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Kate W. C. Chang, Denise Justice, Kevin C. Chung and Lynda J. S. Yang

Object

Neonatal brachial plexus palsy (NBPP) affects 0.4–2.6 newborns per 1000 live births in the US. Many infants recover spontaneously, but for those without spontaneous recovery, nerve and/or secondary musculoskeletal reconstruction can restore function to the affected arm. This condition not only manifests in a paretic/paralyzed arm, but also affects the overall health and psychosocial condition of the children and their parents. Currently, measurement instruments for NBPP focus primarily on physical ability, with limited information regarding the effect of the disablement on activities of daily living and the child's psychosocial well-being. It is also difficult to assess and compare overall treatment efficacy among medical (conservative) or surgical management strategies without consistent use of evaluation instruments. The purpose of this study is to review the reported measurement evaluation methods for NBPP in an attempt to provide recommendations for future measurement usage and development.

Methods

The authors systematically reviewed the literature published between January 1980 and February 2012 using multiple databases to search the keywords “brachial plexus” and “obstetric” or “pediatrics” or “neonatal” or “congenital.” Original articles with primary patient outcomes were included in the data summary. Four types of evaluation methods (classification, diagnostics, physical assessment, and functional outcome) were distinguished among treatment management groups. Descriptive statistics and 1-way ANOVA were applied to compare the data summaries among specific groups.

Results

Of 2836 articles initially identified, 307 were included in the analysis, with 198 articles (9646 patients) reporting results after surgical treatment, 70 articles (4434 patients) reporting results after medical treatment, and 39 articles (4247 patients) reporting results after combined surgical and medical treatment. Among medical practitioners who treat NBPP, there was equivalence in usage of classification, diagnostic, and physical assessment tools (that focused on the Body Function and Structures measure of the International Classification of Functioning, Disability, and Health [ICF]). However, there was discordance in the functional outcome measures that focus on ICF levels of Activity and Participation. Of the 126 reported evaluation methods, only a few (the Active Movement Scale, Toronto Scale Score, Mallet Scale, Assisting Hand Assessment, and Pediatric Outcomes Data Collection Instrument) are specifically validated for evaluating the NBPP population.

Conclusions

In this review, the authors demonstrate disparities in the use of NBPP evaluation instruments in the current literature. Additionally, valid and reliable evaluation instruments specifically for the NBPP population are significantly lacking, manifesting in difficulties with evaluating the overall impact and effectiveness of clinical treatments in a consistent and comparative manner, extending across the various subspecialties that are involved in the treatment of patients with NBPP. The authors suggest that all ICF domains should be considered, and future efforts should include consideration of spontaneous (not practitioner-elicited) use of the affected arm in activities of daily living with attention to the psychosocial impact of the disablement.

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Alexandra L. Mathews, Guang Yang, Kate Wan-Chu Chang and Kevin C. Chung

OBJECTIVE

The effectiveness of contralateral C-7 (CC7) transfer is controversial, yet this procedure has been performed around the world to treat brachial plexus injuries. The authors performed a systematic review to study whether Asian countries reported better outcomes after CC7 transfer compared with “other” countries.

METHODS

A systematic literature search using PubMed, EMBASE, and 3 Chinese databases was completed. Patient outcomes of CC7 transfer to the median and musculocutaneous (MC) nerves were collected and categorized into 2 groups: Asia and “other” countries. China was included as a subcategory of Asia because investigators in China published the majority of the collected studies. To compare outcomes among studies, we created a normalized Medical Research Council (MRC) scale.

RESULTS

For median nerve outcomes, Asia reported that 41% of patients achieved an MRC grade of ≥ M3 of wrist flexion compared with 62% in “other” countries. For finger flexion, Asia found that 41% of patients reached an MRC grade of ≥ M3 compared with 38% in “other” countries. Asia reported that 60% of patients achieved ≥ S3 sensory recovery, compared with 32% in “other” countries. For MC nerve outcomes, 75% of patients from both Asia and “other” countries reached M4 and M3 in elbow flexion.

CONCLUSIONS

Current data did not demonstrate that studies from Asian countries reported better outcomes of CC7 transfer to the median and MC nerves. Future studies should focus on comparing outcomes of different surgical strategies for CC7 transfer.

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Brandon W. Smith, Nicholas J. Chulski, Ann A. Little, Kate W. C. Chang and Lynda J. S. Yang

OBJECTIVE

Neonatal brachial plexus palsy (NBPP) continues to be a problematic occurrence impacting approximately 1.5 per 1000 live births in the United States, with 10%–40% of these infants experiencing permanent disability. These children lose elbow flexion, and one surgical option for recovering it is the Oberlin transfer. Published data support the use of the ulnar nerve fascicle that innervates the flexor carpi ulnaris as the donor nerve in adults, but no analogous published data exist for infants. This study investigated the association of ulnar nerve fascicle choice with functional elbow flexion outcome in NBPP.

METHODS

The authors conducted a retrospective study of 13 cases in which infants underwent ulnar to musculocutaneous nerve transfer for NBPP at a single institution. They collected data on patient demographics, clinical characteristics, active range of motion (AROM), and intraoperative neuromonitoring (IONM) (using 4 ulnar nerve index muscles). Standard statistical analysis compared pre- and postoperative motor function improvement between specific fascicle transfer (1–2 muscles for either wrist flexion or hand intrinsics) and nonspecific fascicle transfer (> 2 muscles for wrist flexion and hand intrinsics) groups.

RESULTS

The patients’ average age at initial clinic visit was 2.9 months, and their average age at surgical intervention was 7.4 months. All NBPPs were unilateral; the majority of patients were female (61%), were Caucasian (69%), had right-sided NBPP (61%), and had Narakas grade I or II injuries (54%). IONM recordings for the fascicular dissection revealed a donor fascicle with nonspecific innervation in 6 (46%) infants and specific innervation in the remaining 7 (54%) patients. At 6-month follow-up, the AROM improvement in elbow flexion in adduction was 38° in the specific fascicle transfer group versus 36° in the nonspecific fascicle transfer group, with no statistically significant difference (p = 0.93).

CONCLUSIONS

Both specific and nonspecific fascicle transfers led to functional recovery, but that the composition of the donor fascicle had no impact on early outcomes. In young infants, ulnar nerve fascicular dissection places the ulnar nerve at risk for iatrogenic damage. The data from this study suggest that the use of any motor fascicle, specific or nonspecific, produces similar results and that the Oberlin transfer can be performed with less intrafascicular dissection, less time of surgical exposure, and less potential for donor site morbidity.

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Brandon W. Smith, Kate W. C. Chang, Lynda J. S. Yang and Mary Catherine Spires

OBJECTIVE

The incorporation of ancillary testing in the preoperative setting for patients with neonatal brachial plexus palsy (NBPP) remains controversial, but the recommendation for early nerve reconstruction when a baby has a preganglionic lesion at the lower nerve roots is generally accepted. At some specialty centers, nerve surgeons use preoperative electrodiagnostic testing (EDX) and imaging to aid in lesion localization and the preoperative planning of the nerve reconstruction. EDX and imaging have been evaluated for their abilities to detect pre- and postganglionic lesions, but their accuracies have never been compared directly in the same set of patients. The aim of the present study was to evaluate the accuracy of imaging and EDX in an NBPP population.

METHODS

A retrospective review was conducted of 54 patients with operative NBPP seen between 2007 and 2017. The patients underwent EDX and imaging: EDX was performed, and the results were reviewed by board-certified electrodiagnosticians, and imaging was reviewed by board-certified neuroradiologists. The gold standard was considered to be the findings at surgical exploration. Descriptive and analytical statistics were utilized to compare the accuracies of imaging and EDX.

RESULTS

The mean age at surgery was 6.94 mos (± 4 mos). Fifteen patients (28%) were Narakas grade I–II, and 39 (72%) were Narakas grade III–IV. For all nerve roots, the overall accuracy of detecting preganglionic lesions was 74% for EDX and 69% for imaging. The overall sensitivity of detecting preganglionic lesions by EDX was 31%, but the specificity was 90%. The overall sensitivity of detecting preganglionic lesions by imaging was 66%, and the overall specificity was 70%. However, at C8, EDX was 37.5% sensitive and 87.5% specific, whereas imaging was 67.7% sensitive but only 29.4% specific.

CONCLUSIONS

EDX outperformed imaging with regard to specificity and accuracy of identifying preganglionic injuries. This finding is especially relevant in the lower nerve roots, given that lower plexus preganglionic lesions are an accepted indication for early intervention.

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Thomas J. Wilson, Kate W. C. Chang, Suneet P. Chauhan and Lynda J. S. Yang

OBJECTIVE

Neonatal brachial plexus palsy (NBPP) occurs due to the stretching of the nerves of the brachial plexus before, during, or after delivery. NBPP can resolve spontaneously or become persistent. To determine if nerve surgery is indicated, predicting recovery is necessary but difficult. Historical attempts explored the association of recovery with only clinical and electrodiagnostic examinations. However, no data exist regarding the neonatal and peripartum factors associated with NBPP persistence.

METHODS

This retrospective cohort study involved all NBPP patients at the University of Michigan between 2005 and 2015. Peripartum and neonatal factors were assessed for their association with persistent NBPP at 1 year, as defined as the presence of musculoskeletal contractures or an active range of motion that deviated from normal by > 10° (shoulder, elbow, hand, and finger ranges of motion were recorded). Standard statistical methods were used.

RESULTS

Of 382 children with NBPP, 85% had persistent NBPP at 1 year. A wide range of neonatal and peripartum factors was explored. We found that cephalic presentation, induction or augmentation of labor, birth weight > 9 lbs, and the presence of Horner syndrome all significantly increased the odds of persistence at 1 year, while cesarean delivery and Narakas Grade I to II injury significantly reduced the odds of persistence.

CONCLUSIONS

Peripartum/neonatal factors were identified that significantly altered the odds of having persistent NBPP at 1 year. Combining these peripartum/neonatal factors with previously published clinical examination findings associated with persistence should allow the development of a prediction algorithm. The implementation of this algorithm may allow the earlier recognition of those cases likely to persist and thus enable earlier intervention, which may improve surgical outcomes.

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Eric W. Franz, J. Nicole Bentley, Patricia P. S. Yee, Kate W. C. Chang, Jennifer Kendall-Thomas, Paul Park and Lynda J. S. Yang

OBJECT

Patient outcome measures are becoming increasingly important in the evaluation of health care quality and physician performance. Of the many novel measures currently being explored, patient satisfaction and other subjective measures of patient experience are among the most heavily weighted. However, these subjective measures are strongly influenced by a number of factors, including patient demographics, level of understanding of the disorder and its treatment, and patient expectations. In the present study, patients referred to a neurosurgery clinic for degenerative spinal disorders were surveyed to determine their understanding of lumbar spondylosis diagnosis and treatment.

METHODS

A multiple-choice, 6-question survey was distributed to all patients referred to a general neurosurgical spine clinic at a tertiary care center over a period of 11 months as a quality improvement initiative to assist the provider with individualized patient counseling. The survey consisted of questions designed to assess patient understanding of the role of radiological imaging in the diagnosis and treatment of low-back and leg pain, and patient perception of the indications for surgical compared with conservative management. Demographic data were also collected.

RESULTS

A total of 121 surveys were included in the analysis. More than 50% of the patients indicated that they would undergo spine surgery based on abnormalities found on MRI, even without symptoms; more than 40% of patients indicated the same for plain radiographs. Similarly, a large proportion of patients (33%) believed that back surgery was more effective than physical therapy in the treatment of back pain without leg pain. Nearly one-fifth of the survey group (17%) also believed that back injections were riskier than back surgery. There were no significant differences in survey responses among patients with a previous history of spine surgery compared with those without previous spine surgery.

CONCLUSIONS

These results show that a surprisingly high percentage of patients have misconceptions regarding the diagnosis and treatment of lumbar spondylosis, and that these misconceptions persist in patients with a history of spine surgery. Specifically, patients overemphasize the value of radiological studies and have mixed perceptions of the relative risk and effectiveness of surgical intervention compared with more conservative management. These misconceptions have the potential to alter patient expectations and decrease satisfaction, which could negatively impact patient outcomes and subjective valuations of physician performance. While these results are preliminary, they highlight a need for improved communication and patient education during surgical consultation for lumbar spondylosis.

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Joseph Domino, Connie McGovern, Kate W. C. Chang, Noelle E. Carlozzi and Lynda J. S. Yang

Object

Perinatal disorders are prone to malpractice litigation. Neonatal brachial plexus palsy (NBPP) results from stretching the nerves in the perinatal period and may lead to paresis or paralysis and sensory loss in the affected arm. Little is known about the key factors associated with malpractice litigation by families of patients with NBPP and whether these factors reflect the practice environment or are inherent to the condition. In this study, the authors documented the percentage of families of NBPP patients at a specialty center that had filed a malpractice suit and described the key factors associated with that pursuit of legal action.

Methods

The families/caregivers of 51 patients with NBPP who had presented to the University of Michigan Interdisciplinary Brachial Plexus Program participated in this study. A qualitative research design was applied using both a questionnaire to examine psychosocial factors and a dynamic tool to measure health outcomes from the patient perspective via parent proxy (Patient-Reported Outcomes Measurement Information System [PROMIS] assessment instruments). Statistical analysis included the Fisher exact test, chi-square test, and Student t-test. The study protocol was approved by the University of Michigan institutional review board.

Results

Forty-seven percent of the families pursued malpractice litigation. In comparing patient families that had pursued legal action with those that had not, significant differences were revealed in the perception that the sustained birth injury was unnecessary (p = 0.002), the information received in the perinatal period was inadequate (p = 0.003), family concerns were ignored in the perinatal period (p = 0.005), and family concerns were not adequately addressed (p < 0.001). Sixty-six percent of the families received external advice to pursue legal action. The PROMIS survey revealed significant group differences in depressive symptoms (p = 0.008), fatigue (p = 0.02), pain (p = 0.01), and anger (p = 0.004). In contrast, the extent of NBPP was not associated with malpractice litigation (p = 0.18). Age, sex, and race were not significantly different between litigation and nonlitigation groups.

Conclusions

Physician-controllable factors, such as communication in the perinatal period, are associated with malpractice litigation in NBPP. The perceived level of global disability may affect the pursuit of malpractice litigation, whereas the isolated extent of nerve root involvement and/or upper extremity dysfunction are not significant factors in pursuing litigation. Identifying and ameliorating these factors within the practice environment may decrease the animosity between families and health care providers and improve overall outcome for patients with NBPP.

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Robert C. Rennert, Kate T. Carroll, Mir Amaan Ali, Thomas Hamelin, Leon Chang, Brian P. Lemkuil and Clark C. Chen

OBJECTIVE

Stereotactic laser ablation (SLA) is typically performed in the setting of intraoperative MRI or in a staged manner in which probe insertion is performed in the operating room and thermal ablation takes place in an MRI suite.

METHODS

The authors describe their experience, in which SLA for glioblastoma (GBM) treatment was performed entirely within a conventional MRI suite using the SmartFrame stereotactic device.

RESULTS

All 10 patients with GBM (2 with isocitrate dehydrogenase 1 mutation [mIDH1] and 8 with wild-type IDH1 [wtIDH1]) were followed for > 6 months. One of these patients underwent 2 independent SLAs approximately 12 months apart. Biopsies were performed prior to SLA for all patients. There were no perioperative morbidities, wound infections, or unplanned 30-day readmissions. The average time for a 3-trajectory SLA (n = 3) was 436 ± 102 minutes; for a 2-trajectory SLA (n = 4) was 321 ± 85 minutes; and for a single-trajectory SLA (n = 4) was 254 ± 28 minutes. No tumor recurrence occurred within the blue isotherm line ablation zone, although 2 patients experienced recurrence immediately adjacent to the blue isotherm ablation line. Overall survival for the patient cohort averaged 356 days, with the 2 patients who had mIDH1 GBMs exhibiting the longest survival (811 and 654 days).

CONCLUSIONS

Multitrajectory SLA for treatment of GBM can be safely performed using the SmartFrame stereotactic device in a conventional MRI suite.

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Kelly L. VanderHave, Karen Bovid, Hilary Alpert, Kate Wan-Chu Chang, Douglas J. Quint, James A. Leonard Jr. and Lynda J. S. Yang

Object

The rate of neonatal brachial plexus palsy (NBPP) remains 0.4%–4% despite improvements in perinatal care. Among affected children, the extent of brachial plexus palsy differs greatly, as does the prognosis. Controversial elements in management include indications and timing of nerve repair as well as type of reconstruction in patients in whom function will ultimately not be recovered without surgical intervention. Differentiating preganglionic (avulsion) from postganglionic (rupture) lesions is critical because preganglionic lesions cannot spontaneously recover motor function. Distinguishing between these lesions at initial presentation based on clinical examination alone can be difficult in infants. The purpose of the present study was to determine the sensitivity of preoperative electrodiagnostic studies (EDSs) and CT myelography (CTM) in determining the presence of nerve root rupture and avulsions in infants with NBPP.

Methods

After receiving institutional review board approval, the authors conducted a retrospective review of patients referred to the Neonatal Brachial Plexus Program between 2007 and 2010. Inclusion criteria included children who underwent brachial plexus exploration following preoperative EDSs and CTM. The CTM scans were interpreted by a staff neuroradiologist, EDSs were conducted by a single physiatrist, and intraoperative findings were recorded by the operating neurosurgeon. The findings from the preoperative EDSs and CTM were then compared with intraoperative findings. The sensitivities and 95% confidence intervals were determined to evaluate performance accuracy of each preoperative measure.

Results

Twenty-one patients (8 male amd 13 female) met inclusion criteria for this study. The sensitivity of EDSs and CTM for detecting a postganglionic rupture was 92.8% (CI 0.841–0.969) and 58.3% (CI 0.420–0.729), respectively. The sensitivity for EDSs and CTM for preganglionic nerve root avulsion was 27.8% (CI 0.125–0.509) and 72.2% (CI 0.491–0.875), respectively. In cases in which both CTM and EDSs gave concordant results, the sensitivity for both modalities combined was 50.0% (CI 0.237–0.763) for avulsion and 80.8% (CI 0.621–0.915) for rupture. Overall, EDSs were most useful in identifying ruptures, particularly in the upper plexus, whereas CTM was most sensitive in identifying avulsions in the lower plexus.

Conclusions

Knowledge of the spinal nerve integrity is critical for early management of patients with NBPP. Surgical management, in the form of nerve repair/reconstruction, and optimal prognostication of NBPP depend on the accurate diagnosis of the level and type of lesion. Both EDSs and CTM scans must always be interpreted in the context of a comprehensive evaluation of the patient. They provide supplemental information (in addition to the physical examination) for early detection of nerve root rupture and avulsion injuries, aiding surgical decision making and preoperative planning for NBPP. Continued advances in imaging, EDSs, and microsurgical nerve repair techniques will allow surgeons to achieve greater success for functional recovery in management of NBPP.

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Kate W. C. Chang, Thomas J. Wilson, Miriana Popadich, Susan H. Brown, Kevin C. Chung and Lynda J. S. Yang

OBJECTIVE

The use of nerve transfers versus nerve grafting for neonatal brachial plexus palsy (NBPP) remains controversial. In adult brachial plexus injury, transfer of an ulnar fascicle to the biceps branch of the musculocutaneous nerve (Oberlin transfer) is reportedly superior to nerve grafting for restoration of elbow flexion. In pediatric patients with NBPP, recovery of elbow flexion and forearm supination is an indicator of resolved NBPP. Currently, limited evidence exists of outcomes for flexion and supination when comparing nerve transfer and nerve grafting for NBPP. Therefore, the authors compared 1-year postoperative outcomes for infants with NBPP who underwent Oberlin transfer versus nerve grafting.

METHODS

This retrospective cohort study reviewed patients with NBPP who underwent Oberlin transfer (n = 19) and nerve grafting (n = 31) at a single institution between 2005 and 2015. A single surgeon conducted intraoperative exploration of the brachial plexus and determined the surgical nerve reconstruction strategy undertaken. Active range of motion was evaluated preoperatively and postoperatively at 1 year.

RESULTS

No significant difference between treatment groups was observed with respect to the mean change (pre- to postoperatively) in elbow flexion in adduction and abduction and biceps strength. The Oberlin transfer group gained significantly more supination (100° vs 19°; p < 0.0001). Forearm pronation was maintained at 90° in the Oberlin transfer group whereas it was slightly improved in the grafting group (0° vs 32°; p = 0.02). Shoulder, wrist, and hand functions were comparable between treatment groups.

CONCLUSIONS

The preliminary data from this study demonstrate that the Oberlin transfer confers an advantageous early recovery of forearm supination over grafting, with equivalent elbow flexion recovery. Further studies that monitor real-world arm usage will provide more insight into the most appropriate surgical strategy for NBPP.