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Stiff Person Syndrome

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Serum osteoprotegerin level is positively associated with peripheral artery disease in patients with peritoneal dialysis.

Wei-Chen Lin, Jen-Pi Tsai, Yu-Hsien Lai, Yu-Li Lin, Chiu-Huang Kuo, Chih-Hsien Wang, Bang-Gee Hsu,

Osteoprotegerin (OPG) is a potential biomarker of cardiovascular disease complications and severity. Peripheral arterial disease (PAD) is associated with an increased risk of death in patients on peritoneal dialysis (PD). Therefore, this study aimed to evaluate the relationship between serum OPG levels and PAD by measuring the ankle-brachial index (ABI) ... Read more >>

Ren Fail (Renal Failure)
[2020, 42(1):131-136]

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Cerebellar Ataxia Followed by Stiff Person Syndrome in a Patient with Anti-GAD Antibodies.

Sinali O Seneviratne, Katherine A Buzzard, Belinda Cruse, Mastura Monif,

Anti-GAD antibody syndrome is a result of the production of antibodies against glutamic acid decarboxylase (GAD), the main enzyme responsible for the production of gamma-aminobutyric acid (GABA). Several neurological manifestations including cerebellar ataxia and stiff person syndrome have been reported in association with anti-GAD antibodies. In this paper, we present ... Read more >>

Case Reports Immunol (Case reports in immunology)
[2020, 2020:8454532]

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Improvement of stiff-person syndrome symptoms in pregnancy: Case series and literature review.

Megan E Esch, Scott D Newsome,

OBJECTIVE:To describe 2 cases from a single academic institution of improvement in stiff-person syndrome (SPS) symptoms during pregnancy and to review the clinical outcomes of SPS in 6 additional pregnancies described in the literature. METHODS:Evaluation of clinical symptoms and treatment changes of disease state during pregnancy. RESULTS:Seven patients with 9 ... Read more >>

Neurol Neuroimmunol Neuroinflamm (Neurology(R) neuroimmunology & neuroinflammation)
[2020, 7(3):]

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Stiff person spectrum disorders: An illustrative case series of their phenotypic and antibody diversity.

Francisco Bernardo, Leonor Rebordão, André Rêgo, Sara Machado, João Passos, Cristina Costa, Simão Cruz, Amélia Nogueira Pinto, Mariana Santos,

Stiff person spectrum disorders (SPSD) are a broad group of immune-mediated disorders. Clinical presentations include classical stiff person syndrome (SPS), focal SPS, and progressive encephalomyelitis with rigidity and myoclonus (PERM). The most frequently associated antibodies are anti-GAD65, anti-GlyR, anti-amphiphysin, and anti-DPPX. Immunotherapy is the primary treatment modality. We present an ... Read more >>

J. Neuroimmunol. (Journal of neuroimmunology)
[2020, 341:577192]

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Intravenous methylprednisolone or immunoglobulin for anti-glutamic acid decarboxylase 65 antibody autoimmune encephalitis: which is better?

Tao-Ran Li, Yu-Di Zhang, Qun Wang, Xiao-Qiu Shao, Zhi-Mei Li, Rui-Juan Lv,

BACKGROUND:Patients positive for anti-glutamic acid decarboxylase 65 (GAD65) antibodies have attracted increasing attention. Their clinical manifestations are highly heterogeneous and can be comorbid with tumors. Currently, there is no consensus on the therapeutic regimen for anti-GAD65-associated neurological diseases due to the clinical complexity, rarity and sporadic distribution. We reported six ... Read more >>

BMC Neurosci (BMC neuroscience)
[2020, 21(1):13]

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Description of orthotic properties and effect evaluation of ankle-foot orthoses in non-spastic calf muscle weakness.

Niels F J Waterval, Merel-Anne Brehm, Jaap Harlaar, Frans Nollet,

OBJECTIVE:To describe the orthotic properties and evaluate the effects of ankle-foot orthoses for calf muscle weakness in persons with non-spastic neuromuscular disorders compared with shoes-only. DESIGN:Cross-sectional study. SUBJECTS:Thirty-four persons who used ankle-foot orthoses for non-spastic calf muscle weakness. METHODS:The following orthotic properties were measured: ankle-foot orthosis type, mass, and ankle ... Read more >>

J Rehabil Med (Journal of rehabilitation medicine)
[2020, 52(3):jrm00026]

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Stiff Person Syndrome and Acetylcholine Receptor Ganglionic Neuronal Antibodies.

Jacobo Lester, Jimmy Cojab, Enrique Klériga,

The association between stiff person syndrome and paraneoplastic syndromes has been described, linking intracellular or extracellular antibodies. We describe the case of a 64-year-old woman with stiff person syndrome and positivity for acetylcholine receptor ganglionic neuronal antibodies, which can also be seen in muscular hyperexcitability conditions, as well as other ... Read more >>

Case Rep Neurol (Case reports in neurology)
[2020, 12(1):24-26]

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Intranasal midazolam for treating acute respiratory crises in a woman with stiff person syndrome.

Giuseppe Cosentino, Marcello Romano, Margherita Algeri, Filippo Brighina, Enrico Alfonsi, Cristina Tassorelli, Grazia Crescimanno,

Neurol Neuroimmunol Neuroinflamm (Neurology(R) neuroimmunology & neuroinflammation)
[2020, 7(4):]

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Low-Dose Naltrexone reduces symptoms in Stiff-Person Syndrome.

Mauro Zappaterra, Elizabeth Shouse, Reed Loring Levine,

Stiff-Person Syndrome (SPS) is a rare neurologic disorder characterized by severe and progressively worsening muscle stiffness and rigidity. SPS can be very painful due to unpredictable muscle spasms which can be triggered by various stimuli, such as noise, touch, or emotional experiences. There is thought to be an autoimmune component ... Read more >>

Med. Hypotheses (Medical hypotheses)
[2020, 137:109546]

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Ultrasound elastography in the evaluation of peripheral neuropathies: a systematic review of the literature.

Jakub Zakrzewski, Katarzyna Zakrzewska, Katarzyna Pluta, Oleg Nowak, Anna Miłoszewska-Paluch,

Peripheral neuropathy is associated with an increase in intraneural pressure, and hence ultrasound elastography seems to be an ideal method to detect early stages of this condition based on changes in the affected nerve stiffness. The aim of this systematic review was to analyse the applicability of strain elastography (SE) ... Read more >>

Pol J Radiol (Polish journal of radiology)
[2019, 84:e581-e591]

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Retinal pathology occurs in stiff-person syndrome.

Jeffrey Lambe, Alissa Rothman, Jerry Prince, Shiv Saidha, Peter A Calabresi, Scott D Newsome,

OBJECTIVE:To evaluate whether structural and functional changes occur in the afferent visual system of patients with stiff-person syndrome (SPS) and whether these changes correlate with disease burden, given the high concentration of γ-aminobutyric acid receptors, which are generally thought to be involved in SPS pathogenesis, in the retina. METHODS:In this ... Read more >>

Neurology (Neurology)
[2020, :]

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Autoimmune musicogenic epilepsy associated with anti-glutamic acid decarboxylase antibodies and Stiff-person syndrome.

Joana Jesus-Ribeiro, Alireza Bozorgi, Modhi Alkhaldi, Mahmoud Shaqfeh, Guadalupe Fernandez-Baca Vaca, Bashar Katirji,

Epilepsy should be suspected in patients with Stiff-person syndrome and new onset paroxysmal episodes. Musicogenic epilepsy may be a manifestation of anti-GAD-Ab spectrum, supporting an autoimmune workup in these patients. Appropriate treatment is not well established, and immunotherapy should be considered in patients with only partial response to antiepileptic drugs. ... Read more >>

Clin Case Rep (Clinical case reports)
[2020, 8(1):61-64]

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Chronic intestinal pseudo-obstruction with dilated biliary tract as a spectrum of stiff person syndrome in a nondiabetic patient.

Alexandre Nguyen, Hubert de Boysson, Fohlen Audrey, Seydou Yameogo, Achille Aouba,

Stiff person syndrome (SPS) is a rare and challenging neuromuscular junction disorder with typical musculoskeletal manifestations associated with anti-GAD65 antibodies, extra rheumatological manifestations, including neuropsychiatric symptoms and severe dysautonomic troubles. Chronic intestinal pseudo-obstruction (CIPO) is also a rare condition corresponding to a sub-occlusive syndrome, resulting from the functional or structural ... Read more >>

J Musculoskelet Neuronal Interact (Journal of musculoskeletal & neuronal interactions)
[2019, 19(4):526-530]

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Improving Ambulation and Minimizing Disability with Therapeutic Plasma Exchange in a Stiff-person Syndrome Patient with Recurrent Falls.

Kyle Sanchez, Aqsa Ullah, Alexandria R Waler, Yassar Chakfe,

Stiff-person syndrome (SPS) is a rare, autoimmune, neuromuscular disorder that manifests with axial and proximal muscle stiffness, rigidity, and painful muscle spasms, often causing progressive disability due to limited movement. First-line therapies comprise symptomatic management with γ-aminobutyric acid-modulating drugs such as benzodiazepines and baclofen. Patients resistant to these treatments are often ... Read more >>

Cureus (Cureus)
[2019, 11(11):e6209]

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Experienced complaints, activity limitations and loss of motor capacities in patients with pure hereditary spastic paraplegia: a web-based survey in the Netherlands.

Bas J H van Lith, Hans C J W Kerstens, Laura A C van den Bemd, Maria W G Nijhuis-van der Sanden, Vivian Weerdesteyn, Rob J E M Smeets, Klemens Fheodoroff, Bart P C van de Warrenburg, Alexander C H Geurts,

BACKGROUND:Hereditary spastic paraplegia (HSP) is a group of inherited disorders characterized by progressive spastic paresis of the lower limbs. Treatment is often focused on reducing spasticity and its physical consequences. To better address individual patients' needs, we investigated a broad range of experienced complaints, activity limitations, and loss of motor ... Read more >>

Orphanet J Rare Dis (Orphanet journal of rare diseases)
[2020, 15(1):64]

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A Case of Anti-glutamic Acid Decarboxylase-65 Antibody Positive Stiff Person Syndrome Presenting Initially as Acute Peripheral Vestibulopathy, Leading to Delayed Diagnosis After Multiple Hospitalizations.

Yash Nene, Tejas Mehta, Sanjay Pawar, Gajanan Patil, Nasli R Ichaporia,

Stiff person syndrome (SPS), with a prevalence of one to two per million, is an extremely rare neurological condition that is characterized by axial muscle stiffness and rigidity along with intermittent painful muscle spasms. It is often associated with psychiatric co-morbidities such as anxiety and depression. The pathophysiology, although poorly ... Read more >>

Cureus (Cureus)
[2019, 11(11):e6083]

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Rituximab improves not only back stiffness but also "stiff eyes" in stiff person syndrome: Implications for immune-mediated treatment.

Satoshi Kodama, Shin-Ichi Tokushige, Yusuke Sugiyama, Kazuya Sato, Juuri Otsuka, Yuichiro Shirota, Masashi Hamada, Atsushi Iwata, Tatsushi Toda, Shoji Tsuji, Yasuo Terao,

OBJECTIVE:Stiff person syndrome (SPS) is usually characterized by truncal muscle rigidity and episodic painful spasms, but it sometimes appears with ocular symptoms called "stiff eyes". We recorded saccade movements in an SPS patient manifesting with "stiff eyes" conditions with slow saccade velocity and evaluated the effect of immunotherapy including rituximab ... Read more >>

J. Neurol. Sci. (Journal of the neurological sciences)
[2020, 408:116506]

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Knee joint kinematics of the pendulum test in children with and without Down syndrome.

Diego M Ferreira, Huaqing Liang, Jianhua Wu,

BACKGROUND:The Wartenberg pendulum test is a common clinical test that is used to measure stiffness about the knee in persons with and without disabilities such as cerebral palsy and Down syndrome (DS). Adults and adolescents with DS show fewer number of swing cycles and a lower relaxation index than healthy ... Read more >>

Gait Posture (Gait & posture)
[2020, 76:311-317]

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A stiff person syndrome misdiagnosed as a psychiatric illness.

Mohamed-Anis Souissi, Syrine Bellakhal, Emna Gharbi, Istabrak Abdelkefi, Mohamed-Hedi Douggui,

A 36-year-old man from presented with an 11-year history of progressive stiffness and painful spasms of his both legs, with recent worsening of his condition overthe last year resulting in a considerable difficulty of standing up and walking. As the patient developed phobic symptoms, he was considered as having a ... Read more >>

Curr Rheumatol Rev (Current rheumatology reviews)
[2019, :]

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Neurologic syndromes related to anti-GAD65: Clinical and serologic response to treatment.

Amaia Muñoz-Lopetegi, Marienke A A M de Bruijn, Sanae Boukhrissi, Anna E M Bastiaansen, Mariska M P Nagtzaam, Esther S P Hulsenboom, Agnita J W Boon, Rinze F Neuteboom, Juna M de Vries, Peter A E Sillevis Smitt, Marco W J Schreurs, Maarten J Titulaer,

OBJECTIVE:Antibodies against glutamic acid decarboxylase 65 (anti-GAD65) are associated with a number of neurologic syndromes. However, their pathogenic role is controversial. Our objective was to describe clinical and paraclinical characteristics of anti-GAD65 patients and analyze their response to immunotherapy. METHODS:Retrospectively, we studied patients (n = 56) with positive anti-GAD65 and ... Read more >>

Neurol Neuroimmunol Neuroinflamm (Neurology(R) neuroimmunology & neuroinflammation)
[2020, 7(3):]

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Ultrasound elastography as a potential method to evaluate entrapment neuropathies in elite athletes: a mini-review.

Agnieszka Dąbrowska-Thing, Jakub Zakrzewski, Oleg Nowak, Żaneta Nitek,

Entrapment neuropathies constitute a significant albeit still neglected problem in athletes. Currently available diagnostic options in patients with suspected neuropathies, nerve conduction studies, and magnetic resonance imaging have some drawbacks, mostly related to their invasive character, high cost, and limited availability. This paper reviews published articles documenting the use of ... Read more >>

Pol J Radiol (Polish journal of radiology)
[2019, 84:e625-e629]

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Modifying ankle foot orthosis stiffness in patients with calf muscle weakness: gait responses on group and individual level.

Niels F J Waterval, Frans Nollet, Jaap Harlaar, Merel-Anne Brehm,

BACKGROUND:To improve gait, persons with calf muscle weakness can be provided with a dorsal leaf spring ankle foot orthosis (DLS-AFO). These AFOs can store energy during stance and return this energy during push-off, which, in turn, reduces walking energy cost. Simulations indicate that the effect of the DLS-AFO on walking ... Read more >>

J Neuroeng Rehabil (Journal of neuroengineering and rehabilitation)
[2019, 16(1):120]

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Utility of Botulinum Injections in Stiff-Person Syndrome.

L M Conners, A Betcher, A Shahinian, P Janda,

Stiff-person syndrome (SPS) is an uncommon neurological disorder characterized by significant rigidity and muscle spasms primarily affecting the truncal and proximal musculature. Furthermore, a wide-based gait with functional impairment is generally seen. High-dose benzodiazepines or baclofen are widely considered the optimal initial therapy; however, major adverse effects often preclude adequate ... Read more >>

Case Rep Neurol Med (Case reports in neurological medicine)
[2019, 2019:9317916]

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Fibromyalgia associated with repeated gadolinium contrast-enhanced MRI examinations.

Silvia Maria Lattanzio, Francesca Imbesi,

We report a case of a fibromyalgia (FM) patient with an history of brain-cancer presenting signs and symptoms of gadolinium toxicity following repeated administrations of a macrocyclic contrast agent, Gadovist. In the present report, we provide evidence supporting the hypothesis of a causal relationship linking gadolinium deposition to a clinical ... Read more >>

Radiol Case Rep (Radiology Case Reports)
[2020, 15(5):534-541]

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Yoga therapy for fibromyalgia syndrome: A case report.

Anita Verma, Sanjay U Shete, Gururaj Doddoli,

Fibromyalgia is a rheumatologic syndrome leading to increased pain sensitivity, sleep disturbance, fatigue, stiffness, and tenderness of joints, muscles, and tendons due to dysregulation of neurophysiological functions. In the present case report, a 42-year-old, non-Indian, nonsmoking, nonalcoholic, female presented with complaints of severe difficulty in walking, joint pains, and generalized ... Read more >>

J Family Med Prim Care (Journal of family medicine and primary care)
[2020, 9(1):435-438]

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