Publications by authors named "Jennifer Stimec"

40 Publications

Tibia stress injury and the imaging appearance of stress fracture in juvenile dermatomyositis: six patients' experiences.

Pediatr Rheumatol Online J 2021 Feb 17;19(1):17. Epub 2021 Feb 17.

Division of Rheumatology, Department of Pediatrics, The Hospital for Sick Children, 555 University Ave, M5G 1X8, Toronto, ON, Canada.

Background: Tibial stress injuries are frequent injuries of the lower extremity and the most common causes of exercise-induced leg pain among athletes and military recruits. They sometimes occur in patients with pathological conditions of bone metabolism such as osteoporosis or rheumatoid arthritis, but there are previously no cases reported in juvenile dermatomyositis (JDM). Here we report 6 JDM patients who presented with shin pain, and the imaging appearance of tibial stress fractures or stress reactions.

Case Presentation: All 6 patients with JDM presented with shin pain or tenderness in the anterior tibia without any evidence of excessive exercise or traumatic episode. They were diagnosed with tibial stress injuries based on a combination of radiographs, three-phase bone scans, and magnetic resonance imaging (MRI), and 5 out of 6 patients had been treated with prednisone and/or methotrexate at onset of tibial stress injuries. In one patient, we could not find any abnormalities in his radiograph, but the subsequent MRI showed tibial stress reaction. In all 6 patients, the tibial stress injuries improved with only rest and/or analgesics.

Conclusion: We experienced 6 children with JDM who presented with shin pain, and who were diagnosed with tibial stress fractures or stress reactions. Their underlying disease and weakness, treatment with glucocorticoid and methotrexate, or inactivity may have resulted in these tibial injuries, and made these patients more predisposed than other children. In addition to preventing JDM patients from getting osteoporosis, we need to consider stress reactions when children with JDM complain of sudden shin pain.
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http://dx.doi.org/10.1186/s12969-021-00501-9DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7890837PMC
February 2021

Discrete Choice Experiment on a Magnetic Resonance Imaging Scoring System for Temporomandibular Joints in Juvenile Idiopathic Arthritis.

Arthritis Care Res (Hoboken) 2021 Feb 8. Epub 2021 Feb 8.

The Hospital for Sick Children, Toronto, ON, Canada.

Objective: To determine the relative importance weights of items and grades of a newly developed additive outcome measure called the juvenile idiopathic arthritis (JIA) magnetic resonance imaging (MRI) scoring system for temporomandibular joints (TMJ, JAMRIS-TMJ).

Methods: An adaptive partial-profile discrete choice experiment (DCE) survey using the 1000Minds platform was independently completed by members of an expert group consisting of radiologists and non-radiologist clinicians to determine the group-averaged relative weights for JAMRIS-TMJ. Subsequently, an image-based vignette ranking exercise was done, during which experts individually rank-ordered 14 patient vignettes for disease severity while blinded to the weights and unrestricted to JAMRIS-TMJ assessment criteria. Validity of the weighted JAMRIS-TMJ was tested by comparing the consensus-graded, DCE-weighted JAMRIS-TMJ score of the vignettes with their unrestricted image-based ranks provided by the experts.

Results: Nineteen experts completed the DCE survey and 21 completed the vignette ranking exercise. Synovial thickening and joint enhancement showed higher weights per raw score compared to bone marrow items and effusion in the inflammatory domain, while erosions and condylar flattening showed non-linear and higher weights compared to disk abnormalities in the damage domain. The weighted JAMRIS-TMJ score of the vignettes correlated highly with the ranks from the unrestricted comparison method, with median Spearman's rho of 0.92 (intra-quartile range: 0.87-0.95) for the inflammation and 0.93 (0.90-0.94) for the damage domain.

Conclusions: A DCE survey was used to quantify the importance weights of the items and grades of the JAMRIS-TMJ. The weighted score showed high convergent validity with an unrestricted, holistic vignette ranking method.
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http://dx.doi.org/10.1002/acr.24577DOI Listing
February 2021

Home Management Versus Primary Care Physician Follow-up of Patients With Distal Radius Buckle Fractures: A Randomized Controlled Trial.

Ann Emerg Med 2021 02 21;77(2):163-173. Epub 2020 Oct 21.

Division of Emergency Medicine, Department of Pediatrics, Hospital for Sick Children and University of Toronto, Toronto, Ontario, Canada. Electronic address:

Study Objective: In patients with a distal radius buckle fracture, we determine whether home removal of a splint and physician follow-up as needed (home management) is noninferior to primary care physician follow-up in 1 to 2 weeks with respect to functional recovery. We also compare groups with respect to health care and patient-level costs.

Methods: This was a noninferiority randomized controlled trial conducted at a tertiary care children's hospital. Eligible patients were randomized to home management versus primary care physician follow-up and received telephone contact at 3 and 6 weeks after the index ED visit. Functional recovery was measured with the Activities Scale for Kids-performance, and participants reported wrist-injury-related health care interventions and expenses. The primary outcome was a comparison of the performance score between groups at 3 weeks.

Results: We enrolled 149 patients with mean age 9.5 years (SD 2.7 years), and 81 (54.4%) were male patients. Of the 133 patients (89.3%) with completed 3-week follow-up, the mean Activities Scale for Kids-performance score was 95.4% in the home management group (n=66) and 95.9% in the primary care physician follow-up group (n=67) (mean difference -0.4%; lower bound of the 95% confidence interval -2.4%). There was a mean costs savings of -$100.10 (95% confidence interval -$130.0 to -$70.20) in health care and -$28.2 (95% confidence interval -$49.6 to -$7.0) in patient costs in the home management versus primary care physician follow-up group.

Conclusion: In patients with distal radius buckle fractures, home management is at least as good as primary care physician follow-up with respect to functional recovery. Implementation of the home management strategy also demonstrated significant cost savings.
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http://dx.doi.org/10.1016/j.annemergmed.2020.07.039DOI Listing
February 2021

Radiological Assessment and Outcome of Local Disease Progression after Neoadjuvant Chemotherapy in Children and Adolescents with Localized Osteosarcoma.

J Clin Med 2020 Dec 17;9(12). Epub 2020 Dec 17.

Division of Hematology/Oncology, Hospital for Sick Children, Toronto, ON M5G 1X8, Canada.

Objective: We examined the interobserver reliability of local progressive disease (L-PD) determination using two major radiological response evaluation criteria systems (Response evaluation Criteria in Solid Tumors (RECIST) and the European and American Osteosarcoma Study (EURAMOS)) in patients diagnosed with localized osteosarcoma (OS). Additionally, we describe the outcomes of patients determined to experience L-PD.

Materials And Methods: Forty-seven patients diagnosed with localized OS between 2000 and 2012 at our institution were identified. Paired magnetic resonance imaging of the primary tumor from diagnosis and post-neoadjuvant chemotherapy were blindly assessed by two experienced radiologists and determined L-PD as per RECIST and EURAMOS radiological criteria. Interobserver reliability was measured using the kappa statistic (κ). The Kaplan Meier method and log-rank test was used to assess differences between groups.

Results: Of 47 patients (median age at diagnosis 12.9 years), 16 (34%) had L-PD (by RECIST or EURAMOS radiological definition). There was less agreement between the radiologists using EURAMOS radiological criteria for L-PD (80.9%, κ = 0.48) than with RECIST criteria (97.9%, κ = 0.87). Patients with radiologically defined L-PD had a 5-year progression-free survival (PFS) of 55.6%, compared to a 5 year-PFS of 82.7% in the group of patients without L-PD ( = 31) (Log rank = 0.0185).

Conclusions: The interobserver reliability of L-PD determination is higher using RECIST than EURAMOS. RECIST can be considered for response assessment in OS clinical trials. The presence of L-PD was associated with worse outcomes.
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http://dx.doi.org/10.3390/jcm9124070DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7767085PMC
December 2020

Whole-Body MRI Quantification for Assessment of Bone Lesions in Chronic Nonbacterial Osteomyelitis Patients Treated With Pamidronate: A Prevalence, Reproducibility, and Responsiveness Study.

J Rheumatol 2020 Sep 15. Epub 2020 Sep 15.

J. Panwar, MD, FRCR, Department of Radiodiagnosis, Christian Medical College and Hospital, Vellore, India and Department of Diagnostic Imaging, The Hospital for Sick Children, Department of Medical Imaging, University of Toronto, Toronto, Ontario; M. Tolend, BSc, A.S. Doria, MD, PhD, MSc, MBA, J. Stimec, MD, FRCPC, Department of Diagnostic Imaging, The Hospital for Sick Children, Department of Medical Imaging, University of Toronto, Toronto, Ontario; L. Lim, MD, FRCPC, Division of Pediatric Rheumatology, Department of Pediatrics, University of Alberta, Edmonton, Alberta; S.M. Tse, MD, FRCPC, R.M. Laxer, MDCM, FRCPC, Division of Rheumatology, The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada. The authors declare no conflicts of interest. Address correspondence to Dr. J. Panwar, Department of Radiodiagnosis, Christian Medical College and Hospital, Vellore 632004, India, and Department of Diagnostic Imaging, The Hospital for Sick Children, Department of Medical Imaging, University of Toronto, Toronto, ON M5G 1X8, Canada. Email: Accepted for publication September 5, 2020.

Objective: The purpose of this study was (1) to assess the interreader reliability in detecting and scoring the inflammatory bone lesions in pediatric patients with chronic nonbacterial osteomyelitis (CNO) by using whole-body magnetic resonance imaging (WB-MRI), and (2) to evaluate the responsiveness of the MRI-detected CNO lesions to pamidronate therapy.

Methods: Eighty-eight WB-MRI examinations were independently reviewed and scored by 2 radiologists blinded to clinical details in 32 retrospectively enrolled pediatric patients with CNO. Inflammatory bone lesions, soft tissue abnormality, and bony structural changes were scored before and after pamidronate therapy. Lesion responsiveness was calculated by using standardized response mean and interreader reliability was assessed by k statistics.

Results: There was good to excellent interreader agreement for the detection and quantification of bone lesions. After the first cycle of pamidronate in all 32 patients, 96 of the 279 lesions (34%; after excluding 108 lesions of hand and feet) resolved, whereas in a subset of 11 patients with 2 or more cycles, 76% of lesions resolved after the second cycle. Twenty-one (7.5%) lesions worsened and 46 (16.4%) new lesions developed after 1 cycle in all 32 patients. In these 11 patients, the number of worsened lesions reduced to 2 (2%) and new lesions to 14 (14.9%) after the second cycle as detected on MRI. Vertebral lesions had the highest response to treatment.

Conclusion: WB-MRI is a reliable tool for objective quantification and assessment of response to treatment of pediatric CNO bone lesions and could be used to monitor disease activity for clinical and research purposes.
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http://dx.doi.org/10.3899/jrheum.200329DOI Listing
September 2020

Monogenic autoinflammatory diseases in children: single center experience with clinical, genetic, and imaging review.

Insights Imaging 2020 Jul 31;11(1):87. Epub 2020 Jul 31.

Department of Diagnostic Imaging, The Hospital for Sick Children, University of Toronto, Toronto, Canada.

Purpose: 1. To review the contemporary literature and present a list of the imaging findings for patients with autoinflammatory diseases from our hospital. All these patients are found to have a genetic mutation that is responsible for their disease. 2. To present follow-up imaging findings, when available, and correlate those with symptoms and type of treatment administered in approximately 40 patients with autoinflammatory diseases of a single tertiary pediatric health care center including familial Mediterranean fever, Cryopyrin-associated autoinflammatory syndrome, PAPA (pyogenic arthritis, pyoderma gangrenousum, and acne) syndrome, and more. These findings are related to disease progression, treatment response, or treatment-induced changes.

Conclusion: Autoinflammatory diseases are relatively rare entities that can affect any system of the body. Given the many nonspecific imaging features, awareness of these diseases and good communication with clinicians aid in reaching an accurate diagnosis.
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http://dx.doi.org/10.1186/s13244-020-00889-0DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7394994PMC
July 2020

Checking the basis of intraosseous access-Radiological study on tibial dimensions in the pediatric population.

Paediatr Anaesth 2020 10 28;30(10):1116-1123. Epub 2020 Aug 28.

Department of Anesthesia, University Children's Hospital, Zurich, Switzerland.

Background: Malposition of intraosseous needles in pediatric patients is frequently reported. Incorrect needle length and penetration depth related to the puncture site and level are possible causes.

Aims: Aim of this study was to analyze anatomic dimensions of the proximal tibia in the pediatric population with respect to intraosseous needle placement and needle tip position.

Methods: Plain lower leg radiographs of children aged from birth to 16 years of age were analyzed. Pretibial tissue layer, cortical bone thickness, and the diameter of the medullary cavity were measured at two different puncture levels. Data were analyzed as descriptive statistics and by polynomial regression plots and set in context to commonly used EZ-IO needle lengths of 15 and 25 mm.

Results: Radiographs from 190 patients (104 boys/86 girls) were included. When fully inserted to skin level, up to 10.5% of needles do not reach medullary cavity at one and 18.5% at two patient's fingerbreadths distal to tibial tuberosity. The opposite cortical wall is touched or penetrated in 16% and 25%, respectively. Up to 96% of too deep needle tip positions occur in children younger than 24 months, as do too superficial tip positions in 59%.

Conclusions: Puncture level and needle length have a great influence on potential needle tip positions. Infants and toddlers are at highest risk for malpositioning. Due to relevant growth-related differences in tibial anatomy, an age-related and well-reflected approach is crucial to successfully establish intraosseous access.
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http://dx.doi.org/10.1111/pan.13979DOI Listing
October 2020

Image guided sacroiliac joint corticosteroid injections in children: an 18-year single-center retrospective study.

Pediatr Rheumatol Online J 2020 Jun 17;18(1):52. Epub 2020 Jun 17.

Division of Image Guided Therapy, Department of Diagnostic Imaging, The Hospital for Sick Children, University of Toronto, 555 University Avenue, Toronto, ON, M5G 1X8, Canada.

Background: Sacroiliitis is commonly seen in enthesitis-related arthritis (ERA), a subtype of juvenile idiopathic arthritis (JIA). Sacroiliitis is characterized by the inflammation of the sacroiliac (SI) joints (+/- adjacent tissues). The treatment options include systemic therapy with or without corticosteroid SI joint injections. Image guided SI joint injections are frequently requested in pediatric patients with sacroiliitis. The purpose of this study was to evaluate the feasibility and efficacy of SI joint injections in children with sacroiliitis.

Methods: A retrospective study of patients referred to Interventional Radiology (IR) for SI joint corticosteroid injections (2000-2018). Clinical information was collected from Electronic Patient Charts and procedural details from PACS. Efficacy was determined clinically, by MRI, or both when available.

Results: 50 patients (13.8 years; M:F = 35:15) underwent image-guided SI joint corticosteroid injections. Most common indications were JIA (84%) and inflammatory bowel disease (14%). 80% had bilateral injections. 80% were performed under general anesthesia and 20% under sedation. The corticosteroid of choice was triamcinolone hexacetonide in 98% of patients. Needle guidance and confirmation was performed using CT and fluoroscopy (54%), Cone Beam CT (CBCT, 46%), with initial ultrasound assistance in 34%. All procedures were technically successful without any complications. 32/50 patients had long-term follow-up (2 years); 21/32 (66%) had clinical improvement within 3-months. Of 15 patients who had both pre- and post-procedure MRIs, 93% showed short-term improvement. At 2 years, 6% of patients were in remission, 44% continued the same treatment and 47% escalated treatment.

Conclusion: Image-guided SI joint injections are safe and technically feasible in children. Imaging modalities for guidance have evolved, with CBCT being the current first choice. Most patients showed short-term clinical and imaging improvement, requiring long-term maintenance or escalation of medical treatment.
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http://dx.doi.org/10.1186/s12969-020-00435-8DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7301971PMC
June 2020

Critical appraisal of the International Prophylaxis Study Group magnetic resonance image scale for evaluating haemophilic arthropathy.

Haemophilia 2020 Jul 4;26(4):565-574. Epub 2020 Jun 4.

Department of Diagnostic Imaging, The Hospital for Sick Children, University of Toronto, Toronto, Canada.

A goal of the International Prophylaxis Study Group (IPSG) is to provide an accurate instrument to measure MRI-based disease severity of haemophilic arthropathy at various time points, so that longitudinal changes in disease severity can be identified to support decisions on treatment management. We review and discuss in this paper the evaluative purpose of the IPSG MRI scale in relation to its development and validation processes so far. We also critically appraise the validity, reliability and responsiveness of using the IPSG MRI scale in different clinical and research settings, and whenever applicable, compare these clinimetric properties of the IPSG MRI scale with those of its precursors, the compatible additive and progressive MRI scales.
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http://dx.doi.org/10.1111/hae.14032DOI Listing
July 2020

Severe Primary Hyperparathyroidism Caused by Parathyroid Carcinoma in a 13-Year-Old Child; Novel Findings From HRpQCT.

JBMR Plus 2020 Mar 2;4(3):e10324. Epub 2020 Jan 2.

Department of Pediatrics, Division of Endocrinology Toronto Canada.

Primary hyperparathyroidism is a condition that occurs infrequently in children. Parathyroid carcinoma, as the underlying cause of hyperparathyroidism in this age group, is extraordinarily rare, with only a few cases reported in the literature. We present a 13-year-old boy with musculoskeletal pain who was found to have brown tumors from primary hyperparathyroidism caused by parafibromin-immunodeficient parathyroid carcinoma. Our patient had no clinical, biochemical, or radiographic evidence of pituitary adenomas, pancreatic tumors, thyroid tumors, pheochromocytoma, jaw tumors, renal abnormalities, or testicular lesions. Germline testing for , , , , , , and the gene showed no pathological variants, and a microarray of did not reveal deletion or duplication. He was managed with i.v. fluids, calcitonin, pamidronate, and denosumab prior to surgery to stabilize hypercalcemia. After removal of a single parathyroid tumor, he developed severe hungry bone syndrome and required 3 weeks of continuous i.v. calcium infusion, in addition to oral calcium and activated vitamin D. Histopathological examination identified an angioinvasive parathyroid carcinoma with global loss of parafibromin (protein encoded by )HRpQCT and DXA studies were obtained prior to surgery and 18-months postsurgery. HRpQCT showed a resolution of osteolytic lesions combined with structural improvement of cortical porosity and an increase in both cortical thickness and density compared with levels prior to treatment. These findings highlight the added value of HRpQCT in primary hyperparathyroidism. In addition to our case, we have provided a review of the published cases of parathyroid cancer in children. © 2019 The Authors. published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.
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http://dx.doi.org/10.1002/jbm4.10324DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7059826PMC
March 2020

Toward Developing a Semiquantitative Whole Body-MRI Scoring for Juvenile Idiopathic Arthritis: Critical Appraisal of the State of the Art, Challenges, and Opportunities.

Acad Radiol 2021 02 3;28(2):271-286. Epub 2020 Mar 3.

Department of Diagnostic Imaging, The Hospital for Sick Children, Department of Medical Imaging, University of Toronto, Toronto, ON, Canada. Electronic address:

With powerful new therapies available for management of juvenile idiopathic arthritis (JIA), early diagnosis leading to appropriate treatment may prevent long-term structural joint damage. Although magnetic resonance imaging (MRI) is typically used to assess individual body parts, indications for whole body (WB) MRI are increasing. Its utility as a diagnostic and monitoring tool has already been widely investigated in adult rheumatology patients, but less so in pediatric rheumatologic patients. This paper is a comprehensive review of scoring systems and a proposal for the conceptual development of a WB-MRI scoring system for the evaluation of JIA. In this review we identify, summarize, and critically appraise the available literature on the use of WB-MRI in inflammatory arthritis, addressing relevant considerations on components of a classification system that can lead to the development of a future pediatric WB-MRI scoring system for use in children with JIA. We also discuss advantages and challenges of developing such a WB-MRI scoring system for assessment of JIA and outline next steps toward the conceptual development of this scoring system.
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http://dx.doi.org/10.1016/j.acra.2020.01.022DOI Listing
February 2021

Establishing Safe Zones to Avoid Nerve Injury in the Approach to the Humerus in Pediatric Patients: A Magnetic Resonance Imaging Study.

J Bone Joint Surg Am 2019 Dec;101(23):2101-2110

University Hospitals Bristol, Bristol, England.

Background: The surgical anatomy of upper-extremity peripheral nerves in adults has been well described as "safe zones" or specific distances from osseous landmarks. In pediatrics, relationships between nerves and osseous landmarks remain ambiguous. The goal of our study was to develop a model to accurately predict the location of the radial and axillary nerves in children to avoid iatrogenic injury when approaching the humerus in this population.

Methods: We conducted a retrospective review of 116 magnetic resonance imaging (MRI) scans of entire humeri of skeletally immature patients; 53 of these studies met our inclusion criteria. Two independent observers reviewed all scans. Arm length was measured as the distance between the lateral aspect of the acromion and the lateral epicondyle. We then calculated the distances (defined as the percentage of arm length) between the radial nerve and distal osseous landmarks (the medial epicondyle, transepicondylar line, and lateral epicondyle) as well between the axillary nerve and the most lateral aspect of the acromion.

Results: The axillary nerve was identified at a distance equaling 18.6% (95% confidence interval [CI], ±0.62%) of arm length inferior to the lateral edge of the acromion. The radial nerve crossed (1) the medial cortex of the posterior part of the humerus at a distance equaling 63.19% (95% CI: ±0.942%) of arm length proximal to the medial epicondyle, (2) the middle of the posterior part of the humerus at a distance equaling 53.9% (95% CI: ±1.08%) of arm length proximal to the transepicondylar line, (3) the lateral cortex of the posterior part of the humerus at a distance equaling 45% (95% CI: ±0.99%) of arm length proximal to the lateral epicondyle, and (4) from the posterior to the anterior compartment at a distance equaling 35.3% (95% CI: ±0.92%) of arm length proximal to the lateral epicondyle. A strong linear relationship between these distances and arm length was observed, with an intraclass correlation coefficient of >0.9 across all measurements.

Conclusions: The positions of the radial and axillary nerves maintain linear relationships with arm lengths in growing children. The locations of these nerves in relation to palpable osseous landmarks are predictable.

Clinical Relevance: Knowing the locations of upper-extremity peripheral nerves as a proportion of arm length in skeletally immature patients may help to avoid iatrogenic injuries during surgical approaches to the humerus.
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http://dx.doi.org/10.2106/JBJS.19.00019DOI Listing
December 2019

Adverse Events from Emergency Physician Pediatric Extremity Radiograph Interpretations: A Prospective Cohort Study.

Acad Emerg Med 2020 02 15;27(2):128-138. Epub 2019 Dec 15.

Division of Pediatric Emergency Medicine, Department of Pediatrics, Child Health Evaluative Sciences, Research Institute, Hospital for Sick Children and University of Toronto, Toronto, Ontario, Canada.

Objectives: We determined how often emergency physician pediatric musculoskeletal (MSK) radiograph interpretations were discordant to that of a radiologist and led to an adverse event (AE). We also established the variables independently associated with this outcome.

Methods: This prospective cohort study was conducted in an urban, tertiary care children's emergency department (ED). We enrolled children who presented to an ED with an extremity injury and received radiographs. ED physicians documented their radiograph interpretation, which was compared to a radiology reference standard. Patients received telephone follow-up and had institutional medical records reviewed in 3 weeks. An AE occurred if there were clinical sequelae and/or repeat health care visits due to a delay in correct radiograph interpretation.

Results: We enrolled 2,302 children (mean [±SD] age = 9.0 [4.4] years; 1,288 (56.0%) male]. Of these, 180 (7.8%; 95% confidence interval = 6.8 to 9.0) ED physician discordant interpretations resulted in an AE. Specifically, there were no negative clinical outcomes; however, relative to cases diagnosed correctly at the index ED, patients whose fracture was not initially identified encountered 77.2% more subsequent ED visits, while those falsely diagnosed with a fracture experienced 41.5% additional orthopedic clinic visits. Odds of an ED discrepant interpretation was significantly higher if a physician's pretest probability of a fracture was ≤ 20% versus> 20% (adjusted odds ratio [aOR] = 1.6), patient's pain score was ≤ 2 versus> 2 (aOR = 1.6), and injury was located in a joint versus other location (aOR = 1.7).

Conclusions: Emergency physician discordant pediatric MSK radiograph interpretations that resulted in an AE occurred with regular frequency in a pediatric ED setting. AEs were primarily an increase in subsequent health care visits. Importantly, a low clinical suspicion for a fracture or injury located in the joint were risk factors for ED physician discordant interpretations.
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http://dx.doi.org/10.1111/acem.13884DOI Listing
February 2020

Development of a Scoring Tool for Chronic Nonbacterial Osteomyelitis Magnetic Resonance Imaging and Evaluation of its Interrater Reliability.

J Rheumatol 2020 05 1;47(5):739-747. Epub 2019 Oct 1.

From the Seattle Children's Hospital, Department of Pediatrics, and Department of Radiology, University of Washington; Center for Clinical and Translational Research, Seattle Children's Research Institute, Seattle, Washington; Department of Radiology, University of Iowa Carver College of Medicine, Iowa City, Iowa, USA; Musculoskeletal Statistics Unit, The Parker Institute, Bispebjerg and Frederiksberg Hospital, Copenhagen; The Rheumatology Research Unit, Department of Rheumatology, Odense University Hospital and University of Southern Denmark, Odense, Denmark; Department of Diagnostic and Interventional Radiology, University Hospital of Ulm, Ulm, Germany; Department of Radiology, Stony Brook University Hospital, Stony Brook, New York; Department of Radiology, Hackensack University Medical Center, Hackensack, New Jersey, USA; Department of Medical Imaging, Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada; Department of Radiology, Christian Medical College and Hospital, Vellore, India; Department of Imaging, Bambino Gesù Children's Hospital, Institute for Research and Health Care (IRCCS), Rome, Italy; Pediatric Rheumatology, Children's Healthcare of Atlanta, Emory University, Atlanta, Georgia; Department of Pediatrics, University of Iowa Carver College of Medicine, Iowa City, Iowa, USA.

Objective: Serial magnetic resonance imaging (MRI) examinations are often needed in chronic nonbacterial osteomyelitis (CNO) to determine the objective response to treatment. Our objectives in this study were (1) to develop a consensus-based MRI scoring tool for clinical and research use in CNO; and (2) to evaluate interrater reliability and agreement using whole-body (WB)-MRI from children with CNO.

Methods: Eleven pediatric radiologists discussed definitions and grading of signal intensity, size of signal abnormality within bone marrow, and associated features on MRI through monthly conference calls and a consensus meeting, using a nominal group technique in July 2017. WB-MRI scans from children with CNO were deidentified for training reading and an interrater reliability study. The reading by each radiologist was conducted in a randomized order. Interrater reliability for abnormal signal and severity were assessed using free-marginal κ statistics.

Results: Radiologists reached a consensus on grading CNO-specific MRI findings and on describing bone units based on anatomy. A total of 45 sets of WB-MRI scans, including 4 sets of non-CNO MRI examinations, were selected for the final reading. The mean κ of each category of bones was > 0.7 with majority > 0.9 demonstrating substantial/almost perfect interrater reliability of readings among radiologists. The agreement on signal intensity and the size of signal abnormality within the most commonly affected bones (femur and tibia) were lower than those of other bones.

Conclusion: The chronic nonbacterial osteomyelitis magnetic resonance imaging scoring (CROMRIS) tool is a comprehensive standardized scoring tool for MRI in children with CNO. Our interrater study demonstrated good interrater reliability and agreement of readings.
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http://dx.doi.org/10.3899/jrheum.190186DOI Listing
May 2020

Osteofibrous Dysplasia of the Tibia in Children: Outcome Without Resection.

J Pediatr Orthop 2019 Sep;39(8):e614-e621

The Hospital for Sick Children, Toronto, ON, Canada.

Background: The proposed association between osteofibrous dysplasia and adamantinoma has led some to advocate resection of the entire lesion, which can require major subsequent reconstruction. However, this link remains unproven and there is some support in more recent literature for a less aggressive approach. This study aims to describe our experience managing pediatric tibial osteofibrous dysplasia with such an approach and to report functional outcomes in children treated thus.

Methods: A total of 28 cases of osteofibrous dysplasia in 25 patients were managed at a referral center for pediatric bone tumors with observation in the first instance, then limited surgical intervention if required to address pain and deformity. Surgery aimed to restore stability and alignment without excising the lesion. Clinical records provided basic clinical outcome measures involving walking, recreation, orthoses and school/work participation and patients provided a Musculoskeletal Tumour Society score (MSTS) where contactable.

Results: Mean age at presentation was 6.0 years and mean follow-up was 8.3 years. Only 8 patients required surgery. According to basic outcome measures, 13 patients were symptom-free. About 15 patients (17 cases) provided a MSTS and the mean score was 24 of 30. No transformation to adamantinoma was observed. Those who presented at a younger age and with bilateral disease more often required surgery and remained symptomatic.

Conclusions: A less aggressive approach to pediatric tibial osteofibrous dysplasia achieves good functional outcomes and patient satisfaction in most cases. Surgery is required in the minority of cases. Transformation to adamantinoma was not observed in this series. We recommend patient education, clinical observation and reactive intervention if required, rather than proactive resection and reconstruction.

Level Of Evidence: Level IV-case series.
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http://dx.doi.org/10.1097/BPO.0000000000001116DOI Listing
September 2019

Building Emergency Medicine Trainee Competency in Pediatric Musculoskeletal Radiograph Interpretation: A Multicenter Prospective Cohort Study.

AEM Educ Train 2019 Jul 12;3(3):269-279. Epub 2019 Mar 12.

Division of Pediatric Emergency Medicine Department of Pediatrics Hospital for Sick Children and University of Toronto Toronto Ontario Canada.

Objectives: As residency programs transition from time- to performance-based competency standards, validated tools are needed to measure performance-based learning outcomes and studies are required to characterize the learning experience for residents. Since pediatric musculoskeletal (MSK) radiograph interpretation can be challenging for emergency medicine trainees, we introduced Web-based pediatric MSK radiograph learning system with performance endpoints into pediatric emergency medicine (PEM) fellowships and determined the feasibility and effectiveness of implementing this intervention.

Methods: This was a multicenter prospective cohort study conducted over 12 months. The course offered 2,100 pediatric MSK radiographs organized into seven body regions. PEM fellows diagnosed each case and received feedback after each interpretation. Participants completed cases until they achieved a performance benchmark of at least 80% accuracy, sensitivity, and specificity. The main outcome measure was the median number of cases completed by participants to achieve the performance benchmark.

Results: Fifty PEM fellows from nine programs in the US and Canada participated. There were 301 of 350 (86%) modules started and 250 of 350 (71%) completed to the predefined performance benchmark during the study period. The median (interquartile range [IQR]) number of cases to performance benchmark per participant was 78 (60-104; min = 56, max = 1,333). Between modules, the median number of cases to achieve the performance benchmark was different for the ankle versus other modules (ankle 366 vs. other 76; difference = 290, 95% confidence interval [CI] = 245 to 335). The performance benchmark was achieved for 90.7% of participants in all modules except the ankle/foot, where 34.9% achieved this goal (difference = 55.8%, 95% CI = 45.3 to 66.3). The mean (95% CI) change in accuracy, sensitivity, and specificity from baseline to performance benchmark was +14.6% (13.4 to 15.8), +16.5% (14.8 to 18.1), and +12.6% (10.7 to 14.5), respectively. Median (IQR) time on each case was 31.0 (21.0-45.3) seconds.

Conclusions: Most participants completed the modules to the performance benchmark within 1 hour and demonstrated significant skill improvement. Further, there was a large variation in the number of cases completed to achieve the performance endpoint in any given module, and this impacted the feasibility of completing specific modules.
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http://dx.doi.org/10.1002/aet2.10329DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6637005PMC
July 2019

Thiemann disease and familial digital arthropathy - brachydactyly: two sides of the same coin?

Orphanet J Rare Dis 2019 06 27;14(1):156. Epub 2019 Jun 27.

Division of Clinical and Metabolic Genetics, The Hospital for Sick Children and University of Toronto, Toronto, ON, M5G 1X8, Canada.

Background: Familial digital arthropathy-brachydactyly (FDAB) and Thiemann disease are non-inflammatory digital arthropathies with many phenotypic similarities. Thirty-three cases of Thiemann disease have been described so far (Mangat et al, Ann Rheum Dis 64:11-2, 2005; Ha et al, Thiemann's disease: a case Report, 2017) but no gene variants have been identified as causative to date. FDAB is reported in only a few patients and has been associated with three heterozygous missense variants in the Transient receptor potential vanilloid 4 (TRPV4) gene. We report a TRPV4 variant in a father and son referred with a diagnosis of Thiemann disease and compare the clinical and radiological features of Thiemann disease with Familial digital arthropathy-brachydactyly (FDAB). We hypothesize that these two entities may be one and the same.

Methods: We describe a father and son referred with a diagnosis of Thiemann disease who were subsequently identified with a heterozygous variant (c.809G > T) in TRPV4. The identical genetic variant was previously reported to cause FDAB. A PUBMED® database search was conducted to retrieve articles related to Thiemann disease and FDAB. We were able to review the clinical and radiological findings of nineteen individuals affected by Thiemann disease and compare them with three families affected by FDAB.

Results: Thiemann disease initially affects the proximal interphalangeal joints and primarily the middle phalangeal bases. In FDAB, the distal phalangeal joints are first affected with the middle phalangeal heads being the primary site of changes. Radial deviation has only been described in FDAB. Our analysis determined that 5 of 20 individuals affected by Thiemann disease have clinical and radiological findings that also fit well with FDAB.

Conclusion: FDAB and Thiemann disease are non-inflammatory digital arthropathies with phenotypic overlap. Although more extensive joint involvement, a distal hand joint preponderance and brachydactyly are expected in FDAB, there are striking clinical and radiological similarities between the two entities. Our analysis suggests that these two phenotypes may represent phenotypic variability of the same entity. Despite many attempts to identify other reported patients affected by Thiemann disease, we were not able to procure DNA from any of the cases to verify our findings. Genetic testing of an affected individual will be crucial in order to provide accurate reproductive genetic counselling about the autosomal dominant nature of this condition.
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http://dx.doi.org/10.1186/s13023-019-1138-xDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6598251PMC
June 2019

The effect of testing and feedback on the forgetting curves for radiograph interpretation skills.

Med Teach 2019 07 2;41(7):756-764. Epub 2019 May 2.

h Department of Emergency Medicine, Division of Learning Analytics , NYU School of Medicine , New York , NY , USA.

Forgetting curves plot skill decay over time. After exposure to a simulation-based radiograph interpretation learning system, we determined the rate of learning decay and how this was impacted by testing (with and without feedback). Further, we examined the association of initial learning parameters on the forgetting curve. This was a multicenter, four-arm randomized control trial. Medical trainees completed 80 elbow radiographs and a 20-case post-test. Group 1 had no testing until 12 months; Groups 2-4 had testing every 2 months until 12 months. At 6 months, Group 3 testing was feedback-enhanced, while Group 4 had feedback-enhanced testing at 2, 6, and 10 months. There were 106 participants ( = 42 Group 1;  = 22 Groups 2 and 3;  = 20 Group 4). Group 1 showed an -8.1% learning decay at 12-months relative to other groups. In Groups 2, 3, and 4, there was no significant learning decay (+0.8%), and there were no differences in skill decay between these groups. Initial score and learning curve slope were predictive of retained skill. Learning decay was mitigated by exposure to 20 test cases (with and without feedback) every two months. Initial learning parameters predicted learning retention and may inform refresher education scheduling.
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http://dx.doi.org/10.1080/0142159X.2019.1570098DOI Listing
July 2019

Diagnostic Accuracy of MRI-Based Sacroiliitis Scoring Systems: A Systematic Review.

AJR Am J Roentgenol 2019 Mar 12:1-14. Epub 2019 Mar 12.

1 Department of Medical Imaging, University of Toronto, Toronto, ON, Canada.

Objective: Accurate and reproducible MRI assessment of the sacroiliac joint (SIJ) is challenging. Numerous scoring systems have been proposed to facilitate consistent SIJ assessment. The purpose of this article is to evaluate the diagnostic accuracy and reliability of existing MRI-based SIJ scoring systems for the evaluation of spondyloarthropathy.

Conclusion: Among existing methods, there is fair (grade B) evidence to recommend the Spondyloarthropathy Research Consortium of Canada scoring systems as tools for MRI evaluation of the SIJ. However, limited data on criterion validity limit assessment of scoring system accuracy.
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http://dx.doi.org/10.2214/AJR.17.19429DOI Listing
March 2019

Disruption of the PTHLH regulatory landscape results in features consistent with hyperparathyroid disease.

Am J Med Genet A 2019 04 25;179(4):663-667. Epub 2019 Feb 25.

Division of Clinical and Metabolic Genetics and University of Toronto, Department of Paediatrics, The Hospital for Sick Children, Toronto, Ontario, Canada.

Parathyroid hormone like hormone (PTHLH) signaling is essential for the proper formation of bone and its elevation or disruption has been directly implicated in several different skeletal dysplasias. We report a patient with a 2.802 Mb deletion upstream of the PTHLH coding sequence who presents with multiple fractures, metaphyseal changes, and overall features consistent with hyperparathyroid like disease. Analysis of the deleted region revealed the loss of putative regulatory regions adjacent to PTHLH and the possible gain of a limb enhancer. Furthermore, PTHLH expression appeared to be mis-regulated in fibroblasts derived from the patient. Altogether, we find that the disruption of the regulatory landscape of PTHLH likely results in its inappropriate expression and this novel clinical presentation.
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http://dx.doi.org/10.1002/ajmg.a.61071DOI Listing
April 2019

Preliminary Definitions for Sacroiliac Joint Pathologies in the OMERACT Juvenile Idiopathic Arthritis Magnetic Resonance Imaging Score (OMERACT JAMRIS-SIJ).

J Rheumatol 2019 09 15;46(9):1192-1197. Epub 2019 Feb 15.

From the Institute of Medical Sciences, Faculty of Medicine, University of Toronto; Department of Diagnostic Imaging, The Hospital for Sick Children; Department of Translational Medicine, SickKids Research Institute, Peter Gilgan Center for Research and Learning; Dalla Lana School of Public Health, University of Toronto; Division of Rheumatology, The Hospital for Sick Children, University of Toronto; Department of Medical Imaging, University of Toronto; Department of Rheumatology, Center for Prognosis Studies in Rheumatologic Diseases, Toronto Western Hospital, Toronto, Ontario; Department of Rheumatology, University of Alberta; Department of Radiology and Diagnostic Imaging, University of Alberta; Division of Pediatric Rheumatology, Department of Pediatrics, University of Alberta, Edmonton, Alberta, Canada; Leeds Institute of Rheumatic and Musculoskeletal Medicine, University of Leeds; UK National Institute for Health Research (NIHR) Leeds Biomedical Research Centre, Leeds Teaching Hospitals National Health Service (NHS) Trust, Leeds, UK; Department of Rheumatology, Leiden University Medical Center, Leiden; Reade | Emma Children's Hospital/Academic Medical Center, Amsterdam, the Netherlands; University of Pennsylvania Perelman School of Medicine, Division of Rheumatology, Children's Hospital of Philadelphia and Departments of Pediatric and Epidemiology, Philadelphia, Pennsylvania; Department of Radiology, Nemours Children's Hospital and Health System, Orlando, Florida, USA; Department of Radiology, National Institute of Geriatrics, Rheumatology and Rehabilitation, Warsaw, Poland; Department of Radiology and Medical Imaging, Ghent University Hospital, Ghent, Belgium; Department of Radiology, Hospital Sant Joan de Deu, Barcelona, Spain; Department of Radiology, Rikshospitalet, Oslo University Hospital, Oslo, Norway; Pediatric Rheumatology Research Institute, Bad Bramstedt, Germany; Department of Clinical Immunology and Rheumatology, Christian Medical College, Vellore, India; State University of Campina-UNICAMP, Department of Internal Medicine, Cidade Universitaria, Sao Paulo, Brazil.

Objective: To develop definitions for the assessment of magnetic resonance imaging (MRI) pathologies of the sacroiliac joints (SIJ) in juvenile idiopathic arthritis.

Methods: An Outcome Measures in Rheumatology (OMERACT) consensus-driven methodology consisting of iterative surveys and focus group meetings within an international group of rheumatologists and radiologists.

Results: Two domains, inflammation and structural, were identified. Definitions for bone marrow edema, joint space inflammation, capsulitis, and enthesitis were derived for joint inflammation; sclerosis, erosion, fatty lesion, and ankylosis were defined for assessing structural joint changes.

Conclusion: Preliminary consensus-driven definitions for inflammation and structural elements have been derived, underpinning the ongoing development of the OMERACT Juvenile Arthritis MRI SIJ scoring system (OMERACT JAMRIS-SIJ).
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http://dx.doi.org/10.3899/jrheum.181115DOI Listing
September 2019

Spondyloarthritis Research Consortium of Canada Scoring System for Sacroiliitis in Juvenile Spondyloarthritis/Enthesitis-related Arthritis: A Reliability, Validity, and Responsiveness Study.

J Rheumatol 2019 06 1;46(6):636-644. Epub 2019 Feb 1.

From the Joint Department of Medical Imaging, University Health Network, University of Toronto; Department of Diagnostic Imaging, The Hospital for Sick Children, University of Toronto; Division of Pediatric Rheumatology, The Hospital for Sick Children, University of Toronto, Toronto, Ontario; Division of Pediatric Rheumatology, Department of Pediatrics, The University of Alberta, Edmonton, Alberta; Kanchi Kamakoti CHILDS Trust Hospital, Chennai; Department of Radiology, Christian Medical College, Vellore, India; Makassed Hospital, Jerusalem, Israel; Department of Family and Community Medicine, University of Toronto, Toronto, Ontario, Canada.

Objective: Intra- and interreader reliability, construct validity, and responsiveness of the Spondyloarthritis Research Consortium of Canada (SPARCC) magnetic resonance imaging (MRI) scoring system were investigated for scoring sacroiliitis in patients with juvenile spondyloarthritis (JSpA)/enthesitis-related arthritis (ERA) who have received biologic and/or nonbiologic treatment.

Methods: Ninety whole-body MRI examinations with dedicated oblique coronal planes of the sacroiliac joints in 46 patients were independently reviewed and scored by 2 pediatric musculoskeletal radiologists, blinded to clinical details, using the SPARCC system. Intra- and interreader reliability was assessed by intraclass correlation coefficients (ICC). Construct validity testing was done by (1) correlating the SPARCC MRI scores of sacroiliitis with clinical disease activity indicators (cross-sectional validity), and (2) correlating the change in the MRI score with the change in clinical indicators before and after treatment (longitudinal validity). Responsiveness of the MRI and clinical indicators was also evaluated, grouped by biologic and nonbiologic treatment.

Results: When applied in children with JSpA/ERA, the SPARCC showed almost perfect intra- and interreader reliability (ICC 0.79-1.00). There was poor cross-sectional and longitudinal correlation between clinical assessment indicators and MRI scoring. SPARCC scores showed higher responsiveness to treatment-related change than most clinical outcome measures. Three clinical outcome measures correlated longitudinally with SPARCC score in nonbiologic treatment: active joint count (r = 0.72, p < 0.001), FABER (Flexion, Abduction, External Rotation) test (r = 0.58, p = 0.012), and physician's global assessment (r = 0.61, p = 0.034).

Conclusion: The SPARCC MRI scoring system is a reliable tool with relatively higher responsiveness than clinical indicators and is suitable for objective quantification of sacroiliitis when applied to pediatric patients with JSpA/ERA.
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http://dx.doi.org/10.3899/jrheum.180222DOI Listing
June 2019

Concurrent chronic recurrent multifocal osteomyelitis and familial Mediterranean fever. A case report.

Clin Exp Rheumatol 2018 Nov-Dec;36(6 Suppl 115):151. Epub 2018 Dec 14.

The Hospital for Sick Children and University of Toronto, Canada.

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March 2019

Imaging findings of sterile pyogenic arthritis, pyoderma gangrenosum and acne (PAPA) syndrome: differential diagnosis and review of the literature.

Pediatr Radiol 2019 01 17;49(1):23-36. Epub 2018 Sep 17.

Department of Diagnostic Imaging, The Hospital for Sick Children, Toronto, ON, Canada.

Pyogenic arthritis, pyoderma gangrenosum and acne (PAPA) syndrome is a rare autosomal-dominant autoinflammatory disease of incomplete penetrance and variable expression. PAPA syndrome is the result of a mutation in the proline serine threonine phosphatase-interacting protein 1 (PSTPIP1/CD2BP1) gene located on chromosome 15, which results in an abnormal overproduction of the pro-inflammatory cytokine interleukin-1β (IL-1). This syndrome clinically manifests as early onset of recurrent episodes of acute aseptic inflammation of the joints, generally occurring in the first two decades of life, followed by manifestation of characteristic skin lesions in the third decade, after an obvious decline in the joint symptoms. Although uncommon, the potential clinical implications of PAPA syndrome warrant an appropriate diagnosis in a timely fashion.
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http://dx.doi.org/10.1007/s00247-018-4246-1DOI Listing
January 2019

Prevalence of tuberculosis infection and disease in children referred for tuberculosis medical surveillance in Ontario: a single-cohort study.

CMAJ Open 2018 Jul-Sep;6(3):E365-E371. Epub 2018 Aug 28.

Faculty of Medicine (Yang), Dalla Lana School of Public Health (Yasseen, Rea), Department of Pediatrics (Waters, Morris, Kitai) and Faculty of Nursing (Lam), University of Toronto, Toronto, Ont.; Children's Hospital of Eastern Ontario Research Institute (Yasseen), Ottawa, Ont.; Department of Diagnostic Imaging (Stimec) and Division of Infectious Diseases (Waters, Morris, Kitai), The Hospital for Sick Children; Toronto Public Health (Rea), Toronto, Ont.

Background: There are few data about the utility of the Canadian tuberculosis medical surveillance system for detecting tuberculosis in children and adolescents. We sought to assess the prevalence of tuberculosis infection and disease in children and adolescents referred by the tuberculosis medical surveillance program who were evaluated at The Hospital for Sick Children (SickKids) tuberculosis program.

Methods: We retrospectively studied clinical records, radiographic findings and results of interferon-γ release assays (IGRAs) of all children less than 18 years of age referred by the tuberculosis medical surveillance program and evaluated at SickKids between November 2012 and June 2016.

Results: The median age of the 216 children was 10.0 years. Most were born in the Philippines (157 [72.7%]) or India (39 [18.0%]). Of the 216, 166 (76.8%) had a history of prior treatment for tuberculosis, and 34 (15.7%) were federal-sponsored refugees from settings with a high tuberculosis burden. Negative IGRA results were found in 110/130 (84.6%) of those with prior tuberculosis treatment. Thirty-one children (14.4%) had any chest radiographic abnormality, of whom 4 had changes thought to be due to tuberculosis. No child received a diagnosis of active tuberculosis at assessment or during follow-up; 3 (1.4%) were treated for latent tuberculosis infection following IGRA testing at SickKids. A positive IGRA result was associated with contact with infectious tuberculosis (odds ratio [OR] 5.97, 95% confidence interval [CI] 2.06-17.52) and older age at first clinic visit (OR 2.98, 95% CI 1.24-8.30) but not with radiographic abnormalities or history of prior tuberculosis treatment.

Interpretation: Most children were referred because of a history of prior treatment for tuberculosis; few had clinical or laboratory evidence of infection or prior disease. The tuberculosis medical surveillance process did not identify any children who required treatment for active disease and requires improvement.
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http://dx.doi.org/10.9778/cmajo.20180043DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6182122PMC
August 2018

Vitamin D Supplementation in Pregnancy and Lactation and Infant Growth.

N Engl J Med 2018 Aug;379(6):535-546

From the Department of Pediatrics (D.E.R., S.K.M., S.Z., E.P., J.K., J. Shi, R.W., P.S.S., L.G.P., H.Q.) and the Dalla Lana School of Public Health (A.R.W.), University of Toronto, and the Centre for Global Child Health (D.E.R., S.K.M., S.Z., E.P., J.K., J. Shi, R.W., L.G.P., H.Q.), the Department of Diagnostic Imaging (J. Stimec), and the Ontario Child Health Support Unit (A.R.W.), Hospital for Sick Children, and the Departments of Pediatrics (P.S.S.), and Obstetrics and Gynecology (K.E.M.), Mt. Sinai Hospital - all in Toronto; the Department of Nutritional Sciences, Penn State University, University Park, PA (A.D.G.); and the Maternal and Child Health Training Institute (I.J.) and the Nutrition and Clinical Services Division, International Center for Diarrheal Disease Research, Bangladesh (T.A., S.S.S., M.M.I., F.K.K., M.M., Q.S.R., A.A.M.), Dhaka, Bangladesh.

Background: It is unclear whether maternal vitamin D supplementation during pregnancy and lactation improves fetal and infant growth in regions where vitamin D deficiency is common.

Methods: We conducted a randomized, double-blind, placebo-controlled trial in Bangladesh to assess the effects of weekly prenatal vitamin D supplementation (from 17 to 24 weeks of gestation until birth) and postpartum vitamin D supplementation on the primary outcome of infants' length-for-age z scores at 1 year according to World Health Organization (WHO) child growth standards. One group received neither prenatal nor postpartum vitamin D (placebo group). Three groups received prenatal supplementation only, in doses of 4200 IU (prenatal 4200 group), 16,800 IU (prenatal 16,800 group), and 28,000 IU (prenatal 28,000 group). The fifth group received prenatal supplementation as well as 26 weeks of postpartum supplementation in the amount of 28,000 IU (prenatal and postpartum 28,000 group).

Results: Among 1164 infants assessed at 1 year of age (89.5% of 1300 pregnancies), there were no significant differences across groups in the mean (±SD) length-for-age z scores. Scores were as follows: placebo, -0.93±1.05; prenatal 4200, -1.11±1.12; prenatal 16,800, -0.97±0.97; prenatal 28,000, -1.06±1.07; and prenatal and postpartum 28,000, -0.94±1.00 (P=0.23 for a global test of differences across groups). Other anthropometric measures, birth outcomes, and morbidity did not differ significantly across groups. Vitamin D supplementation had expected effects on maternal and infant serum 25-hydroxyvitamin D and calcium concentrations, maternal urinary calcium excretion, and maternal parathyroid hormone concentrations. There were no significant differences in the frequencies of adverse events across groups, with the exception of a higher rate of possible hypercalciuria among the women receiving the highest dose.

Conclusions: In a population with widespread prenatal vitamin D deficiency and fetal and infant growth restriction, maternal vitamin D supplementation from midpregnancy until birth or until 6 months post partum did not improve fetal or infant growth. (Funded by the Bill and Melinda Gates Foundation; ClinicalTrials.gov number, NCT01924013 .).
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http://dx.doi.org/10.1056/NEJMoa1800927DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6004541PMC
August 2018

Imaging of temporomandibular joint abnormalities in juvenile idiopathic arthritis with a focus on developing a magnetic resonance imaging protocol.

Pediatr Radiol 2018 06 8;48(6):792-800. Epub 2018 May 8.

Department of Maxillofacial Radiology, Institute of Clinical Dentistry, University of Oslo, Oslo, Norway.

Inflammation and damage in the temporomandibular joint (TMJ) often develop without clinical symptoms but can lead to severe facial growth abnormalities and impaired health-related quality of life, making early diagnosis of TMJ changes crucial to identify. Inflammatory and osteochondral changes detectable through magnetic resonance imaging (MRI) occur in TMJs of approximately 40% of children with juvenile idiopathic arthritis (JIA), and no other imaging modality or physical method of examination can reliably detect these changes. Therefore contrast-enhanced MRI is the diagnostic standard for diagnosis and interval monitoring of JIA. However the specific usage of MRI for TMJ arthritis is not standardized at present. There is a recognized need for a consensus effort toward standardization of an imaging protocol with required and optional sequences to improve detection of pathological changes and shorten study time. Such a consensus imaging protocol is important for providing maximum information with minimally necessary sequences in a way that allows inter-site comparison of results of clinical trials and improved clinical management. In this paper we describe the challenges of TMJ imaging and present expert-panel consensus suggestions for a standardized TMJ MRI protocol.
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http://dx.doi.org/10.1007/s00247-017-4005-8DOI Listing
June 2018

Qualitative and semi-quantitative assessment of temporomandibular joint MRI protocols for juvenile idiopathic arthritis at 1.5 and 3.0T.

Eur J Radiol 2018 Jan 31;98:90-99. Epub 2017 Oct 31.

Peter Gilgan Centre for Research and Learning, The Hospital for Sick Children, University of Toronto, 686 Bay Street, Toronto, ON M5G1A4, Canada; Department of Medical Imaging, The Hospital for Sick Children, University of Toronto, 555 University Avenue, Toronto, ON M5G1X8, Canada. Electronic address:

Objective: Temporomandibular joints (TMJs) frequently develop silent inflammatory and osteochondral changes in children with juvenile idiopathic arthritis. Data-driven recommendations for TMJ imaging protocol are needed to reduce measurement error and scanning time. This study compares the impact of different protocols, imaging coils, and magnet strength on the reliability of image assessment and the subjective quality of images.

Materials And Methods: Three groups of bilateral TMJ MR studies were retrospectively collected from two institutions, including 24 1.5T and 19 3.0T studies using dedicated TMJ surface coils, and 23 1.5T studies with head coil. Post-contrast sequences were re-compiled from the full protocol to create minimum protocol studies for the three groups. Two radiologists and two non-radiologists first scored the three minimum protocol images according to pre-specified definitions, then scored the full protocol images. Minimum-to-full protocol agreement, inter-reader agreement, and subjective item visibility scores were assessed.

Results: With dedicated TMJ dual surface coils, minimum-vs-full protocol agreement was moderate to good (0.5-0.8 intraclass correlation coefficients or kappa) for most items, and was not influenced by the magnet strength. Inter-reader reliability was more significantly influenced by the imaging coil and reader's training background than by protocol length or magnet strength differences. Sagittal and coronal planes weighted on PD, T2 Fat Suppressed and T1 Fat Suppressed -postcontrast adequately visualized all the different features, whereas the axial plane was more limited to visualizing synovium.

Conclusion: Inter-reader reliability and qualitative measure of image quality improved more consistently with the coil offering the higher resolution, rather than increased magnet strength.
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http://dx.doi.org/10.1016/j.ejrad.2017.10.024DOI Listing
January 2018

Toward Establishing a Standardized Magnetic Resonance Imaging Scoring System for Temporomandibular Joints in Juvenile Idiopathic Arthritis.

Arthritis Care Res (Hoboken) 2018 05 12;70(5):758-767. Epub 2018 Apr 12.

University Children's Hospital, Zürich, Switzerland.

Objective: The temporomandibular joints (TMJs) are frequently affected in children with juvenile idiopathic arthritis (JIA). Early detection is challenging, as major variation is present in scoring TMJ pathology on magnetic resonance imaging (MRI). Consensus-driven development and validation of an MRI scoring system for TMJs has important clinical utility in timely improvement of diagnosis and serving as an outcome measure. We report on a multi-institutional collaboration toward developing a TMJ MRI scoring system for JIA.

Methods: Seven readers independently assessed MRI scans from 21 patients (42 TMJs, from patients ages 6-16 years) using 3 existing MRI scoring systems from American, German, and Swiss institutions. Reliability scores, scoring system definitions, and items were discussed among 10 JIA experts through 2 rounds of Delphi surveys, nominal group voting, and subsequent consensus meetings to create a novel TMJ MRI scoring system.

Results: Average-measure absolute agreement intraclass correlation coefficients (avICCs) for the total scores of all 3 scoring systems were highly reliable at 0.96 each. Osteochondral items showed higher reliability than inflammatory items. An additive system was deemed preferable for assessing minor joint changes over time. Eight items were considered sufficiently reliable and/or important for integration into the consensus scoring system: bone marrow edema and enhancement (avICC 0.57-0.61, smallest detectable difference [SDD] ± 45-63% prior to redefining), condylar flattening (avICC 0.95-0.96, SDD ± 23-28%), effusions (avICC 0.85-0.88, SDD ± 25-26%), erosions (avICC 0.94, SDD ± 20%), synovial enhancement and thickening (previously combined, avICC 0.90-0.91, SDD ± 33%), and disk abnormalities (avICC 0.90, SDD ± 19%).

Conclusion: A novel TMJ MRI scoring system was developed by consensus. Further iterative refinements and reliability testing are warranted in upcoming studies.
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http://dx.doi.org/10.1002/acr.23340DOI Listing
May 2018

Accuracy of Point-of-Care Ultrasonography for Pediatric Ankle Sprain Injuries.

Pediatr Emerg Care 2018 Dec;34(12):842-847

Division of Emergency Medicine, Department of Pediatrics, The Hospital for Sick Children and the University of Toronto, Toronto, Ontario, Canada.

Objectives: In children with radiograph fracture-negative lateral ankle injuries, the main objective of this pilot study was to explore the accuracy, sensitivity, and specificity of point-of-care ultrasound (POCUS) performed by a pediatric emergency physician in diagnosing anterior talofibular ligament injuries, radiographically occult distal fibular fractures, and effusions compared with reference standard magnetic resonance imaging (MRI).

Methods: This was a prospective cohort pilot study. Children aged 5 to 17 years with an isolated, acute lateral ankle injury and fracture-negative ankle radiographs were eligible for enrolment. Within 1 week of the injury, enrolled children returned for MRI and POCUS of both ankles.

Results: Seven children were enrolled, with a mean age 12.1 (SD, 3.0) years. Overall, POCUS agreed with MRI with respect to anterior talofibular ligament injury in 4 (57%) of 7 cases. Of the 2 cases with MRI-confirmed ligament damage, POCUS accurately identified and graded the extent of ligament damage in 1 case. Point-of-care ultrasound falsely identified ligament injuries in 2 cases. Both imaging modalities confirmed the absence of cortical fractures in all 7 cases. For all findings, POCUS sensitivity and specificity were 57% and 86%, respectively.

Conclusions: In this pilot study, we established that POCUS diagnosed the specific pathology of radiograph-negative lateral ankle injuries with poor sensitivity but good specificity. Thus, POCUS could act as a tool to exclude significant ligamentous and radiographically occult bony injury in these cases. A larger study is needed to validate the utility of POCUS for this common injury.
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http://dx.doi.org/10.1097/PEC.0000000000001130DOI Listing
December 2018