You are on page 1of 5

Child's Nervous System

https://doi.org/10.1007/s00381-019-04400-z

ORIGINAL ARTICLE

Safety and efficacy of independent allied healthcare professionals


in the assessment and management of plagiocephaly patients
Yahya Khormi 1 & Michelle Chiu 2,3 & Ronette Goodluck Tyndall 4 & Patricia Mortenson 5,6 & David Smith 7 &
Paul Steinbok 8,9

Received: 14 December 2018 / Accepted: 27 September 2019


# Springer-Verlag GmbH Germany, part of Springer Nature 2019

Abstract
Background The incidence of positional plagiocephaly has increased significantly over the last two decades, which has caused a service
delivery challenge for pediatric neurosurgeons. As a potential solution to the long waitlists for abnormal head shape, a plagiocephaly clinic
was established at BC Children’s Hospital (BCCH) in Vancouver, Canada. This clinic was supervised by an occupational therapist who
had been trained by a neurosurgeon to independently assess and manage patients with a referring diagnosis of positional plagiocephaly.
Objectives To determine the efficiency of the BCCH Plagiocephaly Clinic in the management of positional plagiocephaly patients and to
investigatetheclinic’sabilitytoappropriatelyidentifyandreferpatientswithcraniosynostosistopediatricneurosurgeonsforfurtherassessment.
Methods A retrospective chart review was conducted to identify patients who were assessed and managed at the BCCH
Plagiocephaly Clinic between 2008 and 2014. Data on patient demographics, head shape measurements, and treatment recom-
mendations were collected, and the BC Children’s neurosurgical database was cross-referenced to identify craniosynostosis cases
missed by the Plagiocephaly Clinic. A descriptive analysis of the clinic’s average wait times, severity of the patients’
plagiocephaly, and recommended interventions was conducted. In addition, the sensitivity and specificity of the clinic’s ability
to appropriately refer craniosynostosis patients to pediatric neurosurgery were calculated.
Results Of 1752 patients seen in the BC Children’s Plagiocephaly Clinic between 2008 and 2014, 66% of patients received counseling
about repositioning, 34% were referred for head banding, 19% were referred to physiotherapy for torticollis, and 1.4% were referred to the
BC Children’s Pediatric Neurosurgery Clinic for suspicion of craniosynostosis. The mean time from referral to first assessment by the
Plagiocephaly Clinic was 41 days, and time from referral by the plagiocephaly clinic to diagnosis of craniosynostosis by a pediatric
neurosurgeon was 8 days. Pediatric neurosurgeons requested imaging for 6 of the referred patients (25% ). The sensitivity and specificity
of the plagiocephaly clinic for referral of craniosynostosis patients to the Pediatric Neurosurgery Clinic were 100 and 99%, respectively.
Conclusion The BC Children’s Plagiocephaly Clinic is efficient and safe for the initial evaluation and treatment of patients with
positional plagiocephaly. The clinic’s model decreases wait times, appropriately manages patients with positional plagiocephaly,
screens for craniosynostosis with high sensitivity and specificity, and takes pressure off outpatient neurosurgical clinics. This
model for assessment of plagiocephaly could be considered in other medical centers.

Keywords Plagiocephaly clinic . Positional plagiocephaly . Craniosynostosis . Outpatient management

* Yahya Khormi 5
Department of Occupational Therapy, British Columbia Children’s
khormins@gmail.com Hospital, Vancouver, BC, Canada

6
1
Department of Occupational Science and Occupational Therapy,
Division of Neurosurgery, Department of Surgery, Faculty of University of British Columbia, Vancouver, Canada
Medicine, Jazan University, Gizan, Saudi Arabia
2 7
Division of Neurology, University of British Columbia, Department of Pediatrics, British Columbia Children’s Hospital,
Vancouver, BC, Canada Vancouver, BC, Canada
3
Department of Pediatrics, British Columbia Children’s Hospital, 8
Division of Pediatric Neurosurgery, University of British Columbia,
Vancouver, Canada Vancouver, BC, Canada
4
Division of Neurosurgery, Department of Surgery, University of
9
West Indies and University Hospital of West Indies, Department of Surgery, British Columbia Children’s Hospital,
Kingston, Jamaica Vancouver, BC, Canada
Childs Nerv Syst

Background been assessing patients with positional plagiocephaly who had


been referred for consideration of head banding by the pediatric
Plagiocephaly is a common reason for referral to pediatric neurosurgeons. Prior to starting the new Plagiocephaly Clinic,
neurosurgeons. It is characterized by an asymmetry of the the OT received an additional 15-min training session from one
skull and may be caused by craniosynostosis (premature fu- of the pediatric neurosurgeons (PS) at BCCH on how to differ-
sion of the cranial bone sutures) or much more commonly entiate positional plagiocephaly from craniosynostosis on clinical
may be positional. Positional plagiocephaly is defined as a examination. The OT was advised to immediately refer patients,
deformation of the skull produced by extrinsic forces acting who did not have a head shape typical of positional
on an intrinsically normal skull. The greatest amount of defor- plagiocephaly, to a pediatric neurosurgeon.
mation occurs in the first few months of life when the skull is The Plagiocephaly Clinic was initiated as a half-day clinic
most malleable and when an infant spends the majority of time held once a week, with 0.1 FTE (full-time equivalent) of the
lying on the back. Infants may also develop skull flattening in OT’s time assigned for the clinic. Referrals to the clinic came
the prenatal period due to positioning in utero [1–3]. primarily from family physicians and pediatricians, but occasion-
While positional plagiocephaly does not affect brain devel- ally from pediatric craniofacial plastic surgeons. Referrals to the
opment, parents may be concerned with the cosmetic appear- Pediatric Neurosurgery Clinic for assessment of positional
ance of their child’s head. In addition, it is essential to distin- plagiocephaly were directed to the Plagiocephaly Clinic. The
guish positional plagiocephaly from other head shape anom- number of referrals to the Plagiocephaly Clinic rose rapidly. To
alies, especially craniosynostosis which may require surgical increase the clinic’s capacity in the most cost-effective way, a
management [4–7]. A medical professional can usually distin- part-time general pediatrician joined the clinic in 2011 after un-
guish between positional plagiocephaly and craniosynostosis dergoing similar training by the pediatric neurosurgeon.
through clinical examination. Rarely, an X-ray or CT head is The Plagiocephaly Clinic continues to be run independently
required to confirm a diagnosis of craniosynostosis and comes by an OT and general pediatrician with administrative support, as
with the downsides of radiation exposure, sedation, and finan- required. Clinics are held once to twice a week in the BCCH
cial costs to the healthcare system [4, 8, 9]. Occupational Therapy Department. The clinic offers group sem-
Positional plagiocephaly usually improves with conserva- inars for families, which review the types of plagiocephaly, its
tive measures, such as appropriate caregiver counseling on causes and risk factors, repositioning strategies with a focus on
sleep positioning and tummy time, physiotherapy for treat- safe sleep, indications for head banding, and outcomes of posi-
ment of any associated torticollis, and growth over time tional plagiocephaly. In addition, for each patient, there is a thor-
[10–12]. In patients who do not respond sufficiently, a ough clinical assessment, including history of pregnancy, birth,
custom-made cranial molding orthotic may be used as a cos- and general health; review of risk factors; visual assessment of
metic treatment option if parents desire, although recent re- head shape and head measurements; and clinical screening for
ports question the long-term efficacy [13–16]. craniosynostosis, torticollis, and delayed motor development.
The reported incidence of positional plagiocephaly has in- There are a number of possible recommended intervention
creased dramatically in the last two decades [17] and a recent options based on clinical assessment and parental preferences,
Canadian study demonstrated that 47% of infants aged 7 to 12 including:
weeks have some degree of positional plagiocephaly [18]. This
increase is likely due to the Back to Sleep campaign in 1992, which (a) Counseling on repositioning, with a focus on safe sleep
recommended that infants be placed in a supine position during (b) Cranial orthotic if appropriate given the patient’s age and
sleep in order to reduce the incidence of sudden infant death syn- severity of plagiocephaly
drome(SIDS)[19–21].Otherriskfactorsfordevelopingpositional (c) Referral to physiotherapy for underlying torticollis and
plagiocephaly include multiple births, intrauterine constraint, pre- on occasion to the Infant Development Program for de-
mature birth, assisted delivery, torticollis, tummy time less than velopmental delays
three times per day, and slow motor development [1, 3, 22]. (d) Referral to a pediatric neurosurgeon for suspicion of
The number of referrals for abnormal head shape has cor- craniosynostosis
respondingly risen in the past two decades at many centers (e) Follow-up at the Plagiocephaly Clinic, usually within 4
[23, 24], including the Pediatric Neurosurgery Clinic at BC to 6 weeks
Children’s Hospital (BCCH). At BCCH, this caused a service
delivery challenge and increased the wait times for patients to
be seen by a pediatric neurosurgeon.
As a potential solution to this problem, the BC Children’s Objectives
Plagiocephaly Clinic was established in 2005, as a clinic run
independently by an occupational therapist (OT) with a specific The objectives of this study were to determine the efficiency
interest and expertise in positional plagiocephaly. This OT had of the clinic’s management of positional plagiocephaly and to
Childs Nerv Syst

investigate its ability to identify and refer patients with cranio- patients were seen once (77%) or twice (19%) by the
synostosis to pediatric neurosurgeons for further assessment. Plagiocephaly Clinic, and only 4% required three or more
appointments.
Counseling about repositioning was provided to 66% of
Methods patients. Cranial orthotic treatment was recommended in
34% of patients. Referrals were made to physiotherapy for
After institutional ethics approval was obtained, a retrospec- possible torticollis in 19% of patients.
tive chart review was conducted to identify all patients who Twenty-five patients (1.4 %) were referred by the
were independently assessed and managed at the Plagiocephaly Clinic to the BC Children’s Pediatric
Plagiocephaly Clinic between 2008 and 2014. The following Neurosurgery clinic for suspicion of craniosynostosis, with a
variables were collected: patients’ age at first clinic assess- mean wait time of 8 days. Of these patients, the pediatric
ment, date of referral and date of first clinic assessment, neurosurgeon ruled out craniosynostosis based on clinical
oblique diagonal difference (ODD) at first clinic assessment evaluation in 12 patients (50%), while 4 patients (17%) re-
(defined as the difference between the two diagonal measure- ceived head imaging because the diagnosis was not clear clin-
ments of frontozygomaticus to opposite parietal eminence), ically. Eight patients (33%) were diagnosed with craniosynos-
and recommended interventions. Since all infants in the prov- tosis, 6 based on clinical evaluation, and the other 2 after head
ince of British Columbia (BC) who are diagnosed with cra- imaging. The pediatric neurosurgeon suspected lambdoid cra-
niosynostosis are assessed at BCCH by a neurosurgeon, the niosynostosis in one additional referred patient and recom-
BCCH Pediatric Neurosurgery comprehensive database was mended a CT scan, but the family refused and there was no
cross-referenced to identify any patients who were initially follow-up.
misdiagnosed as having positional plagiocephaly by the On the other hand, there were no patients who were diag-
Plagiocephaly Clinic and later identified as having craniosyn- nosed by the Plagiocephaly Clinic as having positional
ostosis by a pediatric neurosurgeon. plagiocephaly who subsequently required a craniosynostosis
The data was reported as a descriptive analysis of the operation. Therefore, the Plagiocephaly Clinic’s sensitivity
following: and specificity for referral of craniosynostosis patients to a
pediatric neurosurgeon were 100% and 99%, respectively.
& Efficiency of the clinic as measured by average wait times
for assessment
& Characteristics of the patients seen by the clinic, including Discussion
the severity of plagiocephaly, based on their ODD and the
interventions that were recommended The BCCH Plagiocephaly Clinic is independently staffed by
& Accuracy of the clinic in identifying appropriate cases for an occupational therapist and a general pediatrician. In this
neurosurgical referral. Specifically, we determined the study, this innovative service delivery model was shown to
percentage of patients referred to the BCCH Pediatric be both efficient and safe in managing patients referred by
Neurosurgery clinic for suspicion of craniosynostosis, family physicians and pediatricians for abnormal head shape,
the percentage of referred patients for whom the neurosur- with a presumed diagnosis of positional plagiocephaly. OTs
geon requested a CT scan, and the percentage of referred and general pediatricians are grounded in knowledge of infant
patients who required surgical correction of craniosynos- development and, with a short period of additional training, in
tosis. The sensitivity and specificity of the clinic in refer- this case 15 min, are particularly well-suited to provide first-
ring craniosynostosis patients were calculated. line assessment and intervention of such infants.
The BCCH Plagiocephaly Clinic screens and manages 250
patients per year with a mean wait time of only 41 days,
compared with the typical wait time of approximately 3
Results months for non-urgent referrals to a pediatric neurosurgeon
at BCCH. This dramatic reduction in the burden to the
A total of 1752 children were seen between 2008 and 2014 at Pediatric Neurosurgery Clinic not only allows infants with
the BC Children’s Hospital Plagiocephaly Clinic by either the abnormal head shapes to be managed quickly and appropri-
occupational therapist or the general pediatrician. The charts ately but also improves access for children with other con-
before 2008 were not readily available. The mean wait time cerns that require neurosurgical assessment.
from referral to first assessment at the clinic was 41 days. The The Plagiocephaly Clinic safely screened for craniosynos-
average age at first assessment was 6.3 months and the per- tosis, which requires different management strategies from
centages of mild, moderate, and severe positional positional plagiocephaly, including surgical intervention. In
plagiocephaly were 47%, 33%, and 20 %, respectively. Most this study, the clinic referred only a minority of patients to a
Childs Nerv Syst

pediatric neurosurgeon for assessment of craniosynostosis compared with the traditional model of direct referral to pedi-
(1.4%), with a very high sensitivity and specificity since all atric neurosurgery.
patients with craniosynostosis and only few of non- For other centers considering this model of care, it should
craniosynostosis patients were referred to neurosurgery clinic. be noted that while our Plagiocephly Clinic is run indepen-
This is the first study in Canada to report the safety and dently by an OT and pediatrician, there are built in protections.
efficacy of independent allied health professionals in Firstly, while the additional training to screen for synostosis
assessing and managing plagiocephaly patients. Literature re- was brief, both these health care professionals had previous
view revealed one study in OR, USA, that discussed pediatric familiarity seeing infants with plagiocephaly and screening for
nurse practitioners as the initial evaluators of children with developmental delays. In addition, the clinic is supported by
plagiocephaly [25], which concluded that this was safe and the Division of Neurosurgery, with consult readily available.
effective with a high sensitivity (100%) but relatively low Secondly, both our clinicians are regulated professionals and
specificity (21%). Furthermore, the pediatric nurse practi- have no conflict of interest for provision of orthotic treatment.
tioners reported enhanced professional satisfaction and inte- Other regulated health care professionals with similar training
gration into a patient-centered, disease-focused subspecialty and scope of practice may also be appropriate, such as phys-
team. iotherapists and nurse practitioners. Finally, this retrospective
However, there are important differences between the review included up until 2014 and prior to further RCT and
BCCH Plagiocephaly Clinic and the one in OR. First, the prognostic evidence questioning the long-term benefit of or-
nurse practitioners had 1 year of training, while the OT and thotic treatment [13, 15]. Anecdotally, our clinic has signifi-
general pediatrician at BCCH had only a 15-min additional cantly lowered the percentage of children referred for orthotic
training session with comparable safety results. Second, the treatment since this time.
nurse practitioners had the ability to order CT imaging for
patients. A total of 45% of patients received imaging in
Oregon and only 9.8% were diagnosed with craniosynostosis, Conclusion
which raises the concern about overuse of imaging. In the
BCCH Plagiocephaly Clinic, neither the OT nor general pedi- The BCCH Plagiocephaly Clinic is efficient and safe for the
atrician ordered CT scans; instead, they referred atypical initial evaluation and treatment of patients with presumed po-
cases, suspected as having craniosynostosis, to a pediatric sitional plagiocephaly. The clinic’s model, in which allied
neurosurgeon, which resulted in a small number of patients health professionals are trained to see patients independently,
requiring imaging. Finally, referral patterns between the two could be considered in other medical centers because it de-
centers differed greatly: in OR, 48% of patients were referred creases wait times, appropriately manages patients with posi-
by a nurse practitioner to a pediatric neurosurgeon for further tional plagiocephaly, and screens for craniosynostosis with
assessment, while only 1.4% of patients seen at the BCCH high sensitivity and specificity.
Plagiocephaly Clinic were referred.
Although this is a large patient-based study with adequate Acknowledgments The authors would like to acknowledge Alexander
sample size to detect any missed cases of craniosynostosis, Cheong for his contributions during the drafting of the manuscript.
multiple limitations must be considered. The data was collect-
ed retrospectively, which raises the concern for data complete- Compliance with ethical standards
ness and correctness. Many patients were seen only once in
the Plagiocephaly Clinic with a lack of long-term follow-up. Conflict of interest On behalf of all authors, the corresponding author
states that there is no conflict of interest.
However, the list of patients seen at the Plagiocephaly Clinic
was cross-referenced with the comprehensive BCCH prospec-
tive neurosurgical database, which contains all children who
have been seen for consultation or have received surgical in- References
tervention by a pediatric neurosurgeon in the province of BC.
If Plagiocephaly Clinic patients were subsequently seen by 1. Bialocerkowski AE, Vladusic SL, Wei Ng C (2008) Prevalence,
risk factors, and natural history of positional plagiocephaly: a sys-
pediatric neurosurgeons in other provinces or countries, they
tematic review. Developmental medicine and child neurology 50:
would not have been captured in this study, but the probability 577–586
of that occurring is considered very low. 2. Joganic JL, Lynch JM, Littlefield TR, Verrelli BC (2009) Risk
Given the study’s sample size and the comprehensiveness factors associated with deformational plagiocephaly. Pediatrics
of the neurosurgical database, these limitations are unlikely to 124:e1126–e1133
3. van Vlimmeren LA, van der Graaf Y, Boere-Boonekamp MM,
invalidate the results of this study. In future studies, other L’Hoir MP, Helders PJ, Engelbert RH (2007) Risk factors for de-
potential benefits of the Plagiocephaly Clinic could be ex- formational plagiocephaly at birth and at 7 weeks of age: a prospec-
plored, including a financial analysis of this model’s costs tive cohort study. Pediatrics 119:e408–e418
Childs Nerv Syst

4. Agrawal D, Steinbok P, Cochrane DD (2007) Significance of beat- standards of care. The Cleft palate-craniofacial journal : official
en copper appearance on skull radiographs in children with isolated publication of the American Cleft Palate-Craniofacial Association
sagittal synostosis. Child’s nervous system : ChNS : official journal 53:394–403
of the International Society for Pediatric Neurosurgery 23:1467– 15. van Wijk RM, van Vlimmeren LA, Groothuis-Oudshoorn CG, Van
1470 der Ploeg CP, Ijzerman MJ, Boere-Boonekamp MM (2014) Helmet
5. Kapp-Simon KA, Speltz ML, Cunningham ML, Patel PK, Tomita therapy in infants with positional skull deformation: randomised
T (2007) Neurodevelopment of children with single suture cranio- controlled trial. Bmj 348:g2741
synostosis: a review. Child’s nervous system : ChNS : official jour- 16. Weissler EH, Sherif RD, Taub PJ (2016) An Evidence-Based
nal of the International Society for Pediatric Neurosurgery 23:269– Approach to Nonsynostotic Plagiocephaly. Plastic and reconstruc-
281 tive surgery 138:682e–689e
6. Renier D, Sainte-Rose C, Marchac D, Hirsch JF (1982) Intracranial 17. Branch LG, Kesty K, Krebs E, Wright L, Leger S, David LR (2015)
pressure in craniostenosis. Journal of neurosurgery 57:370–377 Deformational plagiocephaly and craniosynostosis: trends in diag-
7. Tamburrini G, Caldarelli M, Massimi L, Santini P, Di Rocco C nosis and treatment after the “back to sleep” campaign. The Journal
(2005) Intracranial pressure monitoring in children with single su- of craniofacial surgery 26:147–150
ture and complex craniosynostosis: a review. Child’s nervous sys- 18. Mawji A, Vollman AR, Hatfield J, McNeil DA, Sauve R (2013)
tem : ChNS : official journal of the International Society for The incidence of positional plagiocephaly: a cohort study.
Pediatric Neurosurgery 21:913–921 Pediatrics 132:298–304
8. Agrawal D, Steinbok P, Cochrane DD (2006) Diagnosis of isolated
19. Laughlin J, Luerssen TG, Dias MS, Committee on P, Ambulatory
sagittal synostosis: are radiographic studies necessary? Child’s ner-
Medicine SoNS (2011) Prevention and management of positional
vous system : ChNS : official journal of the International Society for
skull deformities in infants. Pediatrics 128:1236–1241
Pediatric Neurosurgery 22:375–378
9. Cho MJ, Borchert LL, Kane AA (2017) Diagnostic yield of routine 20. Martinez-Lage JF, Arraez Manrique C, Ruiz-Espejo AM, Lopez-
skull radiographs in infants with deformational plagiocephaly. The Guerrero AL, Almagro MJ, Galarza M (2012) Positional cranial
Cleft palate-craniofacial journal : official publication of the deformations: a clinical-epidemiological study. Anales de pediatria
American Cleft Palate-Craniofacial Association 54:497–501 77:176–183
10. Aarnivala H, Vuollo V, Harila V, Heikkinen T, Pirttiniemi P, 21. Mildred J, Beard K, Dallwitz A, Unwin J (1995) Play position is
Valkama AM (2015) Preventing deformational plagiocephaly influenced by knowledge of SIDS sleep position recommendations.
through parent guidance: a randomized, controlled trial. European Journal of paediatrics and child health 31:499–502
journal of pediatrics 174:1197–1208 22. Hutchison BL, Hutchison LA, Thompson JM, Mitchell EA (2004)
11. Hutchison BL, Stewart AW, Mitchell EA (2011) Deformational Plagiocephaly and brachycephaly in the first two years of life: a
plagiocephaly: a follow-up of head shape, parental concern and prospective cohort study. Pediatrics 114:970–980
neurodevelopment at ages 3 and 4 years. Archives of disease in 23. Aarnivala HE, Valkama AM, Pirttiniemi PM (2014) Cranial shape,
childhood 96:85–90 size and cervical motion in normal newborns. Early human devel-
12. van Vlimmeren LA, van der Graaf Y, Boere-Boonekamp MM, opment 90:425–430
L’Hoir MP, Helders PJ, Engelbert RH (2008) Effect of pediatric 24. Peitsch WK, Keefer CH, LaBrie RA, Mulliken JB (2002) Incidence
physical therapy on deformational plagiocephaly in children with of cranial asymmetry in healthy newborns. Pediatrics 110:e72
positional preference: a randomized controlled trial. Archives of 25. Kuang AA, Bergquist C, Crupi L, Oliverio M, Selden NR (2013)
pediatrics & adolescent medicine 162:712–718 Effectiveness and safety of independent pediatric nurse practi-
13. Collett BR, Leroux BG, Wallace ER, Gallagher E, Shao J, Speltz tioners in evaluating plagiocephaly. Plastic and reconstructive sur-
ML (2018) Head shape at age 36 months among children with and gery 132:414–418
without a history of positional skull deformation. Journal of neuro-
surgery Pediatrics 21:204–213
Publisher’s note Springer Nature remains neutral with regard to jurisdic-
14. Lin RS, Stevens PM, Wininger M, Castiglione CL (2016) Orthotic
tional claims in published maps and institutional affiliations.
management of deformational plagiocephaly: consensus clinical

You might also like