Editorial
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World J Gastrointest Pharmacol Ther. Oct 6, 2010; 1(5): 94-101
Published online Oct 6, 2010. doi: 10.4292/wjgpt.v1.i5.94
Z α-1 antitrypsin deficiency and the endoplasmic reticulum stress response
Catherine M Greene, Noel G McElvaney
Catherine M Greene, Noel G McElvaney, Respiratory Research Division, Department of Medicine, Royal College of Surgeons in Ireland, Education and Research Centre, Beaumont Hospital, Dublin 9, Ireland
Author contributions: Greene CM and McElvaney NG wrote this paper.
Supported by The U.S. Alpha One Foundation, the Health Research Board of Ireland, the Medical Research Charities Group, the Programmes for Research in Third Level Institutes administered by the Higher Education Authority and the Children’s Medical and Research Centre, Crumlin Hospital.
Correspondence to: Catherine Greene, PhD, Senior Lecturer, Respiratory Research Division, Department of Medicine, Royal College of Surgeons in Ireland, Education and Research Centre, Beaumont Hospital, Dublin 9, Ireland. cmgreene@rcsi.ie
Telephone: +353-1-8093800 Fax: +353-1-8093808
Received: April 1, 2010
Revised: September 20, 2010
Accepted: September 27, 2010
Published online: October 6, 2010
Abstract

The serine proteinase inhibitor α-1 antitrypsin (AAT) is produced principally by the liver at the rate of 2 g/d. It is secreted into the circulation and provides an antiprotease protective screen throughout the body but most importantly in the lung, where it can neutralise the activity of the serine protease neutrophil elastase. Mutations leading to deficiency in AAT are associated with liver and lung disease. The most notable is the Z AAT mutation, which encodes a misfolded variant of the AAT protein in which the glutamic acid at position 342 is replaced by a lysine. More than 95% of all individuals with AAT deficiency carry at least one Z allele. ZAAT protein is not secreted effectively and accumulates intracellularly in the endoplasmic reticulum (ER) of hepatocytes and other AAT-producing cells. This results in a loss of function associated with decreased circulating and intrapulmonary levels of AAT. However, the misfolded protein acquires a toxic gain of function that impacts on the ER. A major function of the ER is to ensure correct protein folding. ZAAT interferes with this function and promotes ER stress responses and inflammation. Here the signalling pathways activated during ER stress in response to accumulation of ZAAT are described and therapeutic strategies that can potentially relieve ER stress are discussed.

Keywords: α-1 antitrypsin; Unfolded protein response; Endoplasmic reticulum stress; Apoptosis; Autophagy; NFκB