U.S. flag

An official website of the United States government

Format

Send to:

Choose Destination

Griscelli syndrome type 1(GS1)

MedGen UID:
347092
Concept ID:
C1859194
Disease or Syndrome
Synonyms: Griscelli syndrome with neurologic impairment; Griscelli syndrome, cutaneous and neurologic type; GS1; Partial albinism and primary neurologic disease without hemophagocytic syndrome; Pigmentary dilution of the skin and hair, the presence of large clumps of pigment in hair shafts
SNOMED CT: Griscelli syndrome type 1 (1254946006); Hypopigmentation-immunodeficiency disease type 1 (1254946006)
 
Gene (location): MYO5A (15q21.2)
 
Monarch Initiative: MONDO:0008962
OMIM®: 214450
Orphanet: ORPHA79476

Definition

Griscelli syndrome type 1 (GS1) is a rare autosomal recessive disorder that results in pigmentary dilution of the skin and hair, the presence of large clumps of pigment in hair shafts, and an accumulation of melanosomes in melanocytes. In addition to the characteristic silvery-gray appearance of hair and pigmentary defects of skin, GS1 is characterized by primary neurologic deficits that usually are apparent in early infancy and include hypotonia, developmental delay, intellectual disability, and seizures. Immune impairment is not present (summary by Abd Elmaksoud et al., 2020). Bahadoran et al. (2003) characterized GS1 as comprising hypomelanosis and severe central nervous system dysfunction, corresponding to the 'dilute' phenotype in the mouse, and GS2 as comprising hypomelanosis and lymphohistiocytic hemophagocytosis, corresponding to the 'ashen' phenotype in mouse. Anikster et al. (2002), Menasche et al. (2002), Huizing et al. (2002), and Bahadoran et al. (2003, 2003) suggested that Elejalde neuroectodermal melanolysosomal syndrome (256710) in some patients and GS1 represent the same entity. Genetic Heterogeneity of Griscelli Syndrome Griscelli syndrome type 2 (GS2; 607624), characterized by hypomelanosis with immunologic impairment, is caused by mutation in the RAB27A gene (603868). Griscelli syndrome type 3 (GS3; 609227), characterized by hypomelanosis with no immunologic or neurologic manifestations, is caused by mutation in the melanophilin (MLPH; 606526) gene. [from OMIM]

Additional description

From MedlinePlus Genetics
Unusually light skin and hair coloring are the only features of Griscelli syndrome type 3. People with this form of the disorder do not have neurological abnormalities or immune system problems.

People with Griscelli syndrome type 2 have immune system abnormalities in addition to having hypopigmented skin and hair. Affected individuals are prone to recurrent infections. They also develop an immune condition called hemophagocytic lymphohistiocytosis (HLH), in which the immune system produces too many activated immune cells called T-lymphocytes and macrophages (histiocytes). Overactivity of these cells can damage organs and tissues throughout the body, causing life-threatening complications if the condition is untreated. People with Griscelli syndrome type 2 do not have the neurological abnormalities of type 1.

Griscelli syndrome type 1 involves severe problems with brain function in addition to the distinctive skin and hair coloring. Affected individuals typically have delayed development, intellectual disability, seizures, weak muscle tone (hypotonia), and eye and vision abnormalities. Another condition called Elejalde disease has many of the same signs and symptoms, and some researchers have proposed that Griscelli syndrome type 1 and Elejalde disease are actually the same disorder.

Griscelli syndrome is an inherited condition characterized by unusually light (hypopigmented) skin and light silvery-gray hair starting in infancy. Researchers have identified three types of this disorder, which are distinguished by their genetic cause and pattern of signs and symptoms.  https://medlineplus.gov/genetics/condition/griscelli-syndrome

Clinical features

From HPO
Seizure
MedGen UID:
20693
Concept ID:
C0036572
Sign or Symptom
A seizure is an intermittent abnormality of nervous system physiology characterized by a transient occurrence of signs and/or symptoms due to abnormal excessive or synchronous neuronal activity in the brain.
Global developmental delay
MedGen UID:
107838
Concept ID:
C0557874
Finding
A delay in the achievement of motor or mental milestones in the domains of development of a child, including motor skills, speech and language, cognitive skills, and social and emotional skills. This term should only be used to describe children younger than five years of age.
Intellectual disability
MedGen UID:
811461
Concept ID:
C3714756
Mental or Behavioral Dysfunction
Intellectual disability, previously referred to as mental retardation, is characterized by subnormal intellectual functioning that occurs during the developmental period. It is defined by an IQ score below 70.
Hypotonia
MedGen UID:
10133
Concept ID:
C0026827
Finding
Hypotonia is an abnormally low muscle tone (the amount of tension or resistance to movement in a muscle). Even when relaxed, muscles have a continuous and passive partial contraction which provides some resistance to passive stretching. Hypotonia thus manifests as diminished resistance to passive stretching. Hypotonia is not the same as muscle weakness, although the two conditions can co-exist.
Recurrent tonsillitis
MedGen UID:
1781351
Concept ID:
C0740402
Disease or Syndrome
Inflammation of the tonsils that has occurred repeatedly. The definition of recurrent may vary somewhat, but the criteria used recently as a measure of severity were five or more episodes of true tonsillitis per year, symptoms recurring for at least a year, and episodes that are disabling and that prevent normal functioning. In some cases recurrent tonsillitis may be related to immunosusceptibility. Evidence exists for a genetic predisposition for recurrent tonsillitis.
Hypopigmentation of the skin
MedGen UID:
102477
Concept ID:
C0162835
Disease or Syndrome
A reduction of skin color related to a decrease in melanin production and deposition.
Silver-gray hair
MedGen UID:
322949
Concept ID:
C1836576
Finding
Hypopigmented hair that appears silver-gray.
White eyelashes
MedGen UID:
332275
Concept ID:
C1836736
Finding
White color (lack of pigmentation) of the eyelashes.
White eyebrow
MedGen UID:
373165
Concept ID:
C1836737
Finding
White color (lack of pigmentation) of the eyebrow.
Accumulation of melanosomes in melanocytes
MedGen UID:
375180
Concept ID:
C1843389
Finding
Melanin pigment aggregation in hair shafts
MedGen UID:
375181
Concept ID:
C1843390
Finding
Large clumps of pigment irregularly distributed along hair shaft
MedGen UID:
870855
Concept ID:
C4025315
Finding

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVGriscelli syndrome type 1
Follow this link to review classifications for Griscelli syndrome type 1 in Orphanet.

Suggested Reading

Recent clinical studies

Etiology

Gironi LC, Zottarelli F, Savoldi G, Notarangelo LD, Basso ME, Ferrero I, Timeus F, Fagioli F, Maiuri L, Colombo E, Savoia P
Medicina (Kaunas) 2019 Mar 25;55(3) doi: 10.3390/medicina55030078. PMID: 30934652Free PMC Article
Ridaura-Sanz C, Durán-McKinster C, Ruiz-Maldonado R
Pediatr Dermatol 2018 Nov;35(6):780-783. Epub 2018 Oct 18 doi: 10.1111/pde.13624. PMID: 30338556
Amayiri N, Al-Zaben A, Ghatasheh L, Frangoul H, Hussein AA
Pediatr Transplant 2013 Jun;17(4):394-402. doi: 10.1111/petr.12081. PMID: 23692601

Diagnosis

Gupta J, Sharma P, Ameta P, Mathur DK, Gupta A
Indian J Pediatr 2023 Feb;90(2):195-196. Epub 2022 Dec 13 doi: 10.1007/s12098-022-04422-7. PMID: 36512299
Castaño-Jaramillo LM, Lugo-Reyes SO, Cruz Muñoz ME, Scheffler-Mendoza SC, Duran McKinster C, Yamazaki-Nakashimada MA, Espinosa-Padilla SE, Saez-de-Ocariz Gutierrez MDM
Scand J Immunol 2021 Jun;93(6):e13034. Epub 2021 Mar 20 doi: 10.1111/sji.13034. PMID: 33660295
Ridaura-Sanz C, Durán-McKinster C, Ruiz-Maldonado R
Pediatr Dermatol 2018 Nov;35(6):780-783. Epub 2018 Oct 18 doi: 10.1111/pde.13624. PMID: 30338556
Sahu C, Netam SS, Bhutada BR, Jaiswal SJ
Neurol India 2017 Jul-Aug;65(4):869-870. doi: 10.4103/neuroindia.NI_762_16. PMID: 28681765
Saini AG, Nagaraju S, Sahu JK, Rawat A, Vyas S, Singhi P
Neurology 2014 Apr 8;82(14):e122-3. doi: 10.1212/WNL.0000000000000288. PMID: 24711539

Therapy

Unal S, Sag E, Kuskonmaz B, Kesici S, Bayrakci B, Ayvaz DC, Tezcan I, Yalnızoglu D, Uckan D
Pediatr Blood Cancer 2014 May;61(5):928-30. Epub 2013 Oct 18 doi: 10.1002/pbc.24799. PMID: 24307660

Prognosis

Christen M, de le Roi M, Jagannathan V, Becker K, Leeb T
Genes (Basel) 2021 Sep 23;12(10) doi: 10.3390/genes12101479. PMID: 34680875Free PMC Article
Castaño-Jaramillo LM, Lugo-Reyes SO, Cruz Muñoz ME, Scheffler-Mendoza SC, Duran McKinster C, Yamazaki-Nakashimada MA, Espinosa-Padilla SE, Saez-de-Ocariz Gutierrez MDM
Scand J Immunol 2021 Jun;93(6):e13034. Epub 2021 Mar 20 doi: 10.1111/sji.13034. PMID: 33660295
Sahu C, Netam SS, Bhutada BR, Jaiswal SJ
Neurol India 2017 Jul-Aug;65(4):869-870. doi: 10.4103/neuroindia.NI_762_16. PMID: 28681765
Saini AG, Nagaraju S, Sahu JK, Rawat A, Vyas S, Singhi P
Neurology 2014 Apr 8;82(14):e122-3. doi: 10.1212/WNL.0000000000000288. PMID: 24711539
Cağdaş D, Ozgür TT, Asal GT, Tezcan I, Metin A, Lambert N, de Saint Basile G, Sanal O
Eur J Pediatr 2012 Oct;171(10):1527-31. Epub 2012 Jun 19 doi: 10.1007/s00431-012-1765-x. PMID: 22711375

Clinical prediction guides

Christen M, de le Roi M, Jagannathan V, Becker K, Leeb T
Genes (Basel) 2021 Sep 23;12(10) doi: 10.3390/genes12101479. PMID: 34680875Free PMC Article
Ridaura-Sanz C, Durán-McKinster C, Ruiz-Maldonado R
Pediatr Dermatol 2018 Nov;35(6):780-783. Epub 2018 Oct 18 doi: 10.1111/pde.13624. PMID: 30338556

Supplemental Content

Table of contents

    Clinical resources

    Practice guidelines

    • Bookshelf
      See practice and clinical guidelines in NCBI Bookshelf. The search results may include broader topics and may not capture all published guidelines. See the FAQ for details.

    Recent activity

    Your browsing activity is empty.

    Activity recording is turned off.

    Turn recording back on

    See more...