Literature DB >> 9486701

Partial albinism with immunodeficiency: Griscelli syndrome: report of a case and review of the literature.

A J Mancini1, L S Chan, A S Paller.   

Abstract

Partial albinism with immunodeficiency (Griscelli syndrome) is an uncommon disorder characterized by pigmentary dilution and variable immunodeficiency. Features include a silvery-gray sheen to the hair, large clumped melanosomes in hair shafts, and prominent mature melanosomes in cutaneous melanocytes with sparse pigmentation of adjacent keratinocytes. Immunologic abnormalities most often include impaired natural killer cell activity, absent delayed-type hypersensitivity, and impaired responses to mitogens. Impaired helper T cell function and hypogammaglobulinemia have also been described. The syndrome can be differentiated from Chediak-Higashi syndrome by pathognomonic light and electron microscopic features in skin and hair, and absence of consistent granulocyte abnormalities, but similarly carries a poor prognosis without bone marrow transplantation. We describe a patient with Griscelli syndrome who presented with hepatosplenomegaly, hepatitis, pancytopenia, and silvery hair in the newborn period.

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Mesh:

Year:  1998        PMID: 9486701     DOI: 10.1016/s0190-9622(98)70568-7

Source DB:  PubMed          Journal:  J Am Acad Dermatol        ISSN: 0190-9622            Impact factor:   11.527


  18 in total

Review 1.  Normal and abnormal secretion by haemopoietic cells.

Authors:  J C Stinchcombe; G M Griffiths
Journal:  Immunology       Date:  2001-05       Impact factor: 7.397

2.  Oculocutaneous albinism accompanied by minor morphologic stigmata and reduced number and function of NK cells. A new variant of NK cell defect?

Authors:  Susanne Reich; Rolf Keitzer; Reinhold E Schmidt; Roland Jacobs; Verena Varnholt; Dietke Buck; Ralf Herold; Harald Renz
Journal:  Eur J Pediatr       Date:  2008-01-17       Impact factor: 3.183

3.  Chediak-higashi syndrome presented as accelerated phase: case report and review of the literature.

Authors:  Amina Bouatay; Sondes Hizem; Amel Tej; Wided Moatamri; Lamia Boughamoura; Mondher Kortas
Journal:  Indian J Hematol Blood Transfus       Date:  2014-01-31       Impact factor: 0.900

4.  A point mutation in the cargo-binding domain of myosin V affects its interaction with multiple cargoes.

Authors:  Natasha Pashkova; Natalie L Catlett; Jennifer L Novak; Lois S Weisman
Journal:  Eukaryot Cell       Date:  2005-04

5.  Black hair follicular dysplasia in Large Münsterländer dogs: clinical, histological and ultrastructural features.

Authors:  Wolf von Bomhard; Elizabeth A Mauldin; Sheila M Schmutz; Tosso Leeb; Margret L Casal
Journal:  Vet Dermatol       Date:  2006-06       Impact factor: 1.589

Review 6.  Oculocutaneous albinism: epidemiology, genetics, skin manifestation, and psychosocial issues.

Authors:  Emily Z Ma; Albert E Zhou; Karl M Hoegler; Amor Khachemoune
Journal:  Arch Dermatol Res       Date:  2022-02-25       Impact factor: 3.017

7.  Griscelli disease: genotype-phenotype correlation in an array of clinical heterogeneity.

Authors:  Ozden Sanal; Fúgen Ersoy; Ilhan Tezcan; Ayşe Metin; Leman Yel; Gaël Ménasché; Aytemiz Gürgey; Izzet Berkel; Geneviève de Saint Basile
Journal:  J Clin Immunol       Date:  2002-07       Impact factor: 8.317

8.  Griscelli syndrome.

Authors:  Sanjeev Rath; Vivek Jain; R K Marwaha; Amita Trehan; L S Rajesh; Vijay Kumar
Journal:  Indian J Pediatr       Date:  2004-02       Impact factor: 1.967

9.  Silvery grey hair: clue to diagnose immunodeficiency.

Authors:  Ms Sahana; S Sacchidanand; R Hiremagalore; Gs Asha
Journal:  Int J Trichology       Date:  2012-04

Review 10.  Approach to the patient with recurrent infections.

Authors:  Mark Ballow
Journal:  Clin Rev Allergy Immunol       Date:  2008-04       Impact factor: 10.817

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