113 Biomed Pap Med Fac Univ Palacky Olomouc Czech Repub. 2006, 150(1):113–116.
© I. Cernakova, M. Kvasnicova, Z. Lovasova, N. Badova, J. Drabek, K. Bouchalova, R. Trojanec, M. Hajduch
A DUPLICATION dup(4)(q28q35.2) DE NOVO IN A NEWBORN
Iveta Cernakova
a
, Marta Kvasnicova
b
, Zuzana Lovasova
b
, Nora Badova
b
, Jiri Drabek
c
,
Katerina Bouchalova
c
, Radek Trojanec
c
, *Marian Hajduch
c
a
Institute of Biology and Medical Genetics, General Faculty Hospital and The 1
st
Faculty of Medicine, Charles University,
Albertov 4, 128 00 Prague 2, Czech Republic
b
Department of Clinical Genetics, Roosewelt Hospital, 975 15 Banska Bystrica, Slovak Republic
c
Laboratory of Experimental Medicine, Department of Paediatrics, Faculty of Medicine, Palacky University and University
Hospital in Olomouc, Puskinova 6, 775 20 Olomouc, Czech Republic
e-mail: hajduchm@atlas.cz
Received: March 21, 2006; Accepted: April 18, 2006
Key words: Duplication 4q syndrome/Nijmegen breakage syndrome/Cytogenetics/G-band technique/Fluorescence in situ
hybridization (FISH)/Polymerase chain reaction (PCR)
We report here a case of a newborn with hypotrophy and somatic stigmatization: microcephaly, facial dysmorphism,
heart defect and immunodeficiency syndrome. The proband’s karyotype was 46,XY,dup(4)(q28q35.2) de novo with
chromosomal breaks in 4% of metaphases. We demonstrate the usefulness of a combination of physical examination,
classical cytogenetics, FISH and PCR techniques in order to establish correct diagnosis because of overlap of some clini-
cal and cytogenetic features of Nijmegen breakage syndrome (NBS) and duplication 4q in our patient. Although FISH
technique detected translocation t(14q;21q) in 4 metaphases, deletion 657del5 in exon 6 of the NBS1 gene associated
with NBS in Slavic population was not confirmed. We compare in this report similarity of the clinical picture of our
patient, NBS cases and other patients carrying a duplication of the distal part of 4q as described in the literature.
INTRODUCTION
Duplication of the long arm of chromosome 4 has
been already described in more than 60 patients
1, 2
. Most
of them result from the meiotic segregation of a paren-
tal balanced translocation and associate with a partial
monosomy of various segments of other chromosomes.
Patients with partial trisomy 4q have variable clinical
stigmatization, which is both related to the size of dupli-
cated segment of the chromosomal arm 4q and to spe-
cific associated monosomy. A review of the literature
2
revealed approximately 20 cases of a pure duplication
of 4q spanning the region from q12 to q35. Although
simple or pure forms of duplications are less common
they serve as a basis for a better understanding of relation-
ships between chromosomal anomalies and their clinical
manifestations.
In this communication we describe clinical, cytoge-
netic, FISH and PCR findings in a male newborn with
an overlap in some clinical and cytogenetic features of
Nijmegen breakage syndrome and duplication 4q. We il-
lustrate here the usefulness of the combined views of the
clinical geneticist, the cytogeneticist and the molecular
geneticist in establishing the correct diagnosis. To our
knowledge this is the second case of the pure de novo
duplication 4q.
MATERIAL AND METHODS
Cytogenetic and FISH analysis were performed on
cultured PHA-stimulated peripheral blood lymphocytes
of the propositus and his parents. Classical cytogenetics
was carried out using G-banding. The whole chromosome
painting probes 4, 7, 14 and 21 (Vysis) and chromosome
specific telomeric probe 4q (Vysis) were used for detec-
tion of (the) suspected rearrangements by FISH.
PCR diagnosis was performed using a DNA extracted
by salting-out method from peripheral blood as described
previously
3
.
RESULTS
Clinical report
The boy was born in the 40
th
week of physiologic preg-
nancy. His birth weight was 2600 g and his length was 52
cm. The child was referred for genetic counselling at the
age of 1 month because of somatic stigmatisation.
The newborn was hypotrophic and manifested slight
hypertonic syndrome. Physical examination at the age
of 1 week revealed microcephaly with head circumfer-
ence of 31 cm (– 4 SD). The facial features included an-
timongoloid slanted palpebral fissures, epicanthic folds,
strabismus, broad nasal bridge, prominent beaked nose
with anteverted nostrils, low-set malformed ears, high
arched palate, micrognathia and a pigmented naevus in