Data-driven automated classification algorithms for acute health conditions: applying PheNorm to COVID-19 disease.

OBJECTIVES: Automated phenotyping algorithms can reduce development time and operator dependence compared to manually developed algorithms. One such approach, PheNorm, has performed well for identifying chronic health conditions, but its performance for acute conditions is largely unknown. Herein, we implement and evaluate PheNorm applied to symptomatic COVID-19 disease to investigate its potential feasibility for rapid phenotyping of acute health conditions. MATERIALS AND METHODS: PheNorm is a general-purpose automated approach to creating computable phenotype algorithms based on natural language processing, machine learning, and (low cost) silver-standard training labels. We applied PheNorm to cohorts of potential COVID-19 patients from 2 institutions and used gold-standard manual chart review data to investigate the impact on performance of alternative feature engineering options and implementing externally trained models without local retraining. RESULTS: Models at each institution achieved AUC, sensitivity, and positive predictive value of 0.853, 0.879, 0.851 and 0.804, 0.976, and 0.885, respectively, at quantiles of model-predicted risk that maximize F1. We report performance metrics for all combinations of silver labels, feature engineering options, and models trained internally versus externally. DISCUSSION: Phenotyping algorithms developed using PheNorm performed well at both institutions. Performance varied with different silver-standard labels and feature engineering options. Models developed locally at one site also worked well when implemented externally at the other site. CONCLUSION: PheNorm models successfully identified an acute health condition, symptomatic COVID-19. The simplicity of the PheNorm approach allows it to be applied at multiple study sites with substantially reduced overhead compared to traditional approaches.

The Association of Organizational Readiness With Lung Cancer Screening Utilization.

INTRODUCTION: Lung cancer screening is widely underutilized. Organizational factors, such as readiness for change and belief in the value of change (change valence), may contribute to underutilization. The aim of this study was to evaluate the association between healthcare organizations' preparedness and lung cancer screening utilization. METHODS: Investigators cross-sectionally surveyed clinicians, staff, and leaders at10 Veterans Affairs from November 2018 to February 2021 to assess organizational readiness to implement change. In 2022, investigators used simple and multivariable linear regression to evaluate the associations between facility-level organizational readiness to implement change and change valence with lung cancer screening utilization. Organizational readiness to implement change and change valence were calculated from individual surveys. The primary outcome was the proportion of eligible Veterans screened using low-dose computed tomography. Secondary analyses assessed scores by healthcare role. RESULTS: The overall response rate was 27.4% (n=1,049), with 956 complete surveys analyzed: median age of 49 years, 70.3% female, 67.6% White, 34.6% clinicians, 61.1% staff, and 4.3% leaders. For each 1-point increase in median organizational readiness to implement change and change valence, there was an associated 8.4-percentage point (95% CI=0.2, 16.6) and a 6.3-percentage point increase in utilization (95% CI= -3.9, 16.5), respectively. Higher clinician and staff median scores were associated with increased utilization, whereas leader scores were associated with decreased utilization after adjusting for other roles. CONCLUSIONS: Healthcare organizations with higher readiness and change valence utilized more lung cancer screening. These results are hypothesis generating. Future interventions to increase organizations' preparedness, especially among clinicians and staff, may increase lung cancer screening utilization.

Two-year clinical outcomes after successful transcatheter aortic valve implantation with balloon-expandable versus self-expanding valves: A subanalysis of the GALILEO trial.

BACKGROUND: Midterm data comparing clinical outcomes after successful implantation of self-expanding and balloon-expandable transcatheter heart valves (THV) are limited. We aimed to compare 2-year outcomes after successful transcatheter aortic valve implantation (TAVI) with the Edwards balloon-expandable or the Medtronic self-expanding THV. METHODS: Two-year outcomes were analyzed according to the implanted THV in the GALILEO trial. Major adverse cardiac and cerebrovascular events (MACCE) was a composite of all-cause death or thromboembolic events including stroke, myocardial infarction, symptomatic valve thrombosis, systemic embolism, deep-vein thrombosis, or pulmonary embolism. RESULTS: Among 1644 patients recruited in 136 centers across 16 countries between 2015 and 2018, 499 received a self-expanding and 757 patients received a balloon-expandable THV. Patients treated with a self-expanding THV were more likely to be female, and had higher surgical risk, lower hemoglobin levels, and more frequent valve-in-valve procedures than those with a balloon-expandable THV. After multivariable adjustment, there were no significant differences in major clinical outcomes between self-expanding versus balloon-expandable THV: MACCE (17.0% vs. 13.4%, adjusted-hazard ratios [HR] 1.18, 95% confidence intervals [CI]: 0.82-1.69); all-cause death (11.4% vs. 9.3%, adjusted-HR 1.26; 95% CI: 0.78-2.05); cardiovascular death (8.5% vs. 4.0%, adjusted-HR 1.53; 95% CI: 0.82-2.86), any stroke (5.1% vs. 3.7%, adjusted-HR 0.86; 95% CI: 0.43-1.73); major or life-threatening bleeding (5.9% vs. 6.8%, adjusted-HR 0.93; 95% CI: 0.53-1.63). CLINICAL TRIAL REGISTRATION: https://www. CLINICALTRIALS: gov. NCT02556203. CONCLUSIONS: Two-year follow-up data from the GALILEO trial indicate that successful TAVI either with self-expanding or balloon-expandable THVs according to physician discretion did not show difference in rates of MACCE.

Characterization of quantitative flow ratio and fractional flow reserve discordance using doppler flow and clinical follow-up.

The physiological mechanisms of quantitative flow ratio and fractional flow reserve disagreement are not fully understood. We aimed to characterize the coronary flow and resistance profile of intermediate stenosed epicardial coronary arteries with concordant and discordant FFR and QFR. Post-hoc analysis of the DEFINE-FLOW study. Anatomical and Doppler-derived physiological parameters were compared for lesions with FFR+QFR- (n = 18) vs. FFR+QFR+ (n = 43) and for FFR-QFR+ (n = 34) vs. FFR-QFR- (n = 139). The association of QFR results with the two-year rate of target vessel failure was assessed in the proportion of vessels (n = 195) that did not undergo revascularization. Coronary flow reserve was higher [2.3 (IQR: 2.1-2.7) vs. 1.9 (IQR: 1.5-2.4)], hyperemic microvascular resistance lower [1.72 (IQR: 1.48-2.31) vs. 2.26 (IQR: 1.79-2.87)] and anatomical lesion severity less severe [% diameter stenosis 45.5 (IQR: 41.5-52.5) vs. 58.5 (IQR: 53.1-64.0)] for FFR+QFR- lesions compared with FFR+QFR+ lesions. In comparison of FFR-QFR+ vs. FFR-QFR- lesions, lesion severity was more severe [% diameter stenosis 55.2 (IQR: 51.7-61.3) vs. 43.4 (IQR: 35.0-50.6)] while coronary flow reserve [2.2 (IQR: 1.9-2.9) vs. 2.2 (IQR: 1.9-2.6)] and hyperemic microvascular resistance [2.34 (IQR: 1.85-2.81) vs. 2.57 (IQR: 2.01-3.22)] did not differ. The agreement and diagnostic performance of FFR using hyperemic stenosis resistance (> 0.80) as reference standard was higher compared with QFR and coronary flow reserve. Disagreement between FFR and QFR is partly explained by physiological and anatomical factors. Clinical Trials Registration https://www.clinicaltrials.gov ; Unique identifier: NCT01813435. Changes in central physiological and anatomical parameters according to FFR and QFR match/mismatch quadrants.

Fifteen-minute consultation: Childhood rumination syndrome.

Rumination syndrome is a functional bowel disorder where stomach contents effortlessly regurgitate into the mouth. It is an entirely separate entity to gastro-oesophageal reflux disease and requires behavioural rather than pharmaceutical management. Symptoms usually respond well to treatment, though in extreme cases, patients may experience significant weight loss and require inpatient admission.

Microwave Imaging of Breast Tumor Using Time-Domain UWB Circular-SAR Technique.

This paper explores the competency of the time domain ultra-wideband (UWB)-circular synthetic aperture radar (CSAR) to image the breast and detect tumors. The image reconstruction is performed using a time domain global back projection technique adapted to the circular trajectory data acquisition. This paper also proposes a sectional image reconstruction method to compensate for the group velocity changes in different layers of a multilayer medium. Experiments on an advanced breast phantom examines the suitability of this technique for breast tumor imaging. The advanced breast phantom is designed based on a MRI of a real patient, fabricated using 3D printing technology, and filled with liquids that emulate normal and cancerous tissues. The measurement results, compared with MRI imaging of the phantom, demonstrate the suitability of the UWB-CSAR method for breast tumor imaging. This method can be a tool for early diagnosis as well as for treatment monitoring during chemotherapy or radiotherapy.

Proposed Therapeutic Range of Treosulfan in Reduced Toxicity Pediatric Allogeneic Hematopoietic Stem Cell Transplant Conditioning: Results From a Prospective Trial.

Treosulfan is given off-label in pediatric allogeneic hematopoietic stem cell transplant. This study investigated treosulfan's pharmacokinetics (PKs), efficacy, and safety in a prospective trial. Pediatric patients (n = 87) receiving treosulfan-fludarabine conditioning were followed for at least 1 year posttransplant. PKs were described with a two-compartment model. During follow-up, 11 of 87 patients died and 12 of 87 patients had low engraftment (≤ 20% myeloid chimerism). For each increase in treosulfan area under the curve from zero to infinity (AUC(0-∞) ) of 1,000 mg hour/L the hazard ratio (95% confidence interval) for mortality increase was 1.46 (1.23-1.74), and the hazard ratio for low engraftment was 0.61 (0.36-1.04). A cumulative AUC(0-∞) of 4,800 mg hour/L maximized the probability of success (> 20% engraftment and no mortality) at 82%. Probability of success with AUC(0-∞) between 80% and 125% of this target were 78% and 79%. Measuring PK at the first dose and individualizing the third dose may be required in nonmalignant disease.

Intrabone hematopoietic stem cell gene therapy for adult and pediatric patients affected by transfusion-dependent ß-thalassemia.

ß-thalassemia is caused by ß-globin gene mutations resulting in reduced (ß+) or absent (ß0) hemoglobin production. Patient life expectancy has recently increased, but the need for chronic transfusions in transfusion-dependent thalassemia (TDT) and iron chelation impairs quality of life1. Allogeneic hematopoietic stem cell (HSC) transplantation represents the curative treatment, with thalassemia-free survival exceeding 80%. However, it is available to a minority of patients and is associated with morbidity, rejection and graft-versus-host disease2. Gene therapy with autologous HSCs modified to express ß-globin represents a potential therapeutic option. We treated three adults and six children with ß0 or severe ß+ mutations in a phase 1/2 trial ( NCT02453477 ) with an intrabone administration of HSCs transduced with the lentiviral vector GLOBE. Rapid hematopoietic recovery with polyclonal multilineage engraftment of vector-marked cells was achieved, with a median of 37.5% (range 12.6-76.4%) in hematopoietic progenitors and a vector copy number per cell (VCN) of 0.58 (range 0.10-1.97) in erythroid precursors at 1 year, in absence of clonal dominance. Transfusion requirement was reduced in the adults. Three out of four evaluable pediatric participants discontinued transfusions after gene therapy and were transfusion independent at the last follow-up. Younger age and persistence of higher VCN in the repopulating hematopoietic cells are associated with better outcome.

Why Do Patients Seek a Spine Surgeon?

STUDY DESIGN: Retrospective review of chief complaints (CCs) of patients seeking care at a specialty spine clinic with the diagnosis of degenerative scoliosis or lumbar stenosis. OBJECTIVES: The purpose of the study was to ascertain why patients seek care from spine surgeons. Specifically, we asked whether pain or deformity was more common. Secondarily, we studied the correlation of progressive curve magnitude with perceived functionality. BACKGROUND: Scant research is available on what leads a patient to be seen in a clinic. Degenerative scoliosis is often correlated with pain in the low back and extremities, symptoms that impinge on quality of life in the elderly. Some research suggests there is no correlation between progressive curve magnitude and perceived functionality. METHODS: Charts and radiographs of 351 consecutive patients were reviewed. Patient inclusion criteria were as follows: 1) they were seen at our spine clinic in one 12-month period, 2) their chief diagnosis was degenerative scoliosis or lumbar stenosis, 3) they were 50 years of age or older, and 4) they had no known prior history of scoliosis. Oswestry Disability Index (ODI) data were recorded. RESULTS: Of 351 patients, 160 reported their CC was combination back + leg pain on the initial visit survey, 123 complained of back pain only, and 42 complained of leg pain only. Ten complained of deformity or deformity + pain. Patients with degenerative scoliosis + spinal stenosis represented 25% of the study population; 11% were diagnosed with degenerative scoliosis only; 64% with stenosis only. Of the 122 patients with a Cobb angle of greater than 10°, only 10 complained of deformity or deformity + pain on the initial visit survey. CONCLUSIONS: Patients most often presented because of pain, specifically back, leg, or a combination of both. Patients seldom complained of deformity only, even among patients exhibiting a Cobb angle of greater than 30 degrees.

The role of diabetes mellitus in the composition of coronary thrombi in patients presenting with acute ST-segment elevation myocardial infarction undergoing primary percutaneous coronary intervention.

BACKGROUND: Although diabetes mellitus (DM) is a predictor of poor outcomes in patients with ST-segment elevation myocardial infarction (STEMI), few studies have analyzed the impact of DM on the constituency of coronary thrombi. OBJECTIVES: Comparing morphologic and histopathologic aspects of coronary thrombi in STEMI patients with and without DM who underwent primary percutaneous coronary intervention. METHODS: All consecutive patients with STEMI admitted to our institution between April 2010 and December 2012 (n = 1,548) were considered for inclusion. Thrombus material was obtained by aspiration thrombectomy; morphologic and histopathologic aspects were assessed by 3 independent pathologists blinded to clinical characteristics and outcomes. Patients with DM were compared with those without DM. A sensitivity analysis was performed using a propensity score. RESULTS: During the study period, coronary thrombi material from 259 patients was obtained, of whom 19% (n = 49) had diabetes. Diabetic patients were older (P = .10), had a higher frequency of hypertension (P < .01) and dyslipidemia (P = .03), and had a trend to a longer time from the onset of chest pain to hospital arrival (P = .08). The number of retrieved fragments, the size of the thrombi and its composition (leukocytes, fibrin, and erythrocytes percent), and thrombus age and color were similar between patients with or without DM. There were also no statistically significant differences in thrombus constituency of the propensity score-matched patients (n = 92). CONCLUSIONS: In this study, morphologic and histopathologic constituency of coronary thrombi in the setting of a ST-elevation myocardial infarction was not significantly different between patients with or without DM. This finding was intriguing and deserves further investigation.

Hybrid coronary revascularization versus coronary artery bypass surgery with bilateral or single internal mammary artery grafts.

OBJECTIVE: Hybrid coronary revascularization (HCR) combines minimally invasive left internal mammary artery (LIMA)-left anterior descending artery (LAD) bypass with percutaneous intervention of non-LAD vessels. The purpose of this study was to compare outcomes of HCR to conventional coronary artery bypass graft (CABG) surgery with single internal mammary artery (SIMA) or bilateral internal mammary artery (BIMA) grafting. METHODS: Between October 2003 and September 2013, 306 consecutive patients who underwent HCR were compared with 8254 patients who underwent CABG with SIMA (7381; 89.4%) or BIMA (873; 10.6%) at a US academic center. The primary outcome was a composite of 30-day death, myocardial infarction, and stroke (major cerebrovascular and cardiac event [MACCE]). In addition to multiple logistic and linear regression analysis, a propensity score-matched analysis was used to compare HCR with SIMA and with BIMA. RESULTS: The Society of Thoracic Surgeons-predicted risk of mortality was 1.6% for HCR, 2.1% for SIMA, and 1.1% for BIMA (P < .001). Factors associated with HCR use were older age, lower body mass index, history of percutaneous coronary intervention, and 2-vessel disease. In propensity-matched groups, 30-day MACCE rates were comparable (3.3% for HCR vs 1.3% for BIMA [odds ratio (OR), 2.50; P = .12] and vs 3.6% for SIMA [OR, 1.00; P = 1.00]). In-hospital complications were lower after HCR versus SIMA or BIMA (OR, 0.59; P = .033 and OR, 0.55; P = .015, respectively), as was the need for blood transfusion (OR, 0.44; P < .001 and OR, 0.57; P < .001). HCR was associated with shorter hospital stay compared with SIMA (OR, 1.28; P = .038) or BIMA (OR, 1.40; P = .006). No survival difference between matched groups was found at midterm follow-up (HCR vs SIMA: hazard ratio [HR], 0.66; 95% confidence interval [CI], 0.32-1.38; P = .66; HCR vs BIMA: HR, 1.05; 95% CI, 0.48-2.29; P = .91). CONCLUSIONS: HCR may represent a safe, less invasive alternative to conventional CABG in carefully selected patients, with similar short-term and midterm outcomes as CABG performed with either SIMA or BIMA grafting.

Designing and implementing maturity models in hospitals: An experience report from 5 years of research.

In recent years, a wide range of generic and domain-specific maturity models have been developed in order to improve organizational design and learning of healthcare organizations. While many of these studies describe methods on how to measure dedicated aspects of a healthcare organization's "maturity," little evidence exists on how to effectively implement and deploy them into practice. This article therefore delineates the encountered challenges during the design and implementation of three maturity models for distinct improvement areas in hospitals. On the one hand, this study's findings may serve as basis for refining existing maturity model design approaches. On the other hand, it may facilitate further research in domain-specific organizational design with maturity models.

Opportunities to diagnose chronic obstructive pulmonary disease in routine care in the UK: a retrospective study of a clinical cohort.

BACKGROUND: Patterns of health-care use and comorbidities present in patients in the period before diagnosis of chronic obstructive pulmonary disease (COPD) are unknown. We investigated these factors to inform future case-finding strategies. METHODS: We did a retrospective analysis of a clinical cohort in the UK with data from Jan 1, 1990 to Dec 31, 2009 (General Practice Research Database and Optimum Patient Care Research Database). We assessed patients aged 40 years or older who had an electronically coded diagnosis of COPD in their primary care records and had a minimum of 3 years of continuous practice data for COPD (2 years before diagnosis up to a maximum of 20 years, and 1 year after diagnosis) and at least two prescriptions for COPD since diagnosis. We identified missed opportunites to diagnose COPD from routinely collected patient data by reviewing patterns of health-care use and comorbidities present before diagnosis. We assessed patterns of health-care use in terms of lower respiratory consultations (infective and non-infective), lower respiratory consultations with a course of antibiotics or oral steroids, and chest radiography. If these events did not lead to a diagnosis of COPD, they were deemed to be missed opportunities. This study is registered with ClinicalTrials.gov, number NCT01655667. FINDINGS: We assessed data for 38,859 patients. Opportunities for diagnosis were missed in 32,900 (85%) of 38,859 patients in the 5 years immediately preceding diagnosis of COPD; in 12,856 (58%) of 22,286 in the 6-10 years before diagnosis, in 3943 (42%) of 9351 in the 11-15 years before diagnosis; and in 95 (8%) of 1167 in the 16-20 years before diagnosis. Between 1990 and 2009, we noted decreases in the age at diagnosis (0·05 years of age per year, 95% CI 0·03-0·07) and yearly frequency of lower respiratory prescribing consultations (rate ratio 0·982 opportunities per year, 95% CI 0·979-0·985). Prevalence of all comorbidities present at COPD diagnosis increased except for asthma and bronchiectasis, which decreased between 1990 and 2007, from 281 (33·4%) of 842 patients to 451 of 1465 (30·8%) for asthma, and from 53 of 842 (6·3%) to 53 of 1465 (3·6%) for bronchiectasis. In the 2 years before diagnosis, of 6897 patients who had had a chest radiography, only 2296 (33%) also had spirometry. INTERPRETATION: Opportunities to diagnose COPD at an earlier stage are being missed, and could be improved by case-finding in patients with lower respiratory tract symptoms and concordant long-term comorbidities. FUNDING: UK Department of Health, Research in Real Life.

Epithelial attachment and downgrowth on dental implant abutments--a comprehensive review.

UNLABELLED: The soft tissues around dental implants are enlarged compared with the gingiva because of the longer junctional epithelium and the hemidesmosonal attachments are fewer, suggestive of a poorer quality attachment. Inflammatory infiltrates caused by bacterial colonization of the implant-abutment interface are thought to be one of the factors causing epithelial downgrowth and subsequent peri-implant bone loss. Gold alloys and dental ceramics as well as the contamination of the implant surface with amino alcohols, appear to promote epithelial downgrowth. Physical manipulaton of the abutment surfaces, including concave abutment designs, platform switching, and microgrooved surfaces are believed to inhibit epithelial downgrowth and minimizes bone loss at the implant shoulder. This paper reviews the factors that are believed to influence the migration of epithelial attachment the dental implant and abutment surfaces. Exploration of innovative computer-aided design/computer-aided manufacturing-based concepts such as "one abutment-one time" and their effect on epithelial downgrowth are discussed. CLINICAL SIGNIFICANCE: Based on the review of current literature, the authors recommend inserting definitive abutments at the time of surgical uncovering. To implement this concept, registration of the implant position should to be taken at the time of surgical implant placement.

Changing strategies of the retrograde approach for chronic total occlusion during the past 7 years.

OBJECTIVE: We reviewed the technical changes and results achieved with the retrograde approach since we introduced it 7 years ago. SUBJECTS AND METHODS: The subjects were 1,268 patients who were treated for CTO between January 2004 and December 2010. They were investigated with respect to the success rate, the frequency of employing the retrograde approach and its outcome, and other factors. RESULTS: The retrograde approach was employed in ∼30% of chronic total occlusion (CTO) patients (n = 281) and the retrograde guidewire success rate was 81.1%. The kissing wire technique was substituted for the retrograde approach in 126 of the 281 patients, with antegrade crossing of a guidewire being successful in 88 of them (70%). The retrograde approach was combined with the CART and reverse controlled antegrade retrograde tracking (CART) techniques in 22 and 21 patients, respectively. Among 83 patients treated with Corsair catheters, crossing of the CTO was achieved in 63. The overall procedural success rate was 79.7% (224 patients). Complications of the retrograde approach included collateral channel dissection (2.1%), channel perforation (1.7%), CTO perforation (1.7%), and donor artery occlusion (1.1%). CONCLUSION: The success rate and safety of the retrograde approach are both satisfactory if the appropriate devices and techniques are selected.

Screening of healthcare workers for tuberculosis: development and validation of a new health economic model to inform practice.

BACKGROUND: Methods for determining cost-effectiveness of different treatments are well established, unlike appraisal of non-drug interventions, including novel diagnostics and biomarkers. OBJECTIVE: The authors develop and validate a new health economic model by comparing cost-effectiveness of tuberculin skin test (TST); blood test, interferon-gamma release assay (IGRA) and TST followed by IGRA in conditional sequence, in screening healthcare workers for latent or active tuberculosis (TB). DESIGN: The authors focus on healthy life years gained as the benefit metric, rather than quality-adjusted life years given limited data to estimate quality adjustments of life years with TB and complications of treatment, like hepatitis. Healthy life years gained refer to the number of TB or hepatitis cases avoided and the increase in life expectancy. The authors incorporate disease and test parameters informed by systematic meta-analyses and clinical practice. Health and economic outcomes of each strategy are modelled as a decision tree in Markov chains, representing different health states informed by epidemiology. Cost and effectiveness values are generated as the individual is cycled through 20 years of the model. Key parameters undergo one-way and Monte Carlo probabilistic sensitivity analyses. SETTING: Screening healthcare workers in secondary and tertiary care. RESULTS: IGRA is the most effective strategy, with incremental costs per healthy life year gained of £10 614-£20 929, base case, £8021-£18 348, market costs TST £45, IGRA £90, IGRA specificities of 99%-97%; mean (5%, 95%), £12 060 (£4137-£38 418) by Monte Carlo analysis. CONCLUSIONS: Incremental costs per healthy life year gained, a conservative estimate of benefit, are comparable to the £20 000-£30 000 NICE band for IGRA alone, across wide differences in disease and test parameters. Health gains justify IGRA costs, even if IGRA tests cost three times TST. This health economic model offers a powerful tool for appraising non-drug interventions in the market and under development.

Congenital thoracic scoliosis with unilateral unsegmented bar, convex hemivertebrae, and fused concave ribs with severe progression after posterior fusion at age 2: 40-year follow-up after revision anterior and posterior surgery at age 8.

STUDY DESIGN: Case report with 40-year follow-up after definitive surgery. OBJECTIVE: To show that extensive spine fusion in a young child can control the curve and does not necessarily lead to early death. SUMMARY OF BACKGROUND DATA: Recent efforts to avoid early spine fusion by using either progressive lengthening of spinal or rib cage implants are based on the theory that early spine fusion is deleterious and results in early death due to pulmonary compromise. Unfortunately, there is little to no documentation to support this theory. METHODS: This is a single case report of a child who at the age of 3 months had a 32° congenital thoracic congenital scoliosis with a unilateral unsegmented bar, concave fused ribs, and convex hemivertebrae. By age 2 years, the curve had progressed to 64°, so a posterior fusion was done from T5 to T12. By age 8 years, the curve had dramatically increased, and she was referred to the author. She underwent a double-wedge osteotomy, both anteriorly and posteriorly, plus anterior fusion T4-L3 and posterior fusion from T1 to L3. A halo cast was used for correction. RESULTS: At a 40-year follow-up after her definitive surgery at the age of 8 years, she is still alive and functioning well, although her vital capacity is poor. CONCLUSION: Extensive thoracic spine fusion at an early age did not result in early death, but the patient is far from ideal.