Exploration of Caregiver Interrater Agreement and Test-Retest Reliability on the Infant Cleft Observer Outcomes (iCOO).

Caregiver and observer-reported measures are frequently used as outcomes for research on infants and young children who are unable to report on their own health. Our team developed the Infant with Clefts Observation Outcomes Instrument (iCOO) for infants with cleft lip with or without cleft palate. This exploratory study compared test-retest and interrater reliabilities to inform whether differences in caregiver perspective might affect the iCOO.This study is a secondary analysis comparing caregiver interrater agreement to test-retest reliability. Twenty-five pairs of caregivers completed the iCOO before surgery, 1 week later for test-retest reliability, 2 days after surgery, and 2 months after surgery. Reliability was assessed using intraclass correlations (ICCs) and t-tests were used to compare ratings between caregivers.Infants had cleft lip (28%) or cleft lip and palate (72%). Primary caregivers were predominantly mothers (92%) and secondary caregivers were predominantly fathers (80%). Test-retest reliability met psychometric standards for most items on the iCOO (81%-86% of items). Caregiver agreement on the iCOO items was lower than test-retest reliability (33%-46% of items met psychometric standards). Caregivers did not systematically differ in whether they rated infants as healthier or less healthy than the other caregiver (5%-16% of items had statistically significant differences).Caregivers used the measure consistently, but had different experiences and perceptions of their infant's health and functioning. Future studies are needed to explore mechanisms for the differences in test-retest and interrater reliability. Whenever possible, the same caregiver should provide ratings of the infant, including on the iCOO.

Caregiver Observations of Infant Well-Being Before and After Cleft Lip Surgery.

OBJECTIVE: To evaluate the sensitivity to change of daily ratings of the comfort (COMF) and behavioral/emotional health (BEH) domains of the Infants with Clefts Observation Outcomes Instrument (iCOO) at 3 time points, and to assess the association of post-surgical interventions on iCOO ratings. DESIGN: The COMF and BEH domains were completed by caregivers before (T0), immediately after (T1), and 2-months after (T2) cleft lip (CL) surgery. Analyses included descriptive statistics, correlations, t-tests, and generalized estimating equations. PARTICIPANTS: Caregivers (N = 140) of infants with CL with/without cleft palate. MAIN OUTCOME MEASURES: The COMF and BEH domain scores of the iCOO: Scale (SCALE), a summary of observable signs; and Global Impression (IMPR), a single item measuring caregivers' overall impression. RESULTS: Daily COMF and BEH SCALE and IMPR scores changed significantly during T1 (P's < 0.001) but not T0 or T2. Day 1 and 7 T0 scores were significantly higher than Day 1 and 7 T1 scores (P's <0.001 to <0.012) but similar at T2 (P's > 0.05). After CL surgery, the combined use of immobilizers and nasal stents and the use of multiple feeding methods with treatment for gastroesophageal reflux were associated with lower daily scores in COMF and BEH SCALE and IMPR (P's: 0.040 to <0.001). CONCLUSIONS: COMF and BEH iCOO scores were sensitive to daily changes in infant well-being following CL surgery. Future studies should further investigate impact of post-surgical treatments on infant well-being.

Infant with Clefts Observation Outcomes Instrument (iCOO): A New Outcome for Infants and Young Children with Orofacial Clefts.

OBJECTIVE: We evaluated the measurement properties for item and domain scores of the Infant with Clefts Observation Outcomes Instrument (iCOO). DESIGN: Cross-sectional (before lip surgery) and longitudinal study (preoperative baseline and 2 days and 2 months after lip surgery). SETTING: Three academic craniofacial centers and national online advertisements. PARTICIPANTS: Primary caregivers with an infant with cleft lip with or without cleft palate (CL ± P) scheduled to undergo primary lip repair. There were 133 primary caregivers at baseline, 115 at 2 days postsurgery, and 112 at 2 months postsurgery. MAIN OUTCOME MEASURE(S): Caregiver observation items (n = 61) and global impression of health and function items (n = 8) across eight health domains. RESULTS: Mean age at surgery was 6.0 months (range 2.7-11.8 months). Five of eight iCOO domains have scale scores, with Cronbach's alphas ranging from 0.67 to 0.87. Except for the Facial Skin and Mouth domain, iCOO scales had acceptable intraclass correlation coefficients (ICCs) ranging from 0.76 to 0.84. The internal consistency of the Global Impression items across all domains was 0.90 and had acceptable ICCs (range 0.76-0.91). Sixteen out of 20 (nonscale) items had acceptable ICCs (range 0.66-0.96). As anticipated, iCOO scores 2 days postoperatively were generally lower than baseline and scores 2 months postsurgery were consistent with baseline or higher. The iCOO took approximately 10 min to complete. CONCLUSIONS: The iCOO meets measurement standards and may be used for assessing the impact of cleft-related treatments in clinical research and care. More research is needed on its use in various treatment contexts.

Development of an Outcome Measure of Observable Signs of Health and Well-Being in Infants With Orofacial Clefts.

OBJECTIVE: To develop an outcomes instrument that assesses observations that can be reliably reported by caregivers and can be used to assess health of infants with a cleft lip or cleft lip and cleft palate (CL±P) and impacts of treatments. DESIGN: Cross-sectional, mixed methods study. SETTING: Caregivers and health-care providers were recruited from 3 academic craniofacial centers and national advertisements. Most interviews were conducted by telephone, and surveys were completed online. PARTICIPANTS: Caregivers had a child less than 3 years of age with CL±P and spoke either English or Spanish. Health-care providers were members of a cleft team. Caregivers (n = 492) and health-care professionals (n = 75) participated in at least one component of this study. MAIN OUTCOME MEASURE(S): Caregivers and health-care providers participated in tasks related to instrument development: concept elicitation for items within relevant health domains, prioritization of items, and item review. RESULTS: We identified 295 observations of infant well-being across 9 health areas. Research staff and specialists evaluated items for clarity, specificity to CL±P, and responsiveness to treatment. Caregivers and health-care providers rated the resulting list of 104 observations and developed the final instrument of 65 items. CONCLUSIONS: In this phase of development of the Infant with Clefts Observation Outcomes (iCOO) instrument, items were developed to collect caregiver observations about indicators of children's health and well-being across multiple domains allowing for psychometric testing, sensitivity to changes associated with treatment, and documentation of the effects of treatment.

Facilitating Positive Psychosocial Outcomes in Craniofacial Team Care: Strategies for Medical Providers.

OBJECTIVE: Psychosocial issues associated with craniofacial diagnoses and the ongoing burden of care can impact the quality of life of patients and families, as well as treatment adherence and outcomes. Utilizing available literature and clinical expertise across 6 centers, the present article summarizes key psychosocial issues for the benefit of nonmental health medical providers and offers suggestions as to how all members of craniofacial teams can promote positive psychosocial outcomes. RESULTS: Family adjustment across developmental phases is outlined, with strategies to support adaptive parental coping. Teasing is a common concern in craniofacial populations and medical providers can promote coping and social skills, as well as link families to mental health services when needed. Academic issues are described, alongside suggestions for medical providers to assist families with school advocacy and ensure access to appropriate services within the school setting. Medical providers are key in preparing patients and families for surgery, including consideration of medical, social, and logistical supports and barriers. As craniofacial care spans infancy to adulthood, medical providers are instrumental in assisting patients and families to navigate treatment transition periods. In addition to ongoing clinical team assessments, medical providers may utilize screening measures to identify and track patient and family adjustment in multiple areas of team care. CONCLUSIONS: Multidisciplinary providers play an important role in supporting positive adjustment in patients affected by craniofacial conditions and their families.

Parent Observations of the Health Status of Infants With Clefts of the Lip: Results From Qualitative Interviews.

OBJECTIVE: To explore, using semistructured qualitative interviews, parent observations of their infant's health as they relate to having a cleft lip or cleft lip and cleft palate (CL±P) and/or associated treatments. DESIGN: Cross-sectional, qualitative study across 3 sites. SETTING: Parents were recruited from 3 academic craniofacial centers. Most interviews were conducted over the telephone, audio-recorded, and transcribed. PARTICIPANTS: Forty-one parents (31 English-, 10 Spanish-speaking) were interviewed. Parents had a child ages 1 to 35 months diagnosed with CL±P. MAIN OUTCOME MEASURE(S): Twelve domains reflecting infant health and well-being, likely affected by a CL±P and/or associated treatments, were identified from clinical experience and literature review. Study investigators conducted semistructured interviews based on 12 identified domains. After transcripts of the interviews were reviewed, our multidisciplinary team selected illustrative quotes from each domain that reflected consistent observations made by parents. RESULTS: Parents' responses covered all 12 domains. Specifically, parents discussed the domain of feeding most frequently, followed by observations about sleep, development, comfort, breathing, and vocalization. CONCLUSIONS: We found that parents of infants with clefts provided caregiver-centered language useful in describing their daily observations. No additional domains of infant health were noted by parents. We will use these observations and language to develop and validate a parent-reported observation diary that can augment assessments of the impacts of interventions on infants who are receiving care for CL±P.

What difference can a minute make? Social skills and first impressions of youth with craniofacial differences.

OBJECTIVE: To determine whether raters' first impressions of youth with craniofacial differences are modifiable. DESIGN: Observational study of the association between first impressions and social skills as related to youth aged 11 to 18 years with craniofacial differences. SETTING: University research offices and clinics. PARTICIPANTS: Youth aged 11 to 18 years with (n  =  29) and without (n  =  31) craniofacial differences; adults (n  =  40), dental/medical students (n  =  46), and education students (n  =  29), all without craniofacial differences. Participants were recruited from medical clinics and through community advertising at all three study sites. OUTCOME MEASURES: The First Impressions Rating Scale. RESULTS: After viewing 1-minute portrayals of positive social skills by actors with craniofacial differences, raters' perceptions moved significantly in the positive direction for all 26 attributes on the First Impressions Rating Scale; whereas, after viewing negative social skills, ratings moved significantly in the negative direction for 25 of 26 First Impressions Rating Scale attributes. CONCLUSIONS: It appears that first impressions others have of youth with craniofacial differences are significantly affected by how these youth present themselves in social situations, suggesting that positive social skills may help reduce the amount of stigma that youth with craniofacial differences encounter.

Anticipating benefits and decreasing burdens: the responsibility inherent in pediatric plastic surgery.

Working with child patients and their families in the practice of plastic and reconstructive surgery is rewarding and risky. Technical training is understandably intensive. However, the importance of the work that is needed before an operation does not always attract the same attention. Surgery that is done to change a child's body results in a change in that person's identity and cannot be taken lightly. A review of the overall benefit and burden of both the untreated condition and the proposed changes must be considered to support a change so that it does not damage an individual's identity development. By taking the time to understand what the patient's perspective is, surgical change can become a process that a person is part of, instead of it being something that is done to them. This helps to set up a situation where the change will be directly beneficial to the patient, and that change will be sustained over a long period. When evaluating surgical options with children, the influences and pressures that exist within families and throughout society must also be considered. A young child and his/her future must not get lost in the difficult negotiations between parents/guardians, surgeons, and society. By taking more time to consider the tremendous meaning that your work has outside the operating room and in the context of who the patient is becoming, you will have a greater chance of not just enhancing someone's appearance, but also improving his/her quality of life.

Craniofacial summer cAMP: an educational experience for campers, cAMP staff, and the craniofacial team.

Children with congenital or acquired craniofacial conditions often have complex medical and surgical healthcare needs. To provide holistic care to this unique pediatric population, we must seek methods to address the psychosocial needs of children living with a craniofacial difference. Our multidisciplinary craniofacial team concluded our second-year participating in overnight summer camps for our patients with cleft-craniofacial conditions. We worked with 2 different organizations to host the camps each year. Over the course of 2 years, we enrolled more than 100 children, 7-15 years of age, in 1-week, overnight camp experiences. Campers participated in activities to promote team building, self-esteem, confidence, and social awareness. We share the perspectives of the campers and their parents, the camp staff, and the craniofacial team members.

What the patients and parents do not tell you-recollections from families following external LeFort III midface distraction.

OBJECTIVE: The purpose of this study was to document the experience of patients and parents of patients who had recently undergone LeFort III midface distraction using an external halo-based device. DESIGN: Cross-sectional study. SETTING: A craniofacial center in a pediatric tertiary care medical center. SUBJECTS: Eight children who had undergone midface distraction within 1 year of the interview and their caregivers. INTERVENTION: Semi-structured interview. MAIN OUTCOME MEASURE: Transcripts of the interviews were rendered anonymous and analyzed by our multi-disciplinary team. Consistent themes in the subjects' experience during and after midface distraction were identified. RESULTS: (1) Family participation in the decision to undergo distraction and pre-operative preparation was recognized as valuable, but parents identified that there are inherent limitations; (2) home-care tasks seemed daunting pre-operatively but were easier than expected; (3) discomfort, sleeping, and interaction with peers were considered well accommodated, but feeding was challenging; (4) individualized pre-operative plans for community support was important; (5) parents and patients were impressed by the change in appearance, specifically in the peri-orbital region; (6) access to team members and to parents of patients who had participated in the distraction process was invaluable. CONCLUSION: External midface distraction is a valuable clinical technique, but requires intensive preparation and support from a multi-disciplinary team. We provide suggestions for consideration by centers initiating and refining patient care plans for this surgery.

Measuring the quality of life of youth with facial differences.

OBJECTIVE: To describe the Facial Differences Module of the Youth Quality of Life Instruments (YQOL-FD) and present results evaluating domain structure, internal consistency, reproducibility, validity, and respondent burden. DESIGN: A multisite observational study of youth aged 11 to 18 years with acquired or congenital facial differences. PARTICIPANTS: Three hundred seven youth recruited through clinics at four U.S. sites and one U.K. site. Eligible youth were aged 11 to 18 years, had a noticeable facial difference, could read at the fifth-grade level, and, for youth with facial burns, were at least 2 years posttrauma. MEASURES: Included were the newly developed YQOL-FD, the generic Youth Quality of Life Instrument, the Children's Depression Inventory, and demographics. RESULTS: Principal components analysis showed five significant factors that closely matched the domain structure hypothesized a priori. Domain scores of the YQOL-FD showed acceptable internal consistency and reproducibility. Scores were more strongly correlated with the Children's Depression Inventory score than with self-rating of health as predicted. All domain scores showed adequate discrimination among levels of general quality of life (Wilks lambda = 0.84, p = .001). The median time to complete the module was 10 minutes. DISCUSSION: The YQOL Facial Differences Module augmented information obtained from the generic YQOL measure by addressing specific concerns. The module was well received by youth and showed acceptable measurement properties for evaluating the perceived quality-of-life status of youth facial differences. Future use in longitudinal studies and clinical trials is anticipated to evaluate the ability to detect change.

Community orthodontists and craniofacial care: results of a Washington state survey.

BACKGROUND: Little is known about community orthodontists' previous training in, experience with, or receptivity to caring for children with craniofacial disorders. OBJECTIVES: (1) To characterize the current level of participation by Washington state orthodontists in craniofacial care; and (2) to identify factors that promote or impede community orthodontists' involvement in caring for children with craniofacial conditions. DESIGN: Mail survey. METHODS: A 26-item questionnaire was designed and mailed to all active orthodontists in Washington state (N = 230). Question topics included practice characteristics, training and experience with craniofacial conditions, concerns related to public and private insurance, and communication with craniofacial teams. RESULTS: Of eligible respondents, 68% completed the survey. Most orthodontists' patient panels were made up of patients who either have private insurance or pay cash for services. On average, 2% of respondents' patients were Medicaid beneficiaries. Only 20% of respondents had seen more than three patients with cleft lip and/or palate in the past 3 years. Although a minority of orthodontists receive referrals from (27%) or are affiliated with (11%) craniofacial teams, most orthodontists perceived craniofacial care positively and were interested to learn more about craniofacial care and to accept additional patients with these conditions. CONCLUSIONS: Results of this survey can inform potential strategies to increase access to orthodontic care for children with craniofacial disorders. These would include developing an organized training, referral, and communication system between community orthodontists and state craniofacial teams and considering a case-management approach to facilitate this process.

Approaches to craniofacial-specific quality of life assessment in adolescents.

OBJECTIVE: To ascertain the domains that adolescents aged 11 to 18 years with congenital and acquired craniofacial differences (CFDs) consider important to their quality of life (QoL) to create a craniofacial-specific module. DESIGN: Interviews and inductive qualitative methods were used to guide the development of a conceptual and measurement model of QoL among adolescents with CFDs. SETTING: The Craniofacial Center at Children's Hospital and Regional Medical Center in Seattle, Washington. PATIENTS, PARTICIPANTS: Thirty-three in-depth interviews with adolescents (aged 11 to 18 years), one young adult interview (age 19 years), 14 in-depth interviews with parents, one young adult focus group, one parent focus group, and one panel of researchers and clinical professionals working in the field. RESULTS: Using the qualitative methodology, grounded theory, seven domains that adolescents with CFDs perceive are important to having a good QoL were found. Six of the domains (coping, stigma and isolation, intimacy and trust, positive consequences, self-image, and negative emotions) comprised the Youth Quality of Life Instrument-Facial Differences module. One other domain, surgery, was a salient issue for many of the youth, but not all, so it was made into a separate module, the Youth Quality of Life Instrument- Craniofacial Surgery module. This module relates to the experience of surgery, outcomes of surgery, and preferences for future surgery. CONCLUSIONS: Using an established qualitative methodology, two QoL modules specific to adolescents with CFDs were developed and are ready for psychometric validation. Potential uses of the instruments are discussed.

"The visible skeleton series": the art of Laura Ferguson.

Laura Ferguson's The Visible Skeleton Series constitutes an artistic self-representation of scoliosis. Ferguson employs high-tech medical imaging and innovative graphical techniques to suggest a novel view of her skeletal "deformity." This section presents introductions to the series by a historian of medicine and by the artist herself, and offers personal and professional reflections on the work from an orthopedic surgeon, a social worker with lived experience of cleft lip, and a retired pediatric forensic pathologist.

Dealing with the prenatal diagnosis of clefting: a parent's perspective.

OBJECTIVES: Prenatal testing and information is available on an increasing basis for a variety of reasons. There are conflicting standards of care in the delivery of this type of evaluation and even more confusion around access to such services. As physicians and patients evaluate which tests will generate useful information with acceptable levels of risk, the rules of the game constantly shift. Presenting the situation from the viewpoints of both a professional and a parent, issues revolving around uncertainty and reassurance are evaluated. A description of the personal experience of the impact of the discovery of a cleft prenatally is provided. CONCLUSIONS: Finally, efforts are made to raise questions about our motivations behind conducting prenatal testing and also developing some suggestions about how to support families better in our regular clinical work. A challenge is made to providers and patients to do more work prior to examination to explore how the unexpected will be handled. The dilemma about how to facilitate informed consent through full disclosure without overwhelming the patient is also discussed.