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Journal of Musculoskeletal Pain Volume 18, 2010 - Issue 2
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CASE REPORT

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

Ignacio Blanco Blanco Ignacio Blanco Blanco, MD, and Hortensia Canto Argíz, PhD, Department of Internal Medicine, Unit of Respiratory Diseases, Valle del Nalón Hospital, Principado de Asturias, Spain.
, MD,
Aurora Astudillo Aurora Astudillo, PhD, Department of Pathology, Hospital Universitario Central de Asturias, Principado de Asturias, Spain.
, PhD,
Francisco Domínguez Iglesias Francisco Domínguez Iglesias, PhD, Department of Pathology, Hospital Valle del Nalón, Principado de Asturias, Spain.
, PhD,
Sabina Janciauskiene Sabina Janciauskiene, PhD, Department of Medicine, Malmö University Hospital, Malmö, Sweden.
, PhD,
Victoriano Cárcaba Fernández Victoriano Cárcaba Fernández, PhD, and César Gallo Alvaro, MD, Department of Internal Medicine, Valle del Nalón Hospital, Principado de Asturias, Spain.
, PhD,
César Gallo Alvaro Victoriano Cárcaba Fernández, PhD, and César Gallo Alvaro, MD, Department of Internal Medicine, Valle del Nalón Hospital, Principado de Asturias, Spain.
, MD,
Hortensia Canto Argíz Ignacio Blanco Blanco, MD, and Hortensia Canto Argíz, PhD, Department of Internal Medicine, Unit of Respiratory Diseases, Valle del Nalón Hospital, Principado de Asturias, Spain.
, PhD,
Frederick Joseph de Serres Frederick Joseph de Serres, PhD, Center for the Evaluation of Risks to Human Reproduction, Environmental Toxicology Program, National Institute of Environmental Health Sciences, Research Triangle Park, North Carolina, USA.
, PhD &
Enrique Fernández Bustillo Enrique Fernández Bustillo, PhD, Biostatistics Unit, Central University Hospital of Asturias, Principado de Asturias, Spain.
, PhD show all
Pages 167-172 | Accepted 08 Apr 2009, Published online: 13 May 2010

REFERENCES

  • Blanco I, Canto H, de Serres FJ, Fernandez-Bustillo E, Rodriguez MC: Alpha 1-antitrypsin replacement therapy controls fibromyalgia symptoms in 2 patients with PI ZZ alpha 1-antitrypsin deficiency. J Rheumatol 31(10): 2082–2085, 2004.
  • Blanco I, de Serres FJ, Bustillo EF, Al Kassam D, Arbesú D, Rodríguez C, : Alpha 1-Antitrypsin and fibromyalgia: New data in favour of the inflammatory hypothesis of fibromyalgia. Med Hypotheses (Oxford) 64: 759–769, 2005.
  • Blanco I, Janciauskiene S, Astudillo A, Domínguez F, Bustillo EF, Nita I: Fibromyalgia and alpha-1 antitrypsin, chapter 1. New Research in Fibromyalgia. Nova Science Publisher, New York, 2006.
  • Blanco I, Arbesú D, Al Kassam D, de Serres F, Fernández-Bustillo E, Rodríguez Menéndez C: Alpha 1-antitrypsin polymorphism in fibromyalgia patients from the Asturias Province in Northern Spain: A significantly higher prevalence of the PI*Z deficiency allele in patients than in the general population. J Musculoske Pain 14(3): 5–12, 2006.
  • Russell IJ: Alpha 1-antitrypsin deficiency in the fibromyalgia syndrome. J Musculoske Pain 14(3): 1–3, 2006.
  • Blanco I, De Serres FJ, Janciauskiene S, Arbesú D, Bustillo EF, Cárcaba V, : Estimates of the prevalence and number of fibromyalgia syndrome patients and their alpha-1 antitrypsin phenotypic distribution in ten countries. J Musculoske Pain 15(4): 9–23, 2007.
  • Russell IJ: Pathogenesis of the fibromyalgia syndrome. J Musculoske Pain 15(4): 5–8, 2007.
  • Aldonyte R, Jansson L, Ljungberg O, Larsson S, Janciauskiene S: Polymerized alpha-antitrypsin is present on lung vascular endothelium. New insights into the biological significance of alpha-antitrypsin polymerization. Histopathology 45(6): 587–592, 2004.
  • Janciauskiene S, Stevens T, Blanco I: New insights into the biology of alpha 1-antitrypsin and its role in chronic obstructive pulmonary disease. Curr Respir Med Rev 3: 147–158, 2007.
  • Vergnolle N, Bunnett NW, Sharkey KA, Brussee V, Compton SJ, Grady EF, : Proteinase-activated receptor-2 and hyperalgesia: A novel pain pathway. Nat Med 7(7): 821–826, 2001.
  • Vergnolle N: Modulation of visceral pain and inflammation by protease-activated receptors. Br J Pharmacol 141(8): 1264–1274, 2004.
  • Oikonomopoulou K, Hansen KK, Saifeddine M, Vergnolle N, Tea I, Blaber M, : Kallikrein-mediated cell signalling: targeting proteinase-activated receptors (PARs). Biol Chem 387(6): 817–824, 2006.

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