Keywords
cleft lip and palate - burden of care - NAM - preoperative orthodontics procedures
- cleft lip revision
Introduction
India has a population of 1.2 billion people (20% of the world’s population) and a
high birth rate. An estimated 7.9% of total births worldwide are born with a birth
defect (March of Dimes Global Report, 2006)[1] and the vast majority of these occur in low-to-middle income countries. Approximate
35,000 children are born with cleft lip and palate in India every year. Burden of
care is highly relevant in the management of cleft lip and palate children because
it demands multiple surgeries/interventions and a long-term follow-up. Increasing
number of interventions without scientific proof of long-term benefits substantially
increases the burden of care. World Health Organization (WHO) Bulletin 2014 states
that a substantial burden of care increases stress on patients and families and affects
negatively in the patient’s emotional status.[2]
Burden of Care
This is a commonly used terminology at present day in medical care. This is to titrate
and balance how much patient and family commit to their time, compromise of quality
of life, undergo multiple interventions, and take risk weighing against the benefits
the child and family receive. As responsible cleft team professionals, it is very
essential that we understand burden of care. New procedures, interventions, and surgical
procedures are introduced, with promises of better results in short term. Very often
these procedures and interventions have not been followed long enough time to weigh
the advantages and disadvantages. Nevertheless, it is likely that these interventions
and procedures will be tried on these children, probably increasing the burden of
care.
Healthcare Economics Its Effects
In India, the public health expenditure is ~1.3% of gross domestic product (GDP) from
2008 to 2015 without any augmentation from center or state government. In 2016 to
2017, it has marginally increased to 1.4% of GDP. It has been proposed to increase
to 2.5% GDP by 2025, and still, it is less than the world average of 6%.[3]
In India, the total health expenditure is estimated at 3.9% of GDP, which includes
government and private sectors. Only 30% of this total health expenditure is contributed
by the public sector, which is low in compared with Brazil (46%), China (56%), Indonesia
(39%), United States (48%), and United Kingdom (83%). This suggests that 70% of health
expenditure is borne by the individual consumer. Individual consumer pays 95% of times
from out of pocket and 5% of times covered by the insurance or other health schemes.
Malnutrition, preventive measures, and infective/chronic diseases are the focus of
the Government of India and not congenital deformities. Similarly, insurances tend
to cover in-patient expenditure for acute and chronic diseases but not visible congenital
defect, which is cleft lip and palate. This will clearly define that the treatment
for cleft lip and palate will be often out-of-pocket expenditure from parents.
Literature mentioned about “medical poverty trap” that means high out-of-pocket expenses
on illness of a patient can destroy the financial stability of family and rushing
them in poverty. The recovery from this economic burden is challenging for the families.
Approximately 100 million people around the world are poor due to healthcare costs
as per WHO estimate. It also mentioned another 150 million suffer due to catastrophic
expenditure on health. In India, many publications and studies have stated that families
have been pushed into poverty due to out-of-pocket expenses on healthcare. A household
confronted with an illness is obliged to meet varied expenses—the cost of treatment
and transport, opportunity costs for the sufferer, and caregivers and the cost of
caring, besides other routine household expenses. The process of meeting these costs
can impact household consumption expenditure and the standard of living. Households
may simultaneously adopt coping strategies such as borrowing money at high interests,
cutting back on food consumption, and sale of assets, which potentially push them
into a cascading cycle of poverty.[4]
Recently government has come out with program (Pradhan Mantri Jan Arogya Yojana) supporting
healthcare; however, eligibility is for people below poverty line and not lower middle
or middle-income group. Many charitable organizations are supporting free treatment
for cleft lip and palate children through private or governmental organization. However,
majority of them support only surgical interventions. Some charitable organizations
are trying to provide the comprehensive care with limited resources.[5] These organization definitely reached to many by providing free surgical interventions.
Often, these organization provides the funds according to number of surgical interventions.
This provides temptation of unnecessary revision surgeries subjecting children to
major psychological trauma. The burden of care is not only financial but also the
family disruption, psychological and physical stress put on family and potential risk
of complications of intervention, specially surgery. There are many studies about
post-traumatic stress disorder(PTSD) in children undergoing surgical intervention
at younger age that is as severe as war zone exposure, sexual abuse, or loss of family
member.[6] The subject of PTSD has been neglected and needs to be emphasized more when children
are subjected to surgical intervention for not life-threatening conditions like revision
of cleft lip and palate at the request of parents and family members!
Protocol, Morbidity, and Burden of Care in the Management of Cleft
Keeping in mind the long-term management of majority of children coming from middle
or lower socioeconomic class, burden of care becomes is extremely relevant. The appropriate
protocol is suggested in [Table 1].
Table 1
Protocol of essential interventions and other additional interventions increasing
the burden of care
|
Protocol intervention
|
|
|
Unnecessary interventions
|
Burden
|
|
Abbreviations: AMD, anterior maxillar distraction; NAM, naso-alveolar molding; PSOP,
pre-surgical orthognathic procedures; VPI, velo-pharyngeal incomptence.
|
|
After birth (0–3 mo)
|
Feeding advice
|
Feeding plates, NAM, PSOP
|
Multiple visits (12–20) loss of work days complication
|
|
3–5 mo
|
Cleft lip surgery
|
|
|
|
Till palate surgery
|
No regular follow-up only on demand
|
Frequent follow-up to review scars
|
Travel cost, loss of work
|
|
10–12 mo
|
Cleft palate surgery
Council for speech and dental care
|
|
|
|
2–5 y
|
Yearly follow-up for speech and dental checkup and home program (institutional intervention
when necessary)
|
Institutional speech therapy and dental checkup (institutional-based therapy)
|
Travel cost, loss of work days, and school days
|
|
3–4 y
|
Surgery for functional fistula, and preferably to combine with VPI surgery
|
–Preschool rhinoplasty
–Minor lip correction
–Nonfunctional fistula repair
|
–Psychological trauma to children
–Cost
–More scaring
–Risk of anesthesia
|
|
5–7 y
|
VPI evaluation and management (often fistula repaired combined)+ speech therapy
|
|
|
|
7–9 y
|
No orthodontics, except to prepare for ABG
|
Mixed dentition orthodontics intervention
|
Poor oral hygiene
More dental issues demand long maintenance
|
|
9–10 y
|
Alveolar bone graft
|
|
|
|
10–15 y
|
Revision lip nose correction on demand by patients
|
–Multiple cleft lip nose revisions
–Orthodontic
–Surgical intervention like AMD to correct class III
|
–Cost
–Risk of anesthesia
–High relapse
|
|
15–19 y
|
Correction of Class III occlusion
Orthodontic Treatment
Rhinoplasty on demand of patients
|
|
|
While the total burden of disease in terms of both mortality and morbidity in birth
defects including craniofacial anomalies is unknown, the services to deal with the
care of children born with cleft lip and palate have been developing rapidly in recent
years. The Clinical Standards Advisory Group study an audit that was performed to
examine standards in the provision of cleft services across the United Kingdom (CSAG
references, 2001)[7] presented problems and difficulties of management of cleft. In this study, the approximate
percentage of various morbidity and issues associated with management of cleft lip
and palate are as follows:
Speech problems—5 to 30%
Fistula—5 to 20%
Poor dental arch relationship—40 to 50%
Orthognathic surgery—25 to 40%
Poor aesthetics of the nasolabial region (lip and nose)—20 to 40%
Teasing in school—75%
Psychological and psychosocial adjustment problems—20 to 40%
WHO consensus reports in management of cleft lip and palate that the following factors
are particularly relevant to developing countries and to India. Briefly summarized
report included the following[8]:
-
The burden of care throughout India has not been quantified and remains unknown.
-
The disparity in healthcare systems and access to primary surgery means that there
is a high unmet need in many parts of India.
-
It is recognized that repair of adult clefts often requires different surgical protocols
influenced not only by age of the presenting patient but also by difficulties with
longitudinal care due to difficulties in achieving recall or follow-up.
-
Many cleft surgeons spend too much time carrying out revisions of poorly repaired
clefts of the lip and palate.
-
The objectives of primary surgery in the developing world may also differ in that
the unmet need requires strategies that produce functional repairs and address quantity
as well as quality.
A report also highlighted an important message from the developed world to clinicians
and surgeons in the developing world that “simple care can achieve equivalent or superior
outcome to complex care at less human and economic cost”[8] (Shaw et al, 1992, Severns et al, 1998).
Common Causes of Burden of Care
Common Causes of Burden of Care
Donabedian[9] stated that patient satisfaction should be the “ultimate validator of the quality
of care.” There are recognized difficulties in assessing satisfaction with respect
to definitions, measurements (reliability and validity), ceiling effects, and cultural
differences.[10] With over more than two centuries when management of cleft lip and palate has been
evolving, many scientific studies and experience of experts have put the following
procedures/interventions increasing the burden of care.
NAM and Preoperative Orthopaedics
There are many articles written, mainly about technique and short-term outcome. Taiwan
group, which followed up for long term, did not show significant improvement in nasolabial
appearance in a long term. Dutch study, which conducted randomized controlled trial
on preoperative plate molding, did not show any improvement in feeding, aesthetic
outcome, speech, and growth outcome.[11] In addition, often, the complication of nasoalveolar molding (NAM) that a newborn
baby faces ranges as high as 25 to 40%, which are though minor, never presented with
outcome results.[12] Too much aggressive forces on arches lead to permanent bending of vomer that has
never been followed up to know long-term consequences.[13] On personal communication, with Prof. David Huang (Taiwan) mentioned that role of
NAM is to support surgeon if they are not capable of handling wide cleft. This fundamentally
suggested that improvement in skills and knowledge of surgeon would reduce major burden
of care of preoperative orthopaedic.
Multiple Cleft Lip Nose Revisions
Cleft lip nose revision surgery is commonly performed when results of primary surgeries
do not produce aesthetically acceptable results.[14] Any surgical intervention is a major event for patients and families. Cleft lip
nose revision puts financial burden on families and increases burden of care.[15]
[16]
[17] This includes cost of surgical procedure and indirect cost of travel and lost productivity
for patients and their families. Furthermore, cleft lip nose revision does not improve
aesthetic outcome in all patients all the times. And, it can even become worse than
before.[18] Meta-analysis of 45 studies out of 3,034 in 2016 on cleft lip revision surgeries
in unilateral cleft lip has eye-opening observation. This study outcome shows that
the revision surgery ranges from 0 to 100% in different centers with extremely variable
outcomes![19] Quality of primary cleft lip nose repair with specific treatment protocol and threshold
of surgeon to do revision surgery contributes to a great extent to this wide variation
in incidence of revision surgeries.[7] Access to healthcare and insurance status, socioeconomic status, and health literacy
largely increases incidence of revision of surgeries. Furthermore, eagerness of surgeon
to do revision surgery and readiness of patient and family to undergo more surgeries
also plays a significant role.[18]
[19]
[20]
[21] It is obvious that more studies are necessary to understand the variation in number
of revision surgeries and its benefits against burden of care. It is well known fact
that improving outcome of primary cleft lip repair will reduce the number of secondary
revision surgeries.
The Americleft Project
[21] shows that cleft revision surgery may have improved the aesthetic outcome for individual
patient, but in a group of patients, children with added revision surgery did not
have better aesthetic outcome than those had only primary surgeries. This outcome
shows that final result of revision surgeries is highly variable across different
center. And, therefore, revision surgery does not improve aesthetic outcome in all
children and definitely increase the burden of care.
In India, there are many special compulsions and socioeconomic issues that increase
the numbers of cleft lip nose revision, for example, economical incentives, demands
from institutions and organizations of increasing number of interventions, uninformed
patients/parents, and professional falling short of the ethical practices.
Burden of Speech Care
There are nearly no scientific studies to know the burden of care in speech management.
There are too many variables, and literature is full of different types of interventions
and outcome studies without long-term outcome and burden of care. We need to understand
the effectiveness of speech therapy, especially institutional based and its limitation.
We need to respect the patients’/parents’ opinion, their compensation, and their social
milieu rather than enforcing the Western type of institutional based speech therapy
on our patients. Our institutional based work on community-based speech therapy has
made us humble to know how much parents struggle to bring the child for one session
of therapy of 30 minutes by traveling 250 km, changing 3 busses, and walking 10 km
to reach the bus. In addition, we often make parents guilty of not doing enough for
their children, forcing them to disturb family mechanics, and compromising the family
nutrition, income, and stress. Professional needs to take holistic view of patients,
family, and society and provide the best possible without disturbing the dynamics
of their social life.
Burden of Orthodontic Treatment
There are too many variables and too many views regarding additional procedures such
as NAM, mixed dentition orthodontics, mix dentition orthognathic surgeries such as
anterior maxillar distraction (AMD) and other intervention. Often, we see children
under orthodontic care from 8 to 14 years, which is prime and critical time of their
life.
Eurocleft Study[22] is a well-planned long-term outcome study from European centers where all the resources
and supports are available for the cleft children. Eurocleft Study showed that two
centers who had lowest ranking for eventual outcome had highest number of interventions
of early treatment including hospitalization for presurgical orthopaedics. Other centers
having the longest orthodontic interventions and high number of orthodontic visits
showed less favorable outcome. These centers had complex orthodontic protocol and
close follow-up from early childhood till adulthood. These experiences showed that
there is no association between treatment outcome and intensity. And, therefore, simple
protocol with minimum economic burden can provide better or equally good outcome with
less burden of care.
Another astonishing finding of Eurocleft Study was that there is inconsistency between
objectively rated outcome and satisfaction of patient and family. Though center may
have good rating of objective outcome, and this outcome is also associated with the
highest dissatisfaction expressed by the subject. This demands the need to develop
individual protocols that are patient centric and more holistic model of cleft care.[23]
The best way to understand them is from the comments of pioneers who have given their
lifelong commitment to this field.
-
Maria Mazzini
[24]: In her study she presented that children showing early skeletal discrepancy in
childhood were more likely to have final jaw surgeries. At age of 5 years, the outcome
of ANB angle identified 45% of the need for orthognathic surgery. Early or prolong
orthodontic treatment did not change growth pattern or final outcome of craniofacial
pattern.
-
Gunvor Semb
[23]: In her protocol, no presurgical or primary dentition treatment was done. Some received
a short period of orthodontic treatment just before alveolar bone graft (ABG) to facilitate
the operation. Definitive orthodontics performed in the permanent dentition. Patients
who choose to have orthognathic surgery needed a third period of orthodontics in conjunction
with this operation. Independent analysis of a 40-year archive of consecutive patients
with complete clefts from birth to age 21 confirmed the protocol to be acceptable.
The total mean duration of orthodontic treatment was 2.9 years for unilateral cleft
lip and palate (UCLP) and 3.3 years for bilateral cleft lip and palate (BCLP). This
was not very different from the duration of treatment in complex noncleft individuals.
At 21, the occlusion was good in ~70% and fair in 17%; the ABG was good in 97%, and
in 90%, orthodontic space closure without prostheses was possible. Since 1960s the
guiding principle for orthodontic cleft care in Oslo (set down by Olav Bergland) was
to minimize burden of care.
Compulsions Augmenting Burden of Care
Compulsions Augmenting Burden of Care
(Difficult to Accept, but Unconsciously Work)
-
New techniques presented with short-term benefits added without scientifically studies
of long-term benefits and consequences and burden of care.
-
Demand to produce more publications: Series of specific procedure and intervention
in specific number of patients is done to bring out publications.
-
Attraction of more incentive for professionals for providing more care.
-
Medical Council of India (MCI) compulsion of more admissions, surgeries, and interventions.
-
Nongovernmental organizations supported surgeries are easily done than avoided and
often encourage secondary surgeries.
-
Not giving information on alternatives, risks, hardship, and long-term benefits and
lack of evidences to patients and parents.
-
Minimal awareness among the patients about the available options, consequences of
intervention, and associated risks has galvanized today’s practice in doing far more
interventions than necessary.
-
Surgeon’s enthusiasm: With simple statement like “want the best for my patients” or “want my patient to look normal” increases interventions that never take out cleft stigmas. These professionals often
refuse to hear the real issues of patients’ willingness and fail to organize patient-centric
treatment.
….. All above factors do not motivate a surgeon to improve quality outcome of primary
surgeries, which will reduce many of these interventions. It is time that system is
introduced to evaluate the outcome or primary surgery and economical incentive are
planned accordingly!!!!!!! This will be revolutionary!!!!
How Can Cleft Team Reduce the Burden of Care?
How Can Cleft Team Reduce the Burden of Care?
Many cleft centers in India achieve excellent results with much less morbidity than
the figures quoted above, but there is a great deal of variability in quality of care.
How these problems could be addressed? The foremost is we start understanding and
talking about the burden of care in scientific meetings and literature.[23]
-
Creation of “centers of excellence”: The rehabilitation of patients with cleft lip and palate in the best centers in
the world has shown that tremendous benefits and the best outcome can be achieved.
-
Analyzing the outcome results: Studying own outcome, comparing with national and international studies, and improvise
for the better outcome.
-
Intercenter studies: Intercenter studies will help to improve and learn from each other
-
Protocols: Protocols with minimum interventions and adherence to the guidelines.
-
Expertise of team members: Improve individual skills knowledge and commitment of team members to their patients.
Augment the expertise of whole team, as team is as strong as the weakest member of
the team
-
Patient-centric treatment: Learn and look in to child psychology and listen to them. If not able to make judgment
on child psychological issue, please get help from child psychologist.
