Potential Biomarkers for Intellectual Disability: A Gipsy Family Study
Aureli A
1
, Del Beato T
1
, Sebastiani P
1
, Di Rocco M
1
, Marimpietri AE
2
, Graziani A
2
, Sechi E
2
and Di Loreto S
1*
1
Institute of Translational Pharmacology, National Council of Research, L’Aquila, Italy
2
Childhood and Adolescence Neuropsychiatric Clinic, University of L’Aquila, ASL n. 4, L’Aquila, Italy
*
Corresponding author: Silvia Di Loreto, Institute of Translational Pharmacology, National Council of Research, L’Aquila, Italy, Tel: +39 0862 318843; Fax: +39 0862
320750; E-mail: silvia.diloreto@cnr.it
Rec Date: September 15, 2014, Acc Date: November 13, 2014, Pub Date: November 18, 2014
Copyright: © 2014, Aureli A, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted
use, distribution, and reproduction in any medium, provided the original author and source are credited.
Abstract
Intellectual disability (ID) is frequently due to the synergic action of environmental and genetic factors. Here we
describe the particular case of three Gipsy Italian siblings, fifteen, twelve and eleven years old, with ID. Genetic
analysis stated that the parents are not consanguineous, and no human leukocyte antigen (HLA) alleles related to
autism and/or other neurological disorders are present in the 3 ID patients. Instead, a positive association of ID with
brain derived neurotrophic factor (BDNF) (Val66Met and C270T), IL6 (-174) and interleukin 1receptor antagonist
(IL1RA) mspa 11100 polymorphisms was demonstrated in these three ID patients. Moreover, serum levels of
interleukin 1beta (IL1β), interleukin 6 (IL6) were significantly different between the three patients and controls.
Keywords: Intellectual disability; Polymorphism; Cytokine; HLA;
Neurotrophic factor
Introduction
Several genes are involved in ID etiology but the mechanisms by
which these candidates regulate cognitive function remain poorly
understood [1,2]. Cytokines play a central role in CNS functions and,
some of them such as IL-1beta and IL-6 have been associated with
cognitive decline and dementia [3,4] and are released during neuronal
activity, playing a key role in regulating the strength of synaptic [5,6].
Peripheral expression of cytokines can influence both in
pathological and physiological conditions, the hippocampus-related
memory and synaptic plasticity [7-9] and there is growing evidence
that single nucleotide polymorphisms (SNPs) regulate theirs
expression [10,11]. Several polymorphisms of BDNF gene have been
described and in our previous study a positive association between ID
and two BDNF SNPs was demonstrated (Val66Met and C270T) [12].
Current research increasingly also demonstrates a role of the HLA
proteins in the functional interactions of neural cells, in CNS
development [13], and even in neurological disorders [14]. The
phenylketonuria (PKU), a defect in the hepatic enzyme phenylalanine
hydroxylase (PAH), is an inherited disorder that induces delayed
mental and social skills. A higher incidence of PKU was demonstrated
in Gypsies than in other populations [15], and two missense mutations
have been identified in the PAH gene of an Italian PKU patient [16].
Herein, we report the particular case of three Gipsy ID siblings in
which functional cytokine gene polymorphisms, serum levels of
cytokines, HLA class I and II allele polymorphisms, BDNF gene
polymorphisms and two PAH missense mutations have been studied.
Case Report
Three children belonging to a Gipsy family living in the area of the
coast of the Adriatic Sea were enrolled at the Department of
Neuropsychiatric Disorders of the Civil Hospital of L’Aquila, Italy
(Figure 1A).
Patient 1
The first patient is a female born in 1999 without complications
after a normal pregnancy. From birth, her psychomotor development
was delayed, and included feeding problems. She was able to walk at
the age of 18 months. At 5 years old she spoke only a few words and
started intensive linguistic training. A severe intellectual disability was
diagnosed (IQ 35). She is described as an easily irascible although quite
sociable child.
Patient 2
The other female, born in 2002 after an uneventful pregnancy,
developed critical anaemia after the birth and received blood
transfusions. She began walking at 15 months old and a delay in
language development and in the control of the sphincters was
reported. Severe intellectual disability (IQ 34) was noticed early on.
Some spread electric anomalies, particularly in the frontal lobe area,
were observed by her electroencephalogram. She is shy and unable to
integrate with peers.
Patient 3
The younger affected brother was born in 2003 by caesarean
section. The same psychomotor development pattern seen in his
affected sisters was present. His speech was delayed and he started to
speak at 3 years old. A mild intellectual disability was diagnosed (IQ
59). He is short-tempered and does not socialize easily.
According to Diagnostic and Statistical Manual of Mental
Disorders- IV Edition (DSM-IV) criteria, to access ID diagnosis
Wechsler Intelligence Scale for Children-III (WISC-III), a
standardized test of intellectual aptitude for children between ages 6
and 16 years, and the Vineland Adaptive Behaviour Scales (VABS)
were administrated. The WISC-III subscales are used to generate four
composite scores: verbal comprehension, perceptual reasoning,
working memory, and processing speed. The VABS is a widely used
tool for assessing an individual’s ability to care for one’s self personally
and socially. The Child Behaviour CheckList (CBCL), a parent-report
Hereditary Genetics: Current
Research
Aureli, et al., Hereditary Genet 2014, 3:3
http://dx.doi.org/10.4172/2161-1041.1000138
Case Report Open Access
Hereditary Genet
ISSN:2161-1041 HGCR, an open access journal
Volume 3 • Issue 3 • 1000138