Karaağaç AT, Yildirim AI (2016) A Type 3 Klippel Feil Syndrome Case With Atrial Septal Defect and Atrial Septal Aneurysm. Int J Pediat Health Care Adv. 3(1), 13-16.
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International Journal of Pediatric Health Care & Advancements (IJPA)
ISSN 2572-7354
A Type 3 Klippel Feil Syndrome Case With Atrial Septal Defect and Atrial Septal Aneurysm
Case Study
Karaağaç AT
*
, Yildirim AI
Kartal Koşuyolu Research and Training Hospital, Pediatry, Istanbul, Turkey.
Introduction
KFS, irst described by Maurice Klippel and Andre Feil in 1912,
is believed to result from faulty segmentation of the long axis of
developing embryo dring the second to eighth weeks of gestation
[1]. Besides its classical triad, a variety of clinical symptoms such
as scoliosis, rib anomalies, raised scapula (Sprengel’s deformity),
facial asymmetry, cardiac, auditory or urinary anomalies may
accompany KFS [1, 2]. Its incidence was reported as 0.2-0.7 cases
per 1000 people [3].
Case Presentation
Ş.Y, 7-year-old female was referred to our hospital for cardiac
surgery. She was born with a ceserian section, 2700 gr in weight
and 46 cm. in height. She remained in the neonatal intensive care
unit for 10 days due to the respiratory dificulty. Her mother and
father were both 28 years old and 3
rd
degree relatives. They had
no physical abnormality or history of any chronical illness. The
gestational follow up of the mother was normal. She had no
history of cigarette smoking, drug or alcohol use.
On the physical examination in our inpatient clinic, Ş.Y had
facial asymmetry, prominent forehead and occiput, low posterior
hairline, short tilted neck, limited neck motion, scoliosis, kyphosis
and a raised scapula (Figure 1). Her weight was 15 kg(<3
rd
percentile) and height was 105 cm. (3
rd
percentile). There was
a concavity on the left thoracic region of her chest (Figure 2).
Her respiratory sounds were normal and cardiac auscultation
revealed 2/6 systolic murmur on the pulmonic area with 86 heart
beats per minute. Her oxygen saturation was 96% in the room
air. The rest of the systemic examination was normal. On her
laboratory test results there was no abnormality except for a
moderate degree of anemia with a hemoglobin level of 9 gr/dl
and hematocrit (Htc) %26. Her 3 dimensional cervical computed
tomography (CT) showed multiple hemivertebrae anomalies,
fusion of cervical vertebrae and disruption in the atlantoaxial
junction (Figure 3). Spinal magnetic resonance imaging (MRI)
showed multiple fusion anomalies in the cervical and upper
thoracic vertebrae (Figure 4). The plain radiography of the chest
revealed rib fusion anomaly on the left thoracic region (Figure 5).
Echocardiographic examination demonstrated atrial septal defect,
atrial septal aneurysm, tricuspid regurgitation and dilatation in the
right heart chambers. Abdominal ultrasonography was normal
except for a mild hepatomegaly. Fluorescence in situ hybridization
(FISH) analysis performed by D22S75 (Aquarius/Cytocell) probe
showed no chromosome abnormality in our case and her parents
and the karyotype analysis was normal (46,XX).
Abstract
Klippel Feil syndrome (KFS) is a rare congenital disorder characterized by short neck due to abnormal fusion of cervical
vertebrae, limited head and neck motion and low posterior hairline. We present a rare case of type 3 KFS with the fusion
of cervical and thoracic vertebrae, cervical hemivertebrae anomaly, Sprengel’s deformity, rib fusion anomaly, accompanied
by atrial septal defect/septal aneurysm, thereby reviewing the literature about this syndrome.
Keywords: Cervical Vertebrae; Fusion Anomaly; Cardiac Defect; Klippel-Feil Syndrome.
*Corresponding Author:
Aysu Türkmen Karaağaç,
Kartal Koşuyolu Research and Training Hospital, Denizer Cad, Cevizli kavşağı, No:2, 34846, Kartal, Istanbul, Turkey.
Tel: 0(216)5001500-1101
E-mail: aysukaraagac@gmail.com
Received: January 20, 2016
Accepted: January 20, 2016
Published: January 22, 2016
Citation: Karaağaç AT, Yildirim AI (2016) A Type 3 Klippel Feil Syndrome Case With Atrial Septal Defect and Atrial Septal Aneurysm. Int J Pediat Health Care Adv. 3(1), 13-16.
doi: http://dx.doi.org/10.19070/2572-7354-160005
Copyright: Karaağaç AT
©
2016. 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.