Atypical Omenn Syndrome due to Adenosine Deaminase Deficiency

نویسندگان

  • Avni Y. Joshi
  • Erin K. Ham
  • Neel B. Shah
  • Xiangyang Dong
  • Shakila P. Khan
  • Roshini S. Abraham
چکیده

We present here a novel case of an atypical Omenn syndrome (OS) phenotype due to mutations in the ADA gene encoding adenosine deaminase. This case is noteworthy for a significant increase in circulating CD56(bright)CD16- cytokine-producing NK cells after treatment with steroids for skin rash.

برای دانلود رایگان متن کامل این مقاله و بیش از 32 میلیون مقاله دیگر ابتدا ثبت نام کنید

ثبت نام

اگر عضو سایت هستید لطفا وارد حساب کاربری خود شوید

منابع مشابه

Atypical Omenn Syndrome Due to RAG2 Gene Mutation, a Case Report

Severe Combined Immunodeficiency (SCID), characterized by a profound decrease in both the number and function of T cells, is related to more than 20 different mutations. Recombination-activating gene (RAG) 1 and 2 seem to be two of the most common forms presenting with various manifestations, including typical SCID, Omenn syndrome (OS), atypical SCID (AS), or delayed onset combined immunodefici...

متن کامل

Adenosine deaminase deficiency and severe combined immunodeficiency disease.

The nature of the association of adenosine deaminase deficiency and severe combined immunodeficiency disease is reviewed . The basis for the molecular heterogeneity exhibited by adenosine deaminase in human tissue and the mechanisms whereby a deficiency of this activity results in the extreme perturbation of the immune system as observed in severe combined immunodeficiency are critically discus...

متن کامل

Immune deficiency due to adenosine deaminase and purine nucleoside phosphorylase deficiency: a simple diagnostic test.

A simple method is described for diagnosing adenosine deaminase and purine nucleoside phosphorylase deficiency using urine. Cellulose thin layer chromatography of 1 microliter of urine from affected children was performed and deoxyadenosine and deoxyguanosine were easily detected by phosphorescence at the temperature of liquid nitrogen. This test is not expensive and can be done in any laborato...

متن کامل

Caffeine effect on adenosine deaminase catalysis: A new look at the effect of caffeine on adenosine deaminase activity

The effect of physiological concentrations of caffeine (purified from Persian tea) on adenosine deaminase (ADA) activity at physiological and pathological concentrations of adenosine (as substrate) in 50 mM Tris-HCl buffer (pH 7.3) at 37°C was investigated, using UV-VIS spectroscopy. ADA exhibited a bi-phasic activity behavior and both phases showed positive cooperativities indicating adenosine...

متن کامل

Spondylo-mesomelic-acrodysplasia with joint dislocations and severe combined immunodeficiency: a newly recognised immuno-osseous dysplasia.

A newborn girl is described with an association of spondylo-acrodysplasia, mild short limbed dwarfism without significant metaphyseal changes, joint dislocations, and severe immune system dysfunction. This association is distinct from other known immuno-osseous dysplasias, including Schimke dysplasia, ADA deficiency with osseous changes, and Omenn phenotype with short limbed dwarfism.

متن کامل

ذخیره در منابع من


  با ذخیره ی این منبع در منابع من، دسترسی به آن را برای استفاده های بعدی آسان تر کنید

عنوان ژورنال:

دوره 2012  شماره 

صفحات  -

تاریخ انتشار 2012