Amelioration of subacute camptocormia in multiple system atrophy by protirelin tartrate

Takei, Asako; Hamada, Shinsuke; Homma, Sanae; Hamada, Keiko; Tashiro, Kunio; Hamada, Takeshi

doi:10.1002/mds.22699

Cited by 5 publications

(6 citation statements)

References 6 publications

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“…Gabapentin (off‐label) may have symptomatic benefits on ataxia, oscillopsia, and dysarthria according to a case report in 2 MSA‐C patients . Finally, protirelin tartrate (off‐label) has been used for ataxic diseases in Japan, but without any apparent effect in MSA . In concordance with these results, the French cohort of MSA‐C patients consumed vitamin E and buspirone more frequently than MSA‐P or PD patients .…”

Section: Symptomatic Therapiessupporting

confidence: 57%

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Current Concepts in the Treatment of Multiple System Atrophy

Pérez-Lloret

Flabeau

Fernagut

et al. 2015

Movement Disord Clin Pract

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MSA is a progressive neurodegenerative disorder characterized by autonomic failure and a variable combination of poor levodopa‐responsive parkinsonism and cerebellar ataxia (CA). Current therapeutic management is based on symptomatic treatment. Almost one third of MSA patients may benefit from l‐dopa for the symptomatic treatment of parkinsonism, whereas physiotherapy remains the best therapeutic option for CA. Only midodrine and droxidopa were found to be efficient for neurogenic hypotension in double‐blind, controlled studies, whereas other symptoms of autonomic failure may be managed with off‐label treatments. To date, no curative treatment is available for MSA. Recent results of neuroprotective and ‐restorative trials have provided some hope for future advances. Considerations for future clinical trials are also discussed in this review.

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Section: Symptomatic Therapiessupporting

confidence: 57%

“…Whether CC is a dystonic posture, a consequence of axial rigidity, or a focal myopathy is still debated . Protirelin tartrate improved CC dramatically in one case report, presumably by enhancing motoneuronal excitability of the paraspinal muscles . As in focal dystonia, BoNT injections may be effective .…”

Section: Symptomatic Therapiesmentioning

confidence: 99%

Current Concepts in the Treatment of Multiple System Atrophy

Pérez-Lloret

Flabeau

Fernagut

et al. 2015

Movement Disord Clin Pract

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“…Levodopa ( l ‐dopa) is rarely effective and is thought to sometimes induce camptocormia, perhaps as a drug‐induced truncal dystonia. Numerous other treatment options have been tested, including psychotherapy, orthotic use in combination with physical therapy, intrathecal baclofen, corticosteroids, protirelin tartrate, botulinum toxin, immunoglobulins, deep‐brain stimulation (DBS), and orthopedic surgery, but these approaches are not predictably effective . The lack of randomized‐controlled trials and the multifactorial etiology of camptocormia complicates treatment for this condition.…”

Section: Postural Disorders (Spine)mentioning

confidence: 99%

Postural, Bone, and Joint Disorders in Parkinson's Disease

Rabin

Earnhardt

Patel

et al. 2016

Movement Disord Clin Pract

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Background Stooped posture was mentioned in the original description of the characteristic features of Parkinson's disease (PD). Since then, a variety of postural, bone, and joint problems have become recognized as common aspects of the illness and deserve attention. Methods A Medline literature search for the period from 1970 to 2016 was performed to identify articles relevant to this topic. Keywords for the search included posture, spine, bone disorders, fractures, joint disorders, kyphosis, scoliosis, stooping, camptocormia, Pisa syndrome, frozen shoulder, anterocollis, dropped head syndrome, and pain in combination with PD. The articles were then reviewed to summarize clinical features, frequency, impact, pathophysiology, and treatment options for these conditions. Results Postural disorders (kyphoscoliosis, camptocormia, Pisa syndrome, dropped head syndrome), bone mineralization disorders (osteoporosis, bone fractures), and joint disorders (frozen shoulder, dystonia involving joints, joint pain) are often seen in association with PD. Treatment options for these conditions are varied and may include medications, physical therapy, or surgical interventions. Conclusions Posture, bone, and joint disorders are common in patients with PD; they often produce added disability, and they may be treatable.

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“…Arguments for a PNS origin of camptocormia in Parkinson’s disease are that axial muscles may show myopathic features on EMG or muscle biopsy [1] and that MRI of the axial muscles may show atrophy and fatty replacement of the thoracolumbar muscles [33]. CNS disorders associated with camptocormia in addition to Parkinson’s disease include MSA [2,34,35], Alzheimer’s disease [36], basal ganglia disorders [14], and abdominal segmental dystonia [37]. Because of the frequent association of camptocormia with Parkinson’s disease [3,19] or dystonias [28], some authors regard camptocormia even a segmental form of dystonia [14,38].…”

Section: Etiologymentioning

confidence: 99%

Presentation, Etiology, Diagnosis, and Management of Camptocormia

Finsterer

Strobl

2010

Eur Neurol

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Camptocormia (bent spine syndrome, cyphose hystérique) is an abnormality characterized by severe forward flexion of the thoracolumbar spine which typically increases during walking or standing and completely disappears in supine position. Camptocormia can be due to central nervous system diseases, such as Parkinson’s disease, dystonia, multisystem atrophy, or Alzheimer’s disease, due to peripheral nervous system diseases, such as primary myopathy, secondary myopathy, motor neuron disease, myasthenia, or chronic inflammatory demyelinating polyneuropathy, due to side effects of drug treatment, due to disc herniation, arthritis or spinal trauma, or due to paraneoplasia. Only rarely may camptocormia be attributable to psychiatric disease. The diagnosis is based on clinical findings, imaging of the cerebrum or spine, needle electromyography, or muscle biopsy. Treatment options are limited and frequently futile and rely on conservative measures, such as psychotherapy, physiotherapy, use of orthoses, drugs, injection of botulinum toxin, withdrawal of causative drugs, electroconvulsive therapy, or invasive measures, such as surgical correction or deep brain stimulation. The outcome is generally fair. Some patients profit from therapy whereas others do not respond to treatment and become progressively immobile.

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Amelioration of subacute camptocormia in multiple system atrophy by protirelin tartrate

Cited by 5 publications

References 6 publications

Current Concepts in the Treatment of Multiple System Atrophy

Current Concepts in the Treatment of Multiple System Atrophy

Postural, Bone, and Joint Disorders in Parkinson's Disease

Presentation, Etiology, Diagnosis, and Management of Camptocormia

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