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Health Technology Assessment... Oct 2015Dupuytren's disease is a slowly progressive condition of the hand, characterised by the formation of nodules in the palm that gradually develop into fibrotic cords.... (Review)
Review
BACKGROUND
Dupuytren's disease is a slowly progressive condition of the hand, characterised by the formation of nodules in the palm that gradually develop into fibrotic cords. Contracture of the cords produces deformities of the fingers. Surgery is recommended for moderate and severe contractures, but complications and/or recurrences are frequent. Collagenase clostridium histolyticum (CCH) has been developed as a minimally invasive alternative to surgery for some patients.
OBJECTIVES
To assess the clinical effectiveness and cost-effectiveness of collagenase as an alternative to surgery for adults with Dupuytren's contracture with a palpable cord.
DATA SOURCES
We searched all major electronic databases from 1990 to February 2014.
REVIEW METHODS
Randomised controlled trials (RCTs), non-randomised comparative studies and observational studies involving collagenase and/or surgical interventions were considered. Two reviewers independently extracted data and assessed risk of bias of included studies. A de novo Markov model was developed to assess cost-effectiveness of collagenase, percutaneous needle fasciotomy (PNF) and limited fasciectomy (LF). Results were reported as incremental cost per quality-adjusted life-year (QALY) gained. Deterministic and probabilistic sensitivity analyses were undertaken to investigate model and parameter uncertainty.
RESULTS
Five RCTs comparing collagenase with placebo (493 participants), three RCTs comparing surgical techniques (334 participants), two non-randomised studies comparing collagenase and surgery (105 participants), five non-randomised comparative studies assessing various surgical procedures (3571 participants) and 15 collagenase case series (3154 participants) were included. Meta-analyses of RCTs assessing CCH versus placebo were performed. Joints randomised to collagenase were more likely to achieve clinical success. Collagenase-treated participants experienced significant reduction in contracture and an increased range of motion compared with placebo-treated participants. Participants treated with collagenase also experienced significantly more adverse events, most of which were mild or moderate. Four serious adverse events were observed in the collagenase group: two tendon ruptures, one pulley rupture and one complex regional pain syndrome. Two tendon ruptures were also reported in two collagenase case series. Non-randomised studies comparing collagenase with surgery produced variable results and were at high risk of bias. Serious adverse events across surgery studies were low. Recurrence rates ranged from 0% (at 90 days) to 100% (at 8 years) for collagenase and from 0% (at 2.7 years for fasciectomy) to 85% (at 5 years for PNF) for surgery. The results of the de novo economic analysis show that PNF was the cheapest treatment option, whereas LF generated the greatest QALY gains. Collagenase was more costly and generated fewer QALYs compared with LF. LF was £1199 more costly and generated an additional 0.11 QALYs in comparison with PNF. The incremental cost-effectiveness ratio was £10,871 per QALY gained. Two subgroup analyses were conducted for a population of patients with moderate and severe disease and up to two joints affected. In both subgroup analyses, collagenase remained dominated.
LIMITATIONS
The main limitation of the review was the lack of head-to-head RCTs comparing collagenase with surgery and the limited evidence base for estimating the effects of specific surgical procedures (fasciectomy and PNF). Substantial differences across studies further limited the comparability of available evidence. The economic model was derived from a naive indirect comparison and was hindered by a lack of suitable data. In addition, there was considerable uncertainty about the appropriateness of many assumptions and parameters used in the model.
CONCLUSIONS
Collagenase was significantly better than placebo. There was no evidence that collagenase was clinically better or worse than surgical treatments. LF was the most cost-effective choice to treat moderate to severe contractures, whereas collagenase was not. However, the results of the cost-utility analysis are based on a naive indirect comparison of clinical effectiveness, and a RCT is required to confirm or refute these findings.
STUDY REGISTRATION
This study is registered as PROSPERO CRD42013006248.
FUNDING
The National Institute for Health Research Health Technology Assessment programme.
Topics: Adult; Cost-Benefit Analysis; Dupuytren Contracture; Humans; Microbial Collagenase; Postoperative Complications; Quality-Adjusted Life Years
PubMed: 26524616
DOI: 10.3310/hta19900 -
Plastic and Reconstructive Surgery Mar 2014Dupuytren disease is a fibroproliferative disease of palmar fascia of the hand. Its prevalence has been the subject of several reviews; however, an accurate description... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
Dupuytren disease is a fibroproliferative disease of palmar fascia of the hand. Its prevalence has been the subject of several reviews; however, an accurate description of the prevalence range in the general population--and of the relation between age and disease--is lacking.
METHODS
Embase and PubMed were searched using database-specific Medical Subject Headings; titles and abstracts were searched for the words "Dupuytren," "incidence," and "prevalence." Two reviewers independently assessed the articles using inclusion and exclusion criteria, and rated the included studies with a quality assessment instrument. In a meta-analysis, the median prevalence, as a function of age by sex, was estimated, accompanied by 95 percent prediction intervals. The observed heterogeneity in prevalence was investigated with respect to study quality and geographic location.
RESULTS
Twenty-three of 199 unique identified articles were included. The number of participants ranged from 37 to 97,537, and age ranged from 18 to 100 years. Prevalence varied from 0.6 to 31.6 percent. The quality of studies differed but could not explain the heterogeneity among studies. Mean prevalence was estimated as 12, 21, and 29 percent at ages 55, 65, and 75 years, respectively, based on the relation between age and prevalence determined from 10 studies.
CONCLUSIONS
The authors describe a prevalence range of Dupuytren disease in the general population of Western countries. The relation between age and prevalence of Dupuytren disease is given according to sex, including 95 percent prediction intervals. It is possible to determine disease prevalence at a certain age for the total population, and for men and women separately.
Topics: Adolescent; Adult; Aged; Aged, 80 and over; Dupuytren Contracture; Europe; Female; Humans; Male; Middle Aged; Prevalence; Young Adult
PubMed: 24263394
DOI: 10.1097/01.prs.0000438455.37604.0f -
BMC Musculoskeletal Disorders Apr 2013Dupuytren's disease of the hand is a common condition affecting the palmar fascia, resulting in progressive flexion deformities of the digits and hence limitation of... (Review)
Review
BACKGROUND
Dupuytren's disease of the hand is a common condition affecting the palmar fascia, resulting in progressive flexion deformities of the digits and hence limitation of hand function. The optimal treatment remains unclear as outcomes studies have used a variety of measures for assessment.
METHODS
A literature search was performed for all publications describing surgical treatment, percutaneous needle aponeurotomy or collagenase injection for primary or recurrent Dupuytren's disease where outcomes had been monitored using functional measures.
RESULTS
Ninety-one studies met the inclusion criteria. Twenty-two studies reported outcomes using patient reported outcome measures (PROMs) ranging from validated questionnaires to self-reported measures for return to work and self-rated disability. The Disability of Arm, Shoulder and Hand (DASH) score was the most utilised patient-reported function measure (n=11). Patient satisfaction was reported by eighteen studies but no single method was used consistently. Range of movement was the most frequent physical measure and was reported in all 91 studies. However, the methods of measurement and reporting varied, with seventeen different techniques being used. Other physical measures included grip and pinch strength and sensibility, again with variations in measurement protocols. The mean follow-up time ranged from 2 weeks to 17 years.
CONCLUSIONS
There is little consistency in the reporting of outcomes for interventions in patients with Dupuytren's disease, making it impossible to compare the efficacy of different treatment modalities. Although there are limitations to the existing generic patient reported outcomes measures, a combination of these together with a disease-specific questionnaire, and physical measures of active and passive individual joint Range of movement (ROM), grip and sensibility using standardised protocols should be used for future outcomes studies. As Dupuytren's disease tends to recur following treatment as well as extend to involve other areas of the hand, follow-up times should be standardised and designed to capture both short and long term outcomes.
Topics: Clinical Trials as Topic; Dupuytren Contracture; Forecasting; Humans; Practice Guidelines as Topic; Treatment Outcome
PubMed: 23575442
DOI: 10.1186/1471-2474-14-131 -
BMC Musculoskeletal Disorders Jul 2008Splinting after contracture release for Dupuytren's disease of the hand is widely advocated. The purpose of this systematic review was to evaluate the quantity and... (Review)
Review
BACKGROUND
Splinting after contracture release for Dupuytren's disease of the hand is widely advocated. The purpose of this systematic review was to evaluate the quantity and quality of evidence regarding the effectiveness of splinting in the post-surgical management of Dupuytren's contractures.
METHODS
Studies were identified by searching the electronic databases Medline, AMED, CINAHL and EMBASE. Studies were included if they met the following inclusion criteria: prospective or retrospective, experimental, quasi-experimental or observational studies investigating the effectiveness of static or dynamic splints worn day and/or night-time for at least 6 weeks after surgery and reporting either individual joint or composite finger range of motion and/or hand function. The methodological quality of the selected articles was independently assessed by the two authors using the guidelines for evaluating the quality of intervention studies developed by McDermid.
RESULTS
Four studies, with sample sizes ranging from 23 to 268, met the inclusion criteria for the systematic review. Designs included retrospective case review, prospective observational and one controlled trial without randomisation. Interventions included dynamic and static splinting with a mean follow-up ranging from 9 weeks to 2 years. Pooling of results was not possible due to the heterogeneity of interventions (splint type, duration and wearing regimen) and the way outcomes were reported.
CONCLUSION
There is empirical evidence to support the use of low load prolonged stretch through splinting after hand surgery and trauma, however only a few studies have investigated this specifically in Dupuytren's contracture. The low level evidence regarding the effect of post-operative static and dynamic splints on final extension deficit in severe PIP joint contracture (>40 degrees ) is equivocal, as is the effect of patient adherence on outcome. Whilst total active extension deficit improved in some patients wearing a splint there were also deficits in composite finger flexion and hand function. The lack of data on the magnitude of this effect makes it difficult to interpret whether this is of clinical significance. There is a need for well designed controlled trials with proper randomisation to evaluate the short-term and long-term effectiveness of splinting following Dupuytren's surgery.
Topics: Adult; Aged; Dupuytren Contracture; Female; Hand Deformities, Acquired; Humans; Male; Middle Aged; Postoperative Care; Splints; Treatment Outcome
PubMed: 18644117
DOI: 10.1186/1471-2474-9-104 -
Anales de Pediatria (Barcelona, Spain :... Aug 2006Neonatal tumors, which represent only 1.5-2% of all pediatric tumors, have distinctive features. Their incidence is imprecise, as selection criteria vary and there are... (Review)
Review
BACKGROUND
Neonatal tumors, which represent only 1.5-2% of all pediatric tumors, have distinctive features. Their incidence is imprecise, as selection criteria vary and there are few published studies.
OBJECTIVE
To analyze and disseminate findings on the histological, clinical, therapeutic and follow-up characteristics of neonatal tumors.
MATERIAL AND METHODS
We performed a retrospective study of the clinical records of patients diagnosed with neonatal tumors in La Fe University Children's Hospital in Valencia (Spain) between January 1990 and December 1999. Hamartomas, flat and cavernous cutaneous hemangiomas, nevi, lipomas, subcutaneous fibroma, lymphangiomas, and epidermal cysts were excluded. A systematic review of the literature published in the previous 25 years was performed using Medline, Cancerlit, Index Citation Science, and Embase. The search profile combined neonatal or congenital and tumor or cancer or neoplasm. The most interesting studies, as well as the most relevant references contained in these studies and published before the search period, were selected.
RESULTS
The clinical records of 72 patients with neonatal tumors (40 boys and 32 girls), representing 2.8% of all pediatric tumors, were reviewed. The most frequent tumors were hemangiomas (20.8%, 15 patients), neuroblastomas (16.7%, 12 patients), teratomas (12.5 %, 9 patients), and soft tissue tumors (9.7 %, 7 patients). Eighty-six percent of the patients were symptomatic during the first week of life. Although diagnosis was prenatal in 22.2 % of the patients, the most frequent findings on physical examination were identification of a mass or cutaneous lesion in 24 patients (33.3%) and an abdominal mass or hepatomegaly in 13 patients (18%). An associated disease, malformation or syndrome was found in 15 patients (20.8 %). Treatment included surgery (50% of patients) and drugs as monotherapy or coadjuvant therapy (13.9%). Thirteen patients received irradiation after the neonatal period. Spontaneous complete remission took place in six patients. Twenty patients died (27.8%). Mortality was highest in patients with central nervous system tumors or leukemias (83.3% and 75 % respectively). By contrast, none of the patients with hemangiomas or teratomas died. Mortality was low in patients with neuroblastoma (8.3%). At the time of the study, survival was 73 %, with a median follow-up of 8 years.
CONCLUSIONS
Due to their biological features, neonatal tumors represent a distinctive subgroup in pediatric oncohematology. The concept of neonatal tumor should be unified to allow the results of different research groups to be analyzed and compared. Despite the methodological limitations found, the clinical, diagnostic, therapeutic, and follow-up characteristics of our patients are similar to those of other published series. The differences found could be explained by the diverse selection criteria employed.
Topics: Female; Hospitals, Pediatric; Hospitals, University; Humans; Infant, Newborn; Male; Neoplasms; Retrospective Studies; Spain
PubMed: 16948973
DOI: 10.1157/13091478 -
Annals of Oncology : Official Journal... Feb 2003Despite the use of surgery and radiotherapy, 20-35% of patients with aggressive fibromatosis (AF) will have local recurrence. The purpose of this review was to collect... (Review)
Review
BACKGROUND
Despite the use of surgery and radiotherapy, 20-35% of patients with aggressive fibromatosis (AF) will have local recurrence. The purpose of this review was to collect and analyze all available information regarding the role of non-cytotoxic and cytotoxic chemotherapy in AF that has been accumulated over the past few decades.
PATIENTS AND METHODS
A systematic review of published clinical trials, studies and case series was carried out using the Medline Express Databases and the Cochrane Collaboration Database from 1970 to October 2000.
RESULTS
Most studies published in the literature are in the form of successful case reports and single-arm series with small patient numbers. Most commonly used agents include hormonal agents, non-steroidal anti-inflammatory drugs (NSAIDs), interferons and cytotoxics. The literature data support the use of hormonal agents. Several questions, however, remain unresolved, such as which is the most suitable endocrine manipulation and what is the optimal dose and duration of treatment. NSAIDs and interferons have demonstrated activity against AF either alone or in combination with hormone therapy or chemotherapy but the precise mechanism of action is still unknown. Finally, there is growing evidence in the literature that chemotherapy is effective against AF with almost one in two patients being likely to respond.
CONCLUSIONS
The evidence in the literature supports the opinion that both non-cytotoxic and cytotoxic chemotherapies are effective against AF. However, the lack of sufficient patient numbers and randomized trials compromises the validity of the reported results and mandates further investigation with properly designed prospective studies including larger patient numbers, with main end points to include not only tumor response rate and survival but also quality-of-life issues.
Topics: Anti-Inflammatory Agents, Non-Steroidal; Antineoplastic Agents, Hormonal; Antineoplastic Combined Chemotherapy Protocols; Clinical Trials as Topic; Fibroma; Humans; Prognosis; Survival
PubMed: 12562642
DOI: 10.1093/annonc/mdg064