Intravenous Infusions of Purified Alpha-1 Antitripsyn Effectively Controls Symptoms and Reverts Muscle Biopsy Changes in an MZ Alpha-1 Antitripsyn Deficiency and Fibromyalgia Syndrome Patient

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Intravenous Infusions of Purified Alpha-1 Antitripsyn Effectively Controls Symptoms and Reverts Muscle Biopsy Changes in an MZ Alpha-1 Antitripsyn Deficiency and Fibromyalgia Syndrome Patient. / Blanco Blanco, Ignacio; Astudillo, Aurora; Dominguez Iglesias, Francisco; Janciauskiene, Sabina; Carcaba Fernandez, Victoriano; Gallo Alvaro, Cesar; Canto Argiz, Hortensia; de Serres, Frederick Joseph; Fernandez Bustillo, Enrique.

I: Journal of Musculoskeletal Pain, Vol. 18, Nr. 2, 2010, s. 167-172.

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Blanco Blanco, Ignacio ; Astudillo, Aurora ; Dominguez Iglesias, Francisco ; Janciauskiene, Sabina ; Carcaba Fernandez, Victoriano ; Gallo Alvaro, Cesar ; Canto Argiz, Hortensia ; de Serres, Frederick Joseph ; Fernandez Bustillo, Enrique. / Intravenous Infusions of Purified Alpha-1 Antitripsyn Effectively Controls Symptoms and Reverts Muscle Biopsy Changes in an MZ Alpha-1 Antitripsyn Deficiency and Fibromyalgia Syndrome Patient. I: Journal of Musculoskeletal Pain. 2010 ; Vol. 18, Nr. 2. s. 167-172.

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TY - JOUR

T1 - Intravenous Infusions of Purified Alpha-1 Antitripsyn Effectively Controls Symptoms and Reverts Muscle Biopsy Changes in an MZ Alpha-1 Antitripsyn Deficiency and Fibromyalgia Syndrome Patient

AU - Blanco Blanco, Ignacio

AU - Astudillo, Aurora

AU - Dominguez Iglesias, Francisco

AU - Janciauskiene, Sabina

AU - Carcaba Fernandez, Victoriano

AU - Gallo Alvaro, Cesar

AU - Canto Argiz, Hortensia

AU - de Serres, Frederick Joseph

AU - Fernandez Bustillo, Enrique

PY - 2010

Y1 - 2010

N2 - Background: An MZ phenotype alpha-1 antitrypsin [AAT] deficiency and fibromyalgia syndrome [FMS] patient participated in a trial with AAT-intravenous augmentationtherapy [AAT-IV-AT] after failure of conventional therapeutic measures. Three quadricep biopsies were performed at different stages. The first one showed large aggregates of AAT and ubiquitin in myocytes and blood vessels, and moderate muscle atrophy, before AAT-IV-AT. The two remaining biopsies were performed while receiving AAT-IV-AT. Findings: Remarkably good clinical response and histological improvement in the follow-up biopsies. Conclusions: The clinical and histopathological efficacy of AAT-IV-AT evidenced in this patient should open new perspectives of research and management of AAT deficiency and FMS patients.

AB - Background: An MZ phenotype alpha-1 antitrypsin [AAT] deficiency and fibromyalgia syndrome [FMS] patient participated in a trial with AAT-intravenous augmentationtherapy [AAT-IV-AT] after failure of conventional therapeutic measures. Three quadricep biopsies were performed at different stages. The first one showed large aggregates of AAT and ubiquitin in myocytes and blood vessels, and moderate muscle atrophy, before AAT-IV-AT. The two remaining biopsies were performed while receiving AAT-IV-AT. Findings: Remarkably good clinical response and histological improvement in the follow-up biopsies. Conclusions: The clinical and histopathological efficacy of AAT-IV-AT evidenced in this patient should open new perspectives of research and management of AAT deficiency and FMS patients.

KW - alpha-1 antitrypsin augmentation therapy

KW - deficiency

KW - alpha-1 antitrypsin

KW - Fibromyalgia syndrome

U2 - 10.3109/10582452.2010.483962

DO - 10.3109/10582452.2010.483962

M3 - Article

VL - 18

SP - 167

EP - 172

JO - Journal of Musculoskeletal Pain

JF - Journal of Musculoskeletal Pain

SN - 1540-7012

IS - 2

ER -