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Long-Term Benefits of Tailored Exercise in Severe Sarcoidosis: A Case Report.

ABSTRACT: We studied the effects of a supervised, structured exercise program in a severe sarcoidosis patient. After being clinically stable for two years, a 52-year-old woman (stage IV, American Thoracic Society) who originally had irreversible lung fibrosis, pulmonary arterial hypertension (PAH), mild mitral insufficiency, and atrial dilatation, and was candidate for lung transplant, performed a combined high-intensity interval, high load resistance, and inspiratory muscle training for 4.5 years, and was tested (cardiopulmonary exercise testing and dual X-ray absorptiometry) every six months. Cardiorespiratory fitness (CRF) and maximal pulmonary ventilation increased by 44% and 60%, respectively. Ventilatory efficiency also improved (decrease in the ventilatory equivalent for oxygen by 32% and 14% at the ventilatory threshold and respiratory compensation point, respectively). She improved New York Heart Association (NYHA) class (from III to II), and cardiac alterations as well as PAH reversed so that she was not in need of lung transplantation anymore. Likewise, she suffered no more episodes of hemoptysis. Bone health was overall maintained despite the post-menopausal status and the corticoid treatment. A long-term combined exercise intervention safely contributed-at least partly-to improve CRF and NYHA class in a patient with severe sarcoidosis, suggesting a potential coadjuvant effect to attenuate clinical manifestations.

SUBMITTER: Herrera-Olivares AM

PROVIDER: S-EPMC7766182 | biostudies-literature | 2020 Dec

REPOSITORIES: biostudies-literature

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Long-Term Benefits of Tailored Exercise in Severe Sarcoidosis: A Case Report.

Herrera-Olivares Alba M AM García-Manso Juan M JM Rodríguez-Gómez Irene I Ara Ignacio I Lucia Alejandro A Santalla Alfredo A

International journal of environmental research and public health 20201218 24

<h4>Background</h4>We studied the effects of a supervised, structured exercise program in a severe sarcoidosis patient.<h4>Methods</h4>After being clinically stable for two years, a 52-year-old woman (stage IV, American Thoracic Society) who originally had irreversible lung fibrosis, pulmonary arterial hypertension (PAH), mild mitral insufficiency, and atrial dilatation, and was candidate for lung transplant, performed a combined high-intensity interval, high load resistance, and inspiratory mus ...[more]

PMID: 33353138

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Project description:BackgroundCurrent treatments for Alzheimer's disease (AD) modulate global neurotransmission but are neither specific nor anatomically directed. Tailored stimulation of target nuclei will increase treatment efficacy while reducing side effects. We report the results of the first directional deep brain stimulation (dDBS) surgery and treatment of a patient with AD in an attempt to slow the progression of the disease in a woman with multi-domain, amnestic cognitive status.MethodsWe aimed to assess the safety of dDBS in patients with AD using the fornix as stimulation target (primary objective) and the clinical impact of the stimulation (secondary objective). In a registered clinical trial, a female patient aged 81 years with a 2-year history of cognitive decline and diagnoses of AD underwent a bilateral dDBS surgery targeting the fornix. Stimulation parameters were set between 3.9 and 7.5 mA, 90 μs, 130 Hz for 24 months, controlling stimulation effects by 18F-fluoro-2-deoxy-D-glucose (18F-FDG) scans (baseline, 12 and 24 months), magnetoencephalography (MEG) and clinical/neuropsychological assessment (baseline, 6, 12, 18, and 24 months).ResultsThere were no important complications related to the procedure. In general terms, the patient showed cognitive fluctuations over the period, related to attention and executive function patterns, with no meaningful changes in any other cognitive functions, as is shown in the clinical dementia rating scale (CDR = 1) scores over the 24 months. Such stability in neuropsychological scores corresponds to the stability of the brain metabolic function, seen in PET scans. The MEG studies described low functional connectivity at baseline and a subsequent increase in the number of significant connections, mainly in the theta band, at 12 months.ConclusionThe dDBS stimulation in the fornix seems to be a safe treatment for patients in the first stage of AD. Effects on cognition seem to be mild to moderate during the first months of stimulation and return to baseline levels after 24 months, except for verbal fluency.Clinical trial registration[https://clinicaltrials.gov/ct2/show/NCT03290274], identifier [NCT03290274].

Project description:BackgroundInflammatory bowel disease (IBD) is chronic inflammation of the gastrointestinal tract, although its etiology has largely been unclear. Tumor necrosis factor inhibitors (TNF-I) are effective for the treatment. Recently, biosimilars of TNF-I, such as CT-P13, have been developed and are thought to possess equal efficacy and safety to the original TNF-I. Sarcoidosis is also a systemic granulomatous disease of unknown etiology. In steroid-resistant cases of sarcoidosis, TNF-I have been reported effective for achieving resolution. However, the progression of sarcoidosis due to the TNF-I also has been reported. We herein report a case of pulmonary sarcoidosis with a Crohn's disease (CD) patient developed after a long period administration (15 years) of TNF-I.Case presentationsA 37-year-old woman with CD who had been diagnosed at 22 years old had been treated with the TNF-I (original infliximab; O-IFX and infliximab biosimilar; IFX-BS). Fifteen years after starting the TNF-I, she developed a fever and right chest pain. Chest computed tomography (CT) revealed clustered small nodules in both lungs and multiple enlarged hilar lymph nodes. Infectious diseases including tuberculosis were negative. Bronchoscopic examination was performed and the biopsy specimens were obtained. A pathological examination demonstrated noncaseating granulomatous lesions and no malignant findings. TNF-I were discontinued because of the possibility of TNF-I-related sarcoidosis. After having discontinued for four months, her symptoms and the lesions had disappeared completely. Fortunately, despite the discontinuation of TNF-I, she has maintained remission.ConclusionsTo our knowledge, this is the first case in which sarcoidosis developed after switching from O-IFX to IFX-BS. To clarify the characteristics of the cases with development of sarcoidosis during administration of TNF-I, we searched PubMed and identified 106 cases. When developing an unexplained fever, asthenia, uveitis and skin lesions in patients with TNF-I treatment, sarcoidosis should be suspected. Once the diagnosis of sarcoidosis due to TNF-I was made, the discontinuation of TNF-I and administration of steroid therapy should be executed promptly. When re-starting TNF-I, another TNF-I should be used for disease control. Clinicians should be aware of the possibility of sarcoidosis in patients under anti-TNF therapy.

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Long-Term Benefits of Tailored Exercise in Severe Sarcoidosis: A Case Report.

Publications

Long-Term Benefits of Tailored Exercise in Severe Sarcoidosis: A Case Report.

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