Delineating family needs in the transition from hospital to home for children with medical complexity: part 1, a meta-aggregation of qualitative studies.

BACKGROUND: Advances in diagnostic and therapeutic interventions for rare diseases result in greater survival rates, with on the flipside an expanding group of children with medical complexity (CMC). When CMC leave the protective hospital environment to be cared for at home, their parents face many challenges as they take on a new role, that of caregiver rather than care-recipient. However, an overview of needs and experiences of parents of CMC during transition from hospital-to-home (H2H) is lacking, which hampers the creation of a tailored H2H care pathway. Here we address this unmet medical need by performing a literature review to systematically identify, assess and synthesize all existing qualitative evidence on H2H transition needs of CMC parents. METHODS: An extensive search in Medline, PsychINFO and CINAHL (up to September 2022); selection was performed to include all qualitative studies describing parental needs and experiences during H2H transition of CMC. All papers were assessed by two independent investigators for methodological quality before data (study findings) were extracted and pooled. A meta-aggregation method categorized the study findings into categories and formulated overarching synthesized findings, which were assigned a level of confidence, following the ConQual approach. RESULTS: The search yielded 1880 papers of which 25 met eligible criteria. A total of 402 study findings were extracted from the included studies and subsequently aggregated into 50 categories and 9 synthesized findings: (1) parental empowerment: shifting from care recipient to caregiver (2) coordination of care (3) communication and information (4) training skills (5) preparation for discharge (6) access to resources and support system (7) emotional experiences: fatigue, fear, isolation and guilt (8) parent-professional relationship (9) changing perspective: finding new routines and practices. The overall ConQual Score was low for 7 synthesized findings and very low for 2 synthesized findings. CONCLUSIONS: Despite the variability in CMC symptoms and underlying (rare disease) diagnoses, overarching themes in parental needs during H2H transition emerged. We will augment this new knowledge with an interview study in the Dutch setting to ultimately translate into an evidence-based tailored care pathway for implementation by our interdisciplinary team in the newly established 'Jeroen Pit Huis', an innovative care unit which aims for a safe and sustainable H2H transition for CMC and their families.

Objective evaluation of nonsurgical treatment of prominent ears: A systematic review.

Background: The prominent ear is a type of congenital ear deformity that can be corrected by a variety of nonsurgical treatments, such as splinting and the taping method. However, there is no objective evaluation method that is universally accepted. The aim of this review is to evaluate objective measurement methods that are used in the available literature to analyze nonsurgical treatment of prominent ears. Methods: A systematic review was performed in the MEDLINE and Embase databases in December 2022 and updated on April 2023 according to Preferred Reporting Items for Systematics and Meta-Analyses (PRISMA) guideline. Any study using objective measurements (continuous variables such as distance and angle) to evaluate the effect of nonsurgical treatment of prominent ears was included. The Joanna Briggs Institute (JBI) critical appraisal for case series was used for quality assessment. Results: A total of 286 studies were screened for eligibility, of which five articles were eligible for inclusion. All of the included studies were case series. The helix mastoid distance (HMD) is the most commonly used parameter to measure treatment outcome. Pinna and cartilage stiffness, length, and width were also used, but without clear statistical relevance. HMD was classified into grading groups (i.e. good, moderate, and poor) to evaluate the treatment's effect. Conclusion: Based on the included studies, objective measurements are rarely used, and when used, they are largely heterogeneous. Although HMD was the most frequent measurement used, all studies used different definitions for the measurement and grouped subsequent outcomes differently. Automated algorithms, based on three-dimensional imaging, could be used for object measurements in the nonsurgical treatment of prominent ears.

Objective quantitative methods to evaluate microtia reconstruction: A scoping review.

Background: Commonly used methods to evaluate auricles are subjective and are therefore not specific, comprehensive, and precise nor effective in the assessment of microtia reconstruction outcomes. This scoping review aimed to summarize the objective methods for the accurate evaluation of microtia reconstruction. Methods: We performed a scoping review of publications that used objective measurement methods to evaluate outcomes of microtia reconstruction according to the PRISMA-ScR guidelines. A systematic literature search was conducted in the Embase, PubMed, Cochrane, CNKI, and VIP databases, and literature references were screened for additional records. Studies that evaluated auricles after microtia reconstruction using quantitative anthropometric methods were included, and data on these methods were collected. Results: Twenty-five publications reported on quantitative objective outcome measurements. Thirteen studies evaluated auricular protrusion, three articles assessed the position or symmetry, and twelve studies reported on auricle size. The quantitative measurements of fine structures, such as the tragus and concha, were described in three studies. All described measurements used manual landmarking, where fifteen studies described well-defined landmarks, fifteen studies described poorly defined landmarks, and four studies used a combination of well and poorly defined landmarks. Conclusion: The objective evaluation of microtia reconstruction outcomes is hindered by significant heterogeneity of measurement methods. The measurement methods used for general auricular measurements (auricular protrusion, auriculocephalic angle, and size) used in microtia reconstruction were abundant, while measurements of auricular position and the fine structures of the auricle were limited. Three-dimensional imaging combined with computer analyses poses promising future alternatives.

Long-term aesthetics, patient-reported outcomes, and auricular sensitivity after microtia reconstruction: A systematic review.

BACKGROUND: Auricular reconstruction for microtia is most frequently performed using autologous costal cartilage (ACC) or porous polyethylene (PPE) implants. Short-term results are generally promising, but long-term results remain unclear. Long-term outcomes were explored in this systematic review, and minimal reporting criteria were suggested for future original data studies. METHODS: A systematic literature search was conducted in MEDLINE, EMBASE, and the Cochrane Central Register of Controlled Trials from inception through October 14, 2020. Articles on auricular reconstruction in patients with microtia using ACC or PPE were included if postsurgical follow-up was at least 1 year. Outcome reporting was split into separate publications, and results on complications were reported previously. This publication focused on long-term aesthetic, patient-reported, and sensitivity outcomes. RESULTS: Forty-one publications reported on these outcomes. Both materials led to aesthetically pleasing results and high rates of patient satisfaction. ACC frameworks grew similarly to contralateral ears, and the anterior surface of auricles regained sensitivity. Furthermore, postoperative health-related quality of life (HRQoL) outcomes were generally good. Data synthesis was limited due to considerable variability between studies and poor study quality. No conclusions could be drawn on the superiority of either method due to the lack of comparative analyses. CONCLUSION: Future studies should minimally report (1) surgical efficacy measured using the tool provided in the UK Care Standards for the Management of Patients with Microtia and Atresia; (2) complications including framework extrusion or exposure, graft loss, framework resorption, wire exposure and scalp/auricular scar complications and (3) HRQoL before and after treatment using the EAR-Q patient-reported outcome measure (PROM).

Long-term complications of microtia reconstruction: A systematic review.

BACKGROUND: Microtia is a rare disorder characterized by malformation or even complete absence of the auricle. Reconstruction is often performed using autologous costal cartilage (ACC) or porous polyethylene implants (PPE). However, the long-term outcomes of both methods are unclear. OBJECTIVE: This systematic review aimed to analyze long-term complications and suggest minimal reporting criteria for future original data studies. METHODS: A systematic literature search was conducted in MEDLINE, EMBASE and the Cochrane Central Register of Controlled Trials from inception through October 14, 2020. Articles on auricular reconstruction in patients with microtia using ACC or PPE were included provided that the follow-up period was at least one year. This publication focused on long-term complications reported in patients with a postoperative follow-up period of at least one year. RESULTS: Twenty-nine publications reported on complications during long-term follow-up. Overall long-term complication rates were not reported. The incidence of individual complications during long-term follow-up was less than 10% after ACC reconstruction and less than 15% in PPE reconstruction. Framework resorption and wire exposure were reported even after an extended follow-up of more than five years after ACC reconstruction, while reports on the extended long-term results of PPE reconstruction are limited. Data synthesis was limited due to heterogeneity and poor study quality. CONCLUSIONS: Future studies should report on long-term complications including framework exposure or extrusion, graft loss, framework resorption, wire exposure and scalp and auricular scar complications. We recommend a surgical follow-up of at least five years.

Outcomes and measurement instruments used in congenital melanocytic naevi research: A systematic review.

BACKGROUND: Congenital melanocytic naevi (CMN) can have a great impact on patients' lives due to the remarkable appearance and the risk of developing melanoma and neurocutaneous melanosis. Comparison of treatment efficacy is currently hindered by the lack of standard and uniform outcome reporting; this impedes guidance on optimal management policy. To address this, we aim to perform the first step in developing a core outcome set. With this systematic review, we identified a list of domains, outcomes (including patient reported outcomes) and outcome measurement instruments used in CMN research. METHODS: The review was registered in PROSPERO, registration number CRD42018095235. A search was conducted in EMBASE (Ovid), PubMed and the Cochrane Library from 2006 to January 2019. Studies with 10 or more patients, with all sizes of CMN and reporting outcomes on interventional and conservative management were included. RESULTS: A total of 1,285 individual studies was found; 63 studies were included. We extracted 57 different outcomes and 34 outcome measurement instruments showing large heterogeneity. Patient-reported outcomes were included in 38% of studies. Few outcome measurement instruments were described. Moreover, none of the studies reported that the used instruments were ever validated in a CMN population. CONCLUSION: Heterogeneity exists in outcomes and instruments used in CMN research. The development of a core outcome set may reduce this heterogeneity in future research, thereby enabling treatment comparison and eventually facilitating guidance on management. Furthermore, this overview demonstrates a need for the use and validation of (patient reported) outcome measurement instruments for CMN.

Effects of medicinal food plants on impaired glucose tolerance: A systematic review of randomized controlled trials.

BACKGROUND: The objective of this systematic review was to assess available scientific data on the efficacy and safety of medicinal food plants for the treatment of impaired glucose tolerance. METHODS: We included randomized controlled trials (RCTs) with a minimum follow-up period of 6weeks. The diagnosis was determined by fasting plasma glucose values after two-hour oral glucose tolerance testing (OGTT). Two authors independently extracted data and evaluated bias. The Cochrane tool of risk of Bias Tool was used. RESULTS: This review included ten trials. Most studies were highly biased as data were incomplete or reporting was selective. The two-hour fasting plasma glucose after the curcumin extract intervention showed statistical significance after 3, 6 and 9months: p<0.01. Also, glycosylated haemoglobin levels A1c (HbA1c) values after curcumin extract intervention showed statistical significance after 3, 6 and 9months: p<0.01. Insulin resistance (HOMA-IR) after curcumin extract intervention showed statistical significance after 6months and after 9months: p<0.05 and p<0.01. CONCLUSIONS: Curcumin has shown the confident results to be effective for the treatment of impaired glucose tolerance. Fenugreek and flaxseed may also be effective, but due to low quality of these studies the results must be interpreted with caution.