Literature DB >> 28357189

Griscelli syndrome subtype 2 with hemophagocytic lympho-histiocytosis: A case report and review of literature.

Priyanka Minocha1, Richa Choudhary1, Anika Agrawal1, Sadasivan Sitaraman1.   

Abstract

Griscelli syndrome (GS) is a rare autosomal recessive disorder resulting in pigmentary dilution of the skin and hair with variable phenotypes depending upon subtypes. Mutations in 3 distinct genes MYO5A, RAB27A, MLPH are responsible for 3 subtypes (GS1, GS2, and GS3) of GS respectively. GS subtype 2 commonly develops hemophagocytic lymphohistiocytosis (HLH) and recurrent infections due to immunodeficiency. We hereby report a 20 month old male child presenting with silvery gray hair, hypomelanosis and features of hemophagocytosis. The diagnosis of a type 2 GS was made in response to a set of clinical features: hypopigmentation of skin and the silvered reflection of the hair, absence of psychomotor retardation, the occurrence of an accelerated phase (hemophagocytosis) and, above all, a pathognomonic appearance by microscopic examination of a hair. The absence of giant granules in the nucleated cells made it possible to eliminate Chediak-Higashi syndrome, which shares a close clinical spectrum with GS. This case promotes awareness about this rare case of GS as a high indicator of suspicion about this potentially fatal condition and aids in prompt diagnosis and foresees complications. Early bone marrow transplant is the only curative treatment for GS-2.

Entities:  

Keywords:  Griscelli syndrome; chediak-higashi syndrome; hemophagocytic lymphohistiocytosis; immunodeficiency; silvery grey hairs

Year:  2017        PMID: 28357189      PMCID: PMC5359361          DOI: 10.5582/irdr.2016.01084

Source DB:  PubMed          Journal:  Intractable Rare Dis Res        ISSN: 2186-3644


  10 in total

1.  Mutations in RAB27A cause Griscelli syndrome associated with haemophagocytic syndrome.

Authors:  G Ménasché; E Pastural; J Feldmann; S Certain; F Ersoy; S Dupuis; N Wulffraat; D Bianchi; A Fischer; F Le Deist; G de Saint Basile
Journal:  Nat Genet       Date:  2000-06       Impact factor: 38.330

2.  Two genes are responsible for Griscelli syndrome at the same 15q21 locus.

Authors:  E Pastural; F Ersoy; N Yalman; N Wulffraat; E Grillo; F Ozkinay; I Tezcan; G Gediköglu; N Philippe; A Fischer; G de Saint Basile
Journal:  Genomics       Date:  2000-02-01       Impact factor: 5.736

3.  Griscelli syndrome.

Authors:  Amit Kumar Malhotra; G Bhaskar; Mousmee Nanda; Madhulika Kabra; Manoj K Singh; M Ramam
Journal:  J Am Acad Dermatol       Date:  2006-08       Impact factor: 11.527

4.  Griscelli syndrome type 2: A rare and fatal syndrome in a South Indian boy.

Authors:  R Rajyalakshmi; R N B Chakrapani
Journal:  Indian J Pathol Microbiol       Date:  2016 Jan-Mar       Impact factor: 0.740

5.  Hematopoietic stem cell transplantation for hemophagocytic lymphohistiocytosis: a retrospective analysis of data from the Italian Association of Pediatric Hematology Oncology (AIEOP).

Authors:  Simone Cesaro; Franco Locatelli; Edoardo Lanino; Fulvio Porta; Lucia Di Maio; Chiara Messina; Arcangelo Prete; Mimmo Ripaldi; Natasha Maximova; Giovanna Giorgiani; Roberto Rondelli; Maurizio Aricò; Franca Fagioli
Journal:  Haematologica       Date:  2008-09-02       Impact factor: 9.941

6.  A syndrome associating partial albinism and immunodeficiency.

Authors:  C Griscelli; A Durandy; D Guy-Grand; F Daguillard; C Herzog; M Prunieras
Journal:  Am J Med       Date:  1978-10       Impact factor: 4.965

7.  An Indian boy with griscelli syndrome type 2: case report and review of literature.

Authors:  Ankur Singh; Amit Garg; Seema Kapoor; Nita Khurana; Miriam Entesarian; Bianca Tesi
Journal:  Indian J Dermatol       Date:  2014-07       Impact factor: 1.494

8.  Polarized light microscopy of hair shafts aids in the differential diagnosis of Chédiak-Higashi and Griscelli-Prunieras syndromes.

Authors:  Neusa Y S Valente; Maria Cecilia M R Machado; Paula Boggio; Ana Cristina F Alves; Fabiane N Bergonse; Erasmo Casella; Dewton Moraes Vasconcelos; Anete S Grumach; Zilda N P de Oliveira
Journal:  Clinics (Sao Paulo)       Date:  2006-08       Impact factor: 2.365

9.  Griscelli syndrome: hemophagocytic lymphohistiocytosis with silvery hair.

Authors:  Prashant B Mahalingashetti; Manjunatha H Krishnappa; Pinnamaneni Shameem Pawan Kalyan; Ramaswamy A Subramanian; Suneeta Padhy
Journal:  J Lab Physicians       Date:  2012-07

Review 10.  Clinical, laboratory and molecular signs of immunodeficiency in patients with partial oculo-cutaneous albinism.

Authors:  Laura Dotta; Silvia Parolini; Alberto Prandini; Giovanna Tabellini; Maddalena Antolini; Stephen F Kingsmore; Raffaele Badolato
Journal:  Orphanet J Rare Dis       Date:  2013-10-17       Impact factor: 4.123

  10 in total
  1 in total

1.  Case series on Silvery Hair Syndromes: Single Center Experience.

Authors:  Sirisharani Siddiahgari; Santosh Kumar Soma; Chandravathi Penmetcha; Sandhya Vaddadi; Varshini Bandi; Lokesh Lingappa
Journal:  Indian J Dermatol       Date:  2022 Mar-Apr       Impact factor: 1.757

  1 in total

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