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Multiple system atrophy

Multiple system atrophy (MSA) is a rare neurodegenerative disorder characterized by tremors, slow movement, muscle rigidity, postural instability, autonomic dysfunction and ataxia. This is caused by progressive degeneration of neurons in several parts of the brain including the basal ganglia, inferior olivary nucleus, and cerebellum. MSA was first described in 1960 by Milton Shy and Glen Drager and is better known as Shy–Drager syndrome.

Signs and symptoms
MSA is characterized by the following: Autonomic and at least one Motor (clinically established MSA criteria 2022) • autonomic dysfunction: Post-void urinary residual volume ≥100 mL (usually by ultrasound); Unexplained urinary urge incontinence; or Neurogenic orthostatic hypotension (≥20/10 mmHg blood pressure drop) within 3 minutes (usually by head‐up tilt) • parkinsonism (muscle rigidity +/ tremor and slow movement: MSA-P) • cerebellar ataxia (Poor coordination/unsteady walking: MSA-C) A variant with combined features of MSA and dementia with Lewy bodies may also exist. There have also been occasional instances of frontotemporal lobar degeneration associated with MSA. Initial presentation The most common first sign of MSA is the appearance of an "akinetic-rigid syndrome" (i.e. slowness of initiation of movement resembling Parkinson's disease) found in 62% at first presentation. Other common signs at onset include problems with balance (cerebellar ataxia) found in 22% at first presentation, followed by genito-urinary symptoms (9%): both men and women often experience urgency, frequency, incomplete bladder emptying, or an inability to pass urine (retention). About 1 in 5 MSA patients experience a fall in their first year of disease. For men, the first sign can be erectile dysfunction. Women have also reported reduced genital sensitivity. Progression As the disease progresses, one of three groups of symptoms predominates. These are: • Parkinsonism – slow, stiff movement, writing becomes small and spidery • Cerebellar dysfunction – difficulty coordinating movement and balance • Autonomic nervous system dysfunction – impaired automatic body functions, including one, some, or all of the following: ::* postural or orthostatic hypotension, resulting in dizziness or fainting upon standing up ::* urinary incontinence or urinary retention ::* impotence ::* constipation ::* vocal cord paralysis ::* dry mouth and skin ::* trouble regulating body temperature due to sweating deficiency in all parts of the body ::* loud snoring, abnormal breathing or inspiratory stridor during sleep ::* other sleep disorders including sleep apnea, REM behavior disorder ::* double vision ::*muscle twitches ==Genetics==
Genetics
One study found a correlation between the deletion of genes in a specific genetic region and the development of MSA in a group of Japanese patients. The region in question includes the SHC2 gene which, in mice and rats, appears to have some function in the nervous system. The authors of this study hypothesized that there may be a link between the deletion of the SHC2 and the development of MSA. A follow-up study was unable to replicate this finding in American MSA patients. The authors of the study concluded that "Our results indicate that SHC2 gene deletions underlie few, if any, cases of well-characterized MSA in the US population. This is in contrast to the Japanese experience reported by Sasaki et al., likely reflecting heterogeneity of the disease in different genetic backgrounds." Another study investigated the frequency of RFC1 intronic repeat expansions, a phenomenon implicated in CANVAS; a disease with a diagnostic overlap with MSA. The study concluded that these repeats were absent in pathologically confirmed MSA, suggesting an alternative genetic cause. ==Pathophysiology==
Pathophysiology
The defining pathologic feature of multiple system atrophy is the presence of inclusion bodies (known as glial cytoplasmic inclusions or Papp-Lantos bodies) consisting of alpha-synuclein in oligodendrocytes. In addition, neurons are lost in several regions of the nervous system, particularly in the basal ganglia, inferior olivary nuclei, cerebellum, pons, and spinal cord. Reactive astrocytes and microglia are prominent in damaged areas of the central nervous system, especially in regions with abundant oligodendroglial inclusions. and a variety of other potential genetic and environmental risk factors have been proposed. The origin of the alpha-synuclein that forms inclusions in oligodendrocytes is uncertain. Compared to neurons, oligodendrocytes produce little or no alpha-synuclein themselves, suggesting that these cells take up the protein that is generated by neurons. For example, it has been proposed that the α-synuclein inclusions found in oligodendrocytes result from the pruning and engulfment of diseased axonal segments containing aggregated α-synuclein, i.e., of Lewy neurites. Research has shown that the strain of the protein generated by oligodendrocytes causes a more aggressive type of disease than does the Lewy body strain. ==Diagnosis==
Diagnosis
Clinical Clinical diagnostic criteria were defined in 1998 and updated in 2007 Certain signs and symptoms of MSA also occur with other disorders, such as Parkinson's disease, making the diagnosis more difficult. Features characteristic of OPCA include progressive cerebellar ataxia, leading to clumsiness in body movements, veering from midline when walking, wide-based stance, and falls without signs of paralysis or weakness. Clinical presentation can vary greatly between patients, but mostly affects speech, balance and walking. Other possible neurological problems include spasmodic dysphonia, hypertonia, hyperreflexia, rigidity, dysarthria, dysphagia and neck dystonic posture. Diagnosis may be based on a thorough medical exam; the presence of signs and symptoms; imaging studies; various laboratory tests; and an evaluation of the family history. Radiologic Both MRI and CT scanning may show a decrease in the size of the cerebellum and pons in those with cerebellar features (MSA-C). The putamen is hypointense on T2-weighted MRI and may show an increased deposition of iron in the Parkinsonian (MSA-P) form. In MSA-C, a "hot cross bun" sign is sometimes found; it reflects atrophy of the pontocerebellar tracts that give T2 hyper intense signal intensity in the atrophic pons. MRI changes are not required to diagnose the disease as these features are often absent, especially early in the course of the disease. Additionally, the changes can be quite subtle and are usually missed by examiners who are not experienced with MSA. Pathologic Pathological diagnosis can only be made at autopsy by finding abundant glial cytoplasmic inclusions (GCIs) on histological specimens of the central nervous system. Olivopontocerebellar atrophy can be used as a pathological term to describe degeneration of neurons in specific areas of the brain – the cerebellum, pons, and inferior olivary nucleus. OPCA is present in several neurodegenerative syndromes, including inherited and non-inherited forms of ataxia (such as the hereditary spinocerebellar ataxia known as Machado–Joseph disease) and MSA, with which it is primarily associated. MSA presents with extensive pathological α-synuclein inclusions in the cytosol of oligodendrocytes (glial cytoplasmic inclusions), with limited pathology in neurons. rather than the brainstem, limbic and cortical regions typically effected in Lewy inclusion diseases. One group revealed robust α-synuclein pathology in the pontine nuclei and medullary inferior olivary nucleus upon histological analysis of neurological tissue from MSA patients. In 2020, researchers at The University of Texas Health Science Center at Houston concluded that protein misfolding cyclic amplification could be used to distinguish between two progressive neurodegenerative diseases, Parkinson's disease and multiple system atrophy, being the first process to give an objective diagnosis of Multiple System Atrophy instead of just a differential diagnosis. Classification MSA is one of several neurodegenerative diseases known as synucleinopathies: they have in common an abnormal accumulation of alpha-synuclein protein in various parts of the brain. Other synucleinopathies include Parkinson's disease, the Lewy body dementias, and other more rare conditions. Old terminology Historically, many terms were used to refer to this disorder, based on the predominant systems presented. These terms were discontinued by consensus in 1996 and replaced with MSA and its subtypes, but awareness of these older terms and their definitions is helpful to understanding the relevant literature prior to 1996. These include striatonigral degeneration (SND), olivopontocerebellar atrophy (OPCA), and Shy–Drager syndrome. A table describing the characteristics and modern names of these conditions follows: The term olivopontocerebellar atrophy was originally coined by Joseph Jules Dejerine and André Thomas. It was subdivided as: Non-hereditary diseases formerly categorized as olivopontocerebellar atrophy have were reclassified as forms of MSA as well as to four hereditary types, that have been currently reclassified as four different forms of spinocerebellar ataxia: Current terminology The current terminology and diagnostic criteria for the disease were established at a 2007 conference of experts and set forth in a position paper. This Second Consensus Statement defines two categories of MSA, based on the predominant symptoms of the disease at the time of evaluation. These are: • MSA with predominant parkinsonism (MSA-P) – defined as MSA where extrapyramidal features predominate. It is sometimes termed striatonigral degeneration, a parkinsonian variant. • MSA with cerebellar features (MSA-C) – defined as MSA in which cerebellar ataxia predominates. It is sometimes termed sporadic olivopontocerebellar atrophy. ==Management==
Management
Supervision Ongoing care from a neurologist specializing in movement disorders is recommended, because the complex symptoms of MSA are often not familiar to less-specialized neurologists. Hospice/homecare services can be very useful as disability progresses. Drug therapy Levodopa (L-Dopa), a drug used in the treatment of Parkinson's disease, improves parkinsonian symptoms in a small percentage of MSA patients. A recent trial reported that only 1.5% of MSA patients experienced any improvement at all when taking levodopa, their improvement was less than 50%, and even that improvement was a transient effect lasting less than one year. Poor response to L-Dopa has been suggested as a possible element in the differential diagnosis of MSA from Parkinson's disease. The drug riluzole is ineffective in treating MSA or PSP. Instructing patients in gait training will help to improve their mobility and decrease their risk of falls. A physiotherapist may also prescribe mobility aids such as a cane or a walker to increase the patient's safety. Another common drug treatment is the alpha-agonist midodrine. Non-drug treatments include "head-up tilt" (elevating the head of the whole bed by about 10 degrees), salt tablets or increasing salt in the diet, generous intake of fluids, and pressure (elastic) stockings. Avoidance of triggers of low blood pressure, such as hot weather, alcohol, and dehydration, are crucial. Support Social workers and occupational therapists can also help with coping with disability through the provision of equipment and home adaptations, services for caregivers and access to healthcare services, both for the person with MSA as well as family caregivers. ==Prognosis==
Prognosis
The average lifespan after the onset of symptoms in patients with MSA is 6–10 years. ==Epidemiology==
Epidemiology
Multiple system atrophy is estimated to affect approximately 5 per 100,000 people. At autopsy, many patients diagnosed during life with Parkinson's disease are found actually to have MSA, suggesting that the actual incidence of MSA is higher than that estimate. While some suggest that MSA affects slightly more men than women (1.3:1), others suggest that the two sexes are equally likely to be affected. The condition most commonly presents in persons aged 50–60. ==Research==
Research
Mesenchymal stem cell therapy may delay the progression of neurological deficits in patients with MSA-cerebellar type. == Notable cases ==
Notable cases
Nikolai Andrianov was a Soviet/Russian gymnast who held the record for men for the most Olympic medals at 15 (7 gold medals, 5 silver medals, 3 bronze medals) until Michael Phelps surpassed him at the 2008 Beijing Summer Olympics. • Todd J. Campbell (1956–2021), United States district judge and counsel to former Vice President Al Gore. • Singer and songwriter Johnny Cash wrote in his autobiography that he was diagnosed with Shy–Drager in 1997. • Ronald Green (1944–2012), American-Israeli basketball player • Joseph C. Howard Sr. (1922–2000) was the first African American to serve as a United States district judge of the United States District Court for the District of Maryland. • Kenneth More British actor, originally diagnosed with Parkinson's disease. • Chef Kerry Simon died from complications of MSA. • David Colin Sherrington FRS (1945–2014), noted polymer chemist, who was diagnosed in 2012 and died from pneumonia two years later. • Karsten Heuer (1968–2024) Canadian Biologist, Conservationist, Filmmaker and Author. ==See also==
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