Purpose
Adoption status has been investigated in numerous populations, identifying negative and positive effects on patient outcomes. This study compared parent-reported outcomes in adopted and nonadopted individuals with congenital upper-extremity (UE) anomalies to determine whether there was a measurable difference in parent perceptions of psychosocial health and functioning.
Methods
The Congenital Upper Limb Differences registry identified adopted individuals from a large cohort of patients. Each adopted patient was matched to 2 nonadopted patients by sex, age, and diagnosis of UE difference using the Oberg-Manske-Tonkin classification. Parent-reported scores on functional and psychosocial subjective outcomes were compared between the 2 cohorts using the Pediatric Outcome Data Collection Instrument.
Results
Ninety-one adopted pediatric patients with congenital UE differences were identified. Sixty had complete matches for comparison, and the remaining 31 patients were not able to be matched and were excluded from the analysis. Pediatric Outcome Data Collection Instrument scores for the UE function domain were worse in the adopted cohort as judged by minimally clinically important difference. In contrast, scores for Pediatric Outcome Data Collection Instrument domains of transfer and mobility, sports and physical function, pain and comfort, and happiness did not exceed the minimally clinically important difference, although the adopted cohort scores were all relatively lower than the nonadopted cohort scores.
Conclusions
In pediatric patients with UE differences, adoption status is seen to affect only the parent-reported domain of UE function. While differences by specific congenital diagnosis call for additional investigation, overall, these findings should be reassuring to families planning to adopt children with UE differences, and they should be supported in their expectations of UE functioning.
Type of study/level of evidence
Prognostic III.
Key words
An estimated 5 million Americans are adoptees, including approximately 2.5% of all children under 18 years of age.
1
Over the past 50 years, researchers have studied many aspects of the process and implications of adoption.Herman E. Adoption History in Brief. The Adoption History Project. Accessed January 3, 2019. https://pages.uoregon.edu/adoption/topics/adoptionhistbrief.htm
Classically, adoption studies have focused on disentangling the forces of nature and nurture in shaping a particular trait. Adoption studies have examined the relative impact of genetic and environmental influences on substance use conditions by comparing patterns of behavior in adopted and nonadopted children of parents with a history of alcohol misuse or smoking.
2
, 3
, 4
, 5
Adoption studies have also assessed the impact of adoption on development, intelligence, and academic achievement. Additionally, studies examining the effect of adoption on mental health have revealed increased rates of suicide, depression, and anxiety in cohorts of adopted individuals.6
, 7
, 8
These investigations have not included individuals with congenital orthopedic differences. Health outcomes, including mental health, in patients with congenital upper-extremity (UE) differences have demonstrated high self-esteem and quality of life, similar to the normal population; yet, the impact of adoption on mental health in this population has not been evaluated.
9
, 10
, 11
This study sought to determine whether there was a measurable difference between adopted and nonadopted caregivers’ perceptions of their children’s mental health and functioning using parent-reported outcomes. We hypothesized that the perceptions of adoptive patients would yield more negative scores than those of non-adoptive parents.Materials and Methods
After institutional review board approval (Washington University School of Medicine), the Congenital Upper Limb Differences (CoULD) Registry, a multicenter registry of pediatric patients seen for evaluation of an UE congenital difference, was searched to identify all adopted individuals.
9
,12
We included all adopted individuals enrolled into the registry between June 2016 and February 2019. These adopted patients were each matched to 2 nonadopted patients from approximately 1,400 children in the registry. All adopted and nonadopted patients were coded by sex and age range (2–3 years, 4–6 years, 7–10 years, 11–13 years, and 14–17 years). Using the Oberg-Manske-Tonkin classification, diagnosis categories (Table 1) were developed to group individuals and facilitate the matching process.13
Controls were not matched for severity of diagnosis. When more than 2 nonadopted patients were equal candidates for a match, a random draw was performed.Table 1Diagnosis Categories Used for Matching
| Diagnosis Category |
|---|
| Entire upper limb: proximal/distal axis |
| Entire upper limb: anterior/posterior axis |
| Entire upper limb: dorsal/ventral axis |
| Entire upper limb: unspecified axis |
| Hand plate: proximal/distal axis |
| Hand plate: anterior/posterior axis |
| Hand plate: dorsal/ventral axis |
| Hand plate: unspecified axis |
| Deformations |
| Dysplasia: whole limb hypertrophy |
| Dysplasia: partial limb hypertrophy |
| Skeletal tumorous: solitary osteochondromatosis |
| Skeletal tumorous: multiple osteochondromatosis |
| Syndromes |
Adopted and nonadopted matches were then compared using parent-reported outcomes. The Pediatric Outcomes Data Collection Instrument (PODCI) was chosen for parent-reported outcomes because this instrument has been used in children aged 2 years and older.
14
The PODCI questionnaire was developed to assess outcomes in the pediatric orthopedic patient and consists of multiple domains. The domains analyzed in this study were UE function, transfer and mobility, sports/physical function, pain and comfort, and happiness. Patients were included in analysis only if they had scorable PODCI domains. Parent-reported PODCI scores ranged from 0 to 100, with 100 being the best score possible, and lower scores indicating greater disability. The normative scores for these domains in the pediatric population with no congenital anomaly range from mid-80s to high-90s.- Daltroy L.H.
- Liang M.H.
- Fossel A.H.
- Goldberg M.J.
The POSNA pediatric musculoskeletal functional health questionnaire: report on reliability, validity, and sensitivity to change. Pediatric Outcomes Instrument Development Group. Pediatric Orthopaedic Society of North America.
J Pediatr Orthop. 1998; 18: 561-571
15
To assess a minimally clinically important difference (MCID) between PODCI scores in the 2 groups, we used published MCID values for a medium effect size (0.5) in an ambulatory pediatric cerebral palsy cohort, given that there are no published MCID values for congenital differences.16
Cerebral palsy is similar to congenital UE differences in that it is a birth defect and can have both functional limitations and psychosocial effects. MCID values for domains were: UE function 3.4, mobility 4.0, pain 11.2, happiness 9.7, and sports/physical function 4.3.16
Descriptive statistics were used to report group demographics.
Results
A total of 60 adopted patients were identified and included in the study. The adoption cohort included 27 females and 33 males with an average age of 6.3 (2–17) years. Average age of the nonadopted controls was 6.5 (2–17) years. All patients were matched to 2 nonadopted controls by gender, age range, and diagnosis category.
The mean UE PODCI score for adopted patients was 70.8 versus 76.3 for nonadopted patients, which was slightly greater than the estimated MCID of 3.4. Mean PODCI score for transfer and mobility for adopted patients was 86.8 compared to 90.6 for nonadopted patients, sports and physical function was 77.3 for adopted patients and 81.1 for nonadopted patients, pain and comfort was 88.0 for adopted patients and 89.5 for nonadopted patients, and happiness was 86.8 for adopted patients and 90.4 for nonadopted patients (Table 2). Except for UE function, all differences were less than the associated MCIDs.
Table 2Mean PODCI scores in Adopted Versus Nonadopted Patients
| Domain | Adopted | Nonadopted | Difference |
|---|---|---|---|
| Upper-extremity function | 70.8 | 76.3 | 5.5 |
| Transfer and mobility | 86.8 | 90.6 | 3.8 |
| Sports and physical functioning | 77.3 | 81.1 | 3.8 |
| Pain/comfort | 88.0 | 89.5 | 1.5 |
| Happiness | 86.8 | 90.4 | 3.6 |
∗ Greater than MCID.
Comparisons were also made between subgroups based on the anatomic region of the UE that was involved, diagnosis categories, and bilaterality (Table 3). Of note, the average PODCI scores for functional domains were lower in adopted patients with a unilateral hand plate diagnosis compared to matched nonadopted patients. Patients with amniotic band had multiple domains that revealed lower scores in the nonadopted cohort. Notably there was no diagnosis category associated with a difference between adopted and nonadopted patients that exceeded the MCID in the domain of happiness.
Table 3Mean PODCI Scores in Adopted Versus Nonadopted Patients by Diagnosis Category
| Diagnosis | Group (No. of patients) | Upper-Extremity Function (MCID = 3.4) | Transfer and Mobility (MCID = 4.0) | Sports and Physical Functioning (MCID = 4.3) | Pain/Comfort (MCID = 11.2) | Happiness (MCID = 9.7) |
|---|---|---|---|---|---|---|
| Entire upper limb, unilateral | Adopted (9) Nonadopted (18) | 81.2 77.1 | 97.3 96.2 | 90.1 89.3 | 86.9 96.4 | 85.7 93.8 |
| Entire upper limb, bilateral | Adopted (15) Nonadopted (30) | 54.5 60.5 | 64.6 76.7 | 57.9 60.9 | 86.1 88.1 | 81.7 83.3 |
| Hand plate, unilateral | Adopted (10) Nonadopted (20) | 68.3 82.1 | 88.4 97.7 | 73.1 93.3 | 88.9 93.6 | 88.1 91.6 |
| Hand plate, bilateral | Adopted (15) Nonadopted (30) | 76.2 86.0 | 92.4 94.7 | 85.1 87.0 | 95.9 84.1 | 93.2 95.0 |
| Amniotic band | Adopted (2) Nonadopted (4) | 66.5 67.0 | 96.0 86.3 | 96.0 76.3 | 100.0 90.8 | 95.0 88.8 |
| Entire upper limb and hand, unilateral | Adopted (3) Nonadopted (6) | 73.0 71.5 | 99.0 88.7 | 87.3 83.2 | 71.7 96.3 | 87.5 N/A |
| Dysplasia (Osteochrondromatosis) | Adopted (5) Nonadopted (10) | 87.6 90.4 | 93.0 99.1 | 83.6 87.6 | 75.2 81.4 | 82.0 90.0 |
N/A, not applicable.
∗ Difference greater than the MCID and the lower score.
Discussion
Little is known about the impact of adoption status on the outcomes of patients with congenital orthopedic conditions. We hypothesized that adopted children would have worse parent-reported outcomes consistent with the previous literature reports of lower scores in adopted cohorts. However, our hypothesis was not completely proven by our data.
Previous studies have characterized the impact of UE congenital hand differences on physical function, mental health, and well-being in comparison to the general population. In a cross-sectional study, Andersson et al
10
examined self-concept and psychological well-being in 92 children with congenital hand differences who had been treated with reconstructive surgery or a prosthesis. They found that those with whole hand deformities had “good” self-concept, with Piers-Harris Children’s Self-Concept Scale scores like those of children without a congenital hand difference. Interestingly, those with milder conditions had worse scores than those with severe deformities.10
In their cross-sectional study examining quality of life and functioning in pediatric patients with congenital below-the-elbow deficiency, James et al11
found that those patients with congenital below-the-elbow deficiency had similar health-related quality of life in comparison to the general population. Additionally, when comparing PODCI scores, both prosthesis wearers and non-wearers had slightly higher happiness domain scores compared to the general population. Finally, a prior study of CoULD registry patients demonstrated that patients with congenital UE differences tend to have better peer relationships and better outcomes scores for pain, anxiety, and depression.9
The current study took the investigation one step further to assess parent-perceived functioning and mental well-being in adopted and nonadopted patients in this unique cohort. Looking at the entire cohort, only UE function showed a difference between adopted and nonadopted patients that was greater than the MCID. This revealed some parent-perceived limitations to UE function, but in contrast, the perceived psychosocial well-being scores were less than the MCID, which was contrary to our hypothesis. The remaining domain scores (transfer and mobility, sports and physical function, pain and comfort, and happiness) did not show a difference between adopted and nonadopted patients greater than the estimated MCID values. Interestingly, however, when looking at the entire cohort, the adoption group scores were all lower, or worse, than the scores in the nonadopted group. While a larger sample size and more precise matches could provide more insight, it is interesting that these scores were uniformly worse. Perhaps, adoptive parents are more objective in their perception of their child than biological parents who potentially have feelings of guilt and an innate bias to minimize abnormal performance, which could have an impact on their perceptions. However, this finding did not carry over in the subgroup analysis by diagnosis (Table 3). A few of the nonadopted subgroups had scores that were worse than adopted subgroups. Only amniotic band nonadopted patients had 2 domains that were lower than adopted patients, but the differences were small. Overall, it is possible that some differences between the adopted and nonadopted cohorts within the diagnosis subgroups were missed due to insufficient study power, secondary to the limited number of adopted patients in our database and our ability to match them to nonadopted patients.
Looking at the cohort as a whole, the findings refuted the study hypothesis; differences between adopted and nonadopted cohorts in parent-reported outcomes scores were not seen as clinically relevant, except in UE function. These results suggest that adopted pediatric patients with congenital UE conditions, in general, have similar parent-perceived outcomes compared to children born into their families.
Few studies have focused on the influence of adoption status on medical outcomes, and it has been unknown how adoption status influences outcomes for congenital orthopedic conditions. The impact of adoption on surgical outcomes has been investigated in cleft palate repairs. Retrospective studies have found that international adoption is associated with delayed repair of cleft palate. Cleft palate repair is often performed in the United States before the age of 18 months. Due to delays in surgery and the challenges of a new language and social environment, adopted international patients with cleft palate tend to have poorer speech outcomes and greater need for secondary surgeries.
17
, 18
, 19
In contrast to these studies, the present study evaluated subjective outcomes without considering the age of intervention, and all data were collected prior to any surgery.Studies investigating the impact of adoption on development and mental health have demonstrated differences between adopted and nonadopted patients. Internationally adopted toddlers at age 36 months tend to have delays in communication relative to their nonadopted counterparts.
6
In adolescents, adoptees have been shown to experience more difficulties with academics, substance use, and mental health.7
A meta-analysis in 2005 reported comparable IQs with a lag in school performance and cognitive abilities for adopted compared to nonadopted children.8
A study in 2013 found a 4-fold risk of suicide in adopted versus nonadopted participants.20
We did not find evidence of mental health concerns in our cohort, although the PODCI was not a perfect tool for assessing these issues.This study has several limitations. First, despite including all adopted patients in the registry, the study is potentially underpowered to confirm or rule out true differences between groups. Therefore, we chose to use MCID to determine a clinically important difference to assist in assessment of the effect of adoption. Additionally, the use of PODCI scores reflects parent perceptions of patient functioning; therefore, it introduces potential bias in scores. Parents have been shown to underestimate their child’s physical function PODCI domain scores in comparison to self-reported scores, and they tend to underestimate pain relative to what children report.
21
However, the use of PODCI scores allowed for the inclusion of all adopted patients from the proposed study period because other patient-reported outcome measures have age limitations for use. For example, the use of Patient-Reported Outcomes Measurement Information System scores is limited to subjects aged 5 years and older and would exclude many participants from this cohort, which has an average age of 6 years old. Furthermore, these factors are mitigated by the fact that the control group was similarly assessed. Another limitation is the inability to match by severity of involvement. While this is a clear limitation, we believe that parents do not have a full understanding of severity. The actual presence of the congenital difference most likely has a greater impact on the patient and parent than the severity of the deformity as determined by physician-grading systems. Also, we do not have a method to establish, based on any pre-determined measurement, what level of severity should exclude a match and be consistent across diagnoses. Finally, since all PODCI scores were collected prior to any surgical intervention, this study did not examine the effect of surgery on patient outcomes; this is an area for future study.This study demonstrates that parent-reported outcomes of functioning and mental well-being are similar between adopted and nonadopted patients with congenital UE conditions, except for UE functioning. Our findings provide important insight for providers about the expected family perceptions of functional and psychosocial well-being in children with congenital UE differences. Adopted and nonadopted patients should not be viewed differently by providers. UE functional expectations should be reinforced and encouraged, especially with families of patients who have been adopted.
References
Herman E. Adoption History in Brief. The Adoption History Project. Accessed January 3, 2019. https://pages.uoregon.edu/adoption/topics/adoptionhistbrief.htm
- Drinking problems in adopted and nonadopted sons of alcoholics.Arch Gen Psychiatry. 1974; 31: 164-169
- Psychopathology in adopted and nonadopted daughters of alcoholics.Arch Gen Psychiatry. 1977; 34: 1005-1009
- Parent-offspring similarity for drinking: a longitudinal adoption study.Behav Genet. 2014; 44: 620-628
- Parental smoking and adolescent problem behavior: an adoption study of general and specific effects.Am J Psychiatry. 2008; 165: 1338-1344
- Similarities and differences between internationally adopted and nonadopted children in their toddler years: outcomes from a longitudinal study.Am J Orthopsychiatry. 2014; 84: 397-408
- Comparisons of adopted and nonadopted adolescents in a large, nationally representative sample.Child Dev. 2000; 71: 1458-1473
- Adoption and cognitive development: a meta-analytic comparison of adopted and nonadopted children’s IQ and school performance.Psychol Bull. 2005; 131: 301-316
- Functional impact of congenital hand differences: early results from the congenital upper limb differences (CoULD) registry.J Hand Surg Am. 2018; 43: 321-330
- Children with surgically corrected hand deformities and upper limb deficiencies: self-concept and psychological well-being.J Hand Surg Eur Vol. 2011; 36: 795-801
- Impact of prostheses on function and quality of life for children with unilateral congenital below-the-elbow deficiency.J Bone Joint Surg Am. 2006; 88: 2356-2365
- Intraobserver and interobserver reliability of the Oberg-Manske-Tonkin (OMT) classification: establishing a registry on congenital upper limb differences.J Pediatr Orthop. 2018; 38: 69-74
- Developmental biology and classification of congenital anomalies of the hand and upper extremity.J Hand Surg Am. 2010; 35: 2066-2076
- The POSNA pediatric musculoskeletal functional health questionnaire: report on reliability, validity, and sensitivity to change. Pediatric Outcomes Instrument Development Group. Pediatric Orthopaedic Society of North America.J Pediatr Orthop. 1998; 18: 561-571
- The Pediatric Orthopaedic Society of North America pediatric orthopaedic functional health questionnaire: an analysis of normals.J Pediatr Orthop. 2001; 21: 619-621
- Outcome tools used for ambulatory children with cerebral palsy: responsiveness and minimum clinically important differences.Dev Med Child Neurol. 2008; 50: 918-925
- Velopharyngeal insufficiency rates after delayed cleft palate repair: lessons learned from internationally adopted patients.Ann Plast Surg. 2015; 75: 302-305
- International adoptees with cleft lip and/or palate: lessons learned from a growing population.Ann Plast Surg. 2016; 77: 377-382
- Speech and surgical outcomes in children with Veau types III and IV cleft palate: a comparison of internationally adopted and nonadopted children.Cleft Palate Craniofac J. 2018; 55: 396-404
- Risk of suicide attempt in adopted and nonadopted offspring.Pediatrics. 2013; 132: 639-646
- Comparison of self-reports and parent proxy-reports of function and quality of life of children with below-the-elbow deficiency.J Bone Joint Surg Am. 2009; 91: 2852-2859
Article info
Publication history
Published online: May 24, 2021
Accepted:
April 15,
2021
Received:
April 6,
2020
Footnotes
No benefits in any form have been received or will be received related directly or indirectly to the subject of this article.
Identification
Copyright
© 2021 by the American Society for Surgery of the Hand. All rights reserved.
