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Early Human Development
journal homepage: www.elsevier.com/locate/earlhumdev
Positional plagiocephaly from structure to function: Clinical experience of
the service of pediatric osteopathy in Italy
Monica Filisettia,⁎
, Donatella Cattarellia
, Stefano Bonomia,b
a
Desenzano del Garda Hospital (ASST del Garda, Bs), Italy
b
Osteopathic Center for Children Italy (Lonato del Garda, Bs), Italy
A B S T R A C T
Objective: Aim of the study is to evaluate disorders related to positional plagiocephaly and introduce a new model of early intervention based on the osteopathic
integrated approach.
Methods: We review clinical experience of the “Program for Neurodevelopmental Follow-up and Pediatric Osteopathy”, a service dedicated to newborns at risk for
developmental disorders.
Results: We present clinical data of 310 newborns followed during first years of life. Data analysis examines perinatal history, general features and disorders that
could be related to plagiocephaly.
Conclusions: The experience confirms that plagiocephaly is not only a problem regarding the shape of the head, it involves the functions. In our Service most babies
(81%) with positional plagiocephaly showed isolated or associated disorders that had an impact on growth, behavior and development. The early intervention based
on the osteopathic integrated approach is addressed not only to the cranial shape but consider the baby as a whole, and the environment where he lives.
1. Introduction
Positional plagiocephaly is a clinical condition that refers to a
newborn with a flattening of a portion of the occipital region of the
head. The flattening of the parieto-occipital region determine adaptive
modifications too that result parallelogram shaped head [1]. A review
of recent literature suggested positional plagiocephaly is an increasing
problem. Prevalence appears to be age-dependent with a peak in the
first months of life (up to 30%) and a tendency to in the first years [2,3].
Plagiocephaly is caused by the application of external forces on the
malleable bones of the baby's skull that interferes with its intrinsically
normal, symmetric and harmonious development [4].
Positional plagiocephaly could have different pathogenesis:
Prenatal origin
Fetal plasticity and fetal mobility make the fetus sensitive to the
intrauterine molding
Some conditions could exacerbate intrauterine constraint, such as
macrosomia, multiple gestation, oligohydramnios, and maternal con-
ditions such as increased abdominal muscular tone, and anomalies of
the uterus or of the birth canal [5].
Perinatal origin
The skull of the newborn is designed to provide maximum flexibility
during the birth. At birth skull bones are not completely ossified, and
for this reason the applied forces can cause deformation [6].
The bones of the cranium accommodate the size and shape of ma-
ternal pelvis by sliding and overlapping each other at the level of the
sutures. This process is referred to as molding. It results in different
head shapes postpartum, depending on the presentation of the fetal
head and characteristics of maternal pelvis [7]. Some conditions in-
crease the risk of plagiocephaly: assisted vaginal delivery (with use of
forceps or vacuum), prolonged labor, long period of contractions,
unusual birth position [4].
Postnatal origin
The position of the infant head during the first months of life could
interfere with normal development of cranial bones. A prolonged po-
sition of the head may have adverse effects due to environmental fac-
tors such as position of the crib in the room or one sided breastfeeding.
The rapid increase of the problem has been linked to the Back to Sleep
Recommendation campaign, to avoid the risk of sudden infant death
https://doi.org/10.1016/j.earlhumdev.2020.105028
Received 14 February 2020; Received in revised form 22 March 2020; Accepted 24 March 2020
⁎
Corresponding author at: via Padre Marcolini 92, 25017 Lonato del Garda (Bs), Italy.
E-mail address: monica.filisetti@asst-garda.it (M. Filisetti).
Early Human Development 146 (2020) 105028
0378-3782/ © 2020 The Authors. Published by Elsevier B.V. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/BY-NC-ND/4.0/).
T
syndrome, by the American Academy of Pediatrics in 1992. In this re-
gard it is important to note that this policy resulted in a 40% reduction
of the sudden infant death syndrome rate [8].
Identified risk factors for positional plagiocephaly are: male sex
(2:1), premature birth, first born child, multiparity, presence of ce-
phalohematoma at birth, congenital muscular torticollis [2,4,9], ad-
vanced maternal age, ethnicity (Caucasian more than African) [3].
While macrosomia is considered a risk factor for intrauterine molding,
surprisingly the infant's length, birth weight and head circumference
did not correlate with presence of a flat area or other unusual head
shape [10].
Cranial flattening is most frequently observed on the right side,
because the right side of the occiput is more often exposed to stresses
and strains during delivery [3,11].
Positional plagiocephaly is considered a benign condition but could
lead to important consequences
Many studies suggest that positional plagiocephaly can be asso-
ciated with several short and long term disorders.
1.1. Postural and musculoskeletal disorders
Musculoskeletal disorders include a limited range of cervical and
head motion, torticollis and global postural asymmetry. Limited neck
motion may increase the possibility of persistent skull deformation
[12,13]. Other associated anomalies are: hip dysplasia, clubfeet, man-
dibular asymmetry or hypoplasia [5]. Long term consequences include
temporomandibular articulation disorders, occlusal problems and
asymmetries of the spine, including scoliosis [13,14].
1.2. Sensory disorders
Moderate to severe plagiocephaly could lead to facial asymmetry
(anterior shift of the ipsilateral forehead, ear and cheek) with asym-
metric opening of the palpebral fissures and misalignment of the eyes
and/or ears. These asymmetries could interfere with auditory proces-
sing and visual development [15].
1.3. Impact on neurological development
An increasing number of studies show that positional plagiocephaly
is a risk factor for developmental delay in some motor, sensorial and
cognitive areas of development [16–23].
Dr. V. Frymann in 1976 demonstrate that various cranial strain
pattern were observed in children with learning difficulties and in
children with visual problems [24].
Recent study on school age patients highlighted a relationship be-
tween positional plagiocephaly and the need for interventions during
primary school years and special education services including speech
therapy, occupational therapy, and physical therapy. Moreover, an as-
sociation between plagiocephaly and lower cognitive and academic
outcome is demonstrated [25,26].
This implies that children with this condition require early evalua-
tion and early intervention and should be monitored during their
growth [27,28].
The therapeutic approach is multidisciplinary and requires different
treatments such as behavioral interventions, physical therapy, manual
therapy, and helmet [1]. Osteopathy is considered a safe and effective
approach and contributes to modify the cranial shape [13,14,29].
2. Methods
This descriptive study is a review of clinical data about newborns
followed at the Service “Neurodevelopment Follow-up and Pediatric
Osteopathy” at Desenzano del Garda Hospital (Italy). The objectives
are:
- to introduce the program of osteopathic integrated approach pro-
posed on the Service
- to identify babies with positional plagiocephaly that has been fol-
lowed, review clinical data about perinatal history and identify
plagiocephaly related disorders.
- to consider the value of the program for children with positional
plagiocephaly.
The Program is a public health service dedicated to newborns at risk
for developmental disorders and offers a new model of early interven-
tion based on an osteopathic integrated approach.
The professionals who operate at the program are two medical
doctors (MF, SB) with experience in neurodevelopment problems and
pediatric rehabilitation. In addition to that they are also osteopaths
(DO) trained in traditional pediatric osteopathy who followed specific
professional trainings at the Osteopathic Center for Children (San
Diego, California) with Viola M. Frymann, DO, FAAO, an inter-
nationally recognized authority for pediatric osteopathy.
Clinical data related to medical history and pediatric and neurolo-
gical evaluation are reported in the medical records of each child.
Patients are referred to the Service as outpatients. Usually the first
appointment is about 2 weeks after the discharge from the neonatology
department (for preterm babies or babies with congenital problems) or
when the pediatrician found a disorder during the regular follow up of
babies.
Main reasons for referral to the program are: prematurity, dystocic
delivery, positional plagiocephaly, postural and orthopedic disorders
(torticollis, foot deformities), gastrointestinal functional disorders
(gastroesophageal reflux, constipation), neurological disorders (hyper-
excitability, hypotonia, hypertonia, tremors), congenital syndromes
and neurological diseases (Down Syndrome, epilepsy), others (sleep
disorders, visual problems).
2.1. Medical history
During the first appointment we record the age of the baby and we
collected information about ethnic origin, pregnancy, labor, perinatal
period, and health conditions. Moreover during the first meeting we
present to the parents the program of follow up and we introduce the
osteopathic approach.
2.2. Clinical evaluation
2.2.1. Cranial evaluation
The evaluation of the head includes the observation of the head
shape from anterior (frontal), posterior and superior view to determine
the side (right, left, bilateral) and severity of plagiocephaly. Facial
evaluation focused on eyes position, ear alignment, mandibular sym-
metry. The exam included palpation of the fontanellae and cranial su-
tures.
2.2.2. Postural assessment
The assessment of the posture consists in taking time to observe the
global natural position of the body, with attention to the head, trunk
and upper and lower limbs alignemnt.
2.2.3. Motor behavior
Motor assessment requires the observation of spontaneus motor
activity, to determine the fluency and elegance of global movements
and freedom of head and neck movements. Assessment is performed in
supine position and involves staring and visual tracking with age ap-
propriate targets (black and white objects at a distance of 20 cm in
newborns).
2.2.4. Neurological assessment
It consists of an accurate evaluation of muscle tone, reflexes,
M. Filisetti, et al. Early Human Development 146 (2020) 105028
2
neurovegetative signs (hyper excitability, tremors, postural instability)
and developmental assessment. Neurological evolutionary profile
emerge from he repeated evaluation performed at each meeting.
2.3. Intervention
The program of early intervention includes osteopathy and a spe-
cific home program to support child development according to the
principles of neurodevelopmental care.
2.3.1. Osteopathic evaluation and treatment
The osteopathic approach begins with an evaluation based on
standardized assessment of the whole musculoskeletal system and
craniosacral system and includes examination of posture and motor
patterns. A gentle perceptive palpation, appropriate to the delicate
tissues of a newborn, can detect somatic dysfunction. Somatic dys-
function, listed in the International Classification of Diseases (ICD-10
Segmental and Somatic Dysfunction), is a restriction of movement in a
part of the musculoskeletal system [30]. Childbirth frequently results in
somatic dysfunctions in cranial, cervical, lumbar, and sacral regions,
that can predispose the infant to musculoskeletal problems, such as
plagiocephaly [11,31].
Osteopathic manipulative treatment consists in very delicate ma-
nipulations and soft mobilization of the musculoskeletal structures. The
treatment is personalized, based on the somatic dysfunctions found in
each baby. The objective is the restoration of unrestricted, symmetric,
physiologic mobility of all body parts in order to improve postural
setting and movement [14,30,32]. Osteopathic approach follows the
principles of traditional osteopathy, according to the teachings of Dr.
Frymann. Any possible adverse events were reported in the medical
record of each baby.
2.3.2. Home program to promote development
During each session a program which involved parents to create a
good environment was proposed to improve their relationship and
support motor and sensory development during home activities. This is
a program carried out at home by the family day by day. The program
has been developed according to recent discoveries about the im-
portance of the experiences during the first months of life, when the
nervous system is very plastic. Evidence suggests that early intervention
programs positively influence cognitive and social development in
children at risk for developmental impairment [33–35].
The program consists in taking care of posture and of the baby and
stimulates motor skills. Some examples of intervention are: to alternate
the position of the child while is awake (left and right side, supine and
prone position, under the parent supervision); to use adequate toys that
motivate the child to rotate the head in both directions; to limit the
amount of time the infant spends in the car seat; to vary head position
during feeding, especially in case of bottle feeding.
2.3.3. Follow-up
The Program offers services covering the first years of life, the
standard program included appointments at ages of
2–3–6–12–18 months. The approach is customized, if necessary further
treatment sessions are planned. Discharge is expected when the im-
provement of the head shape is satisfactory and the baby shows normal
development. In case of developmental delay, babies are sent to more
local services for specific rehabilitative programs.
We considered newborns followed between 2011 and 2018.
Exclusions criteria are: diagnosis of craniosynosostis, congenital syn-
dromes (e.g. Down Syndrome), severe prematurity (gestational age less
than 32 w), congenital malformations, serious neurological disorders
(e.g. epilepsy, cerebral palsy).
3. Results
During 8 years of experience, the program has taken care of 530
newborns.
We identified 310 infants with positional plagiocephaly. Summary
statistics will be provided for the data.
3.1. General features
Characteristics of newborns are described in Table 1.
3.1.1. Gender
We observed a higher prevalence of male than female children with
plagiocephaly. 198 (64%) were males while 112 (36%) female, with a
relation M:F of 1.7:1.
3.1.2. Ethnic origin
236 (76%) of the families were Italian, while 74 (24%) were of
different origins, according to Italian social landscape.
The average age at the time of the first evaluation was 2 months
(with an interval between 10 days and 6 months of age). This is im-
portant to ensure an early intervention.
3.1.3. Reason for consultation
We examined the reasons why pediatricians and neurologists sent
patients to the Program. 170 babies (55%) were recommended con-
sultation because of a diagnosis of positional plagiocephaly. The re-
maining 140 babies (45%) was sent to the Program due to other reasons
(follow-up of preterm babies or dystocic delivery, minor neurological
disorders, gastrointestinal disorders). In this group the first evaluation
identified the presence of plagiocephaly. Probably the plagiocephaly in
these babies would have been identified if the problem had become
more evident, and they probably would not have received adequate
treatment in time achieve optimal results.
Table 1
Characteristics of newborns with positional plagiocephaly (N. 310).
General features
Gender Male 198 64%
Female 112 36%
Ethnic origin Italian 236 76%
Others 74 24%
Est Europe 28 38%
Marocco and
Northern Africa
20 27%
Asia 19 26%
Central and South
Africa
5 6%
South America 2 3%
Reason for
consultation
Plagiocephaly 170 55%
Others 140 45%
Prematurity 78 56%
Minor
neurological
disorders
24 17%
Gastrointestinal
disorders
14 10%
Torticollis 11 8%
Dystocia 10 7%
Sleep disturbances 3 2%
Side of
plagiocephaly
Right 201 65%
Left 88 28%
Bilateral 21 7%
M. Filisetti, et al. Early Human Development 146 (2020) 105028
3
3.1.4. Side of plagiocephaly
Most of the children had an occipito-parietal plagiocephaly on the
right side (201 babies, 65%); for 87 infants (28%) was on the left side
and in 22 (7%) it was bilateral.
3.2. Perinatal history
Information about perinatal history is described in Table 2.
3.2.1. Delivery
220 babies (71%) were born with spontaneous vaginal delivery; 53
of these (24%) had a dystocic delivery due to the following conditions:
use of vacuum assisted delivery, failure of labor to progress sponta-
neously, fracture of clavicle, fetal acidosis. The remaining 90 (29%)
babies were delivered by cesarean section. 9 cases featured a twin
pregnancy.
3.2.2. Gestational age
The number of preterm babies, born earlier than 38 weeks, was 102
(33%). We considered mild and moderate prematurity (32–38 weeks),
while newborns with severe prematurity (gestational age less than 32
w) were excluded from this study.
3.3. Clinical disorders
Clinical disorders are defined according to the interviews with
parents regarding the well-being, behavior and feeding of the child, and
according to the clinical and neurological evaluations of the babies. We
could determine that from 310 babies with positional plagiocephaly, 58
babies (19%) did not have any other disorders except plagiocephaly.
The remaining 252 babies (81%) showed isolated or associated dis-
orders that had an impact on growth, behavior and development and
could affect the well-being of the child and family. Table 3 shows
clinical disorders showed in 252 babies (81%).
3.3.1. Limited head rotation
Newborns with positional plagiocephaly showed a preferred rota-
tion of the head toward the same side as the flattening. During the
evaluation of neck mobility we found that the rotation of the head was
limited toward the side opposite the flatten occiput in 192 babies
(62%).
3.3.2. Minor neurological signs
In 56 babies (18%) minor neurological symptoms such as alterations
of muscular tone (hypo or hypertonia), delay in some areas of neuro-
development were evident.
3.3.3. Gastrointestinal disorders
Mothers of 98 babies (31%) reported the presence of gastro-
intestinal functional disorders, especially gastroesophageal reflux (62
babies) and constipation (36 babies). In some cases both disorders were
detected. Among the 62 babies with gastroesophageal reflux, 17 babies
(27%) were prescribed pharmacological therapy.
3.3.4. Other disorders
Muscular torticollis was present in 5% of babies. Other disorders
were: sleep disturbances (6%), ocular disorders (strabismus, alterations
in staring and pursuit eye movements 5%), feet anomalies (3%), re-
current otitis (1%).
3.4. Observed outcome
All families accepted the suggested follow-up program and osteo-
pathic treatment.
No child has had any adverse effect. Each child received an average
of 6 evaluations and treatment sessions (for a total of more than 1800
osteopathic treatments). The average age at discharge from the treat-
ment program was 20 months of age. Most of the babies showed a
visible improvement in general head shape, symmetry of occipital
parietal region and facial symmetry.
As far as the freedom of head movements is concerned, in most cases,
we observed a resolution of that problem after the first treatment. Among
192 babies with a limitation in head rotation, 163 babies (85%) showed a
complete rotation freedom of the head on both sides, without limitations,
during the second evaluation, after the first treatment. In the remaining
29 babies (15%) this result was obtained after the second treatment.
The evaluation of postural and motor assessment showed changes
on postural alignment and stability and a progress in motor behavior, in
terms of harmony, fluidity and symmetry. This type of evaluation was
difficult to quantify.
Gastrointestinal functions improved in all children. No child has had
any impact on weight growth and babies that received pharmacological
treatment for gastroesophageal reflux stopped the therapy in 95% of
cases, after 2–3 sessions.
Significant results concerned neurological development. The evo-
lutionary neurodevelopment profile has been described for each baby,
based on the clinical neurological evaluation. 270 babies (87%) showed
a normal developmental profile at age of 12 months, the remaining 40
babies (13%) reached that result at the age of 18 months. We report
that all babies reached a normal development profile and no child
showed delay or impairment. Further evaluations have been pro-
grammed to create developmental profiles as our population of children
progress through life.
4. Discussion
The model of early intervention offered in our Program considers
the baby as a whole. The Program offers a traditional osteopathic
Table 2
Information about perinatal history.
Perinatal history
Delivery Vaginal 220 71%
Normal 167 76%
Dystocic 53 24%
Cesarean
session
90 29%
Urgent 61 67%
Programmed 30 33%
Gestational age Term 208 66%
Preterm 102 33%
Mild (34–37 w) 49 48%
Moderate (32–34
w)
53 52%
Table 3
Clinical disorders in newborns with positional plagiocephaly.
Types of disorders
Limitation on head
rotation
192 62%
Neurological minor
disorders
56 18%
Tonus anomalies 29 9%
Motor anomalies 15 5%
Signs of developmental
delay
12 4%
Gastrointestinal disorders 98 31%
Gastroesophageal reflux 62 20%
Constipation 36 11%
Sleep disturbances 18 6%
Torticollis 17 5%
Ocular disorders 15 5%
Feet anomalies 8 3%
Recurrent otitis 3 1%
M. Filisetti, et al. Early Human Development 146 (2020) 105028
4
approach performed by professionals skilled in pediatric osteopathy,
following the model of Dr. Viola Frymann. No child enrolled in the
study had adverse effects. According to previous studies, the osteo-
pathic treatment can be considered safe for children, and appropriate in
a model of integrated medicine, especially for newborn with positional
plagiocephaly, that is one of the most common reasons for consultation
[36–39].
Small clinical observational pilot studies do suggest that there may
be benefit of osteopathic treatment when applied to children diagnosed
with positional plagiocephaly [13,40].
Our study focused on the unitary vision of the child and showed the
presence of high rates of clinical disorders (about 80%) related to po-
sitional plagiocephaly. Most of those disorders improved or resolved
with the offered intervention.
In most children (62%) the problem of the head shape was asso-
ciated with a limitation of head rotation. Head shape impacts on head
and neck movements. Freedom of head movements is important to
develop visual and sensorial abilities, interact with the environment
and with the parents [41–43]. Moreover it is a main prerequisite for a
harmonic and symmetric neurological development and for feeding.
After the first or second treatment all babies showed a normalization of
the head movement, with freedom of rotation in both directions. This
result can be considered a good support for child development.
Some babies showed disorders in muscular tone and delay in normal
stages of development. Alterations in muscular tone have an impact on
motor behavior and can limit sensorial opportunities; that could in-
terfere with normal development [44,45].
Incidence of gastrointestinal disorders is also significant.
Osteopathic treatment is appropriate to promote good posture, support
motor and self-regulation, improves and stabilizes autonomic functions.
Moreover, increased postural stability and midline orientation support
feeding, digestive and gastrointestinal function, which likely explains
the improvement of gastrointestinal function [29].
All babies reached normal neurological development, this allowed
the discharge from the Program at maximum age of 18 months.
However the study has methodological limitations. We do not have
a control group. Ethical consideration suggests that all infants with
plagiocephaly should receive adequate treatment as soon as possible.
Neurological assessment was performed using accurate clinical eva-
luation without neurological scale. We are working to reassess the 310
babies after leaving the Service for developmental assessment. A further
limitation is that 140 (45%) of babies were referred to the program
because of problems other than plagiocephaly. Therefore, our patient
group may have a higher rate of disorders besides plagiocephaly than
that which may be seen in a non-specialty clinic setting.
5. Conclusions
Pagiocephaly is not only a problem of shape, but also a problem of
function. We have to be aware of the short and long term consequences
of plagiocephaly and work in the field of prevention, to enable every
child to achieve his/her developmental potential. When we find an
asymmetrical head shape in a newborn, we must remember that this
can have an impact on all aspects of his/her life: global posture and
motor development, sensorial development, neurovegetative state and
finally this can influence the baby growth and development.
“The human body does not function in separate units, but as a harmo-
nious whole” [46].
“The baby grows as a unit: the physical, mental and emotional states and
degrees of growth cannot be considered separately, they are very inter-
dependent” [9].
During this 8 year experience in Desenzano del Garda Hospital, 310
babies with positional plagiocephaly were followed for over 1 year and
a half. Our experience confirmed the presence of clinical disorders re-
lated to plagiocephaly. We realized that an appropriate early inter-
vention can improve head shape and support child development. While
further studies are needed to quantify results, the experience we report
supports the value of the proposed intervention, based on the osteo-
pathic integrated approach.
Table of contents summary
Clinical experience encourage to consider the potential of osteo-
pathic integrated approach for babies with positional plagiocephaly.
What's known on this subject
Babies with positional plagiocephaly can show short-and-long term
disorders, and are at risk for delayed or impaired development.
What this study adds
The study confirms the presence of disorders related with positional
plagiocephaly. Osteopathic integrated approach, based on traditional
osteopathy following teaching of Dr. Frymann, could be considered a
safe and appropriate intervention.
Financial disclosure
The authors have no financial relationships relevant to this article to
disclose.
Funding source
The project was done with no specific support.
Declaration of competing interest
The authors have no conflicts of interest relevant to this article to
disclose.
Acknowledgments
The project has been developed thanks to the supervision and
teachings of Dr. Viola M. Frymann, MD, DO (1921-2016), the founder
of the “Osteopathic Center for Children (SAN DIEGO, CALIFORNIA) and
international reference point for pediatric osteopathy.
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Positional plagiocephaly from structure to function: Clinical experience of the service of pediatric osteopathy in Italy

  • 1. Contents lists available at ScienceDirect Early Human Development journal homepage: www.elsevier.com/locate/earlhumdev Positional plagiocephaly from structure to function: Clinical experience of the service of pediatric osteopathy in Italy Monica Filisettia,⁎ , Donatella Cattarellia , Stefano Bonomia,b a Desenzano del Garda Hospital (ASST del Garda, Bs), Italy b Osteopathic Center for Children Italy (Lonato del Garda, Bs), Italy A B S T R A C T Objective: Aim of the study is to evaluate disorders related to positional plagiocephaly and introduce a new model of early intervention based on the osteopathic integrated approach. Methods: We review clinical experience of the “Program for Neurodevelopmental Follow-up and Pediatric Osteopathy”, a service dedicated to newborns at risk for developmental disorders. Results: We present clinical data of 310 newborns followed during first years of life. Data analysis examines perinatal history, general features and disorders that could be related to plagiocephaly. Conclusions: The experience confirms that plagiocephaly is not only a problem regarding the shape of the head, it involves the functions. In our Service most babies (81%) with positional plagiocephaly showed isolated or associated disorders that had an impact on growth, behavior and development. The early intervention based on the osteopathic integrated approach is addressed not only to the cranial shape but consider the baby as a whole, and the environment where he lives. 1. Introduction Positional plagiocephaly is a clinical condition that refers to a newborn with a flattening of a portion of the occipital region of the head. The flattening of the parieto-occipital region determine adaptive modifications too that result parallelogram shaped head [1]. A review of recent literature suggested positional plagiocephaly is an increasing problem. Prevalence appears to be age-dependent with a peak in the first months of life (up to 30%) and a tendency to in the first years [2,3]. Plagiocephaly is caused by the application of external forces on the malleable bones of the baby's skull that interferes with its intrinsically normal, symmetric and harmonious development [4]. Positional plagiocephaly could have different pathogenesis: Prenatal origin Fetal plasticity and fetal mobility make the fetus sensitive to the intrauterine molding Some conditions could exacerbate intrauterine constraint, such as macrosomia, multiple gestation, oligohydramnios, and maternal con- ditions such as increased abdominal muscular tone, and anomalies of the uterus or of the birth canal [5]. Perinatal origin The skull of the newborn is designed to provide maximum flexibility during the birth. At birth skull bones are not completely ossified, and for this reason the applied forces can cause deformation [6]. The bones of the cranium accommodate the size and shape of ma- ternal pelvis by sliding and overlapping each other at the level of the sutures. This process is referred to as molding. It results in different head shapes postpartum, depending on the presentation of the fetal head and characteristics of maternal pelvis [7]. Some conditions in- crease the risk of plagiocephaly: assisted vaginal delivery (with use of forceps or vacuum), prolonged labor, long period of contractions, unusual birth position [4]. Postnatal origin The position of the infant head during the first months of life could interfere with normal development of cranial bones. A prolonged po- sition of the head may have adverse effects due to environmental fac- tors such as position of the crib in the room or one sided breastfeeding. The rapid increase of the problem has been linked to the Back to Sleep Recommendation campaign, to avoid the risk of sudden infant death https://doi.org/10.1016/j.earlhumdev.2020.105028 Received 14 February 2020; Received in revised form 22 March 2020; Accepted 24 March 2020 ⁎ Corresponding author at: via Padre Marcolini 92, 25017 Lonato del Garda (Bs), Italy. E-mail address: monica.filisetti@asst-garda.it (M. Filisetti). Early Human Development 146 (2020) 105028 0378-3782/ © 2020 The Authors. Published by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/BY-NC-ND/4.0/). T
  • 2. syndrome, by the American Academy of Pediatrics in 1992. In this re- gard it is important to note that this policy resulted in a 40% reduction of the sudden infant death syndrome rate [8]. Identified risk factors for positional plagiocephaly are: male sex (2:1), premature birth, first born child, multiparity, presence of ce- phalohematoma at birth, congenital muscular torticollis [2,4,9], ad- vanced maternal age, ethnicity (Caucasian more than African) [3]. While macrosomia is considered a risk factor for intrauterine molding, surprisingly the infant's length, birth weight and head circumference did not correlate with presence of a flat area or other unusual head shape [10]. Cranial flattening is most frequently observed on the right side, because the right side of the occiput is more often exposed to stresses and strains during delivery [3,11]. Positional plagiocephaly is considered a benign condition but could lead to important consequences Many studies suggest that positional plagiocephaly can be asso- ciated with several short and long term disorders. 1.1. Postural and musculoskeletal disorders Musculoskeletal disorders include a limited range of cervical and head motion, torticollis and global postural asymmetry. Limited neck motion may increase the possibility of persistent skull deformation [12,13]. Other associated anomalies are: hip dysplasia, clubfeet, man- dibular asymmetry or hypoplasia [5]. Long term consequences include temporomandibular articulation disorders, occlusal problems and asymmetries of the spine, including scoliosis [13,14]. 1.2. Sensory disorders Moderate to severe plagiocephaly could lead to facial asymmetry (anterior shift of the ipsilateral forehead, ear and cheek) with asym- metric opening of the palpebral fissures and misalignment of the eyes and/or ears. These asymmetries could interfere with auditory proces- sing and visual development [15]. 1.3. Impact on neurological development An increasing number of studies show that positional plagiocephaly is a risk factor for developmental delay in some motor, sensorial and cognitive areas of development [16–23]. Dr. V. Frymann in 1976 demonstrate that various cranial strain pattern were observed in children with learning difficulties and in children with visual problems [24]. Recent study on school age patients highlighted a relationship be- tween positional plagiocephaly and the need for interventions during primary school years and special education services including speech therapy, occupational therapy, and physical therapy. Moreover, an as- sociation between plagiocephaly and lower cognitive and academic outcome is demonstrated [25,26]. This implies that children with this condition require early evalua- tion and early intervention and should be monitored during their growth [27,28]. The therapeutic approach is multidisciplinary and requires different treatments such as behavioral interventions, physical therapy, manual therapy, and helmet [1]. Osteopathy is considered a safe and effective approach and contributes to modify the cranial shape [13,14,29]. 2. Methods This descriptive study is a review of clinical data about newborns followed at the Service “Neurodevelopment Follow-up and Pediatric Osteopathy” at Desenzano del Garda Hospital (Italy). The objectives are: - to introduce the program of osteopathic integrated approach pro- posed on the Service - to identify babies with positional plagiocephaly that has been fol- lowed, review clinical data about perinatal history and identify plagiocephaly related disorders. - to consider the value of the program for children with positional plagiocephaly. The Program is a public health service dedicated to newborns at risk for developmental disorders and offers a new model of early interven- tion based on an osteopathic integrated approach. The professionals who operate at the program are two medical doctors (MF, SB) with experience in neurodevelopment problems and pediatric rehabilitation. In addition to that they are also osteopaths (DO) trained in traditional pediatric osteopathy who followed specific professional trainings at the Osteopathic Center for Children (San Diego, California) with Viola M. Frymann, DO, FAAO, an inter- nationally recognized authority for pediatric osteopathy. Clinical data related to medical history and pediatric and neurolo- gical evaluation are reported in the medical records of each child. Patients are referred to the Service as outpatients. Usually the first appointment is about 2 weeks after the discharge from the neonatology department (for preterm babies or babies with congenital problems) or when the pediatrician found a disorder during the regular follow up of babies. Main reasons for referral to the program are: prematurity, dystocic delivery, positional plagiocephaly, postural and orthopedic disorders (torticollis, foot deformities), gastrointestinal functional disorders (gastroesophageal reflux, constipation), neurological disorders (hyper- excitability, hypotonia, hypertonia, tremors), congenital syndromes and neurological diseases (Down Syndrome, epilepsy), others (sleep disorders, visual problems). 2.1. Medical history During the first appointment we record the age of the baby and we collected information about ethnic origin, pregnancy, labor, perinatal period, and health conditions. Moreover during the first meeting we present to the parents the program of follow up and we introduce the osteopathic approach. 2.2. Clinical evaluation 2.2.1. Cranial evaluation The evaluation of the head includes the observation of the head shape from anterior (frontal), posterior and superior view to determine the side (right, left, bilateral) and severity of plagiocephaly. Facial evaluation focused on eyes position, ear alignment, mandibular sym- metry. The exam included palpation of the fontanellae and cranial su- tures. 2.2.2. Postural assessment The assessment of the posture consists in taking time to observe the global natural position of the body, with attention to the head, trunk and upper and lower limbs alignemnt. 2.2.3. Motor behavior Motor assessment requires the observation of spontaneus motor activity, to determine the fluency and elegance of global movements and freedom of head and neck movements. Assessment is performed in supine position and involves staring and visual tracking with age ap- propriate targets (black and white objects at a distance of 20 cm in newborns). 2.2.4. Neurological assessment It consists of an accurate evaluation of muscle tone, reflexes, M. Filisetti, et al. Early Human Development 146 (2020) 105028 2
  • 3. neurovegetative signs (hyper excitability, tremors, postural instability) and developmental assessment. Neurological evolutionary profile emerge from he repeated evaluation performed at each meeting. 2.3. Intervention The program of early intervention includes osteopathy and a spe- cific home program to support child development according to the principles of neurodevelopmental care. 2.3.1. Osteopathic evaluation and treatment The osteopathic approach begins with an evaluation based on standardized assessment of the whole musculoskeletal system and craniosacral system and includes examination of posture and motor patterns. A gentle perceptive palpation, appropriate to the delicate tissues of a newborn, can detect somatic dysfunction. Somatic dys- function, listed in the International Classification of Diseases (ICD-10 Segmental and Somatic Dysfunction), is a restriction of movement in a part of the musculoskeletal system [30]. Childbirth frequently results in somatic dysfunctions in cranial, cervical, lumbar, and sacral regions, that can predispose the infant to musculoskeletal problems, such as plagiocephaly [11,31]. Osteopathic manipulative treatment consists in very delicate ma- nipulations and soft mobilization of the musculoskeletal structures. The treatment is personalized, based on the somatic dysfunctions found in each baby. The objective is the restoration of unrestricted, symmetric, physiologic mobility of all body parts in order to improve postural setting and movement [14,30,32]. Osteopathic approach follows the principles of traditional osteopathy, according to the teachings of Dr. Frymann. Any possible adverse events were reported in the medical record of each baby. 2.3.2. Home program to promote development During each session a program which involved parents to create a good environment was proposed to improve their relationship and support motor and sensory development during home activities. This is a program carried out at home by the family day by day. The program has been developed according to recent discoveries about the im- portance of the experiences during the first months of life, when the nervous system is very plastic. Evidence suggests that early intervention programs positively influence cognitive and social development in children at risk for developmental impairment [33–35]. The program consists in taking care of posture and of the baby and stimulates motor skills. Some examples of intervention are: to alternate the position of the child while is awake (left and right side, supine and prone position, under the parent supervision); to use adequate toys that motivate the child to rotate the head in both directions; to limit the amount of time the infant spends in the car seat; to vary head position during feeding, especially in case of bottle feeding. 2.3.3. Follow-up The Program offers services covering the first years of life, the standard program included appointments at ages of 2–3–6–12–18 months. The approach is customized, if necessary further treatment sessions are planned. Discharge is expected when the im- provement of the head shape is satisfactory and the baby shows normal development. In case of developmental delay, babies are sent to more local services for specific rehabilitative programs. We considered newborns followed between 2011 and 2018. Exclusions criteria are: diagnosis of craniosynosostis, congenital syn- dromes (e.g. Down Syndrome), severe prematurity (gestational age less than 32 w), congenital malformations, serious neurological disorders (e.g. epilepsy, cerebral palsy). 3. Results During 8 years of experience, the program has taken care of 530 newborns. We identified 310 infants with positional plagiocephaly. Summary statistics will be provided for the data. 3.1. General features Characteristics of newborns are described in Table 1. 3.1.1. Gender We observed a higher prevalence of male than female children with plagiocephaly. 198 (64%) were males while 112 (36%) female, with a relation M:F of 1.7:1. 3.1.2. Ethnic origin 236 (76%) of the families were Italian, while 74 (24%) were of different origins, according to Italian social landscape. The average age at the time of the first evaluation was 2 months (with an interval between 10 days and 6 months of age). This is im- portant to ensure an early intervention. 3.1.3. Reason for consultation We examined the reasons why pediatricians and neurologists sent patients to the Program. 170 babies (55%) were recommended con- sultation because of a diagnosis of positional plagiocephaly. The re- maining 140 babies (45%) was sent to the Program due to other reasons (follow-up of preterm babies or dystocic delivery, minor neurological disorders, gastrointestinal disorders). In this group the first evaluation identified the presence of plagiocephaly. Probably the plagiocephaly in these babies would have been identified if the problem had become more evident, and they probably would not have received adequate treatment in time achieve optimal results. Table 1 Characteristics of newborns with positional plagiocephaly (N. 310). General features Gender Male 198 64% Female 112 36% Ethnic origin Italian 236 76% Others 74 24% Est Europe 28 38% Marocco and Northern Africa 20 27% Asia 19 26% Central and South Africa 5 6% South America 2 3% Reason for consultation Plagiocephaly 170 55% Others 140 45% Prematurity 78 56% Minor neurological disorders 24 17% Gastrointestinal disorders 14 10% Torticollis 11 8% Dystocia 10 7% Sleep disturbances 3 2% Side of plagiocephaly Right 201 65% Left 88 28% Bilateral 21 7% M. Filisetti, et al. Early Human Development 146 (2020) 105028 3
  • 4. 3.1.4. Side of plagiocephaly Most of the children had an occipito-parietal plagiocephaly on the right side (201 babies, 65%); for 87 infants (28%) was on the left side and in 22 (7%) it was bilateral. 3.2. Perinatal history Information about perinatal history is described in Table 2. 3.2.1. Delivery 220 babies (71%) were born with spontaneous vaginal delivery; 53 of these (24%) had a dystocic delivery due to the following conditions: use of vacuum assisted delivery, failure of labor to progress sponta- neously, fracture of clavicle, fetal acidosis. The remaining 90 (29%) babies were delivered by cesarean section. 9 cases featured a twin pregnancy. 3.2.2. Gestational age The number of preterm babies, born earlier than 38 weeks, was 102 (33%). We considered mild and moderate prematurity (32–38 weeks), while newborns with severe prematurity (gestational age less than 32 w) were excluded from this study. 3.3. Clinical disorders Clinical disorders are defined according to the interviews with parents regarding the well-being, behavior and feeding of the child, and according to the clinical and neurological evaluations of the babies. We could determine that from 310 babies with positional plagiocephaly, 58 babies (19%) did not have any other disorders except plagiocephaly. The remaining 252 babies (81%) showed isolated or associated dis- orders that had an impact on growth, behavior and development and could affect the well-being of the child and family. Table 3 shows clinical disorders showed in 252 babies (81%). 3.3.1. Limited head rotation Newborns with positional plagiocephaly showed a preferred rota- tion of the head toward the same side as the flattening. During the evaluation of neck mobility we found that the rotation of the head was limited toward the side opposite the flatten occiput in 192 babies (62%). 3.3.2. Minor neurological signs In 56 babies (18%) minor neurological symptoms such as alterations of muscular tone (hypo or hypertonia), delay in some areas of neuro- development were evident. 3.3.3. Gastrointestinal disorders Mothers of 98 babies (31%) reported the presence of gastro- intestinal functional disorders, especially gastroesophageal reflux (62 babies) and constipation (36 babies). In some cases both disorders were detected. Among the 62 babies with gastroesophageal reflux, 17 babies (27%) were prescribed pharmacological therapy. 3.3.4. Other disorders Muscular torticollis was present in 5% of babies. Other disorders were: sleep disturbances (6%), ocular disorders (strabismus, alterations in staring and pursuit eye movements 5%), feet anomalies (3%), re- current otitis (1%). 3.4. Observed outcome All families accepted the suggested follow-up program and osteo- pathic treatment. No child has had any adverse effect. Each child received an average of 6 evaluations and treatment sessions (for a total of more than 1800 osteopathic treatments). The average age at discharge from the treat- ment program was 20 months of age. Most of the babies showed a visible improvement in general head shape, symmetry of occipital parietal region and facial symmetry. As far as the freedom of head movements is concerned, in most cases, we observed a resolution of that problem after the first treatment. Among 192 babies with a limitation in head rotation, 163 babies (85%) showed a complete rotation freedom of the head on both sides, without limitations, during the second evaluation, after the first treatment. In the remaining 29 babies (15%) this result was obtained after the second treatment. The evaluation of postural and motor assessment showed changes on postural alignment and stability and a progress in motor behavior, in terms of harmony, fluidity and symmetry. This type of evaluation was difficult to quantify. Gastrointestinal functions improved in all children. No child has had any impact on weight growth and babies that received pharmacological treatment for gastroesophageal reflux stopped the therapy in 95% of cases, after 2–3 sessions. Significant results concerned neurological development. The evo- lutionary neurodevelopment profile has been described for each baby, based on the clinical neurological evaluation. 270 babies (87%) showed a normal developmental profile at age of 12 months, the remaining 40 babies (13%) reached that result at the age of 18 months. We report that all babies reached a normal development profile and no child showed delay or impairment. Further evaluations have been pro- grammed to create developmental profiles as our population of children progress through life. 4. Discussion The model of early intervention offered in our Program considers the baby as a whole. The Program offers a traditional osteopathic Table 2 Information about perinatal history. Perinatal history Delivery Vaginal 220 71% Normal 167 76% Dystocic 53 24% Cesarean session 90 29% Urgent 61 67% Programmed 30 33% Gestational age Term 208 66% Preterm 102 33% Mild (34–37 w) 49 48% Moderate (32–34 w) 53 52% Table 3 Clinical disorders in newborns with positional plagiocephaly. Types of disorders Limitation on head rotation 192 62% Neurological minor disorders 56 18% Tonus anomalies 29 9% Motor anomalies 15 5% Signs of developmental delay 12 4% Gastrointestinal disorders 98 31% Gastroesophageal reflux 62 20% Constipation 36 11% Sleep disturbances 18 6% Torticollis 17 5% Ocular disorders 15 5% Feet anomalies 8 3% Recurrent otitis 3 1% M. Filisetti, et al. Early Human Development 146 (2020) 105028 4
  • 5. approach performed by professionals skilled in pediatric osteopathy, following the model of Dr. Viola Frymann. No child enrolled in the study had adverse effects. According to previous studies, the osteo- pathic treatment can be considered safe for children, and appropriate in a model of integrated medicine, especially for newborn with positional plagiocephaly, that is one of the most common reasons for consultation [36–39]. Small clinical observational pilot studies do suggest that there may be benefit of osteopathic treatment when applied to children diagnosed with positional plagiocephaly [13,40]. Our study focused on the unitary vision of the child and showed the presence of high rates of clinical disorders (about 80%) related to po- sitional plagiocephaly. Most of those disorders improved or resolved with the offered intervention. In most children (62%) the problem of the head shape was asso- ciated with a limitation of head rotation. Head shape impacts on head and neck movements. Freedom of head movements is important to develop visual and sensorial abilities, interact with the environment and with the parents [41–43]. Moreover it is a main prerequisite for a harmonic and symmetric neurological development and for feeding. After the first or second treatment all babies showed a normalization of the head movement, with freedom of rotation in both directions. This result can be considered a good support for child development. Some babies showed disorders in muscular tone and delay in normal stages of development. Alterations in muscular tone have an impact on motor behavior and can limit sensorial opportunities; that could in- terfere with normal development [44,45]. Incidence of gastrointestinal disorders is also significant. Osteopathic treatment is appropriate to promote good posture, support motor and self-regulation, improves and stabilizes autonomic functions. Moreover, increased postural stability and midline orientation support feeding, digestive and gastrointestinal function, which likely explains the improvement of gastrointestinal function [29]. All babies reached normal neurological development, this allowed the discharge from the Program at maximum age of 18 months. However the study has methodological limitations. We do not have a control group. Ethical consideration suggests that all infants with plagiocephaly should receive adequate treatment as soon as possible. Neurological assessment was performed using accurate clinical eva- luation without neurological scale. We are working to reassess the 310 babies after leaving the Service for developmental assessment. A further limitation is that 140 (45%) of babies were referred to the program because of problems other than plagiocephaly. Therefore, our patient group may have a higher rate of disorders besides plagiocephaly than that which may be seen in a non-specialty clinic setting. 5. Conclusions Pagiocephaly is not only a problem of shape, but also a problem of function. We have to be aware of the short and long term consequences of plagiocephaly and work in the field of prevention, to enable every child to achieve his/her developmental potential. When we find an asymmetrical head shape in a newborn, we must remember that this can have an impact on all aspects of his/her life: global posture and motor development, sensorial development, neurovegetative state and finally this can influence the baby growth and development. “The human body does not function in separate units, but as a harmo- nious whole” [46]. “The baby grows as a unit: the physical, mental and emotional states and degrees of growth cannot be considered separately, they are very inter- dependent” [9]. During this 8 year experience in Desenzano del Garda Hospital, 310 babies with positional plagiocephaly were followed for over 1 year and a half. Our experience confirmed the presence of clinical disorders re- lated to plagiocephaly. We realized that an appropriate early inter- vention can improve head shape and support child development. While further studies are needed to quantify results, the experience we report supports the value of the proposed intervention, based on the osteo- pathic integrated approach. Table of contents summary Clinical experience encourage to consider the potential of osteo- pathic integrated approach for babies with positional plagiocephaly. What's known on this subject Babies with positional plagiocephaly can show short-and-long term disorders, and are at risk for delayed or impaired development. What this study adds The study confirms the presence of disorders related with positional plagiocephaly. 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