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Abstract. Cerebral palsy (CP) is associated with complex health care needs and, although improved, a continued shortened life
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expectancy. In order to quantify and understand advances in the diagnosis and management of CP, systematic literature searches of
key word groupings in the PubMed database were completed and revealed a recent increased incidence of publications focusing
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on quality of life, physical activity, exercise, and treatment options. Our bibliometric exploration revealed growing emphasis on
function, performance, aging, and health compared to earlier studies when diagnostic features and brain pathology dominated
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research. Our findings highlight the transition from diagnosis and identification to management of specific conditions and providing
guidance for the continuum of needs our patients experience over the course of a lifetime. The field must be prepared to advance
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our understanding of best practices and implement evidence-based interventions and management options.
Keywords: Cerebral palsy, publications, research, bibliometrics, trends, rehabilitation, physical medicine
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2 Much has changed since Dr. William John Little first speculated that this may be due to medical frailty or 19
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3 described the clinical features of cerebral palsy (CP) in limited access to specialized care. 20
4 1860 [1]. CP is estimated to affect 1.5 to more than 4 Physiatrists have played an important role in the care 21
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5 out of every 1,000 live births. The incidence in Canada
6 and the US for recent years has remained stable [2, this area. In particular, physiatrists are instrumental in 23
PM&R, JH 301, 750 East Adams St, Syracuse, NY, 13210, USA. Tel.: While the number of training programs for the spe- 39
+1 315 464 5820; Fax: +1 315 464 8699; E-mail: turkm@upstate.edu. cialty of PRM increased, multiple authors and orga- 40
1874-5393/20/$35.00 c 2020 – IOS Press and the authors. All rights reserved
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2 M.A. Klawonn et al. / Trends in publications about cerebral palsy 1990 to 2020
41 nizations, including the Institute of Medicine (now initial searches, with an emphasis on PRM-related top- 89
42 National Academy of Medicine), called for recogni- ics. We assessed the returned publications to obtain two 90
43 tion of the field of disability and rehabilitation re- pieces of information: the size of and topics in the body 91
44 search and increasing productivity and rigor of that of publications. All searches were completed between 92
45 research [7–11]. These calls and an emphasis on ev- February 15 and March 15, 2020. 93
50 mately associated with the management of individu- ing or decreasing interest in particular subtopics of the 96
51 als with CP. We sought to better understand the broad study of CP. Generally, we found that there has been a 97
52 trends in publications about clinical therapy, medica- marked increase in the number of peer reviewed articles 98
53 tion, physical activity, and comorbidities of those af- published since the 1990–1995 epoch, with the most 99
54 fected by CP through a systematized literature search significant increase over the most recent five year period 100
55 that focused on different time intervals, using selected (see Table 1). We report on the changes noted between 101
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56 metrics. This commentary presents an overview of the the 1990–1995 and 2015–2020 epochs for counts and 102
57 trends in disability and rehabilitation research about CP terms of interest. Five-year epoch specific search results 103
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58 using a variety of techniques and metrics that reveal a
change in focus over the years. Much of this new in- Table 1 displays the large increases of percentages 105
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60 formation has been incorporated into practice, although in the number of articles focusing on comorbidities, 106
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71 to provide an historical and comparative perspective. this affects their life satisfaction. The higher number of 121
72 To minimize any preconceptions, we chose to use se- returned publications from searches such as “cerebral 122
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73 lected techniques used in larger in-depth reviews. Bib- palsy and physical activity or exercise and aging or age” 123
74 liometrics offered that opportunity since it is a quanti- and “cerebral palsy and physical activity or exercise” 124
75 tative overview of the development of a field of study show the increasing awareness and focus on how phys- 125
76 by determining size, frequencies, and trends [12]. To ical activity, exercise, and BMI (body mass index) are 126
77 further limit bias, a systematic literature search using important factors to consider for people with CP. Fig- 127
78 a single database (PubMed) and focusing on key areas ure 1 presents the same data, but allows for easier visual 128
79 in the field of CP was implemented. We directed our comparison of the increases in searches on these topics 129
80 search from the1990s to the present in an effort to in- over time. This is shown by the large relative increases 130
81 clude the most timely and relevant information and sub- in the colored bars corresponding to the searches listed 131
82 grouped findings by 5-year intervals. We piloted our above. Appendix Table A lists the search results by 132
83 approach using broad searches such as “cerebral palsy 5 year epochs, 1990–2020. 133
84 and rehabilitation” and “cerebral palsy and medical in-
85 tervention,” again looking for broad trends. After look- 2.2. Term usage in publications 134
88 using frequently occurring words that we noted in the ement to identify changes in interest in and focus of 136
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M.A. Klawonn et al. / Trends in publications about cerebral palsy 1990 to 2020 3
Table 1
Publication counts for each search performed in the 1990–1995 and 2015–2020 time frames
% Change in number
Article number Article number of publications from
Pubmed search performed
1990–1995 2015–2020 1990–1995 to
2015–2020
Cerebral Palsy AND comorbidities 5 156 3020%
Cerebral palsy AND medical intervention 73 1458 1897%
Cerebral palsy AND (quality of life OR life satisfaction) 27 534 1878%
Cerebral palsy AND (physical activity OR exercise) AND (aging OR age) 22 358 1527%
Cerebral palsy AND (physical activity OR exercise) 70 978 1297%
Cerebral palsy AND access 11 144 1209%
Cerebral palsy AND (sarcopenia OR metabolic syndrome) 2 23 1050%
Cerebral palsy AND (physical activity OR exercise) AND fatigue 3 32 967%
Cerebral palsy AND rehabilitation 377 2749 629%
Cerebral palsy AND spasticity AND (repeated botox injection OR repeated 1 7 600%
botulinum toxin injections)
Cerebral palsy AND pain 68 458 573%
Cerebral palsy AND muscle wasting 7 25 257%
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Cerebral palsy 2062 7131 245%
Cerebral palsy AND spasticity 211 669 217%
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Cerebral palsy AND life expectancy 7 16 128%
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Fig. 1. Rate of increase in publications by search terms for 1990–1995 and 2015–2020. This figure displays the number of publications returned
for each performed search, on a logarithmic scale. When comparing multiple time frames of a single search, an increase of one unit on the y-axis
indicates the number of publications doubled. As an example, because the number of publications for “Cerebral Palsy AND medical intervention”
doubles from ‘90–’95 to ‘95–’00, the corresponding light blue color increases by 1 unit (a relatively minor increase). Significant increases in a
single color indicate the number of publications doubled many times over. However, an increase of one unit on the y-axis for the size of the whole
bar does not correspond to a doubling in the total number of publications, since the composite bar for each time frame constitutes all searches
being added to one another, not multiplied.
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4 M.A. Klawonn et al. / Trends in publications about cerebral palsy 1990 to 2020
Table 2
Top 20 most frequently occurring terms in titles for the results of the
and “adult.” The increased prevalence of the words 161
‘cerebral palsy’ search, comparing 1990–1995 to 2015–2020 “therapy,” “gait” and “function” in particular support 162
palsy search palsy search terms in titles, more PRM diagnoses and treatments. Further, their ap- 164
1990–1995 2015–2020 from 1990–1995 pearance indicates that the way these patients function 165
top 20 words top 20 words epoch and and their level of independence is a highly important 166
in titles in titles 2015–2020 epoch
aspect of their overall care. Similarly, the appearance of 167
Child Child Unchanged
Study Study Unchanged the word “adult” points to the increasing importance of 168
Case Spasticity ↑5→3 treating adults with CP as the life expectancy for these 169
Infant Effect ↑ 20 → 4 patients has increased. The words “infant,” and “brain” 170
Spasticity Outcome ↑8→5
occur higher on the list in the 1990–1995 time period, 171
Patient Review New term
Treatment Motor ↑ 15 → 7 which may represent an increased focus on the diag- 172
Outcome Patient ↓6→8 nosis and initial management of CP. The 1990–1995 173
Syndrome∗ Gait New term words “syndrome,” and “birth” do not even appear in 174
Brain Infant ↓ 4 → 10
Surgery∗ Function New term
2015–2020, again highlighting the focus of earlier lit- 175
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Clinical Therapy New term erature on diagnosis as opposed to management of CP. 176
Birth∗ Case ↓ 3 → 13 Appendix Table B reports the 50 most common terms 177
Disease∗
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Systematic New term appearing in article titles. 178
Motor Preterm New term
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Rhizotomy∗ Clinical ↓ 12 → 16
Nerve∗ Treatment ↓ 7 → 17 2.4. Term context 179
Hip∗ Brain ↓ 10 → 18
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Disorder∗ Adult New term
Quantifying and visualizing term-context is a more 180
Effect Training New term
challenging problem than identifying term-frequency.
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Legend: ∗ indicates 1990–1995 term no longer in top 10 2015–2020
terms. One approach commonly taken in NLP is to develop 182
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137 research and publications. For this initial overview, arti- tor (i.e., a coordinate in some space) that is produced 184
138 cle titles provided the basis of terms analysis. A term’s for each term in a collection of text such that terms 185
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139 frequency is the number of times it appears in the titles, with similar contexts are close together. Representing 186
140 and the context describes how often terms co-occur in terms numerically allows for more direct comparisons 187
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142 2.3. Term frequency dard processing techniques on the results for a ‘cere- 190
bral palsy’ search for both the 1990–1995 and 2015– 191
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143 Term frequency was easily calculated by process- 2020 epochs. While the resulting model assigns high- 192
144 ing the titles of publications returned by the ‘cerebral dimensional vectors to each term, we can visualize each 193
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145 palsy’ query using standard natural language process- term in two dimensions via the t-SNE projection al- 194
146 ing (NLP) techniques. NLP refers to the study of pro- gorithm [14], which reduces each vector to two di- 195
147 cessing, analyzing, and understanding human language mensions while preserving the “closeness” of any two 196
149 An NLP program counted how frequently each The visualization of the top fifty most frequently 198
150 unique term appeared in the resulting collection of text occurring terms (1990–1995 and 2015–2020) and the 199
151 (code available from authors upon request). This pro- context in which they are presented in article titles is 200
152 cess was repeated for the 1990–1995 and the 2015– displayed in Fig. 2 (see Appendix for listing of Top 50 201
153 2020 time frames. Pronouns, punctuation, and words terms). We used these embeddings to identify interest- 202
154 that were determined not to carry clinical significance ing areas for further exploration, using select terms to 203
155 were removed. The top twenty most frequently occur- narrow our search and identify key publications. 204
156 ring words appear in Table 2. In Fig. 2A (1990–1995), the word “functional” is 205
157 In Table 2, some notable key words that are among closely associated with the words “effect,” “infarction,” 206
158 the top twenty most commonly used in article titles and “preterm”, while in Fig. 2B (2015–2020), the word 207
159 in the 2015–2020 time frame but not the 1990–1995 “functional” is associated with “motor” and “inter- 208
160 time frame are “review,” “gait,” “function,” “therapy,” vention.” The difference in word associations suggests 209
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M.A. Klawonn et al. / Trends in publications about cerebral palsy 1990 to 2020 5
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Fig. 2. Bubble chart of terms used in titles. t-SNE projection of learned term vectors, learned from the titles of the 1990–1995 time frame (A), as
compared to those learned from the 2015–2020 time frame (B). The size of each bubble corresponds to the frequency with which each word was
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used in the titles. Relative positions of words are meaningful (closeness implies similar contexts), yet the exact coordinates have no interpretable
meaning.
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210 a shift in the way function is approached in the two in Fig. 2B “disability” is associated closely with the 228
211 time periods. Perhaps interventions to improve func- words “surgery,” and ”treatment.” This suggests a shift 229
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212 tion and motor impairments’ effect on function have in the literature towards intervention for improvement 230
213 become more of a focus in recent literature. As an ex- of function in patients with disability in recent arti- 231
214 ample, searching for “Cerebral Palsy AND Functional” cles and an emphasis on diagnosis and general under- 232
215 in 1990–1995 (n = 163) returned publications such standing of CP characteristics and mechanics in the ear- 233
216 as Ferrari et al. [15], which is a descriptive case series lier time frame. Representative of the search “Cerebral 234
217 study of preterm infants’ movements compared to brain Palsy and Disability” in 1990–1995 (n = 307), Rose et 235
218 pathology and later development of CP; the same search al. [17] discuss muscle structure and energy expenditure 236
219 for the 2015–2020 range (n = 1151) returned publica- while walking, while in 2015–2020 (n = 1262), Lin- 237
220 tions represented by Sakzewski et al. [16], which re- tanf et al. [18] provide a systematic review and meta- 238
221 viewed randomized trials of interventions to improve analysis looking at the effect of ankle foot orthoses on 239
222 upper limb motor function and noted the need for rigor gait in patients with CP, to provide an evidence base for 240
224 In the earlier period, Fig. 2A shows that the word The word “therapy” does not appear in the top 50 242
225 “disability” is in close proximity to a large cluster terms in the former epoch (Fig. 2A), but it is in the latter 243
226 of terms including the words “analysis”, “supranu- epoch (Fig. 2B) and is associated closely with “clin- 244
227 clear,” and “progressive” compared to the latter where ical,” “training,” “spasticity,” “effect,” “muscle,” and 245
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6 M.A. Klawonn et al. / Trends in publications about cerebral palsy 1990 to 2020
246 “child,” likely indicating the increased recognition of ered by our analysis, there has been a marked increase 291
247 the importance of rehabilitative therapies as treatments in the number of publications of original research and 292
248 for people with CP and the importance of initiation of review articles covering CP and the care of individuals 293
249 therapies during childhood. The search “Cerebral Palsy with this condition. 294
250 and Therapy” in 2015–2020 (n = 3606) yields publi- We drew upon the emerging practice of scientomet- 295
251 cations such as Verschuren et al. [19], which provides rics to analyze the scientific literature pertaining to 296
252 recommendations regarding physical activity and exer- CP. Our systematic search of peer reviewed publica- 297
253 cise for children, adolescents, and adults with CP, based tions listed in PubMed highlighted heightened focus on 298
254 on a review and analysis of the literature and expert trends towards assessing functional mobility, improving 299
255 opinion. quality of life, addressing age related changes, and in- 300
257 The increasing volume of review articles since the ized scale has enhanced assessments of affected in- 305
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258 turn of the century (see Table 2) parallels the focus on dividuals by enabling evaluation of mobility at home 306
259 evidence-based medicine and guideline development. and functionality at school and also in the community 307
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260 Guidelines and use of evidence-based approaches have at large [25]. Similarly, clinical gait analysis has been 308
been promoted to improve effectiveness and quality of shown to be a useful tool for examining gait in individu-
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261 309
262 care and to decrease variations in clinical practice [20]. als with CP [26]. Well-defined assessment criteria have
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263 There has been an appeal to improve the rigor and facilitated improved communication between physia- 311
264 quality of research in order to develop well supported trists and orthopedists as well as other members of the 312
systematic searches/reviews and guidelines [21–23]. health care team. Further clarification of interventions
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265 313
266 The Cochrane Database of Systematic Reviews [23] is to maintain and improve mobility in individuals with 314
267 the foremost resource for health-related reviews, how- CP is needed. 315
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268 ever many systematic searches/reviews are published in Review of targetable avenues that improve quality of 316
269 peer-reviewed journals. Within the Cochrane Database life in CP patients reveals that sarcopenia is an impor- 317
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270 are twelve reviews on topics about CP with recommen- tant contributing factor. Identification of determinants 318
271 dations based on those reviews, ranging from 2004 to that can address sarcopenia and help clarify prognosis 319
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272 2019. Regarding guidelines, a search on the Agency offer an opportunity to greatly improve quality of life 320
273 for Healthcare Research and Quality website [24] re- in CP. Although sarcopenia commonly affects individ- 321
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274 turned 742 results. However the vast majority matched uals over 50 years of age, research has highlighted the 322
275 to “cerebral” not “Cerebral Palsy”. Of those related to impact of muscle loss on life expectancy and wellness. 323
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276 CP, most were exploratory and common topics were Because individuals with CP have smaller muscle mass 324
277 feeding/nutrition and transitional care issues. Despite than their peers, effect on gross motor function are par- 325
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278 the increase in research and publications, evidence re- ticularly pronounced with aging. Many individuals who 326
279 mains limited for the majority of interventions typically were once independent lose important skills to maintain 327
280 considered for function and associated or secondary that level independence, which may result in additional 328
281 conditions for people with CP. Caution is warranted for health conditions and earlier change in living arrange- 329
282 recommendations or guidelines that are based solely on ments. A better understanding of the sarcopenic pro- 330
283 expert opinion or rely on multiple levels of evidence cess may reveal novel approaches to enhance function 331
284 equally. High quality guidelines are needed. longer term and improve quality of life for people with 332
CP. 333
285 4. Future research ticity in CP have come into common use since 1990 335
286 Although there have been significant advances in However, the consequences of long-term treatment with 337
287 neonatal care which have improved outcomes and lim- these agents has been suggested to have effects that 338
288 ited the risk of adverse outcomes to very low birth were not anticipated. Botulinum toxin injections may 339
289 weight infants, CP remains a disability that is common change the architecture of muscle cells [27] and re- 340
290 and difficult to manage. Between the time frames cov- duce muscle volume over time [28]. Intrathecal ba- 341
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M.A. Klawonn et al. / Trends in publications about cerebral palsy 1990 to 2020 7
342 clofen pumps have been implicated in accelerating pro- formation Directorate, 525 Brooks Road, Rome, NY 389
343 gression of scoliosis [29], although may contribute to 13441, matthew.klawonn.2@us.af.mil) for his support 390
344 longer lifespan [30]. Further clarification of these con- in use of natural language processing techniques and 391
345 sequences and their impact on function and quality of generating the visualizations and their corresponding 392
348 Our review of publications related to CP has pro- There was no funding to support this work. 395
349 vided an overview using selected techniques, metrics,
All authors participated in the development of this 396
350 and visualizations. Over the past 30 years, there has
manuscript and met the International Committee of 397
351 been significant increases in both numbers and topics,
Medical Journal Editors (ICJME) (http://www.icmje. 398
352 with a change in focus from diagnosis, brain pathol-
org) definition of authorship. 399
353 ogy, and infants to function, performance, aging, and
The authors have no conflicts of interest to report 400
health. This increase in publications supports the report
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354
directly related to this publication. 401
355 on the status of PRM practice in 2017 [31] that notes
Drs. Klawonn and Klamar have no financial disclo- 402
356 greater research engagement among pediatric physi-
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357 atrists. However, the report also notes minimal fund- sures. Dr. Turk’s financial disclosures are Co-Editor-in- 403
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358 ing support for that research and significant geographic
359 variations in practice. Centers for Disease Control and Prevention, National 405
361 ically been comparatively low. Pediatric Physiatrists cooperative agreement #1U19DD001218. 407
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362 remain a small group, primarily focused on clinical
363 service, and must find ways to contribute to the re-
search enterprise, with or without funding. Multidis- References 408
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Appendix 531
Appendix Table A
Full list of search results by 5 year epochs, 1990–2020
% Change in
number of
Article Article Article Article Article Article
publications
Pubmed search performed number number number number number number
from
1990–1995 1995–2000 2000–2005 2005–2010 2010–2015 2015–2020
1990–1995 to
2015–2020
Cerebral palsy AND comorbidities 5 16 43 104 163 156 3020%
Cerebral palsy AND medical intervention 73 148 290 574 1060 1458 1897%
Cerebral palsy AND (quality of life OR life 27 53 140 352 463 534 1878%
satisfaction)
Cerebral palsy AND (physical activity OR 22 34 86 227 368 358 1527%
exercise) AND (aging OR age)
Cerebral palsy AND (physical activity OR 70 103 195 556 970 978 1297%
on
exercise)
Cerebral palsy AND access 11 14 35 71 125 144 1209%
Cerebral palsy AND (sarcopenia OR metabolic 2 9 8 8 26 23 1050%
si
syndrome)
er
Cerebral palsy AND (physical activity OR 3 1 4 17 29 32 967%
exercise) AND fatigue
Cerebral palsy AND rehabilitation 377 413 758 1405 2400 2749 629%
fv
Cerebral palsy AND spasticity AND (repeated 1 2 6 10 16 7 600%
botox injection OR repeated botulinum
oo
toxin injections)
Cerebral palsy AND pain 68 104 197 324 381 458 573%
Cerebral palsy AND muscle wasting 7 7 14 24 27 25 257%
pr
10 M.A. Klawonn et al. / Trends in publications about cerebral palsy 1990 to 2020
Appendix Table B
Top 50 most frequently occurring terms in titles for the results of the
‘cerebral palsy’ search, comparing 1990–1995 to 2015–2020
Pubmed cerebral palsy search Pubmed cerebral palsy search
1990–1995 top 50 words in titles 2015–2020 top 50 words in titles
Child Child
Study Study
Case Spasticity
Infant Effect
Spasticity Outcome
Patient Review
Treatment Motor
Outcome Patient
Syndrome Gait
Brain Infant
Surgery Function
Clinical Therapy
Birth Case
Disease Systematic
on
Motor Preterm
Rhizotomy Clinical
si
Nerve Treatment
Hip Brain
er
Disorder Adult
Effect Training
Disability
fv
Surgery
Perinatal Muscle
Report Disability
oo
Posterior Trial
Selective Assessment
Management Movement
pr
Analysis Unilateral
Progressive Neonatal
Supranuclear Adolescent
ed
Developmental Neurodevelopmental
Neonatal Activity
Development Risk
ct
Functional Disorder
Associated Report
rre
Assessment Intervention
Infantile Analysis
Infarction Functional
co
Preterm Physical
Artery Life
un
Change Pediatric
Mental Factor
Adult Young
Following Hip
Neurodevelopmental Early
Weight Health
Early Randomized
Age Injury
Care Quality
Asphyxia Limb
Due Controlled
Galley Proof 9/06/2020; 16:58 File: prm–1-prm200697.tex; BOKCTP/xjm p. 11
M.A. Klawonn et al. / Trends in publications about cerebral palsy 1990 to 2020 11
on
si
Appendix Fig. 2A. 1990–1995.
er
fv
oo
pr
ed
ct
rre
co
un