247
major malformations and associated
medical problems of each infant are
summarized in the Table. Seven pa-
tients had CHD. Two had anatomic
defects of the palate. Four patients had
neither congenital heart disease nor
cleft palate (33%) (cases 2, 3, 10, and
12). Six patients required evaluation
for cyanotic episodes as newborns
(cases 1, 2, 5, 8, 10, and 12). In 2 cases,
cyanosis was attributed to CHD. Two
patients were evaluated for unex-
plained single cyanotic episodes that
occurred at home. Two patients had
mild upper airway obstruction as
demonstrated by polysomnography.
One patient had chronic lung disease
secondary to prolonged intubation and
complications of tracheal web, and one
had observed obstructive apnea in the
perinatal period secondary to micro-
gnathia (Pierre Robin sequence).
Respiratory illnesses were common.
Six patients (cases 2, 5, 6, 7, 9, and 10)
required hospitalization for presumed
viral respiratory illnesses at ages 3
weeks to 10 months. Four patients had
intermittent wheezing with onset from
5 to 10 months (cases 2, 5, 7, and 10).
Feeding difficulties and nutritional
status led to interventions in 9 patients
(cases 1, 2, 4, 5, 6, 7, 9, 10, and 12).
Most of the feeding interventions were
initiated in the perinatal period and
managed adequately in an outpatient
Velocardiofacial syndrome is a domi-
nantly inherited contiguous gene syn-
drome associated with congenital
heart defects, palatal abnormalities,
and learning disorders.
1
The incidence
of this disorder is estimated at 1/5000
births.
2
Molecular analysis with fluo-
rescence in situ hybridization or poly-
merase chain reaction reveals a mi-
crodeletion of chromosome 22q11.2 in
the majority of cases. With the advent
of sensitive and reliable diagnostic
testing, a greater number of patients
are being identified in infancy. We ret-
rospectively reviewed the medical
records of patients diagnosed with
VCFS during infancy and discovered
Increased need for medical interventions in infants
with velocardiofacial (deletion 22q11) syndrome
Robert J. Hopkin, MD, Elizabeth K. Schorry, MD, Mary Bofinger, MD, and Howard M. Saal, MD
a surprisingly high need for medical in-
terventions and hospitalization.
METHODS
A database of all patients seen in the
Division of Human Genetics at Chil-
dren’s Hospital Medical Center was
searched for patients diagnosed with
VCFS before age 1 year and confirmed
by deletion 22q11.2. Specific data
identified for each patient included age
at diagnosis, range and extent of med-
ical problems, and number of hospital-
izations during the first year of life.
RESULTS
Fluorescence in situ hybridization–
proven deletion 22q11.2 was identified
in 45 patients; 12 of these patients were
diagnosed before the age of 1 year. Age
at diagnosis ranged from birth to 9
months, and 6 patients were diagnosed
in the first postpartum week. The
From the Division of Human Genetics and the Cranio-
facial Center, Children’s Hospital Medical Center,
Cincinnati, Ohio.
Submitted for publication Apr 19, 1999; revi-
sions received Sept 29, 1999, and Mar 28,
2000; accepted Apr 19, 2000.
Reprint requests: Robert J. Hopkin, MD,
Children’s Hospital Medical Center, Division
of Human Genetics, 3333 Burnet Ave,
Cincinnati OH 45229.
Copyright © 2000 by Mosby, Inc
0022-3476/2000/$12.00 + 0 9/22/108272
doi:10.1067/mpd.2000.108272
CHD Congenital heart defects
VCFS Velocardiofacial syndrome
Ten of 12 patients diagnosed with deletion 22q11.2 in infancy required a
total of 26 hospitalizations during their first year of life. After heart disease,
feeding and respiratory problems were the most frequent reasons for inter-
vention. (J Pediatr 2000;137:247-9)
See related articles, p. 145
and p. 158.
CLINICAL AND
LABORATORY
OBSERVATIONS