Effects of adherence to treatment for repositioning therapy, physical therapy, and cranial remolding orthoses in infants with cranial deformation.

Deformational head shapes are most often treated through repositioning therapy (RT) and/or cranial remolding orthotic (CRO) treatment. However, there is conflicting evidence about the effectiveness of each method, and treatment compliance is suspected to affect treatment effectiveness. This study examines participant adherence with these treatment methods and explores if cranial correction is related to compliance. This study also reviews effects of developmental milestones and explores other potential impacts on compliance. A total of 45 infants with cranial deformation were consented and those with congenital muscular torticollis (CMT) concurrently received physical therapy. Infants were followed from 2 to 12 months of age and initially assigned to RT. Caregivers continued RT until the head shape corrected, caregivers chose to switch to a CRO, or infants turned 12 months of age. All participants were scheduled for a final visit at 12 months of age. Throughout treatment, caregiver surveys were used to examine compliance and developmental milestones. Results show promise for future investigation into the relationship between treatment modalities and adherence with treatment for deformational head shapes. Our findings provide preliminary support that treatment adherence may be linked with treatment success and concurrent enrollment in physical therapy increases patient compliance.

Development and Validation of a Prediction Model for the Treatment Time of Deformational Head Shapes Using a Cranial Remolding Orthosis.

The cranial remolding orthosis (CRO) has been shown in previous studies to be an effective method of treatment for deformational head shapes. Many studies have shown younger infants achieve greater correction than older infants and generally have a shorter treatment duration. The goal of this study is to develop and validate a prediction equation for the maximum treatment time for deformational head shapes when utilizing a CRO. This retrospective study included subjects with deformational plagiocephaly (DP), deformational brachycephaly (DB), or deformational asymmetrical brachycephaly (DAB) who began CRO treatment between 3 and 18 months of gestational age. Prediction models were derived from 1250 subjects with DP, DB, and DAB and the validation used data from 210 different subjects. Actual treatment time was less than or equal to predicted treatment time in 85.19% (DP), 56.67% (DB), and 75.40% (DAB) of the cases when rounding the prediction up to the nearest month. The prediction equation has moderate accuracy for predicting the likely maximum amount of CRO treatment time for patients with DP, DB, and DAB and may be used clinically to give caregivers an estimated treatment duration for a patient who is indicated for a CRO, if treatment was initiated immediately.

Side Effects of Cranial Remolding Orthoses: A Multi-Site Review.

ABSTRACT: Infants with deformational head shapes may be treated with a Cranial Remolding Orthosis (CRO). Risks of CROs include skin irritations and other minor side effects. It is important to examine the incidence of complications with CRO treatment to determine if the potential benefits outweigh the risks.This study retrospectively examined surveys provided by both practitioners and caregivers regarding orthotic treatment side effects and wear time at four clinics in Canada. Statistical analysis including the Chi-square test and Fisher's exact tests were performed to examine how the side effects varied by treatment site and by reported orthotic wear time.In total, 5,025 surveys about the CRO treatment of 453 patients were reviewed. The most common side effects were pressure spots, redness lasting for more than 1 hour, excessive sweat , and foul odor; however, the vast majority reported no significant issues (87.08%). Only 2.81% of surveys reported multiple side effects, which was about three times more like to happen when the CRO was reported to have fit problems. Wear time is correlated with the report of side effects; infants who wore the CRO 18 to 23.5 hours had statistically fewer side effects than those who wore the CRO for less time. The reported incidence of side effects may be artificially inflated because infants experiencing side effects are more likely to have more frequent follow-up.Overall, the side effects of CRO treatment reported in this cohort were relatively rare and minimal, indicating CRO treatment is a safe way to treat deformational head shapes.

Significant Factors in Cranial Remolding Orthotic Treatment of Asymmetrical Brachycephaly.

This retrospective chart review focuses on determining the most effective time to begin cranial remolding orthosis (CRO) treatment for infants with asymmetrical brachycephaly. Subjects with asymmetrical brachycephaly started CRO treatment between 3 and 18 months of age. These infants had a cranial vault asymmetry index (CVAI) ≥ 3.5 and a cranial index (CI) ≥ 90. Subjects were excluded if they had any comorbidities affecting growth, dropped out of treatment, were lost to follow-up, or were noncompliant. Factors which were found to statistically influence treatment outcomes were subject initial age, initial CVAI, and initial CI. Overall, younger subjects were more likely to achieve a corrected head shape. The presence of prematurity or torticollis had statistically nonsignificant effects on the success of treatment. Initial CI was found to be a stronger predictor than initial CVAI as to which subjects achieved correction. The less severe the starting CI, the more likely the subject was to achieve full correction. The clinical understanding is that it requires more cranial growth to "round out" a full posterior skull flattening than an asymmetry. Based on the study results, infants with asymmetrical brachycephaly should be treated as early as possible to increase chances of achieving full correction of the deformity.

Effects of Initial Age and Severity on Cranial Remolding Orthotic Treatment for Infants with Deformational Plagiocephaly.

The aim of this study is to review the effects of an infant's presenting age and severity of deformation on cranial remolding orthotic (CRO) treatment outcomes for patients with deformational plagiocephaly. This study is a retrospective chart review of 499 infants with non-synostotic plagiocephaly who completed CRO treatment. Data collected included age at start of treatment, head shape measurements before and after treatment, total months of CRO treatment, and other factors such as presence/absence of prematurity. The infants were divided into subgroups according to age and severity at initiation of treatment and data for subgroups was analyzed to track the change in head shape over the course of treatment, review overall treatment duration, and discuss the rate of change of cranial deformation. Overall, treatment times tended to statistically increase with increasing initial severity and age. Posttreatment asymmetry measurements statistically trended to greater residual deformation in infants who began treatment in the older or more severe subcategories. This indicates that younger and less severe infants have shorter treatment durations and less residual cranial deformation after CRO treatment. Therefore, clinical consideration may need to be taken to treat infants at younger ages or prior to progression of the cranial deformity.

Significant Factors Influencing the Effectiveness of Cranial Remolding Orthoses in Infants With Deformational Plagiocephaly.

Infants with deformational plagiocephaly may be indicated for a cranial remolding orthosis (CRO) depending on the severity of deformation. Studies have found torticollis and prematurity to be risk factors for the development of deformational plagiocephaly and indicate younger infants have faster and greater correction. However, clinical decisions about which infants should be recommended for CRO treatment differ among healthcare providers and insurance coverage criteria vary. In order to provide insight into treatment recommendations, this study aims to examine the influence of 4 specific factors on CRO treatment time and the infant's resultant posttreatment head shape. Results indicate that presenting age, presenting severity, and the presence of torticollis significantly influence treatment duration. Presenting age and severity were found to significantly influence final head shape. The presence of prematurity was not found to be significant when infants were categorized by their corrected postpartum ages.

Pathologic and clinical characteristics of early onset renal cell carcinoma.

The majority of renal cell carcinomas (RCCs) occur within the 7th decade of life, uncommonly arising in adults ≤46 years. We reviewed the clinicopathologic features of early onset RCC and evaluated the role of immunohistochemistry (IHC) in potentially identifying diagnoses of newly recognized RCC subtypes that may have been previously misclassified. A retrospective review was performed from 2011-2016 for cases of RCC. Early onset RCC was defined as ≤46 years of age. Clinicopathologic findings and hematoxylin and eosin (H&E) slides were reviewed on early onset RCC patients. IHC was performed on all cases previously diagnosed as unclassified or papillary. Clinicopathologic findings were compared to a control group of RCC patients >46 years over the same time period. We identified 98/598 (16.4%) early onset RCCs. The median age in the early onset RCC and control group was 38.4 and 62.8 years, respectively. The early onset RCC group contained 33/96 (34.3%) females and 63/96 (65.6%) males, including 52/96 (54.2%) whites, 39/96 (40.6%) African Americans, 4/96 (4.2%) Hispanics, and 1/96 (1%) Asian. Nonwhites were significantly more likely to develop early onset RCC (P=.004). Early onset RCCs included 52% clear cell, 28.6% papillary, 8.2% unclassified, 5.1% chromophobe, 3.1% clear cell papillary(CCP), and 3 other rare tumors. Six unclassified and 26 papillary RCCs had tissue available for IHC. Two of 6 (33.3%) unclassified RCCs were reclassified (1 CCP, 1 Xp11 translocation). One of 26 (3.8%) papillary RCCs was reclassified as CCP. Early onset RCCs were more likely to occur in nonwhites (P=.004), be lower stage (P=.03), and undergo partial nephrectomy (P=.002). Few unclassified and papillary tumors were reclassified with IHC.

New population-based reference values for spinal mobility measures based on the 2009-2010 National Health and Nutrition Examination Survey.

OBJECTIVE: To report population-based percentile reference values for selected spinal mobility measures in a nationally representative sample of 5,001 US adults ages 20-69 years who were examined in the 2009-2010 US National Health and Nutrition Examination Survey (NHANES). METHODS: Occiput-to-wall distance (OWD), thoracic expansion (TE), and anterior lumbar flexion (ALF; by modified Schober test) were measured by trained examiners in a standardized manner. TE was measured at the xiphisternal level, while the lower reference point for ALF was a line marked at the level of the superior margin of the lateral iliac crests. We report reference values based on the 95th percentile for the OWD and the 5th percentile for TE and ALF, as well as other summary statistics for these measures, in the study population. RESULTS: An OWD of >0 was present in 3.8% of the participants, while 8.8% of them had out-of-range values for TE based on the commonly used threshold of 2.5 cm. The 95th percentile of the OWD measurement was 0, while the 5th percentile for TE and ALF were 1.9 cm and 2 cm, respectively. The spinal measures were significantly associated with sex, age, ethnicity, height, and body mass index (BMI). Exclusion of individuals with severe obesity (BMI >35 kg/m(2) ) changed the proposed reference values for TE and ALF to 2.2 cm and 1.9 cm, respectively. CONCLUSION: We verified a reference value of 0 for the OWD in the general population. Using the reported population-based percentile values, new reference values for TE and ALF can be derived.

Biopsychosocial typologies of pain in a cohort of patients with systemic sclerosis.

OBJECTIVE: Despite being a common problem in systemic sclerosis (SSc; scleroderma), the extant literature on pain has primarily focused on biomedical correlates, or bivariate relationships with a few psychological characteristics. There is a need to investigate the more heuristic biopsychosocial model, which incorporates the simultaneous contributions of medical, psychological, and social variables in understanding pain. METHODS: Patients with SSc (n = 333) received clinical examinations and completed self-report surveys at enrollment in the Genetics versus Environment in Scleroderma Outcome Study. Latent profile analysis was used to derive biopsychosocial profiles of patients using skin thickening, percent predicted forced vital lung capacity, perceived physical health, health worry, mental health, and social support. The profiles were examined in relation to pain and pain medication usage. RESULTS: A 3-profile solution provided the best fit to the data. Based on the biopsychosocial indicators, the profiles were characterized as managing (n = 217), resilient (n = 86), and distressed (n = 30). Between-group differences for pain emerged, with the distressed group, whose disease was less severe than the resilient group, reporting the highest pain and the greatest utilization of pain medication. CONCLUSION: Clinicians should consider biopsychosocial characteristics as contributing factors to the experience of pain in patients with SSc. Patients who are similar to those in the distressed profile may be at an increased risk for pain and would likely benefit from a referral to a behavioral health or other ancillary service provider for pain management, rather than relying solely on pharmacologic therapies.

Skin gene expression correlates of severity of interstitial lung disease in systemic sclerosis.

OBJECTIVE: We undertook this hypothesis-generating study to identify skin transcripts correlating with severity of interstitial lung disease (ILD) in systemic sclerosis (SSc). METHODS: Skin biopsy samples from 59 patients enrolled in the Genetics versus Environment in Scleroderma Outcome Study (GENISOS) cohort or an open-label imatinib study (baseline visit) were examined by global gene expression analysis using Illumina HT-12 arrays. Skin transcripts correlating with concomitantly obtained forced vital capacity (FVC) values and the modified Rodnan skin thickness score (MRSS) were identified by quantitative trait analysis. Also, immunofluorescence staining for selected transcripts was performed in affected skin and lung tissue. Plasma levels of CCL2, soluble SELP, and soluble P-selectin glycoprotein ligand 1 (sPSGL-1) were examined in all patients enrolled in the GENISOS cohort (n = 266). RESULTS: Eighty-two skin transcripts correlated significantly with FVC. This gene list distinguished patients with more severe ILD (FVC <70% predicted) in unsupervised hierarchical clustering analysis (P < 0.001). These genes included SELP, CCL2, and matrix metalloproteinase 3, which are involved in extravasation and adhesion of inflammatory cells. Among the FVC correlates, 8 genes (CCL2, HAPLN3, GPR4, ADCYAP1, WARS, CDC25B, PLP1, and STXBP6) also correlated with the MRSS. Immunofluorescence staining revealed that SELP and CCL2 were also overexpressed in affected skin and lung tissue from SSc patients compared to those from controls. Plasma levels of CCL2 and sPSGL-1 correlated with concomitantly obtained FVC values (r = -0.22, P = 0.001 and r = 0.17, P = 0.015, respectively). This relationship was independent of potential confounders (age, sex, ethnicity, smoking status, anti-topoisomerase I positivity, treatment with immunosuppressive agents, MRSS, disease type, and disease duration). CONCLUSION: A limited number of skin transcripts including genes involved in extravasation and adhesion of inflammatory cells correlate with severity of ILD.