians clinical practice guideline and expert panel report. Chest. 2023 Mar 13 [Epub ahead of print].
https://journal.chestnet.org/article/S0012-3692(23)00353-7/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/36921894?tool=bestpractice.com
Symptomatic patients or asymptomatic patients with at least one of: FVC values of less than 50% to 65% of predicted value, or SNIP <40 cm H₂O, or abnormal nocturnal oximetry
Noninvasive ventilation (NIV) is indicated for these patients.[35]Andersen PM, Abrahams S, Borasio GD, et al. EFNS guidelines on the clinical management of amyotrophic lateral sclerosis (MALS) - revised report of an EFNS task force. Eur J Neurol. 2012 Mar;19(3):360-75.
https://onlinelibrary.wiley.com/doi/10.1111/j.1468-1331.2011.03501.x
http://www.ncbi.nlm.nih.gov/pubmed/21914052?tool=bestpractice.com
[36]National Institute for Health and Care Excellence. Motor neurone disease: assessment and management. Jul 2019 [internet publication].
https://www.nice.org.uk/guidance/ng42
[58]Lee CN. Reviewing evidences on the management of patients with motor neuron disease. Hong Kong Med J. 2012 Feb;18(1):48-55.
https://www.hkmj.org/system/files/hkm1202p48.pdf
http://www.ncbi.nlm.nih.gov/pubmed/22302912?tool=bestpractice.com
[72]Khan A, Frazer-Green L, Amin R, et al. Respiratory management of patients with neuromuscular weakness: an American College of Chest Physicians clinical practice guideline and expert panel report. Chest. 2023 Mar 13 [Epub ahead of print].
https://journal.chestnet.org/article/S0012-3692(23)00353-7/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/36921894?tool=bestpractice.com
Historic guidelines have used a threshold for asymptomatic patients of FVC <50% of predicted value for initiation of NIV. More recent expert-based best practice recommendations have suggested earlier initiation of NIV in asymptomatic patients, with a threshold of FVC <65% of predicted, because of evidence suggesting that NIV initiation at FVC around 50% is better than at FVC below 50%.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[72]Khan A, Frazer-Green L, Amin R, et al. Respiratory management of patients with neuromuscular weakness: an American College of Chest Physicians clinical practice guideline and expert panel report. Chest. 2023 Mar 13 [Epub ahead of print].
https://journal.chestnet.org/article/S0012-3692(23)00353-7/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/36921894?tool=bestpractice.com
[74]Gonzalez Calzada N, Prats Soro E, Mateu Gomez L, et al. Factors predicting survival in amyotrophic lateral sclerosis patients on non-invasive ventilation. Amyotroph Lateral Scler Frontotemporal Degener. 2016 Jul-Aug;17(5-6):337-42.
http://www.ncbi.nlm.nih.gov/pubmed/27046344?tool=bestpractice.com
All guidelines suggest NIV initiation for symptomatic patients, even if they have FVC >65%.
When NIV is prescribed, modification in pressure settings to ensure maximum comfort is often required, as is individual determination of the most appropriate interface (contact between ventilator and patient). Anxiolytic medication, such as lorazepam, might help with the process of adjustment to this type of treatment.
NIV is usually used initially at night, but with symptom progression can be used up to 24 hours per day. Patients usually need uptitration of NIV pressures as their respiratory function declines. Also, patients often require different interfaces for day use and night use as they increase their NIV usage, to avoid constant pressure points with a single mask and to allow for better integration with activities.[72]Khan A, Frazer-Green L, Amin R, et al. Respiratory management of patients with neuromuscular weakness: an American College of Chest Physicians clinical practice guideline and expert panel report. Chest. 2023 Mar 13 [Epub ahead of print].
https://journal.chestnet.org/article/S0012-3692(23)00353-7/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/36921894?tool=bestpractice.com
For example, some patients with ALS will still be able to speak if supported by a nasal-only interface for NIV during the day. Successful NIV use has been shown to have a positive impact on quality of life and survival.[65]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: drug, nutritional, and respiratory therapies (an evidence-based review). Neurology. 2009 Oct 13;73(15):1218-26. [Errata in: Neurology. 2009 Dec 15;73(24):2134; Neurology. 2010 Mar 2;74(9):781.]
https://n.neurology.org/content/73/15/1218
http://www.ncbi.nlm.nih.gov/pubmed/19822872?tool=bestpractice.com
[75]Radunovic A, Annane D, Rafiq MK, et al. Mechanical ventilation for amyotrophic lateral sclerosis/motor neuron disease. Cochrane Database Syst Rev. 2017 Oct 6;(10):CD004427.
https://www.cochranelibrary.com/cdsr/doi/10.1002/14651858.CD004427.pub4/full
http://www.ncbi.nlm.nih.gov/pubmed/28982219?tool=bestpractice.com
Diaphragm pacing should not be used for patients with ALS because it is ineffective and may be harmful.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[76]McDermott CJ, Bradburn MJ, Maguire C, et al; DiPALS Writing Committee; DiPALS Study Group Collaborators. Safety and efficacy of diaphragm pacing in patients with respiratory insufficiency due to amyotrophic lateral sclerosis (DiPALS): a multicentre, open-label, randomised controlled trial. Lancet Neurol. 2015 Sep;14(9):883-92.
https://www.thelancet.com/journals/laneur/article/PIIS1474-4422(15)00152-0/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/26234554?tool=bestpractice.com
[77]Gonzalez-Bermejo J, Morélot-Panzini C, Tanguy ML, et al. Early diaphragm pacing in patients with amyotrophic lateral sclerosis (RespiStimALS): a randomised controlled triple-blind trial. Lancet Neurol. 2016 Nov;15(12):1217-27.
http://www.ncbi.nlm.nih.gov/pubmed/27751553?tool=bestpractice.com
Severe respiratory insufficiency not sufficiently treated with NIV
For patients who would consider long-term invasive ventilation, tracheostomy and permanent positive pressure ventilation can be life-prolonging.[65]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: drug, nutritional, and respiratory therapies (an evidence-based review). Neurology. 2009 Oct 13;73(15):1218-26. [Errata in: Neurology. 2009 Dec 15;73(24):2134; Neurology. 2010 Mar 2;74(9):781.]
https://n.neurology.org/content/73/15/1218
http://www.ncbi.nlm.nih.gov/pubmed/19822872?tool=bestpractice.com
[72]Khan A, Frazer-Green L, Amin R, et al. Respiratory management of patients with neuromuscular weakness: an American College of Chest Physicians clinical practice guideline and expert panel report. Chest. 2023 Mar 13 [Epub ahead of print].
https://journal.chestnet.org/article/S0012-3692(23)00353-7/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/36921894?tool=bestpractice.com
[78]McKim DA, Road J, Avendano M, et al; Canadian Thoracic Society Home Mechanical Ventilation Committee. Home mechanical ventilation: a Canadian Thoracic Society clinical practice guideline. Can Respir J. 2011 Jul-Aug;18(4):197-215.
https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3205101
http://www.ncbi.nlm.nih.gov/pubmed/22059178?tool=bestpractice.com
Consideration of treatment should be preceded by revisiting advance directives and a thorough discussion regarding the type of care that is needed for patients with invasive ventilation. This discussion should take place well in advance of respiratory failure.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[62]Taylor LP, Besbris JM, Graf WD, et al. Clinical guidance in neuropalliative care: an AAN position statement. Neurology. 2022 Mar 8;98(10):409-16.
https://n.neurology.org/content/98/10/409
http://www.ncbi.nlm.nih.gov/pubmed/35256519?tool=bestpractice.com
[72]Khan A, Frazer-Green L, Amin R, et al. Respiratory management of patients with neuromuscular weakness: an American College of Chest Physicians clinical practice guideline and expert panel report. Chest. 2023 Mar 13 [Epub ahead of print].
https://journal.chestnet.org/article/S0012-3692(23)00353-7/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/36921894?tool=bestpractice.com
As caring for a patient on permanent mechanical ventilation requires a high level of skill and 24-hour support for management of the ventilator and deep suctioning, most often patients cannot be supported in a home environment and placement in a chronic hospital environment is required. Less than 10% of patients with ALS consider invasive ventilation.
With permanent ventilation in place, patients may survive a variable number of years. Invasive ventilation can be withdrawn any time at the patient's request, although it is recognized that this may become a difficult decision, particularly if the patient has developed cognitive impairment or has progressed to a locked-in state, with no means left to express their wishes.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[79]Moss AH, Casey P, Stocking CB, et al. Home ventilation for amyotrophic lateral sclerosis patients: outcomes, costs, and patient, family, and physician attitudes. Neurology. 1993 Feb;43(2):438-43.
http://www.ncbi.nlm.nih.gov/pubmed/8437718?tool=bestpractice.com
For patients who would not consider long-term invasive ventilation and who have progressed to this degree of severity of respiratory dysfunction, care becomes palliative.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[62]Taylor LP, Besbris JM, Graf WD, et al. Clinical guidance in neuropalliative care: an AAN position statement. Neurology. 2022 Mar 8;98(10):409-16.
https://n.neurology.org/content/98/10/409
http://www.ncbi.nlm.nih.gov/pubmed/35256519?tool=bestpractice.com
Palliative care focuses on comfort, with special emphasis on symptomatic treatment of shortness of breath with opioids and/or benzodiazepines and consideration of concurrent support via NIV. Increased airflow in the room via an open window or fan can also be considered.[59]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: multidisciplinary care, symptom management, and cognitive/behavioral impairment (an evidence-based review). Neurology. 2009 Oct 13;73(15):1227-33.
https://n.neurology.org/content/73/15/1227
http://www.ncbi.nlm.nih.gov/pubmed/19822873?tool=bestpractice.com
[80]Borasio GD, Voltz R, Miller RG. Palliative care in amyotrophic lateral sclerosis. Neurol Clin. 2001 Nov;19(4):829-47.
http://www.ncbi.nlm.nih.gov/pubmed/11854102?tool=bestpractice.com
[81]Simon ST, Higginson IJ, Booth S, et al. Benzodiazepines for the relief of breathlessness in advanced malignant and non-malignant diseases in adults. Cochrane Database Syst Rev. 2016 Oct 20;(10):CD007354.
https://www.cochranelibrary.com/cdsr/doi/10.1002/14651858.CD007354.pub3/full
http://www.ncbi.nlm.nih.gov/pubmed/27764523?tool=bestpractice.com
Mild to moderate symptomatic dysphagia with mild weight loss (<10%)
For weight loss, the first step is diet modification. A dietitian should be involved to suggest nutritionally adequate substitutions. Diet modification with high-calorie supplements is usually effective in maintaining weight for a variable period of time, after which other options must be discussed.[82]Dorst J, Doenz J, Kandler K, et al. Fat-rich versus carbohydrate-rich nutrition in ALS: a randomised controlled study. J Neurol Neurosurg Psychiatry. 2022 Mar;93(3):298-302.
http://www.ncbi.nlm.nih.gov/pubmed/35022317?tool=bestpractice.com
For management of dysphagia, texture modification of food can be suggested by a speech-language pathologist or occupational therapist trained in dysphagia assessment. Early intervention may include avoiding dry and particulate food. As dysphagia worsens, the swallowing assessor may suggest progression to pureed food and thickening of liquids.
Moderate symptomatic dysphagia with significant weight loss (>10%)
For patients who are unable to maintain a stable weight, percutaneous endoscopic gastrostomy (PEG) or radiologically inserted gastrostomy (RIG) tube placement should be suggested. Feeding tube placement should be discussed as part of the decision-making related to long-term care. Nutrition by feeding tube allows for weight stabilization and overall might have a beneficial impact on survival.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[35]Andersen PM, Abrahams S, Borasio GD, et al. EFNS guidelines on the clinical management of amyotrophic lateral sclerosis (MALS) - revised report of an EFNS task force. Eur J Neurol. 2012 Mar;19(3):360-75.
https://onlinelibrary.wiley.com/doi/10.1111/j.1468-1331.2011.03501.x
http://www.ncbi.nlm.nih.gov/pubmed/21914052?tool=bestpractice.com
[65]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: drug, nutritional, and respiratory therapies (an evidence-based review). Neurology. 2009 Oct 13;73(15):1218-26. [Errata in: Neurology. 2009 Dec 15;73(24):2134; Neurology. 2010 Mar 2;74(9):781.]
https://n.neurology.org/content/73/15/1218
http://www.ncbi.nlm.nih.gov/pubmed/19822872?tool=bestpractice.com
[83]Castanheira A, Swash M, De Carvalho M. Percutaneous gastrostomy in amyotrophic lateral sclerosis: a review. Amyotroph Lateral Scler Frontotemporal Degener. 2022 May;23(3-4):176-89.
http://www.ncbi.nlm.nih.gov/pubmed/34196236?tool=bestpractice.com
It has an uncertain impact on patient quality of life.[65]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: drug, nutritional, and respiratory therapies (an evidence-based review). Neurology. 2009 Oct 13;73(15):1218-26. [Errata in: Neurology. 2009 Dec 15;73(24):2134; Neurology. 2010 Mar 2;74(9):781.]
https://n.neurology.org/content/73/15/1218
http://www.ncbi.nlm.nih.gov/pubmed/19822872?tool=bestpractice.com
[83]Castanheira A, Swash M, De Carvalho M. Percutaneous gastrostomy in amyotrophic lateral sclerosis: a review. Amyotroph Lateral Scler Frontotemporal Degener. 2022 May;23(3-4):176-89.
http://www.ncbi.nlm.nih.gov/pubmed/34196236?tool=bestpractice.com
[84]Katzberg, HD, Benatar M. Enteral tube feeding for amyotrophic lateral sclerosis/motor neuron disease. Cochrane Database Syst Rev. 2011 Jan 19;(1):CD004030.
https://www.cochranelibrary.com/cdsr/doi/10.1002/14651858.CD004030.pub3/full
http://www.ncbi.nlm.nih.gov/pubmed/21249659?tool=bestpractice.com
Gastrostomy surgery should be considered before FVC decreases below 50% of its predicted value, to reduce the risks of perioperative morbidity associated with respiratory dysfunction, even if the patient does not have significant dysphagia at that time.[65]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: drug, nutritional, and respiratory therapies (an evidence-based review). Neurology. 2009 Oct 13;73(15):1218-26. [Errata in: Neurology. 2009 Dec 15;73(24):2134; Neurology. 2010 Mar 2;74(9):781.]
https://n.neurology.org/content/73/15/1218
http://www.ncbi.nlm.nih.gov/pubmed/19822872?tool=bestpractice.com
[85]Chio A, Finnochiaro E, Meineri P, et al; ALS Percutaneous Endoscopic Gastrostomy Study Group. Safety and factors related to survival after percutaneous endoscopic gastrostomy in ALS. Neurology. 1999 Sep 22;53(5):1123-5.
http://www.ncbi.nlm.nih.gov/pubmed/10496278?tool=bestpractice.com
If a feeding tube is placed presymptomatically, patients are encouraged to maintain the oral intake as tolerated, while the tube is only flushed with normal saline daily to maintain patency. Even with tube feeding, aspiration precautions should be maintained.
There is insufficient evidence on whether PEG or RIG tube feeding is better in terms of safety and clinical outcomes.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[86]ProGas Study Group. Gastrostomy in patients with amyotrophic lateral sclerosis (ProGas): a prospective cohort study. Lancet Neurol. 2015 Jul;14(7):702-9.
https://www.thelancet.com/journals/laneur/article/PIIS1474-4422(15)00104-0/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/26027943?tool=bestpractice.com
RIG allows placement in patients with more advanced respiratory failure, but RIG tubes have been found to have a high rate of blocking, leaking, and needing to be replaced in the months after insertion.[86]ProGas Study Group. Gastrostomy in patients with amyotrophic lateral sclerosis (ProGas): a prospective cohort study. Lancet Neurol. 2015 Jul;14(7):702-9.
https://www.thelancet.com/journals/laneur/article/PIIS1474-4422(15)00104-0/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/26027943?tool=bestpractice.com
Dysarthria
Most (but not all) patients with ALS experience significant dysarthria progressing to anarthria. Speech therapy is usually not effective, so the most effective intervention consists of finding appropriate communication strategies to substitute for normal speech. This may include writing, as well as use of a variety of augmentative and alternative communication systems that can be activated with hand movements, facial movements, eye gaze, or whatever movement a given patient is able to perform.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[59]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: multidisciplinary care, symptom management, and cognitive/behavioral impairment (an evidence-based review). Neurology. 2009 Oct 13;73(15):1227-33.
https://n.neurology.org/content/73/15/1227
http://www.ncbi.nlm.nih.gov/pubmed/19822873?tool=bestpractice.com
[87]Pattee GL, Plowman EK, Focht Garand KL, et al. Provisional best practices guidelines for the evaluation of bulbar dysfunction in amyotrophic lateral sclerosis. Muscle Nerve. 2019 May;59(5):531-6.
http://www.ncbi.nlm.nih.gov/pubmed/30620104?tool=bestpractice.com
[88]Light J, McNaughton D, Beukelman D, et al. Challenges and opportunities in augmentative and alternative communication: research and technology development to enhance communication and participation for individuals with complex communication needs. Augment Altern Commun. 2019 Mar;35(1):1-12.
http://www.ncbi.nlm.nih.gov/pubmed/30648903?tool=bestpractice.com
As the patient's mobility is progressively changing, close monitoring and regular reassessment is necessary to enable them to maintain effective communication ability.
Sialorrhea
Patients with dysphagia often experience drooling because of inability to swallow saliva. Symptomatic treatments usually consist of anticholinergic drugs such as hyoscyamine hydrobromide, amitriptyline, atropine, or glycopyrrolate.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[72]Khan A, Frazer-Green L, Amin R, et al. Respiratory management of patients with neuromuscular weakness: an American College of Chest Physicians clinical practice guideline and expert panel report. Chest. 2023 Mar 13 [Epub ahead of print].
https://journal.chestnet.org/article/S0012-3692(23)00353-7/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/36921894?tool=bestpractice.com
For patients with refractory sialorrhea, rimabotulinumtoxinB (formerly known as botulinum toxin type B) may be considered.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[59]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: multidisciplinary care, symptom management, and cognitive/behavioral impairment (an evidence-based review). Neurology. 2009 Oct 13;73(15):1227-33.
https://n.neurology.org/content/73/15/1227
http://www.ncbi.nlm.nih.gov/pubmed/19822873?tool=bestpractice.com
[72]Khan A, Frazer-Green L, Amin R, et al. Respiratory management of patients with neuromuscular weakness: an American College of Chest Physicians clinical practice guideline and expert panel report. Chest. 2023 Mar 13 [Epub ahead of print].
https://journal.chestnet.org/article/S0012-3692(23)00353-7/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/36921894?tool=bestpractice.com
[89]James E, Ellis C, Brassington R, et al. Treatment for sialorrhea (excessive saliva) in people with motor neuron disease/amyotrophic lateral sclerosis. Cochrane Database Syst Rev. 2022 May 20;(5):CD006981.
https://www.cochranelibrary.com/cdsr/doi/10.1002/14651858.CD006981.pub3/full
http://www.ncbi.nlm.nih.gov/pubmed/35593746?tool=bestpractice.com
If injected into each parotid and submandibular gland, it can interrupt saliva release; however, localization for injections is variable and the response is also variable. When effective, repeat injections are usually required at approximately 3-month intervals. Maximum benefit is usually experienced at 4 weeks.[90]Stone CA, O'Leary N. Systematic review of the effectiveness of botulinum toxin or radiotherapy for sialorrhea in patients with amyotrophic lateral sclerosis. J Pain Symptom Manage. 2009 Feb;37(2):246-58.
https://www.jpsmjournal.com/article/S0885-3924(08)00301-1/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/18676117?tool=bestpractice.com
Low-dose radiation therapy to the salivary glands is also possibly effective in patients with medically refractory sialorrhea, although evidence is uncertain.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[59]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: multidisciplinary care, symptom management, and cognitive/behavioral impairment (an evidence-based review). Neurology. 2009 Oct 13;73(15):1227-33.
https://n.neurology.org/content/73/15/1227
http://www.ncbi.nlm.nih.gov/pubmed/19822873?tool=bestpractice.com
[72]Khan A, Frazer-Green L, Amin R, et al. Respiratory management of patients with neuromuscular weakness: an American College of Chest Physicians clinical practice guideline and expert panel report. Chest. 2023 Mar 13 [Epub ahead of print].
https://journal.chestnet.org/article/S0012-3692(23)00353-7/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/36921894?tool=bestpractice.com
[89]James E, Ellis C, Brassington R, et al. Treatment for sialorrhea (excessive saliva) in people with motor neuron disease/amyotrophic lateral sclerosis. Cochrane Database Syst Rev. 2022 May 20;(5):CD006981.
https://www.cochranelibrary.com/cdsr/doi/10.1002/14651858.CD006981.pub3/full
http://www.ncbi.nlm.nih.gov/pubmed/35593746?tool=bestpractice.com
It may decrease saliva secretion for up to 6 months.[59]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: multidisciplinary care, symptom management, and cognitive/behavioral impairment (an evidence-based review). Neurology. 2009 Oct 13;73(15):1227-33.
https://n.neurology.org/content/73/15/1227
http://www.ncbi.nlm.nih.gov/pubmed/19822873?tool=bestpractice.com
[90]Stone CA, O'Leary N. Systematic review of the effectiveness of botulinum toxin or radiotherapy for sialorrhea in patients with amyotrophic lateral sclerosis. J Pain Symptom Manage. 2009 Feb;37(2):246-58.
https://www.jpsmjournal.com/article/S0885-3924(08)00301-1/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/18676117?tool=bestpractice.com
[91]Tysnes OB. Treatment of sialorrhea in amyotrophic lateral sclerosis. Acta Neurol Scand Suppl. 2008 May;117(s188):77-81.
http://www.ncbi.nlm.nih.gov/pubmed/18439227?tool=bestpractice.com
Difficulty with mucus secretions
Reduced cough volume and strength may result in inability to expel pulmonary secretions. Mechanical cough assist devices may be very helpful, particularly if peak cough flow is reduced.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[65]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: drug, nutritional, and respiratory therapies (an evidence-based review). Neurology. 2009 Oct 13;73(15):1218-26. [Errata in: Neurology. 2009 Dec 15;73(24):2134; Neurology. 2010 Mar 2;74(9):781.]
https://n.neurology.org/content/73/15/1218
http://www.ncbi.nlm.nih.gov/pubmed/19822872?tool=bestpractice.com
[72]Khan A, Frazer-Green L, Amin R, et al. Respiratory management of patients with neuromuscular weakness: an American College of Chest Physicians clinical practice guideline and expert panel report. Chest. 2023 Mar 13 [Epub ahead of print].
https://journal.chestnet.org/article/S0012-3692(23)00353-7/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/36921894?tool=bestpractice.com
Carbocysteine is a mucolytic that can break down mucus, making it easier to clear, but it is not available in the US.[92]McGeachan AJ, Mcdermott CJ. Management of oral secretions in neurological disease. Pract Neurol. 2017 Apr;17(2):96-103.
https://pn.bmj.com/content/17/2/96
http://www.ncbi.nlm.nih.gov/pubmed/28188210?tool=bestpractice.com
Other mucolytics such as nebulized acetylcysteine or oral guaifenesin can be tried.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
Ensuring sufficient hydration and reduction in anticholinergics can potentially help with management of thick secretions. Occasionally, patients may elect to undergo tracheostomy to allow for more complete suctioning of airways, but this is uncommon.
Pseudobulbar affect
Occasionally patients with significant bulbar upper motor neuron signs will also display pseudobulbar affect, manifested by excessive or inappropriate laughing or crying. Most often, explanation of the etiology of these symptoms and reassurance are sufficient to allow patients to deal with this symptom.
If treatment is needed, the combination of dextromethorphan and quinidine has been found to be efficacious in controlling the pseudobulbar affect and is well tolerated.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[59]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: multidisciplinary care, symptom management, and cognitive/behavioral impairment (an evidence-based review). Neurology. 2009 Oct 13;73(15):1227-33.
https://n.neurology.org/content/73/15/1227
http://www.ncbi.nlm.nih.gov/pubmed/19822873?tool=bestpractice.com
[93]Pioro EP, Brooks BR, Cummings J, et al. Dextromethorphan plus ultra low-dose quinidine reduces pseudobulbar affect. Ann Neurol. 2010;68:693-702.
http://www.ncbi.nlm.nih.gov/pubmed/20839238?tool=bestpractice.com
Alternatively, amitriptyline may be considered for treating sleep problems and pseudobulbar affect, or a selective serotonin-reuptake inhibitor (SSRI) may be used to treat depression and pseudobulbar affect.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
Physical therapy for muscle weakness
Physical therapy is for maintenance of muscle elasticity and joint range of motion, to prevent contractures, and to determine the level of appropriate bracing and the need for ambulation devices. Such devices include ankle foot orthoses, cervical collars (neck weakness), canes, walkers, and wheelchairs. In each case, a range of devices is available, and the choice must be made by an experienced professional, taking into account the patient's needs and preferences.[61]Lazovic M, Nikolic D, Boyer FC, et al. Evidence-based position paper on Physical and Rehabilitation Medicine practice for people with amyotrophic lateral sclerosis. Eur J Phys Rehabil Med. 2022 Apr;58(2):271-9.
https://www.minervamedica.it/en/journals/europa-medicophysica/article.php?cod=R33Y2022N02A0271
http://www.ncbi.nlm.nih.gov/pubmed/34786907?tool=bestpractice.com
Functional decline is an inevitable feature of ALS, so serial evaluations by physical therapists will be necessary throughout the course of the illness.
Individualized exercise programs of moderate intensity are considered safe and beneficial for patients with early disease.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[94]Lui AJ, Byl NN. A systematic review of the effect of moderate intensity exercise on function and disease progression in amyotrophic lateral sclerosis. J Neurol Physical Therapy. 2009 Jun;33(2):68-87.
https://journals.lww.com/jnpt/Fulltext/2009/06000/A_Systematic_Review_of_the_Effect_of_Moderate.2.aspx
http://www.ncbi.nlm.nih.gov/pubmed/19556916?tool=bestpractice.com
[95]Dal Bello-Haas V, Florence JM. Therapeutic exercise for people with amyotrophic lateral sclerosis or motor neuron disease. Cochrane Database Syst Rev. 2013 May 31;(5):CD005229.
https://www.cochranelibrary.com/cdsr/doi/10.1002/14651858.CD005229.pub3/full
http://www.ncbi.nlm.nih.gov/pubmed/23728653?tool=bestpractice.com
These should be home-based and monitored by physical therapists, using telemedicine approaches if necessary.[61]Lazovic M, Nikolic D, Boyer FC, et al. Evidence-based position paper on Physical and Rehabilitation Medicine practice for people with amyotrophic lateral sclerosis. Eur J Phys Rehabil Med. 2022 Apr;58(2):271-9.
https://www.minervamedica.it/en/journals/europa-medicophysica/article.php?cod=R33Y2022N02A0271
http://www.ncbi.nlm.nih.gov/pubmed/34786907?tool=bestpractice.com
Occupational therapy for muscle weakness
The goal is to assess the needs of a patient in order to support their independence in performing activities of daily living and to assist with the care for patients with advanced disease. Examples include devices to assist with transfer from and to a wheelchair (e.g., bed rails, Hoyer-lift) and adapting the environment (e.g., ramps, shower chair, commode, powered hospital-type bed, modifying keyboards, adaptive utensils, accessible clothing).
Serial evaluations by occupational therapists will be required as the disease progresses, in order to define the patient's needs at different points in time.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[36]National Institute for Health and Care Excellence. Motor neurone disease: assessment and management. Jul 2019 [internet publication].
https://www.nice.org.uk/guidance/ng42
Therapy for fully paralyzed patients
Treatment goals relate to maintaining skin integrity and comfort. Motorized beds and water and air mattresses can reduce pressure and vary sites of skin contact, preventing pressure ulcers and aiding in providing adequate pain control.[96]Newrick PG, Langton-Hewer R. Pain in motor neuron disease. J Neurol Neurosurg Psychiatry. 1985 Aug;48(8):838-40.
https://jnnp.bmj.com/content/jnnp/48/8/838.full.pdf
http://www.ncbi.nlm.nih.gov/pubmed/4031936?tool=bestpractice.com
Muscle spasms and spasticity
Muscle spasms are usually a reflection of spasticity, and are treated with exercises and antispasticity medication. Medications that may be considered include baclofen, tizanidine, botulinum toxin, benzodiazepines, and cannabinoids.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[59]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: multidisciplinary care, symptom management, and cognitive/behavioral impairment (an evidence-based review). Neurology. 2009 Oct 13;73(15):1227-33.
https://n.neurology.org/content/73/15/1227
http://www.ncbi.nlm.nih.gov/pubmed/19822873?tool=bestpractice.com
[97]Marvulli R, Megna M, Citraro A, et al. Botulinum toxin type A and physiotherapy in spasticity of the lower limbs due to amyotrophic lateral sclerosis. Toxins (Basel). 2019 Jul 1;11(7):381.
https://www.mdpi.com/2072-6651/11/7/381
http://www.ncbi.nlm.nih.gov/pubmed/31266172?tool=bestpractice.com
[98]Meyer T, Funke A, Münch C, et al. Real world experience of patients with amyotrophic lateral sclerosis (ALS) in the treatment of spasticity using tetrahydrocannabinol:cannabidiol (THC:CBD). BMC Neurol. 2019 Sep 7;19(1):222.
https://bmcneurol.biomedcentral.com/articles/10.1186/s12883-019-1443-y
http://www.ncbi.nlm.nih.gov/pubmed/31493784?tool=bestpractice.com
Antispasticity medications can sometimes worsen ambulation or transfer ability due to the emergence of underlying muscle weakness when limb tone is reduced, and benzodiazepines bring a risk of respiratory suppression.
Physical therapy for spasticity aims to reduce muscle tone, maintain range of motion and mobility, and improve comfort. Modalities include strengthening, stretching, and positioning exercises. Individualized exercise programs of moderate intensity are considered safe and beneficial for patients with early disease.[94]Lui AJ, Byl NN. A systematic review of the effect of moderate intensity exercise on function and disease progression in amyotrophic lateral sclerosis. J Neurol Physical Therapy. 2009 Jun;33(2):68-87.
https://journals.lww.com/jnpt/Fulltext/2009/06000/A_Systematic_Review_of_the_Effect_of_Moderate.2.aspx
http://www.ncbi.nlm.nih.gov/pubmed/19556916?tool=bestpractice.com
[95]Dal Bello-Haas V, Florence JM. Therapeutic exercise for people with amyotrophic lateral sclerosis or motor neuron disease. Cochrane Database Syst Rev. 2013 May 31;(5):CD005229.
https://www.cochranelibrary.com/cdsr/doi/10.1002/14651858.CD005229.pub3/full
http://www.ncbi.nlm.nih.gov/pubmed/23728653?tool=bestpractice.com
Therapy to actively or passively perform range-of-motion movements will reduce tone as effectively as pharmacologic therapy, but is time-consuming and often requires family members or therapists to provide passive movements.
Depression and anxiety
Symptoms of depression and anxiety should be sought at every patient visit. The incidence of depression in patients with ALS is not well studied, but may be higher than in the general population, especially in those with advanced disease.[99]Rabkin JG, Albert SM, Del Bene ML, et al. Prevalence of depressive disorders and change over time in late-stage ALS. Neurology. 2005 Jul 12;65(1):62-7.
https://www.ncbi.nlm.nih.gov/pmc/articles/PMC1201558
http://www.ncbi.nlm.nih.gov/pubmed/16009886?tool=bestpractice.com
Depression is often well treated pharmacologically with an SSRI or serotonin-norepinephrine reuptake inhibitor (SNRI).[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[59]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: multidisciplinary care, symptom management, and cognitive/behavioral impairment (an evidence-based review). Neurology. 2009 Oct 13;73(15):1227-33.
https://n.neurology.org/content/73/15/1227
http://www.ncbi.nlm.nih.gov/pubmed/19822873?tool=bestpractice.com
Because most patients with ALS eventually develop dysphagia, it is appropriate to consider initiating treatment with an antidepressant that can be safely crushed (by avoiding capsules or coated medications).
Evidence for psychological interventions for patients with ALS is limited, but there is some evidence of effectiveness for mindfulness and cognitive behavioral therapy for treating depression and anxiety.[100]Zarotti N, Mayberry E, Ovaska-Stafford N, et al. Psychological interventions for people with motor neuron disease: a scoping review. Amyotroph Lateral Scler Frontotemporal Degener. 2021 Feb;22(1-2):1-11.
http://www.ncbi.nlm.nih.gov/pubmed/32657152?tool=bestpractice.com
[101]British Psychological Society. Psychological interventions for people with Huntington’s disease, Parkinson’s disease, motor neurone disease, and multiple sclerosis. Jan 2021 [internet publication].
https://www.bps.org.uk/guideline/psychological-interventions-people-huntingtons-disease-parkinsons-disease-motor-neurone
Insomnia
If insomnia is a problem, it is important to determine the cause so that appropriate treatment can be started. If insomnia is related to respiratory insufficiency, nocturnal NIV may be an effective treatment. Other possible causes of insomnia include mood disorders and pain.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
Treatment of insomnia with benzodiazepines should generally be avoided in patients with ALS due to the risk of respiratory suppression. Non-benzodiazepine sleep medication such as melatonin or zopiclone can be considered.
Cognitive and behavioral impairment
Up to 45% of patients with ALS have associated cognitive and behavioral impairment, and in approximately 14% of patients these deficits are severe enough to meet criteria for frontotemporal dementia.[102]Pender N, Pinto-Grau M, Hardiman O. Cognitive and behavioural impairment in amyotrophic lateral sclerosis. Curr Opin Neurol. 2020 Oct;33(5):649-54.
http://www.ncbi.nlm.nih.gov/pubmed/32833751?tool=bestpractice.com
Symptoms may precede the onset of motor neuron disease symptoms, or may become evident late in the disease course.[4]Neary D, Snowden JS, Gustafson L, et al. Frontotemporal lobar degeneration: a consensus on clinical diagnostic criteria. Neurology. 1998 Dec;51(6):1546-54.
http://www.ncbi.nlm.nih.gov/pubmed/9855500?tool=bestpractice.com
[103]Cairns NJ, Bigio EH, Mackenzie IR, et al; Consortium for Frontotemporal Lobar Degeneration. Neuropathologic diagnostic and nosologic criteria for frontotemporal lobar degeneration: consensus of the Consortium for Frontotemporal Lobar Degeneration. Acta Neuropathol. 2007 Jul;114(1):5-22.
https://link.springer.com/article/10.1007/s00401-007-0237-2
http://www.ncbi.nlm.nih.gov/pubmed/17579875?tool=bestpractice.com
Screening for behavioral, cognitive, and language dysfunction should be carried out for patients with ALS.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[59]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: multidisciplinary care, symptom management, and cognitive/behavioral impairment (an evidence-based review). Neurology. 2009 Oct 13;73(15):1227-33.
https://n.neurology.org/content/73/15/1227
http://www.ncbi.nlm.nih.gov/pubmed/19822873?tool=bestpractice.com
Interventions are generally supportive, as there is no evidence for effectiveness of pharmacologic therapies.
Palliative care
ALS does not have a cure, and weakness and functional deficits will progress throughout the disease course. Patients living with ALS should be supported using the principles of palliative care and, in particular, the use of a holistic approach to support patients and their families throughout the course of their illness. Key components of palliative care in ALS are goals-of-care discussions, advance directive planning, symptom management, and end-of-life support.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[62]Taylor LP, Besbris JM, Graf WD, et al. Clinical guidance in neuropalliative care: an AAN position statement. Neurology. 2022 Mar 8;98(10):409-16.
https://n.neurology.org/content/98/10/409
http://www.ncbi.nlm.nih.gov/pubmed/35256519?tool=bestpractice.com
There are several models of palliative care that can be followed, including integration of palliative care into the multidisciplinary ALS clinic, separate involvement of a palliative care specialty team, home-based palliative care, telemedicine-supported care, and hospice care.
Advance directives and wishes for end-of-life care should be discussed with the patient and family/caregivers as early as possible, long before hospice care is needed, and should be an ongoing conversation.[62]Taylor LP, Besbris JM, Graf WD, et al. Clinical guidance in neuropalliative care: an AAN position statement. Neurology. 2022 Mar 8;98(10):409-16.
https://n.neurology.org/content/98/10/409
http://www.ncbi.nlm.nih.gov/pubmed/35256519?tool=bestpractice.com
[104]Flemming K, Turner V, Bolsher S, et al. The experiences of, and need for, palliative care for people with motor neurone disease and their informal caregivers: a qualitative systematic review. Palliat Med. 2020 Jun;34(6):708-30.
https://journals.sagepub.com/doi/10.1177/0269216320908775
http://www.ncbi.nlm.nih.gov/pubmed/32286157?tool=bestpractice.com
Revisiting advance directives should precede discussions about starting hospice care and end-of-life management.[11]Shoesmith C, Abrahao A, Benstead T, et al. Canadian best practice recommendations for the management of amyotrophic lateral sclerosis. CMAJ. 2020 Nov 16;192(46):E1453-68.
https://www.cmaj.ca/content/192/46/E1453
http://www.ncbi.nlm.nih.gov/pubmed/33199452?tool=bestpractice.com
[59]Miller RG, Jackson CE, Kasarskis EJ, et al; Quality Standards Subcommittee of the American Academy of Neurology. Practice parameter update: the care of the patient with amyotrophic lateral sclerosis: multidisciplinary care, symptom management, and cognitive/behavioral impairment (an evidence-based review). Neurology. 2009 Oct 13;73(15):1227-33.
https://n.neurology.org/content/73/15/1227
http://www.ncbi.nlm.nih.gov/pubmed/19822873?tool=bestpractice.com
[80]Borasio GD, Voltz R, Miller RG. Palliative care in amyotrophic lateral sclerosis. Neurol Clin. 2001 Nov;19(4):829-47.
http://www.ncbi.nlm.nih.gov/pubmed/11854102?tool=bestpractice.com
End-of-life management should focus on the patient's comfort and dignity.