Il trattamento delle paralisi cerebrali infantili: sperimentazioni cliniche e ricadute assistenziali di nuovi approcci terapeutici e riabilitativi The treatment of infant cerebral palsy: clinical experimental trials of new therapeutic approaches and outcomes on health care

Background In the last decades, several treatment approaches have been used to improve upper limb function in hemiplegic CP. Only recently has Constraint Induced Movement Therapy (CIMT) emerged as a treatment approach for children with hemiplegic CP with the aim of reversing the behavioral suppression of movement in the affected upper limb. To date, evidence on this treatment has been very poor and limited, since all currently available trials reveal methodological limitations and a need for additional research to support the application of this treatment technique. Aim The thesis aims at exploring the safety and efficacy of a new treatment approach, Constraint Induced Movement Therapy has been studied and compared to a bimanual intensive rehabilitation approach and to traditional rehabilitation program. Methods This thesis presents the planning and development of a national mustisite clinical trial started in 2006 and carried out in collaboration with 21 Italian rehabilitations centers belonging to the Italian Group for Cerebral Palsy (GIPCI). The effectiveness and safety of CIMT combined with an intensive rehabilitation program was compared to 2 comparison groups: one treated with an intensive rehabilitation program and the other with standard treatment. Patients with hemiplegic cerebral palsy, aged between 2 and 8 years, who have never undergone constraint therapy have been recruited. Primary outcome measures include 2 major domains: UE motor ability (QUEST) and hand function assessment evaluating both grip function and spontaneous use of the affected side (Besta Scale). Secondary outcome measures concern overall function, behavior, compliance and satisfaction with treatment program of both child and family. Patients’ follow-up assessment was performed at 3, 6 and 12 months after the end of treatment. Collected data have been analyzed through univariate and multivariate statistical analysis. The experimental phase was preceded by a standardized analysis or primary outcome measures (QUEST and Besta Scale) in order to evaluate the agreement among the assessors of all participating centers. All evaluators scored the same 84 video-recorded tests each (42 QUEST and 42 Besta Scales administered to 42 children, 2 per each participating center). The analysis evaluated the inter-rater agreement and the reliability of primary outcome measures. During the clinical trial all the professionals involved in the project have been interviewed to explore their opinion on conducting clinical research in the field of rehabilitation and in their daily practice. Results 105 children have been recruited (39 undergoing CIMT, 33 undergoing bimanual intensive rehabilitation program and 33 undergoing traditional rehabilitation program). The main results demonstrate a significant effect of both intensive programs on the upper limb function. CIMT results particularly effective in ameliorating the grasp function, while bimanual intensive rehabilitation program results particularly efficacious in activity of daily living tasks. The traditional treatment does not show a significant modification of upper limb function. The patterns of brain plasticity and the process cortical reorganization following injury seem to play a crucial role in upper limb function modifications after intense treatment. Secondary outcome measures show the importance of the intensity of treatment and follow-up to reduce family stress and behavioral disturbances in children. The agreement analysis demonstrated an overall good inter-rater agreement and the reliability of both scales in assessing hand and upper limb function. 68 professionals have been involved in the survey regarding research in rehabilitation. The main results show the importance of conducting research in rehabilitation not only to obtain evidence for new therapeutic approaches, but also to standardize the practices and to share and improve the ability to use new and standardized assessment tools. Conclusions The researches conducted and the results obtained seem particularly important for the current rehabilitation practice and for the organization of rehabilitation programs in the dedicated health care service. Further research is needed on these issues, since, if these results will be confirmed they could dramatically change the approach to children with hemiplegic cerebral palsy and modify sensitively their disease’s natural history.Background In the last decades, several treatment approaches have been used to improve upper limb function in hemiplegic CP. Only recently has Constraint Induced Movement Therapy (CIMT) emerged as a treatment approach for children with hemiplegic CP with the aim of reversing the behavioral suppression of movement in the affected upper limb. To date, evidence on this treatment has been very poor and limited, since all currently available trials reveal methodological limitations and a need for additional research to support the application of this treatment technique. Aim The thesis aims at exploring the safety and efficacy of a new treatment approach, Constraint Induced Movement Therapy has been studied and compared to a bimanual intensive rehabilitation approach and to traditional rehabilitation program. Methods This thesis presents the planning and development of a national mustisite clinical trial started in 2006 and carried out in collaboration with 21 Italian rehabilitations centers belonging to the Italian Group for Cerebral Palsy (GIPCI). The effectiveness and safety of CIMT combined with an intensive rehabilitation program was compared to 2 comparison groups: one treated with an intensive rehabilitation program and the other with standard treatment. Patients with hemiplegic cerebral palsy, aged between 2 and 8 years, who have never undergone constraint therapy have been recruited. Primary outcome measures include 2 major domains: UE motor ability (QUEST) and hand function assessment evaluating both grip function and spontaneous use of the affected side (Besta Scale). Secondary outcome measures concern overall function, behavior, compliance and satisfaction with treatment program of both child and family. Patients’ follow-up assessment was performed at 3, 6 and 12 months after the end of treatment. Collected data have been analyzed through univariate and multivariate statistical analysis. The experimental phase was preceded by a standardized analysis or primary outcome measures (QUEST and Besta Scale) in order to evaluate the agreement among the assessors of all participating centers. All evaluators scored the same 84 video-recorded tests each (42 QUEST and 42 Besta Scales administered to 42 children, 2 per each participating center). The analysis evaluated the inter-rater agreement and the reliability of primary outcome measures. During the clinical trial all the professionals involved in the project have been interviewed to explore their opinion on conducting clinical research in the field of rehabilitation and in their daily practice. Results 105 children have been recruited (39 undergoing CIMT, 33 undergoing bimanual intensive rehabilitation program and 33 undergoing traditional rehabilitation program). The main results demonstrate a significant effect of both intensive programs on the upper limb function. CIMT results particularly effective in ameliorating the grasp function, while bimanual intensive rehabilitation program results particularly efficacious in activity of daily living tasks. The traditional treatment does not show a significant modification of upper limb function. The patterns of brain plasticity and the process cortical reorganization following injury seem to play a crucial role in upper limb function modifications after intense treatment. Secondary outcome measures show the importance of the intensity of treatment and follow-up to reduce family stress and behavioral disturbances in children. The agreement analysis demonstrated an overall good inter-rater agreement and the reliability of both scales in assessing hand and upper limb function. 68 professionals have been involved in the survey regarding research in rehabilitation. The main results show the importance of conducting research in rehabilitation not only to obtain evidence for new therapeutic approaches, but also to standardize the practices and to share and improve the ability to use new and standardized assessment tools. Conclusions The researches conducted and the results obtained seem particularly important for the current rehabilitation practice and for the organization of rehabilitation programs in the dedicated health care service. Further research is needed on these issues, since, if these results will be confirmed they could dramatically change the approach to children with hemiplegic cerebral palsy and modify sensitively their disease’s natural history

The overlapping area between Osteogenesis Imperfecta and child physical abuse: a bilbiometric analysis.

Osteogenesis Imperfecta (OI) is a hereditary connective tissue disorder. Its manifestations are fragile bones. multiple bone fractures, bone deformities with a history of minimal or no trauma. This clinical picture may be observed in Child Physical Abuse (CPA). CPA is a relevant differential diagnosis when a child presents unexplained fractures. Aim is to quantify the weight and the trends of the wrold wide literature on OI and CPA, in order to measure if and how the 2 topics overlap and are studied together. A retrospective bibliometric analysis was carried out. Through PubMed, literature was explored and 2 quoted phrases Osteogenesis Imperfecta (OI), Child Physical Abuse (CPA) (CPA) were searched in all available manuscripts. Starting from retrieved papers, a descriptive analysis and a text mining search were performed (SAS software). Search retrieved 7,140 manuscripts, distributed per interval period (1950-2006) per each DB as follows: OI 48% (1950-2006), CPA 52% (1950-2006). English is the leading language of publication: OI 70% and CPA 93%. For both the DBs the top Countries of publication are USA and UK. An overlapping area exists between OI & CPA: 70 manuscripts representing the 1% of the whole (1969-2006), written in English (84%) and published in the US and UK (45% and 29%). Further results on text mining search will follow. Results obtained show that English-written papers predominate and understanding English results fundamental to update oneself. The overlapping area is nearly absent despite the strong correlation of the two issues, it means that bibliographic search on CPA/differential diagnosis could be easily biased retrieving incomplete results, with the continuous risk of falling in a bibliographic cul-de-sac

Can U B A Can Writer? Child Abuse and Neglect: Identikit of the Model Author

Background: Child Abuse and Neglect (CAN) represents a relatively young theme in the world of research. However, more than 50 000 manuscripts dealing with it have been published since its birth, classically considered the Kempe\u2019s paper, \u201cThe battered child syndrome\u201d, published in 1962. Such an amount of papers has allowed the blooming of knowledge, and more and more people got involved in understanding the phenomenon. In the last few years, a decrease is observed in the interest this issue is able to evoke in the scientific community (measurable in the progressively lower number of manuscripts published). Identification of model contributors and estimation of prevalence of group and corporate authorship could represent a good tool to understand how to enhance the number of publications, considered as the currency of academic research. Objective: Aim of the present study is to draw an identikit of the model researcher involved in investigating the issue of child abuse and neglect. Method: A retrospective bibliometric analysis was carried out, utilizing the search engine of NIH, PubMed. The last 10-year literature (1995-2004) was explored and the quoted phrase \u201cChild Abuse\u201d was searched in all available manuscripts. Starting from retrieved papers, information on authors was collected. Particular attention was paid to affiliation, country of origin, preferences shown on country of publication, tendency to publish in groups or corporate authorships. Starting from the Web of Science, 50 most prolific investigators were studied for the number of citation they showed. A descriptive analysis was performed. Results: The bibliographic research retrieved 7369 articles, with a total \u201chorde\u201d of 19075 authors. The main characteristic of average investigator is utilizing English as language of transmission of his/her knowledge (92.4%) and write alone (38% of manuscripts have a single author; mean 2.6 authors per paper, range 0-26; SD of 2). Moreover, the model researcher seems to look for journals published in English speaking countries. In fact, top five countries of publication are the US, the UK, Germany, Australia and Canada (85.8% English speaking, 2.9% German speaking). The trend is to choose traditional journals (or traditional plus e-publishing journals), instead of stand-alone e-publications (99.9% vs 0.1%). Authors show another clear tendency, i.e. to publish in their own homeland, or in a country with whom there is an historical of geographical cooperation (for example Canada and the US, India and England or Japan and Australia). Looking to the most prolific authors, the 1st one have published, during the considered interval period, 41 manuscripts creating his own CV starting moving on the \u201cchessboard\u201d of author\u2019s position during years: 1st author in 10 manuscripts with 60% after 2001, and 2nd author in 23 manuscripts with 50% published after 2001. Further result on most cited authors will follow. Conclusion: CAN seems to be an hot topic for single-author: in fact the at-a-glance picture of the published manuscripts in the considered interval period strongly highlight the scarce frequency of corporate authorships, and much more than multidisciplinary teams. A number of reflections on most prolific authors and leading investigators working-in-the-field can be drawn. The study has pointed out that both being able to speak in English and coming form the US increase the probability to be able to publish a paper on CAN. Is it a choice of authors or a protectionist policy of publishers

Munchausen's Syndrome By Proxy at a Glance: Twisting an Old Tale

Background: Munchausen\u2019s Syndrome By Proxy (MSBP) is a particular and potentially lethal form of child abuse, with reported mortality rates of about 10%. By definition, MSBP occurs when an adult repeatedly produces symptoms and/or signs of illness in a child under his/her responsibility, causing unnecessary medical investigations/treatments/hospitalisations. The syndrome was discovered by R. Meadow in 1977, and since then, more than 400 cases have been reported in the international literature. Objective: Aim of the present study is to evaluate the latest 15-years research on MSBP, outlining main trends of the specific literature and main publishing attitudes. Method: A descriptive bibliometric analysis was performed. Through the MeSH database of NIH PubMed, the definition of MSBP was selected (A phenomenon in which symptoms of a disease are fabricated by an individual other than the patient causing unnecessary, and often painful, physical examinations and treatments. This syndrome is considered a form of CHILD ABUSE, since another individual, usually a parent, is the source of the fabrication of symptoms and presents the child for medical care). From the databank, in the period 1990-2005, all articles strictly referring to this definition were retrieved. For each record retrieved, data were collected on authors, source (journals have been subdivided by subject area), publication year, publication type, language, pages, abstract. All the information was recorded in a dedicated database. Results: In the whole 15-years period considered, 385 articles were extracted from the database MEDLINE/Pubmed. The most profit-bearing year was 1995, with the 12.6% of the whole period production. In the \u201chit-parade\u201d of journals, Child Abuse & Neglect classed first with a 7%, followed by Archives of Diseases in Childhood and BMJ (with 5,6 % and 5,3%, respectively). Mostly North-American and European journals are at stake. The subject area mostly involved is the Medical (83%, mainly general medicine and paediatrics) followed by Nursing (11%), Psychology (3%). Looking at languages, 44% of articles are written in English, 19% German, 5% in Spanish. More than 44% are single-author articles (range=0-10; mean= 2.16; SD=1.6; variance= 2.55). On what attains authorship, the most prolific writers are sir R. Meadow and H. A. Schreier. The mean number of pages per article is 5,3 (range 33-1); evaluating number of pages in time, it is clear that at the beginning there were more articles with a lower number of page, while in the last years the situation is reversed (more pages, less articles) attesting probably a sort of quality knowledge consolidation. Case Report is the most declared type of publication (58.4%), followed by Review (36.7%), Observational Population-Based Study represent less than 1% of all manuscripts. Conclusion: MSBP represents a great challenge in the broad field of child abuse and neglect, diagnosis is frequently delayed mostly for the pleomorphic nature of this disease and, detection is much more difficult, since even if there are few typical aspects of the diseases, there are no exclusive signs or symptoms. MSBP results in being still extremely underreported and understudied and unknown in many of its aspects. Being the manuscripts mainly case reports, only partial conclusions can be drawn on the syndrome, especially from and epidemiological point of view and this extreme fragmentariness of knowledge and the information gap can not be ignored

Mimicking Child Physical Abuse: is there a place in CAN literature for Osteogenesis imperfecta?

Background: Osteogenesis Imperfecta (OI) is a hereditary connective tissue disorder. Typical manifestations are fragile bones with multiple bone fractures and bone deformities. A history of minimal or no trauma and recurrent fractures are features of OI, but they are also typical of Child Physical Abuse (CPA). OI and CPA are relevant differential diagnoses when a child presents with unexplained fractures. Objective: Quantify the weight and the trends of the worldwide literature on OI and CPA, in order to measure if and how the 2 topics overlap and are studied together. Method: A retrospective bibliometric analysis was carried out, utilizing the search engine of NIH, PubMed. The worldwide literature was explored and the 2 quoted phrases Osteogenesis Imperfecta (OI), Child Physical Abuse (CPA) were searched in all available manuscripts. Starting from retrieved papers, information on title, author(s), affiliation, source, journal, publication year, language and abstract were collected and recorded in 2 dedicated databases (DB) that are finally merged all in one in order to identify the overlapping area. A descriptive analysis and a text mining search were performed utilizing SAS software. Results: The search retrieved 7,140 manuscripts, distributed per interval period per each DB as follows: OI 48% (1950-2006), CPA 52% (1950-2006). English is the leading language of publication: OI 70% and CPA 93%. For both the DBs the top Countries of publication are United States of America and UK. An overlapping area exists between OI & CPA: 70 manuscripts representing the 1% of the whole (1969-2006). The prevalent language of publication is English (84%) and the most frequent countries of publication are United States of America and UK (45% and 29%). Further results on typology/journal of publication and text mining search will follow. Conclusion: The results obtained show that on this issue, the Anglophone countries demonstrate a predominant interest and therefore understanding the English language results fundamental to update oneself. Moreover, the nearly absent overlapping area among different DBs shows isolation (from a bibliometric point of view) of issue that are strongly related in reality. Practically, this means that a bibli- ographic search on the broad theme on child physical abuse/differential diagnosis could be easily biased retrieving incomplete results, with the continuous risk of falling in a bibliographic cul-de-sac. There is the need, especially in CAN literature, to implement the issue of differential diagnosis in order to allow the increasing of pro- fessionals\u2019 self education & training

Conoscere, Cercare e Intervenire: il Chemical Abuse come esempio del crescente bisogno di integrazione tra servizi per la tutela dei minori

Il Chemical Abuse \ue8 una forma di maltrattamento sui minori determinata da un\u2019esposizione, attiva o passiva, a sostanze tossiche quali stupefacenti, farmaci, alcol e agenti caustici che pu\uf2 provocare esiti complessi ed irreversibili per la salute del bambino, dai ritardi dello sviluppo, alle lesioni cerebrali, fino alla morte. Lo studio realizzato ha evidenziato come il Chemical Abuse si configuri generalmente a danno di bambini molto piccoli (0 e 3 anni) all\u2019interno di famiglie in cui uno o entrambi i genitori risultano fare uso di sostanze stupefacenti (61%). Questi risultati evidenziano la necessit\ue0 di una diagnosi precoce e conseguentemente di un lavoro integrato tra i servizi specialistici per gli adulti ed i servizi di tutela minori