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Effect of Tranilast on the Frequency of Invasive Treatment for Extra-Abdominal Desmoid Fibromatosis.Journal of Nippon Medical School =... Mar 2023Active surveillance (AS) has been suggested for managing extra-abdominal desmoid fibromatosis (EADF), but a substantial percentage of such patients transitioned to...
BACKGROUND
Active surveillance (AS) has been suggested for managing extra-abdominal desmoid fibromatosis (EADF), but a substantial percentage of such patients transitioned to invasive secondary treatments. The anti-keloid medication tranilast is frequently used in Japan but its effectiveness for EADF is not well understood.
METHODS
We retrospectively analyzed the medical records of EADF patients treated with tranilast between January 2009 and March 2021. EADF has been reported to shrink spontaneously, so the effects of all drugs must be compared with AS. To assess the effect of tranilast, we compared the clinical courses of patients receiving tranilast with those managed by AS (as identified in a systematic review). A systematic review of AS outcomes was conducted on July 22, 2021, in accordance with PRISMA guidelines. The primary endpoint was rate of conversion to secondary treatment. Secondary endpoints were progression-free survival, objective response rate (ORR), disease control rate (DCR), and adverse events. The rates of conversion to secondary treatment, ORRs, and DCRs were compared between the two groups by using the Fisher exact test.
RESULTS
Eighteen patients who received tranilast as initial treatment for EADF were included. Two patients (11.1%) underwent surgical resection for treatment of tumor growth and persistent pain. The rate of conversion to secondary treatment was significantly lower for tranilast than for a pure AS approach (40.1%; p = 0.01). ORR and DCR did not differ between groups.
CONCLUSIONS
Tranilast was better than AS for initial management of EADF.
Topics: Humans; Fibromatosis, Aggressive; Retrospective Studies; Fibroma; Japan
PubMed: 36436917
DOI: 10.1272/jnms.JNMS.2023_90-113 -
BMC Musculoskeletal Disorders May 2019As treatment of Dupuytren disease (DD) is expected to shift towards prevention of progression, the use of imaging in patients with DD becomes more important. In this...
BACKGROUND
As treatment of Dupuytren disease (DD) is expected to shift towards prevention of progression, the use of imaging in patients with DD becomes more important. In this systematic review an overview is given of the different methods for and applications of imaging for DD that have been described.
METHODS
The MEDLINE and EMBASE databases were searched for articles reporting the use of imaging in patients with DD, published before May 17, 2018. Studies were systematically examined in two rounds by two observers according to the PRISMA systematic. All studies containing original data on imaging for DD were considered for inclusion.
RESULTS
Three hundred and seven unique studies were identified, of which 23 were included in the study. Only studies on the use of ultrasound (US) and magnetic resonance imaging (MRI) were identified. Broadly, articles could be divided into 5 categories. Seven studies were found on diagnosis, two on measurement of disease extent, four on measurement of disease activity, seven on guidance of minimally invasive procedures and five studies on evaluation of treatment. According to the Oxford CEBM, the levels of evidence were low, ranging from level 3 to 5.
CONCLUSIONS
A variety of applications for US and MRI for patients with DD has been described. Based on the results of this review, the largest value for imaging lies in the measurement of disease activity and the follow-up of treatment of patients with early stage disease. Unfortunately, the overall level of evidence of the available literature was low. Future research is necessary to define the exact value of US and MRI in the management of patients with DD.
Topics: Disease Progression; Dupuytren Contracture; Humans; Magnetic Resonance Imaging; Ultrasonography
PubMed: 31101038
DOI: 10.1186/s12891-019-2606-0 -
Head and Neck Pathology Sep 2023Granular Cell Odontogenic Fibroma (GCOF) is a rare odontogenic neoplasm reported over time with different names. The purpose of this study is to review all available...
BACKGROUND
Granular Cell Odontogenic Fibroma (GCOF) is a rare odontogenic neoplasm reported over time with different names. The purpose of this study is to review all available data on the GCOF in the scientific literature, with a summary of all reported cases and a report of a new case.
METHODS
This review was conducted following the PRISMA guidelines. An electronic search was performed up to November 2022.
RESULTS
Thirty-nine studies reporting fifty-three cases were included. GCOF is a rare neoplasm among the odontogenic tumors, with a higher prevalence in women of the middle-aged and white population. This lesion occurs mostly on the posterior region of the mandible. Furthermore, based on clinical, radiographic, and histopathologic features, conservative treatment was the most reported choice with recurrence reported in two cases.
CONCLUSION
GCOF remains controversial due to the still unsolved histogenesis.
Topics: Female; Humans; Middle Aged; Fibroma; Mandible; Odontogenic Tumors; Case Reports as Topic
PubMed: 37118351
DOI: 10.1007/s12105-023-01553-x -
Nagoya Journal of Medical Science Nov 2021The efficacy and safety of cyclooxygenase 2 (COX2) inhibitors for the treatment of desmoid-type fibromatosis (DF) are unclear. Therefore, we systematically reviewed...
The efficacy and safety of cyclooxygenase 2 (COX2) inhibitors for the treatment of desmoid-type fibromatosis (DF) are unclear. Therefore, we systematically reviewed related literature to assess the efficacy and safety of COX2 inhibitors for DF treatment. We searched pertinent literature between January 1999 and August 2017 to identify relevant studies using the keywords "Fibromatosis, aggressive" and "Cyclooxygenase inhibitors." Thereafter, we screened and determined the quality of the studies using the Grading of Recommendations Assessment, Development, and Evaluation system and extracted the article data. The critical outcomes selected were the efficacy and adverse effects of COX2 inhibitors. Efficacy was evaluated in terms of clinical benefit when patients showed complete response, partial response, and stable disease. Thirty-one articles were identified from the database search, and one was identified through the reviewers' manual search. Finally, we retrieved six studies, including three case reports, comprising 89 patients after the first and second screenings. Fifty-three patients were excluded because three studies were reported from the same institution; hence, in total, 36 patients were included. Clinical benefit was noted in 64% of the patients. Three adverse effects were identified from the records of the six extracted studies. The strategy of watchful waiting using COX2 inhibitors with few side effects is weakly recommended for DF, especially DF patients with pain.
Topics: Cyclooxygenase 2 Inhibitors; Fibromatosis, Abdominal; Fibromatosis, Aggressive; Humans
PubMed: 34916712
DOI: 10.18999/nagjms.83.4.673 -
Clinical and Experimental Dental... Feb 2023The aim of this study is to evaluate recent evidence-based data that summarize the clinicopathological findings and treatment along with follow-up measures taken in... (Review)
Review
OBJECTIVE
The aim of this study is to evaluate recent evidence-based data that summarize the clinicopathological findings and treatment along with follow-up measures taken in terms of published cases of Juvenile psammomatoid ossifying fibroma (JPOF) of the maxilla and mandible by a systematic review. MATERIALS AND METHODS: The databases searched were PubMed, MEDLINE, Scopus, Google scholar, and Cross references. Only those case reports of JPOFs published in the English language from 2000 to 2022 were considered. All cases included confirmed JPOF lesions histopathologically. The SR-included details like clinical and radiographic data, follow-up details such as recurrence, and the presence of any adverse outcome.
RESULTS
The database search produced 595 articles from 2000 to 2022, among which 22 case reports were included in the systematic review. The mean age of JPOF occurrence in patients was 18 ± 16 years. A male predilection was noted among patients younger than 14 years of age, whereas a female predilection was noted in patients older than 14 years of age. Frequent involvement of the mandible (56%) compared to the maxilla (44%) was reported. The posterior mandible was the most commonly affected site involving numerous adjacent structures. The expansile nature of the JPOF displayed 57% buccolingual expansion, 50% downward displacement or erosion of the lower border of the mandible and 81% of involvement of the maxillary antrum/pterygoid plate/orbital floor. Among the 20 cases reported, the treatment provided included surgical excision in 45% of the patients, jaw resection in 35% of the patients, and enucleation and curettage in 18% of the patients. Follow-up details were provided in 80% of the reports that showed recurrence.
CONCLUSIONS
The diagnosis of JPOF requires correlation of the clinical and radiographic features with key histopathological features. Although long-term follow-up of the case reports has been reported, the data lack information about the long-term outcomes of JPOF.
Topics: Humans; Male; Female; Child, Preschool; Child; Adolescent; Young Adult; Adult; Maxilla; Fibroma, Ossifying; Bone Neoplasms; Mandible
PubMed: 36325758
DOI: 10.1002/cre2.687 -
Journal of Orthopaedic Surgery and... Oct 2020The Dupuytren disease is a benign fibroproliferative disorder that leads to the formation of the collagen knots and fibres in the palmar fascia. The previous studies... (Meta-Analysis)
Meta-Analysis
BACKGROUND
The Dupuytren disease is a benign fibroproliferative disorder that leads to the formation of the collagen knots and fibres in the palmar fascia. The previous studies reveal different levels of Dupuytren's prevalence worldwide; hence, this study uses meta-analysis to approximate the prevalence of Dupuytren globally.
METHODS
In this study, systematic review and meta-analysis have been conducted on the previous studies focused on the prevalence of the Dupuytren disease. The search keywords were Prevalence, Prevalent, Epidemiology, Dupuytren Contracture, Dupuytren and Incidence. Subsequently, SID, MagIran, ScienceDirect, Embase, Scopus, PubMed and Web of Science databases and Google Scholar search engine were searched without a lower time limit and until June 2020. In order to analyse reliable studies, the stochastic effects model was used and the I index was applied to test the heterogeneity of the selected studies. Data analysis was performed within the Comprehensive Meta-Analysis Software version 2.0.
RESULTS
By evaluating 85 studies (10 in Asia, 56 in Europe, 2 in Africa and 17 studies in America) with a total sample size of 6628506 individuals, the prevalence of Dupuytren disease in the world is found as 8.2% (95% CI 5.7-11.7%). The highest prevalence rate is reported in Africa with 17.2% (95% CI 13-22.3%). According to the subgroup analysis, in terms of underlying diseases, the highest prevalence was obtained in patients with type 1 diabetes with 34.1% (95% CI 25-44.6%). The results of meta-regression revealed a decreasing trend in the prevalence of Dupuytren disease by increasing the sample size and the research year (P < 0.05).
CONCLUSION
The results of this study show that the prevalence of Dupuytren disease is particularly higher in alcoholic patients with diabetes. Therefore, the officials of the World Health Organization should design measures for the prevention and treatment of this disease.
Topics: Adult; Aged; Aged, 80 and over; Alcoholism; Comorbidity; Diabetes Mellitus, Type 1; Dupuytren Contracture; Female; Global Health; Humans; Incidence; Male; Middle Aged; Prevalence; Young Adult
PubMed: 33115483
DOI: 10.1186/s13018-020-01999-7 -
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 -
European Journal of Cancer (Oxford,... Sep 2020This study evaluates the results of the active surveillance (AS) approach in adult patients with desmoid-type fibromatosis (DTF) because AS is advocated as a front-line...
BACKGROUND
This study evaluates the results of the active surveillance (AS) approach in adult patients with desmoid-type fibromatosis (DTF) because AS is advocated as a front-line approach for DTF in the European consensus guidelines.
METHODS
A systematic literature search was conducted (December 19th, 2019, updated on April 14th, 2020). Studies describing the outcomes of the AS approach were included. The PRISMA guidelines were used.
RESULTS
Twenty-five articles were included for data retrieval. Forty-two percent of reported patients (1480 of 3527 patients) received AS, the majority were women and the majority had a primary tumour. The median age at diagnosis ranged from 28 to 59 years. Common tumour sites were the extremities/girdles (n = 273), the abdominal wall (n = 253) and the trunk (n = 153). The median reported percentage of progressive disease, stable disease and partial response was 20% (interquartile range [IQR]: 13-35%), 59% (IQR: 37-69%) and 19% (IQR 3-23%), respectively. In 640 patients, the outcome was not specified. The median reported percentage of shifting to an active form of treatment was 29%, most commonly to systemic treatment (n = 195) and surgery (n = 107). The reported median follow-up time ranged between 8 and 73 months. The reported median time to progression and/or initiation of the subgroup shifting from AS to 'active' therapy ranged from 6.3 months to 19.7 months.
CONCLUSION
The majority of patients undergoing AS have either stable disease or a partial response, and about one-third of patients shift to an active form of treatment. Selecting patients who will benefit from active surveillance upfront should be the priority of future studies.
Topics: Adult; Disease Progression; Female; Fibromatosis, Aggressive; Humans; Male; Middle Aged
PubMed: 32738571
DOI: 10.1016/j.ejca.2020.06.022 -
Journal of Clinical and Experimental... Feb 2017The oral examination is an essential part of the multidisciplinary medical care in elderly people. Oral mucosal lesions and normal variations of oral anatomy (OMLs) are...
BACKGROUND
The oral examination is an essential part of the multidisciplinary medical care in elderly people. Oral mucosal lesions and normal variations of oral anatomy (OMLs) are very common in this people, but few studies have examined the frequency and prevalence of these conditions worldwide and less in Chile. The aim of this research was to evaluate the frequency of OMLs in a Chilean elderly population.
MATERIAL AND METHODS
It was conducted a retrospective study (Talca, Chile). Two hundred seventy-seven OMLs were classified in groups and anatomical sites. In order to contextualize our numbers, we made a systematic review using Publish or Perish software, Google Scholar and InteractiVenn.
RESULTS
The most prevalent OMLs groups were soft tissue tumors, epithelial pathology, facial pain and neuromuscular diseases, and dermatologic diseases. The most frequent OMLs included irritation fibroma (30 patients, 10.8%), hemangioma (20, 7.2%), burning mouth syndrome (20 cases, 7.2%), oral lichen planus (12, 4.3%) and epulis fissuratum (12, 4.3%). In the systematic review, 75 OMLs were relevant and the more studied pathologies were traumatic ulcerations (11 of 15 articles), oral lichen planus (10/15), irritation fibroma, melanotic pigmentations, and recurrent aphthous stomatitis (9/10, respectively). Considering all included articles, most frequent OMLs in elderly people included denture-related stomatitis (13.3%), irritation fibroma (8.7%) and fissured tongue (6.3%).
CONCLUSIONS
The results reflect the frequency of OMLs diagnosed in a specialized service in south of Chile and many countries around the world. These numbers will allow the establishment of preventive politics and adequacy of the clinical services. Oral mucosal lesions, elderly people, Chilean population, frequency, systematic review.
PubMed: 28210449
DOI: 10.4317/jced.53427 -
Clinical Medicine (London, England) Nov 2020
The importance of early completion of cardiac investigations after ischaemic stroke: a case and systematic review of reperfusion therapy in stroke due to cardiac fibroelastoma.
Topics: Brain Ischemia; Fibroma; Humans; Ischemic Stroke; Reperfusion; Stroke
PubMed: 33199328
DOI: 10.7861/clinmed.2020-0763