Literature DB >> 22111599

Pulmonary lymphomatoid granulomatosis in Griscelli syndrome type 2.

Aleksandra Szczawinska-Poplonyk1, Zdzislawa Kycler, Anna Breborowicz, Maja Klaudel-Dreszler, Malgorzata Pac, Maria Zegadlo-Mylik, Renata Langfort.   

Abstract

Griscelli syndrome type 2 (GS2) is a rare autosomal-recessive disorder associated with a RAB27A gene mutation, and clinically manifesting as hypopigmentation, disseminated chronic encephalitis, and severe immunological disorders characterized by an accelerated hematological phase, also referred to as hemophagocytic syndrome (HS), or hemophagocytic lymphohistiocytosis (HLH). The authors report the diagnosis of GS2 in an 11-year-old girl with hypopigmentation, immunodeficiency, hepatosplenomegaly, severe neurological impairments, and fatal multiorgan failure. In this patient a diagnosis of pulmonary lymphomatoid granulomatosis (LG), an Epstein-Barr virus (EBV)-related lymphoproliferative disorder, was established from radiological and histological findings. Although EBV-related malignancies are common in immunocompromised patients, this is the first report of a diagnosis of pulmonary LG in a patient with GS2.

Entities:  

Mesh:

Year:  2011        PMID: 22111599     DOI: 10.1089/vim.2011.0034

Source DB:  PubMed          Journal:  Viral Immunol        ISSN: 0882-8245            Impact factor:   2.257


  4 in total

1.  Cutaneous and systemic granulomatosis in ataxia-telangiectasia: a clinico-pathological study.

Authors:  Aleksandra Szczawińska-Popłonyk; Katarzyna Olejniczak; Katarzyna Tąpolska-Jóźwiak; Maciej Boruczkowski; Katarzyna Jończyk-Potoczna; Jadwiga Małdyk; Anna Bręborowicz
Journal:  Postepy Dermatol Alergol       Date:  2020-11-07       Impact factor: 1.837

Review 2.  Host genetics of Epstein-Barr virus infection, latency and disease.

Authors:  Charlotte J Houldcroft; Paul Kellam
Journal:  Rev Med Virol       Date:  2014-11-27       Impact factor: 6.989

3.  Griscelli syndrome: a case report.

Authors:  Seyed Ebrahim Mansouri Nejad; Mohammad Javad Yazdan Panah; Naser Tayyebi Meibodi; Farah Ashraf Zadeh; Javad Akhondian; Mehran Beiraghi Toosi; Hossein Eslamieh
Journal:  Iran J Child Neurol       Date:  2014

4.  Bart's Syndrome Associated Corpus Callosum Agenesis and Choanal Atresia.

Authors:  Muhammad Saeed; Anwar Ul Haq; Khaqan Qadir
Journal:  Iran J Child Neurol       Date:  2014
  4 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.