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The discrete kernel-based regression approach generally provides pointwise estimates of count data that do not account for uncertainty about both parameters and resulting estimates. This work aims to provide probabilistic kernel estimates of count regression function by using Bayesian approach and then allows for a readily quantification of uncertainty. Bayesian approach enables to incorporate prior knowledge of parameters used in discrete kernel-based regression. An application was proposed on count data of condition factor of fish (K) provided from an experimental project that analyzed various pond management strategies. The probabilistic distribution of estimates were contrasted by discrete kernels, as a support to theoretical results on the performance of kernels. More practically, Bayesian credibility intervals of K-estimates were evaluated to compare pond management strategies. Thus, similarities were found between performances of semi-intensive and coupled fishponds, with formulated feed, in comparison with extensive fishponds, without formulated feed. In particular, the fish development was less predictable in extensive fishpond, dependent on natural resources, than in the two other fishponds, supplied in formulated feed.
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Immunoglobulin (Ig) therapy is used to treat a wide range of immunodeficiencies and autoimmune diseases; While, its clinical benefit has been demonstrated in several studies, Ig therapy is associated with a risk of systemic adverse effects. As such, Onset of renal impairment, including acute renal failure, osmotic nephrosis and renal insufficiency, after immunoglobulin administration is rare, but is one of the most significant concerns related to intravenous Ig use at immunomodulatory doses. However, only few studies have investigated the safety of subcutaneous Ig (SCIg) in relation to these rare conditions. The aim of this prospective study is to describe the safety of SCIg (Gammanorm), specifically with regards to renal function, in inflammatory myopathies including mainly polymyositis (PM), dermatomyositis (DM) and inclusion body myositis (IBM). Twenty-four cases were included: 10 patients with PM, 6 with IBM, 5 with DM, 2 with mixed connective-tissue disease (MCTD) and 1 patient with scleromyositis. SCIg was given two to three times per week at 2 g/kg/month in all patients. Patients were treated for a mean duration of 24.6 ± 11.4 months (range 8-37 months) and received a median of 78 SCIg infusions. Renal function was stable over the study period in all patients. High-dose SCIg was well tolerated; the treatment was not withdrawn during the first year in any patient for safety issues. These results suggest that the use of high-dose SCIg is generally feasible and safe in patients with inflammatory myopathies.
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BACKGROUND: Antisynthetase syndrome is a rare and debilitating multiorgan disease characterized by inflammatory myopathy, interstitial lung disease, cutaneous involvement, and frequent chronic inflammation of the joints. Standard treatments include corticosteroids and immunosuppressants. In some cases, treatment resistance may develop. Administration of immunoglobulins intravenously is recommended in patients with drug-resistant antisynthetase syndrome. CASE PRESENTATION: Here, we describe the case of a 56-year-old woman of Algerian origin. She is the first case of a patient with multidrug-resistant antisynthetase syndrome featuring pulmonary involvement and arthropathy, and chronic secondary immune deficiency with recurrent infections, after anti-CD20 treatment, in which her primary antisynthetase syndrome-related symptoms and secondary immune deficiency were treated successfully with subcutaneous administration of immunoglobulin. The administration of immunoglobulin subcutaneously was introduced at a dose of 2 g/kg per month and was well tolerated. Clinical improvement was observed within 3 months of initiation of subcutaneous administration of immunoglobulin. After 22 months of treatment, she showed a significant improvement in terms of muscle strength, pulmonary involvement, arthralgia, and immunodeficiency. Her serum creatine phosphokinase and C-reactive protein levels remained normal. Finally, she was compliant and entirely satisfied with the treatment. CONCLUSIONS: Taken together, these observations suggest that administration of immunoglobulin subcutaneously may be a useful therapeutic approach to tackle steroid-refractory antisynthetase syndrome while ensuring minimal side effects and improved treatment compliance. This treatment also allowed, in our case, for the regression of the chronic immunodeficiency secondary to rituximab treatment.
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Imunoglobulinas/administração & dosagem , Miosite/terapia , Agamaglobulinemia/complicações , Feminino , Humanos , Síndromes de Imunodeficiência/complicações , Imunossupressores/uso terapêutico , Doenças Pulmonares Intersticiais/complicações , Doenças Pulmonares Intersticiais/diagnóstico por imagem , Imageamento por Ressonância Magnética , Metotrexato/uso terapêutico , Pessoa de Meia-Idade , Miosite/complicações , Miosite/diagnóstico por imagem , Absorção SubcutâneaRESUMO
Subcutaneous immunoglobulin (SCIg) therapy is indicated in primary and secondary immunodeficiency diseases. Its use in practice is being extended to autoimmune diseases. Few studies investigated the feasibility and safety of SCIg in these rare conditions. The aim was to describe the use of SCIg in inflammatory myopathies including polymyositis (PM), dermatomyositis (DM) and inclusion body myositis (IBM), in real-life settings. This case series was based upon a retrospective data collection. The primary objective was to assess the feasibility of the SCIg injections for the treatment of autoimmune diseases and adherence to high doses. Secondary objectives included safety and efficacy. Nineteen cases were identified: 7 patients were diagnosed with PM, 7 with IBM, 2 with DM, and 3 with myositis associated with connective tissue disease. Patients were treated and followed-up for a mean duration of 18.8 months (range 4.5-42). They received a median of 64 SCIg infusions and a total of 1215 infusions. Out of 14 patients, 10 showed an improvement in muscle strength, and 7 out of 11 showed an improvement in muscle disability scale. Two patients were lost to follow-up. Few slight adverse reactions were reported including mainly mild headaches and local skin reactions. Any serious adverse event was reported. These results suggest that the use of high-dose SCIg is feasible, beneficial and safe in patients with inflammatory myopathies. SCIg could be an alternative of IVIg in patients with difficult venous access or with insufficient response, and in patients preferring home care setting.
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Imunoglobulinas Intravenosas/uso terapêutico , Miosite/imunologia , Humanos , Infusões Subcutâneas , Força Muscular , Miosite/tratamento farmacológico , Estudos RetrospectivosRESUMO
INTRODUCTION: Inclusion body myositis (IBM) is a slowly progressive degenerative inflammatory disorder affecting both proximal and distal muscles. Immunosuppressive therapies are generally ineffective in the treatment of this disorder, and most patients are resistant to steroid therapy. Some benefits with mild improvement were observed with intravenous immunoglobulin (IVIg), particularly in patients with severe dysphagia. OBJECTIVES: The objective of this review was to describe the use of subcutaneous Ig (SCIg) in patients with IBM and to assess its feasibility. RESULTS: This report reviews 6 cases of IBM treated with SCIg in clinical practice. All patients had received prior treatments for IBM, including immunosuppressive agents and IVIg. SCIg was administered over a long period of time, ranging from 4.5 to 27 months. No patient discontinued the SCIg because of a treatment-related event or safety issues. The 6 cases showed an improvement in muscle strength and resolution of dysphagia. For 2 patients, this improvement persisted for approximately 12 months. CONCLUSIONS: SCIg might be proposed as an alternative therapy to patients with IBM who are resistant to corticoids and immunosuppressive therapies. Our findings suggest that treatment with SCIg (Gammanorm 16.5%, Octapharma AB) is feasible and safe in patients with IBM.
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BACKGROUND AND AIMS: Sarcopenia has been indicated as a reliable marker of frailty and poor prognosis among the oldest individuals. There are only few data on sarcopenia in healthy general population. We evaluated the prevalence of sarcopenia and its association with functional and clinical status in a population of healthy ambulatory subjects over 45 years living at home, in Paris (France). METHODS: This study was conducted selecting all ambulatory participants (n = 1,445) aged 45 years and older from October 2008 to September 2011, consulting in the Institute of Physiology (Institut de Jaeger) from Paris (France) for a functional and muscular evaluation, and did not have limitations to moderate physical exercise. All were healthy people. All subjects performed a medical examination, associated with evaluation of muscle mass (body composition assessment using dual-energy X-ray absorptiometry) and of muscle function (by hand grip strength). Diagnosis of sarcopenia required the documentation of low muscle mass with low muscle strength according to the current international consensus definition of sarcopenia. RESULTS: From 1,421 participants (553 males and 868 females) definitively enrolled, 221 subjects (135 females and 86 males) (15.5 %) were identified as sarcopenic. Results from multivariate logistic regression models showed that sarcopenia was inversely associated with BMI with those participants with BMI higher than 22 kg/m(2) showing a lower risk of sarcopenia relative to those with BMI less than 21 kg/m(2) (OR 0.72; 95 % CI 0.60-0.91). Similarly, probability of sarcopenia was lower among subjects involved in leisure physical activities for 3 h or more per week (OR 0.45; 95 % CI 0.24-0.93). According to the category of age [45-54; 55-64; 65-74; 75-84 and 85 years or more], the prevalence of sarcopenia in women increase from 9.1; 12.7; 14.5; 19.4; to 33.3 %, respectively. For the men, the percentage of sarcopenia increase with aging from 8.6; 15.6; 13.6; 63.8 to 45.5 %, respectively. CONCLUSIONS: The present study suggests that among healthy ambulatory subjects over 45 years living at home, sarcopenia is frequent, even to the youngest subjects of the studied population, taking place from 9 % from 45 years, until 64.3 % for the subjects over 85 years. Our findings support the hypothesis that muscle mass and function are associated with BMI and physical activity, whatever the age of the subject.