Literature DB >> 28058497

[A rare cause of severe panniculitis].

C Fiehn1.   

Abstract

A 58-year-old patient presented with a severe, episodic panniculitis of the upper legs. Necrosis of the fatty tissue and a suspected superinfection led to amputation of one leg. The panniculitis was caused by a hereditary deficiency of alpha-1 antitrypsin (AAT) due to a ZZ mutation of the AAT gene. Neutrophilic panniculitis is found in 0.1% of patients with the ZZ mutation and therefore is the rarest clinical manifestation of AAT deficiency. With the exception of mild COPD, the patient had no other typical clinical symptoms of AAT deficiency. Treatment with colchicum reduced the frequency and severity of the flares.

Entities:  

Keywords:  Alpha-1 antitrypsin; Colchicin; Panniculitis

Mesh:

Year:  2017        PMID: 28058497     DOI: 10.1007/s00393-016-0247-3

Source DB:  PubMed          Journal:  Z Rheumatol        ISSN: 0340-1855            Impact factor:   1.372


  8 in total

Review 1.  alpha1-Antitrypsin deficiency panniculitis (phenotype PiZZ) precipitated postpartum and successfully treated with dapsone.

Authors:  P D Yesudian; C M Dobson; N J Wilson
Journal:  Br J Dermatol       Date:  2004-06       Impact factor: 9.302

Review 2.  Anakinra for difficult-to-treat neutrophilic panniculitis: IL-1 blockade as a promising treatment option for neutrophil-mediated inflammatory skin disease.

Authors:  Dan Lipsker; Cécile Perrigouard; Audrey Foubert; Bernard Cribier
Journal:  Dermatology       Date:  2010-03-02       Impact factor: 5.366

3.  alpha-1 antitrypsin inhibits caspase-3 activity, preventing lung endothelial cell apoptosis.

Authors:  Irina Petrache; Iwona Fijalkowska; Terry R Medler; Jarrett Skirball; Pedro Cruz; Lijie Zhen; Horia I Petrache; Terence R Flotte; Rubin M Tuder
Journal:  Am J Pathol       Date:  2006-10       Impact factor: 4.307

4.  α-1 Antitrypsin regulates human neutrophil chemotaxis induced by soluble immune complexes and IL-8.

Authors:  David A Bergin; Emer P Reeves; Paula Meleady; Michael Henry; Oliver J McElvaney; Tomás P Carroll; Claire Condron; Sanjay H Chotirmall; Martin Clynes; Shane J O'Neill; Noel G McElvaney
Journal:  J Clin Invest       Date:  2010-11-08       Impact factor: 14.808

5.  New Findings in PiZZ alpha1-antitrypsin deficiency-related panniculitis. Demonstration of skin polymers and high dosing requirements of intravenous augmentation therapy.

Authors:  B Gross; M Grebe; M Wencker; J K Stoller; L M Bjursten; S Janciauskiene
Journal:  Dermatology       Date:  2009-02-16       Impact factor: 5.366

Review 6.  Neutrophilic panniculitis associated with alpha-1-antitrypsin deficiency: an update.

Authors:  I Blanco; D Lipsker; B Lara; S Janciauskiene
Journal:  Br J Dermatol       Date:  2016-01-27       Impact factor: 9.302

7.  Alpha-1 antitrypsin augmentation therapy corrects accelerated neutrophil apoptosis in deficient individuals.

Authors:  Killian Hurley; Noreen Lacey; Ciara A O'Dwyer; David A Bergin; Oliver J McElvaney; M Emmet O'Brien; Oisín F McElvaney; Emer P Reeves; Noel G McElvaney
Journal:  J Immunol       Date:  2014-09-12       Impact factor: 5.422

8.  ANCA-associated vasculitis is linked to carriage of the Z allele of α₁ antitrypsin and its polymers.

Authors:  H Morris; M D Morgan; A M Wood; S W Smith; U I Ekeowa; K Herrmann; J U Holle; L Guillevin; D A Lomas; J Perez; C D Pusey; A D Salama; R Stockley; S Wieczorek; A J McKnight; A P Maxwell; E Miranda; J Williams; C O Savage; L Harper
Journal:  Ann Rheum Dis       Date:  2011-08-05       Impact factor: 19.103

  8 in total

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