Functional Impact of Congenital Hand Differences: Early Results From the Congenital Upper Limb Differences (CoULD) Registry

Bae, Donald S.; Canizares, Maria F.; Miller, Patricia E.; Waters, Peter M.; Goldfarb, Charles A.

doi:10.1016/j.jhsa.2017.10.006

Cited by 45 publications

(32 citation statements)

References 21 publications

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“…While previous authors have emphasised difficulties associated with fine motor tasks, such as doing up buttons and zips (Bae et al, 2018;Kelley et al, 2016), overwhelmingly children in our study found writing the greatest functional challenge. Associated pain and fatigue caused significant frustration, sometimes necessitating stopping writing altogether.…”

Section: Discussioncontrasting

confidence: 80%

The psychosocial impact of congenital hand and upper limb differences on children: a qualitative study

McDougall

Kennedy

Coombs

et al. 2020

J Hand Surg Eur Vol

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This qualitative study explored the psychosocial concerns of children born with congenital hand and upper limb differences (CHULDs) from the point of view of these children and their parents. Qualitative, in-depth, semi-structured interviews were conducted face-to-face with eight parent–child dyads. Open format questions allowed spontaneous emergence of relevant themes, followed by guided questioning. Thematic analysis of audio-recorded and transcribed interviews found that children as young as 5 years old had unique and meaningful opinions about their CHULD, of which parents were sometimes unaware. Children reported that unsolicited questions from peers caused significant stress, and this increased around the time of surgery. All children used planned responses to peers as an effective coping mechanism. Children also identified positive aspects of their difference, including an increased sense of determination and an appreciation for being unique. Strategies that may improve psychosocial outcomes for children with CHULDs are discussed. Level of evidence: IV

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Section: Discussioncontrasting

confidence: 80%

The psychosocial impact of congenital hand and upper limb differences on children: a qualitative study

McDougall

Kennedy

Coombs

et al. 2020

J Hand Surg Eur Vol

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“…However, the Pediatric Outcomes Data Collection Instrument and Patient-Reported Outcomes Measurement Information System are being used successfully for continuous and longitudinal data collection within the Congenital Upper Limb Differences database. 24…”

Section: Recommendationsmentioning

confidence: 99%

Correction of Thumb Duplication: A Systematic Review of Surgical Techniques

Miller

Kaempfen

Moledina

et al. 2020

Journal of Hand and Microsurgery

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Surgical intervention for thumb duplication can be divided into three categories: simple excision of the accessory thumb, excision of the accessory thumb with reconstruction from available “spare parts,” and combining the two thumbs into one, as described by Bilhaut. This prospectively PROSPERO registered systematic review evaluates the overall, aesthetic and functional outcomes for the latter two options (reconstruction from spare parts vs. combining two thumbs into one), aiming to facilitate evidence-based decision making when addressing thumb duplication and direct future research. The review was performed in accordance with the Cochrane Handbook of Systematic Reviews and PRISMA statement. Embase, PubMed, Medline, and Cochrane databases were systematically searched. Studies offering comparisons of techniques were included. Risk of bias was assessed using the Risk of Bias In Non-randomized Studies—of Intervention tool. The quality of the evidence was assessed using the Grading of Recommendations, Assessment, Development, and Evaluation. Ten retrospective observational studies were included. Data did not consistently allow analysis by procedure type. Four studies reported similar overall outcomes between techniques, while two specifically reported poor overall outcomes for the Bilhaut procedure. Two studies reported comparatively worse aesthetic outcomes for the Bilhaut procedure with four studies reporting comparatively improved functional outcomes for this procedure. Overall, interpretation of outcomes was challenging with no patient-reported outcome measures used. The quality of the evidence was universally “very low” due to all studies being at risk of methodological bias. Based on the available evidence, surgical techniques for thumb duplication correction appear comparable regarding overall outcome. There is limited evidence suggesting reconstruction with spare parts offers superior aesthetic outcomes at the expense of stability. The level of evidence is III.

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“…Future high-quality evidence will likely arise from prospective multicenter efforts such as the Congenital Upper Limb Differences (COULD) Registry. 3 The presence of an omovertebral bone and a negative glenoid inclination angle are factors that we believe may lead to a greater change in postoperative function; however, this has not yet been proven and should be the focus of the future study. In addition, the future study of patients with Sprengel's deformity should include both physicianand patient-derived measures, prospective documentation of range of motion across time particularly abduction, forward flexion, and shoulder rotation.…”

Section: Discussionmentioning

confidence: 87%

Sprengel's deformity: an analysis of surgically and nonsurgically treated patients

Vuillermin

Wang

Williams

et al. 2021

Journal of Shoulder and Elbow Surgery

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Background: Sprengel's deformity is a congenital anomaly of the scapula and can be associated with reduced shoulder mobility and esthetic concerns. Controversies remain regarding its natural history, radiographic findings, and optimal treatment strategy. Methods: A total of 74 Sprengel's shoulders in 71 patients presenting to a pediatric referral hospital were retrospectively reviewed including 24 surgically treated shoulders. Median age at initial presentation was 3.8 years. Median age at the final follow-up was 9.6 years. Results: Twenty shoulders were analyzed for range of motion across time. The median composite abduction range was initially 105 and at the last time-point 98 , with a median loss of 15 over 5 years (P ¼ .007). A total of 26% of the nonsurgical shoulders reported pain at the latest follow-up, compared with 1 surgical patient (4%) (P ¼ .028). Older age was independently associated with pain (P < .001); this effect was only seen in nonsurgical patients. Radiographically, the glenoid inclination angle (GIA) independently correlated with the abduction range (P ¼ .001). Surgically treated shoulders underwent a modified Woodward procedure at a median 4.8 years of age. At a median follow-up of 41 months, the abduction range improved in the surgical group from 90 preoperatively to 110 postoperatively, with a median gain of 40 (P < .001). The median GIA changed from À2 to 4 with a median gain of 9 (P ¼ .004). Conclusions: The natural history of untreated Sprengel's deformity may be one of gradual decline in the abduction range, with a proportion of patients developing pain in adolescence. The GIA correlates with the shoulder abduction range. Surgery with a modified Woodward procedure may alter the natural history of the condition by improving motion and reducing risk of pain later in life.

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Functional Impact of Congenital Hand Differences: Early Results From the Congenital Upper Limb Differences (CoULD) Registry

Cited by 45 publications

References 21 publications

The psychosocial impact of congenital hand and upper limb differences on children: a qualitative study

The psychosocial impact of congenital hand and upper limb differences on children: a qualitative study

Correction of Thumb Duplication: A Systematic Review of Surgical Techniques

Sprengel's deformity: an analysis of surgically and nonsurgically treated patients

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