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International Journal of Surgery... May 2023Microsurgical treatment options for lymphedema consist mainly of lymphovenous anastomosis (LVA) and vascularized lymph node transfers (VLNTs). There are no standard... (Meta-Analysis)
Meta-Analysis
BACKGROUND
Microsurgical treatment options for lymphedema consist mainly of lymphovenous anastomosis (LVA) and vascularized lymph node transfers (VLNTs). There are no standard measurements of the effectiveness of these interventions and reported outcomes vary among studies.
METHODS
A systematic review and meta-analysis were performed based on a structured search in Embase, Medline, PubMed, Cinahl, Cochrane, and ProQuest in October 2020, with an update in February 2022. Firstly, a qualitative summary of the main reported outcomes was performed, followed by a pooled meta-analysis of the three most frequently reported outcomes using a random effects model. Randomized controlled trials, prospective cohorts, retrospective cohorts, and cross-sectional and case-control studies that documented outcomes following microsurgery in adult patients were included. Studies of other surgical treatments (liposuction, radical excision, lymphatic vessel transplantation) or without reported outcomes were excluded. The study protocol was registered on PROSPERO (International Prospective Register of Systematic Reviews) (ID: CRD42020202417). No external funding was received for this review.
RESULTS
One hundred fifty studies, including 6496 patients, were included in the systematic review. The qualitative analysis highlighted the three most frequently reported outcomes: change in circumference, change in volume, and change in the number of infectious episodes per year. The overall pooled change in excess circumference across 29 studies, including 1002 patients, was -35.6% [95% CI: -30.8 to -40.3]. The overall pooled change in excess volume across 12 studies including 587 patients was -32.7% [95% CI: -19.8 to -45.6], and the overall pooled change in the number of cutaneous infections episodes per year across 8 studies including 248 patients was -1.9 [95% CI: -1.4 to -2.3]. The vast majority of the studies included were case series and cohorts, which were intrinsically exposed to a risk of selection bias.
CONCLUSION
The currently available evidence supports LVA and vascularized lymph node transfers as effective treatments to reduce the severity of secondary lymphedema. Standardization of staging method, outcomes measurements, and reporting is paramount in future research in order to allow comparability across studies and pooling of results.
Topics: Adult; Humans; Retrospective Studies; Cross-Sectional Studies; Lymphedema; Lymphatic Vessels; Treatment Outcome; Randomized Controlled Trials as Topic
PubMed: 37057889
DOI: 10.1097/JS9.0000000000000210 -
Clinical and Experimental Dermatology May 2022Chronic bullous dermatosis of childhood (CBDC) is a rare autoimmune subepidermal blistering disease, which can develop following vaccination or medication, or with an...
BACKGROUND
Chronic bullous dermatosis of childhood (CBDC) is a rare autoimmune subepidermal blistering disease, which can develop following vaccination or medication, or with an autoimmune condition or illness, among other causes.
AIM
To identify and better understand the clinical features of CBDC by performing a systematic review, in line with the Preferred Reporting Items for Systematic Reviews and Meta-analyses guidelines.
METHODS
Eligible studies included publication since 1980, CBDC diagnosis, case studies, subjects aged < 18 years, clinical features and no language restriction. A database search was conducted including Embase, MEDLINE and Scopus on 14 July 2021 (see Appendix for search terms). Data were assessed for risk of bias. Jamovi was used for statistical analysis. Age and sex were compared with mucocutaneous involvement, cutaneous involvement, other symptoms, human leucocyte antigen type and lesion descriptions.
RESULTS
After removing duplicate references using Endnote, 351 papers were identified, of which 91 met the inclusion criteria. These papers included 130 cases of CBDC: 110 children and 20 neonates. The ratio of male : female patients was 19 : 1 for neonates and 74:55 for children. χ² analysis with 1 degree of freedom showed that CBDC in neonates was associated with facial (χ² = 9.67, P < 0.01), mouth (χ² = 31.0, P < 0.001), upper airway (χ² = 52.7, P < 0.001), oesophageal (χ² = 34.6, P < 0.001), ophthalmic (χ² = 6.27, P = 0.01) involvement and with respiratory distress (χ² = 22.7 P < 0.001). CBDC in children was associated with genital (χ² = 3.96, P < 0.05), arm (χ² = 6.99, P < 0.01) and leg (χ² = 7.03, P < 0.01) involvement. CBDC in male patients was associated with facial (χ² = 7.01, P < 0.01), scalp (χ² = 5.96, P < 0.02) and perianal (χ² = 7.22, P < 0.01) involvement.
CONCLUSION
Neonates with CBDC are more likely to have a mucocutaneous distribution of lesions, whereas children are more likely to have cutaneous lesions. The limitations of this study include selection bias, and the small neonate sample size makes the study unrepresentative of the population. The review highlights the need for further research into the clinical features of CBDC in neonates.
Topics: Autoimmune Diseases; Child; Chronic Disease; Female; Graft vs Host Disease; Humans; Immunoglobulin A; Infant, Newborn; Male; Skin Diseases, Vesiculobullous
PubMed: 34919291
DOI: 10.1111/ced.15059 -
The Cochrane Database of Systematic... Aug 2023There is a growing body of research evidence to guide acute stroke care. Receiving care in a stroke unit improves access to recommended evidence-based therapies and... (Review)
Review
BACKGROUND
There is a growing body of research evidence to guide acute stroke care. Receiving care in a stroke unit improves access to recommended evidence-based therapies and patient outcomes. However, even in stroke units, evidence-based recommendations are inconsistently delivered by healthcare workers to patients with stroke. Implementation interventions are strategies designed to improve the delivery of evidence-based care.
OBJECTIVES
To assess the effects of implementation interventions (compared to no intervention or another implementation intervention) on adherence to evidence-based recommendations by health professionals working in acute stroke units. Secondary objectives were to assess factors that may modify the effect of these interventions, and to determine if single or multifaceted strategies are more effective in increasing adherence with evidence-based recommendations.
SEARCH METHODS
We searched CENTRAL, MEDLINE, Embase, CINAHL, Joanna Briggs Institute and ProQuest databases to 13 April 2022. We searched the grey literature and trial registries and reviewed reference lists of all included studies, relevant systematic reviews and primary studies; contacted corresponding authors of relevant studies and conducted forward citation searching of the included studies. There were no restrictions on language and publication date.
SELECTION CRITERIA
We included randomised trials and cluster-randomised trials. Participants were health professionals providing care to patients in acute stroke units; implementation interventions (i.e. strategies to improve delivery of evidence-based care) were compared to no intervention or another implementation intervention. We included studies only if they reported on our primary outcome which was quality of care, as measured by adherence to evidence-based recommendations, in order to address the review aim.
DATA COLLECTION AND ANALYSIS
Two review authors independently selected studies for inclusion, extracted data and assessed risk of bias and certainty of evidence using GRADE. We compared single implementation interventions to no intervention, multifaceted implementation interventions to no intervention, multifaceted implementation interventions compared to single implementation interventions and multifaceted implementation interventions to another multifaceted intervention. Our primary outcome was adherence to evidence-based recommendations.
MAIN RESULTS
We included seven cluster-randomised trials with 42,489 patient participants from 129 hospitals, conducted in Australia, the UK, China, and the Netherlands. Health professional participants (numbers not specified) included nursing, medical and allied health professionals. Interventions in all studies included implementation strategies targeting healthcare workers; three studies included delivery arrangements, no studies used financial arrangements or governance arrangements. Five trials compared a multifaceted implementation intervention to no intervention, two trials compared one multifaceted implementation intervention to another multifaceted implementation intervention. No included studies compared a single implementation intervention to no intervention or to a multifaceted implementation intervention. Quality of care outcomes (proportions of patients receiving evidence-based care) were included in all included studies. All studies had low risks of selection bias and reporting bias, but high risk of performance bias. Three studies had high risks of bias from non-blinding of outcome assessors or due to analyses used. We are uncertain whether a multifaceted implementation intervention leads to any change in adherence to evidence-based recommendations compared with no intervention (risk ratio (RR) 1.73; 95% confidence interval (CI) 0.83 to 3.61; 4 trials; 76 clusters; 2144 participants, I =92%, very low-certainty evidence). Looking at two specific processes of care, multifaceted implementation interventions compared to no intervention probably lead to little or no difference in the proportion of patients with ischaemic stroke who received thrombolysis (RR 1.14, 95% CI 0.94 to 1.37, 2 trials; 32 clusters; 1228 participants, moderate-certainty evidence), but probably do increase the proportion of patients who receive a swallow screen within 24 hours of admission (RR 6.76, 95% CI 4.44 to 10.76; 1 trial; 19 clusters; 1,804 participants; moderate-certainty evidence). Multifaceted implementation interventions probably make little or no difference in reducing the risk of death, disability or dependency compared to no intervention (RR 0.93, 95% CI 0.85 to 1.02; 3 trials; 51 clusters ; 1228 participants; moderate-certainty evidence), and probably make little or no difference to hospital length of stay compared with no intervention (difference in absolute change 1.5 days; 95% CI -0.5 to 3.5; 1 trial; 19 clusters; 1804 participants; moderate-certainty evidence). We do not know if a multifaceted implementation intervention compared to no intervention result in changes to resource use or health professionals' knowledge because no included studies collected these outcomes.
AUTHORS' CONCLUSIONS
We are uncertain whether a multifaceted implementation intervention compared to no intervention improves adherence to evidence-based recommendations in acute stroke settings, because the certainty of evidence is very low.
Topics: Humans; Brain Ischemia; China; Health Personnel; Stroke
PubMed: 37565934
DOI: 10.1002/14651858.CD012520.pub2 -
Psychological Medicine Aug 2020The association between childhood adversity (CA) and psychosis has been extensively investigated in recent years. An increasing body of research has also focused on the...
The association between childhood adversity (CA) and psychosis has been extensively investigated in recent years. An increasing body of research has also focused on the mediating or moderating role of biological and psychological mechanisms, as well as other risk factors that might account for the link between CA and psychosis. We conducted a systematic search of the PsychINFO, Embase, Ovid, and Web of Science databases for original articles investigating the role of genetic vulnerabilities, environmental factors, psychological and psychopathological mechanisms in the association between CA and psychosis up to August 2019. We included studies with individuals at different stages of the psychosis continuum, from subclinical psychotic experiences to diagnosed disorders. From the 28 944 records identified, a total of 121 studies were included in this review. Only 26% of the studies identified met the criteria for methodological robustness. Overall, the current evidence suggests that CA may be associated with psychosis largely independently of genetic vulnerabilities. More consistent and robust evidence supports interaction between early and recent adversities, as well as the mediating role of attachment and mood symptoms, which is suggestive of an affective pathway between CA and psychosis across the continuum from subclinical experiences to diagnosable disorder. This review highlighted numerous methodological issues with the existing literature, including selection bias, heterogeneity of measurement instruments utilised, and lack of control for potential confounders. Future research should address these limitations to more accurately estimate mediation and moderation effects on the CA-psychosis association to inform the development of preventive interventions.
Topics: Adult Survivors of Child Adverse Events; Adverse Childhood Experiences; Effect Modifier, Epidemiologic; Gene-Environment Interaction; Humans; Psychotic Disorders
PubMed: 32624020
DOI: 10.1017/S0033291720002172 -
Journal of Clinical Medicine Nov 2023Low-grade myofibroblastic sarcoma (LGMS) is a rare tumor entity which occurs in the subcutaneous and deep soft tissues; it is less common in the bone with a predilection... (Review)
Review
INTRODUCTION
Low-grade myofibroblastic sarcoma (LGMS) is a rare tumor entity which occurs in the subcutaneous and deep soft tissues; it is less common in the bone with a predilection for the extremities and the head and neck region. As confirming the diagnosis is difficult and treatment strategies are not standardized, we aimed to identify patient and tumor characteristics, and to summarize treatment strategies and their clinical outcomes to guide surgeons.
METHODS
Included were full articles reporting patients with histology of LGMS in the extremities, excluding tumors of the trunk. All patients underwent surgery but with different extend, from marginal to wide resection. Included studies should inform about local recurrence, metastasis, or evidence of disease, depending on the surgical treatment. We conducted a structured search using MEDLINE (via PubMed), Web of Science, EMBASE and Cochrane Central Register of Controlled Trials (CENTRAL) to identify studies on low-grade myofibroblastic sarcoma of the extremities. Study designs like randomized controlled trials, systematic reviews, prospective trials, retrospective studies, and case reports were included. Prospective studies and comparative studies were not available at all. Therefore, meta-analysis was not possible and statistical analysis was purely descriptive.
RESULTS
Of the 789 studies identified from our initial search, 17 studies including 59 cases reported LGMS of the extremities with the surgical treatment and clinical outcome and were therefore analyzed. In addition, we present the rare case and surgical management of a 28-year-old male patient with residual LGMS of the thumb after an initial incomplete resection. The current literature suggests that a wide excision with R0 margins should be considered the standard treatment for LGMS. In cases where surgery leads to significant functional impairment, individual options like free tissue transfer from a donor site have to be considered. Therefore, we also present an illustrative case. For all selected case series and case reports, a high risk of confounding, selection bias, information bias, and reporting bias must be anticipated. Nevertheless, this systematic review provides a comprehensive overview on surgical treatment and clinical outcomes in LGMS surgery of the extremities.
PubMed: 38002641
DOI: 10.3390/jcm12227027 -
Cureus Dec 2020Stroke is a leading cause of death, disability, and dementia worldwide. Strokes can be divided into ischemic strokes and hemorrhagic strokes. At the moment, tissue... (Review)
Review
Stroke is a leading cause of death, disability, and dementia worldwide. Strokes can be divided into ischemic strokes and hemorrhagic strokes. At the moment, tissue plasminogen activator (tPA) is the only FDA-approved drug for ischemic stroke. Minocycline (MC) and Magnesium (Mg) are promising therapies for ischemic stroke, especially in the pre-hospital setting. These drugs are readily available, inexpensive, and generally safe. We decided to investigate these drugs' neuroprotective effects in treating ischemic stroke in the acute and chronic setting. We conducted a systematic review of the published literature on MC and Mg's functional outcome in ischemic stroke. This paper's methodology included only clinical trials published in the last 15 years, using PubMed as a database. The systematic review demonstrated that MC infusion in the pre-hospital and hospital setting improved functional outcomes and disability scores. Furthermore, MC also decreased matrix metalloproteinase 9 (MMP-9) levels. MC might have a more significant effect on men than women because different molecular pathways of cerebral ischemia seem to be involved between both genders. The systematic review showed that patients with ischemic stroke did not benefit from magnesium sulfate infusion in the pre-hospital and hospital setting. Nevertheless, patients with lacunar strokes and patients who supplemented their meals with potassium-magnesium salt in the diet had better functional outcomes. Future studies would need a more significant sample of participants and a better selection to increase the study's power and avoid selection bias, respectively. Further publications could benefit from subcategorizing strokes and investigating the gender role in stroke treatment. These directives could give a more robust conclusion regarding the neuroprotective effects of these drugs.
PubMed: 33520535
DOI: 10.7759/cureus.12339 -
Defining and evaluating the Hawthorne effect in primary care, a systematic review and meta-analysis.Frontiers in Medicine 2022In 2015, we conducted a randomized controlled trial (RCT) in primary care to evaluate if posters and pamphlets dispensed in general practice waiting rooms enhanced...
In 2015, we conducted a randomized controlled trial (RCT) in primary care to evaluate if posters and pamphlets dispensed in general practice waiting rooms enhanced vaccination uptake for seasonal influenza. Unexpectedly, vaccination uptake rose in both arms of the RCT whereas public health data indicated a decrease. We wondered if the design of the trial had led to a Hawthorne effect (HE). Searching the literature, we noticed that the definition of the HE was unclear if stated. Our objectives were to refine a definition of the HE for primary care, to evaluate its size, and to draw consequences for primary care research. We designed a Preferred Reporting Items for Systematic reviews and Meta-Analyses review and meta-analysis between January 2012 and March 2022. We included original reports defining the HE and reports measuring it without setting limitations. Definitions of the HE were collected and summarized. Main published outcomes were extracted and measures were analyzed to evaluate odds ratios (ORs) in primary care. The search led to 180 records, reduced on review to 74 for definition and 15 for quantification. Our definition of HE is "an aware or unconscious complex behavior change in a study environment, related to the complex interaction of four biases affecting the study subjects and investigators: selection bias, commitment and congruence bias, conformity and social desirability bias and observation and measurement bias." Its size varies in time and depends on the education and professional position of the investigators and subjects, the study environment, and the outcome. There are overlap areas between the HE, placebo effect, and regression to the mean. In binary outcomes, the overall OR of the HE computed in primary care was 1.41 (95% CI: [1.13; 1.75]; = 97%), but the significance of the HE disappears in well-designed studies. We conclude that the HE results from a complex system of interacting phenomena and appears to some degree in all experimental research, but its size can considerably be reduced by refining study designs.
PubMed: 36425097
DOI: 10.3389/fmed.2022.1033486 -
Neurourology and Urodynamics Apr 2023Sacral neuromodulation (SNM) is a treatment approved for use in several conditions including refractory overactive bladder (OAB) and voiding dysfunction. Chronic pelvic... (Meta-Analysis)
Meta-Analysis Review
INTRODUCTION
Sacral neuromodulation (SNM) is a treatment approved for use in several conditions including refractory overactive bladder (OAB) and voiding dysfunction. Chronic pelvic pain (CPP) is a debilitating condition for which treatment is often challenging. SNM shows promising effect in patients with refractory CPP. However, there is a lack of clear evidence, especially in long-term outcomes. This systematic review will assess outcomes of SNM for treating CPP.
METHODS
A systematic search of MEDLINE, Embase, Cochrane Central and clinical trial databases was completed from database inception until January 14, 2022. Studies using original data investigating SNM in an adult population with CPP which recorded pre and posttreatment pain scores were selected. Primary outcome was numerical change in pain score. Secondary outcomes were quality of life assessment and change in medication use and all-time complications of SNM. Risk of bias was assessed using the Newcastle Ottawa Tool for cohort studies.
RESULTS
Twenty-six of 1026 identified articles were selected evaluating 853 patients with CPP. The implantation rate after test-phase success was 64.3%. Significant improvement of pain scores was reported in 13 studies; three studies reported no significant change. WMD in pain scores on a 10-point scale was -4.64 (95% confidence interval [CI] = -5.32 to -3.95, p < 0.00001) across 20 studies which were quantitatively synthesized: effects were maintained at long-term follow-up. Mean follow-up was 42.5 months (0-59). Quality of life was measured by RAND SF-36 and EQ-5D questionnaires and all studies reported improvement in quality of life. One hundred and eighty-nine complications were reported in 1555 patients (Clavien-Dindo Grade I-IIIb). Risk of bias ranged from low to high risk. Studies were case series and bias stemmed from selection bias and loss to follow-up.
CONCLUSION
Sacral Neuromodulation is a reasonably effective treatment of Chronic Pelvic Pain and significantly reduces pain and increases patients' quality of life with immediate to long-term effects.
Topics: Adult; Humans; Electric Stimulation Therapy; Quality of Life; Urinary Bladder, Overactive; Treatment Outcome; Pelvic Pain
PubMed: 36877182
DOI: 10.1002/nau.25167 -
The Cochrane Database of Systematic... Nov 2023Fractures of the calcaneus (heel bone) comprise up to 2% of all fractures. These fractures are mostly caused by a fall from a height, and are common in younger adults.... (Review)
Review
BACKGROUND
Fractures of the calcaneus (heel bone) comprise up to 2% of all fractures. These fractures are mostly caused by a fall from a height, and are common in younger adults. Treatment can be surgical or non-surgical; however, there is clinical uncertainty over optimal management. This is an update of a Cochrane Review first published in 2013.
OBJECTIVES
To assess the effects (benefits and harms) of surgical versus conservative treatment of displaced intra-articular calcaneal fractures.
SEARCH METHODS
We searched the Cochrane Bone, Joint and Muscle Trauma Group Specialised Register, CENTRAL, MEDLINE, Embase, and clinical trials registers in November 2022.
SELECTION CRITERIA
We included randomised controlled trials (RCTs) and quasi-RCTs comparing surgical versus non-surgical management of displaced intra-articular calcaneal fractures in skeletally mature adults (older than 14 years of age). For surgical treatment, we included closed manipulation with percutaneous wire fixation, open reduction with internal fixation (ORIF) with or without bone graft, or primary arthrodesis. For non-surgical treatment, we included ice, elevation and rest, or plaster cast or splint immobilisation.
DATA COLLECTION AND ANALYSIS
We used standard Cochrane methodological procedures. We collected data for the following outcomes: function in the short term (within three months of injury) or long term (more than three months after injury), chronic pain, health-related quality of life (HRQoL) and ability to return to normal activities, as well as complications which may or may not have led to an unplanned return to theatre.
MAIN RESULTS
We included 10 RCTs and two quasi-RCTs with 1097 participants. Sample sizes in studies ranged from 29 to 424 participants. Most participants were male (86%), and the mean age in studies ranged from 28 to 52 years. In the surgical groups, participants were mostly managed with ORIF with plates, screws, or wires; one study used only minimally invasive techniques. Participants in the non-surgical groups were managed with a plaster cast, removable splint or a bandage, or with rest, elevation, and sometimes ice. Risk of performance bias was unavoidably high in all studies as it was not possible to blind participants and personnel to treatment; in addition, some studies were at high or unclear risk of other types of bias (including high risk of selection bias for quasi-RCTs, high risk of attrition bias, and unclear risk of selective reporting bias). We downgraded the certainty of all the evidence for serious risk of bias. We also downgraded the certainty of the evidence for imprecision for all outcomes (except for complications requiring return to theatre for subtalar arthrodesis) because the evidence was derived from few participants. We downgraded the evidence for subtalar arthrodesis for inconsistency because the pooled data included high levels of statistical heterogeneity. We found that surgical management may improve function at six to 24 months after injury when measured using the American Orthopaedic Foot and Ankle Society (AOFAS) score (mean difference (MD) 6.58, 95% confidence interval (CI) 1.04 to 12.12; 5 studies, 319 participants; low-certainty evidence). We are not aware of a published minimal clinically important difference (MCID) for the AOFAS score for this type of fracture. Previously published MCIDs for other foot conditions range from 2.0 to 7.9. No studies reported short-term function within three months of injury. Surgical management may reduce the number of people with chronic pain up to 24 months after injury (risk ratio (RR) 0.56, 95% CI 0.37 to 0.84; 4 studies, 175 participants; low-certainty evidence); this equates to 295 per 1000 fewer people with pain after surgical management (95% CI 107 to 422 per 1000). Surgical management may also lead to improved physical HRQoL (MD 6.49, 95% CI 2.49 to 10.48; 2 studies, 192 participants; low-certainty evidence). This outcome was measured using the physical component score of the 36-Item Short Form Health Survey. We used a change in effect of 5% to indicate a clinically important difference for this scoring system and thus judged that the difference in HRQoL between people treated surgically or non-surgically includes both clinically relevant and not relevant changes for those treated surgically. There may be little or no difference in the number of people who returned to work within 24 months (RR 1.26, 95% CI 0.94 to 1.68; 5 studies, 250 participants; low-certainty evidence) or who require secondary surgery for subtalar arthrodesis (RR 0.38, 95% CI 0.09 to 1.53; 3 studies, 657 participants; low-certainty evidence). For other complications requiring return to theatre in people treated surgically, we found low-certainty evidence for amputation (2.4%; 1 study, 42 participants), implant removal (3.4%; 3 studies, 321 participants), deep infection (5.3%; 1 study, 206 participants), and wound debridement (2.7%; 1 study, 73 participants). We found low-certainty evidence that 14% of participants who were treated surgically (7 studies, 847 participants) had superficial site infection.
AUTHORS' CONCLUSIONS
Our confidence in the evidence is limited. Although pooled evidence indicated that surgical treatment may lead to improved functional outcome but with an increased risk of unplanned second operations, we judged the evidence to be of low certainty as it was often derived from few participants in studies that were not sufficiently robust in design. We found no evidence of a difference between treatment options in the number of people who needed late reconstruction surgery for subtalar arthritis, although the estimate included the possibility of important harms and benefits. Large, well-conducted studies that attempt to minimise detection bias and that measure functional outcomes using calcaneal-specific measurement tools would increase the confidence in these findings. Given that minimally invasive surgical procedures are already becoming more prevalent in practice, research is urgently needed to determine whether these newer surgical techniques offer better outcomes with regard to function, pain, quality of life, and postoperative complications for intra-articular displaced calcaneal fractures.
Topics: Male; Adult; Humans; Middle Aged; Female; Fracture Fixation; Chronic Pain; Ice; Fractures, Bone; Bandages
PubMed: 37933733
DOI: 10.1002/14651858.CD008628.pub3 -
International Journal of Dermatology Mar 2023Woringer-Kolopp disease (WKD), also known as localized pagetoid reticulosis, is a rare variant of mycosis fungoides as described by the World Health... (Review)
Review
OBJECTIVE
Woringer-Kolopp disease (WKD), also known as localized pagetoid reticulosis, is a rare variant of mycosis fungoides as described by the World Health Organization-European Organization for Research and Treatment of Cancer (WHO-EORTC) classification system for cutaneous lymphomas. Our objective was to conduct a comprehensive review that describes and evaluates patient demographics, clinical presentation, immunohistochemical findings, management, and outcomes of WKD.
METHODS
The databases PubMed, Embase, and Cochrane Library were searched for relevant literature. Patient demographics, imaging, treatments, and other clinical characteristics were obtained. The literature search was conducted on December 7, 2020. Studies were included if they contained primary data related to WKD. Non-pertinent studies, non-English studies, non-human studies, review articles, or studies with insufficient case information were excluded. The quality of the included studies and the risk of bias were evaluated using the Grades of Recommendation, Assessment, Development, and Evaluation Group (GRADE) criteria (2016), Oxford Centre for Evidence-Based Medicine: Levels of Evidence (OCEBM) (March 2009), and the Methodological Index for Non-Randomized Studies (MINORS) criteria (2003).
RESULTS
A total of 84 studies with 143 patients met the inclusion criteria for this study. The most common chief complaint was an asymptomatic, slow-growing, and erythematous plaque. The average time from initial skin eruption to presentation was 97.6 months. Histologically, 128 cases of WKD displayed epidermotropism (97.7%). Various therapies, including radiotherapy, surgery, and local chemotherapy, were used. In total, 92 (78.6%) cases had complete remission and 11 (9.4%) cases recurred.
CONCLUSIONS
WKD is a rare cutaneous T-cell lymphoma that often presents as a single plaque on the extremities with an indolent course and a favorable prognosis. However, it is often misdiagnosed, leading to delays in treatment. The diagnosis of WKD involves clinical appraisal, a biopsy of suspicious lesions, and immunohistochemistry. Monotherapy appears to be preferred compared to combination therapy for WKD with radiotherapy demonstrating among the highest cure rates and lowest recurrence. Future retrospective and prospective studies are needed to accurately determine the epidemiology, prognosis, and effective treatments for WKD. Limitations include a possibility of missed studies, heterogeneity in reporting methods, publication, and selection bias.
Topics: Humans; Pagetoid Reticulosis; Retrospective Studies; Mycosis Fungoides; Skin Neoplasms; Lymphoma, T-Cell, Cutaneous
PubMed: 35485962
DOI: 10.1111/ijd.16224