a Attribute is significant ( P <.05).
b Not available.
c The corresponding coefficients and P values are not provided.
In total, 2 of the 5 multicountry studies did not report preferences for each country and were therefore excluded from the synthesis of the most important attribute. A total of 53 data points on COVID-19 vaccine preferences were collected from the study population of the corresponding country. In the outcome category, among the 30 attributes examined, effectiveness emerged as the most prominent, accounting for 40% (21/53) of the studies [ 31 , 35 , 36 , 38 - 42 , 48 , 50 - 52 , 57 , 58 , 60 - 62 , 64 - 67 ]. Safety was addressed in 13% (7/53) of the studies [ 33 , 43 , 47 , 56 , 59 , 68 , 69 ], while protection duration was mentioned in 4% (2/53) [ 11 , 50 ]. In the process category, 13 attributes were identified. Brand (1/53, 2%) [ 32 ], region of vaccine manufacturer (1/53, 2%) [ 34 ], and halal content (1/53, 2%) [ 53 ] were associated with vaccine production. In addition, waiting time for COVID-19 vaccination (1/53, 2%) [ 70 ] and vaccine frequency (1/53, 2%) [ 71 ] were considered. Furthermore, 3 (6%) studies on vaccine distribution prioritized vaccination for the medical risk group (1/53, 2%) [ 72 ], those who had a higher COVID-19 mortality risk (6/53, 11%) [ 63 ], and those who had the potential capacity to spread the virus (1/53, 2%) [ 72 ]. In the cost category, personal vaccination cost accounted for 6% (3/53) [ 31 , 37 , 41 ]. Among the other attributes (7/53, 13%), disease risk threat was of particular importance, including possible trends of the epidemic (1/53, 2%) [ 30 ] and COVID-19 mortality rate (1/53, 2%) [ 55 ]. In addition, incentives and penalties for vaccination were identified, including quarantine-free travel (1/53, 2%) [ 33 ] and mandatory testing at own expense if not vaccinated (1/53, 2%) [ 44 ]. Vaccine advice or support included vaccination invitation sender (1/53, 2%) [ 73 ] and recommenders (1/53, 2%) [ 46 ]. The proportion of friends and family members who had received the vaccine (1/53, 2%) [ 26 ] was also among the other attributes influencing decision-making ( Table 2 ).
Although effectiveness remained the most important attribute, it is worth noting that variations in preferences were also observed among different subgroups. A higher proportion of studies conducted in LMICs (4/24, 17%) than in HICs (3/29, 10%) prioritized on safety ( Multimedia Appendix 5 ). In addition, COVID-19 mortality risk was the second most important attribute (6/29, 21%) after effectiveness in HICs. Cost was considered to be another most important attribute (3/24, 13%) in LMICs. Interestingly, many other attributes also became more important as the pandemic progressed. Protection duration (2/24, 8%) emerged as one of the most important attributes during the pandemic wave. COVID-19 mortality risk (5/25, 20%) and cost (3/25, 12%) were considered as the most important attributes after the pandemic wave ( Multimedia Appendix 6 ).
The overall reporting quality was deemed acceptable but there is room for improvement. The PREFS scores of the 47 studies ranged from 2 to 4, with a mean of 3.23 (SD 0.52). No study scored 5. Most studies scored 3 (32/47, 68%) or 4 (13/47, 28%), while 2 studies (2/47, 4%) scored 2 ( Multimedia Appendix 7 [ 11 , 26 , 30 - 74 ]).
This systematic review synthesizes existing data on preference for COVID-19 vaccine using DCE, with the aim of informing improvements in vaccine coverage and vaccine policy development. We identified 47 studies conducted in 29 countries, including 21 HICs and 8 LMICs. HICs had an adequate supply of vaccine since the early emergency availability of COVID-19 vaccine, and HICs had 1.5 times more doses of COVID-19 vaccinations than LMICs by September 2023 [ 85 ]. In total, 19 (40%) studies were conducted in China and 9 (19%) in the United States, demonstrating their significant contribution to the research and their leadership in vaccine research and development. Vaccine effectiveness and safety were the most important attributes in DCEs, although preferences differed among subgroups.
Recent years have seen new trends in the design, implementation, and validation of the DCE. For example, most studies (40/47, 85%) reported that the DCE was administered through web-based surveys, which have become a quick and cost-effective way to collect DCE data [ 66 ]. Almost half of the studies (25/47, 53%) did not report a pilot test. However, piloting in multiple stages throughout the development of a DCE is conducive to identifying appropriate and understandable attributes, considering whether participants can effectively evaluate the full profiles, and producing an efficient design [ 21 , 86 , 87 ].
Overall, vaccine effectiveness and safety have emerged as the most commonly investigated attributes in the outcome category. Despite heterogeneity in preferences across subpopulations, effectiveness remains the primary driver for COVID-19 vaccination across the studies [ 31 , 35 , 36 , 38 - 42 , 48 , 50 , 51 , 57 , 58 , 60 - 62 , 64 - 67 ], similar to the previous findings [ 18 ]. A study conducted in India and Europe found that respondents’ preference for the COVID-19 vaccine increased with effectiveness and peaked at 95% effectiveness [ 45 ]. Another study conducted among university staff and students in South Africa found that vaccine effectiveness not only was a concern but also significantly influenced vaccine choice behavior [ 64 ]. Interestingly, a nationwide stated choice survey in the United States found a strong interaction between effectiveness and other attributes [ 58 ]. These findings support the ongoing efforts to maximize vaccine effectiveness while emphasizing the importance of communicating information on vaccine effectiveness to the target population for promotion [ 62 ].
Safety has also been identified as a crucial factor influencing the acceptance of COVID-19 vaccine [ 33 , 43 , 47 , 56 , 59 , 68 , 69 ]. One study indicated that the likelihood of the general public choosing vaccines with low or moderate side effects increased by 75% and 63%, respectively, compared with vaccines with high side effects. While the likelihood changed within a 30% range when most attributes other than effectiveness and safety were changed [ 69 ]. In addition, respondents in Australia expressed a willingness to wait an additional 0.04 and 1.2 months to reduce the incidence of mild and severe adverse events by 1/10,000, respectively [ 56 ].
Similar to the results of previous systematic reviews of DCEs for various vaccines [ 18 , 19 ], the most common predictors of COVID-19 vaccine acceptance are effectiveness and safety, particularly during the rapid development and rollout of COVID-19 vaccines, which essentially boils down to trust in the vaccine [ 31 ]. Respondents expressed the importance of having a safe and effective COVID-19 vaccine available as soon as possible, but the majority preferred to wait a few months to observe the experience of others rather than be the first in line [ 43 ]. Therefore, collaborating to enhance vaccine effectiveness while reducing the risk of severe side effects could be a highly effective strategy to address vaccine hesitancy and augment vaccine desirability. Dissemination of this important vaccine-related information by governments and health care institutions, along with effective communication by health care professionals, can help build public trust and ultimately increase vaccination rates [ 69 ]. However, these inherent vaccine attributes are typically beyond the control of a vaccination program, and given the ongoing mutations of SARS-CoV-2, it is challenging to predict the effectiveness of the vaccines currently in development [ 66 ]. Global collaboration between scientists and pharmaceutical companies is therefore essential to improve vaccine effectiveness and minimize side effects [ 41 ].
Vaccine production, including its origin, brand, vaccine frequency, and content, are key considerations in the process category. Vaccine brand also has a significant impact on vaccine choice [ 32 ], independent of effectiveness and safety, due to factors such as reputation, country of origin, technological advances, and reported side effects associated with the brands [ 35 ]. For vaccine origin, some studies found that participants preferred domestic vaccines to imported vaccines, which may depend on the availability or the approval of vaccines in different countries [ 31 , 41 , 50 ] or the incidence of side effects among different types of COVID-19 vaccines [ 37 ]. However, some studies found that imported vaccines were more likely to be accepted than domestically produced vaccines, which may be attributed to less trust in domestically produced vaccines [ 57 , 66 ]. A study on vaccine preferences among the Malaysian population found that the composition and production process of the COVID-19 vaccine, which complied with Islamic dietary requirements (ie, halal content) was an important factor for many Malaysians when deciding whether to be vaccinated. This underscores the substantial influence of religion on vaccine choice [ 53 ].
Vaccine frequency was emphasized to play an important role in the choice of COVID-19 vaccine among the US public, while the 90% efficacy with low side effect rate of the COVID-19 vaccine was set. The prospect of vaccinating once to get lifelong immunity was very attractive, reflecting the fact that people were effort minimizers [ 71 ]. This is similar to the nature of the 2 studies referenced in the outcome attribute, where the protection duration is prioritized. Given the threat of COVID-19, people expect the protection duration to be as long as possible [ 11 , 50 ].
When vaccine supply is limited, people tend to prioritize vaccination for those who are more susceptible to the disease, have higher mortality rates from infectious diseases, or have greater potential to spread the virus. A study in Iran found that individuals tend to prioritize vaccination for those in the community with higher potential for virus transmission [ 57 ]. In addition, results from a study in 6 European countries revealed unanimous agreement among respondents that candidates with higher mortality and infection risks should be prioritized for vaccination [ 63 ]. While another study conducted among Belgians also found that respondents would prioritize populations at higher medical risk [ 72 ].
Cost was another important factor influencing COVID-19 vaccine preferences, mostly related to out-of-pocket costs [ 31 , 37 , 41 ]. In 2 studies comparing public preferences for COVID-19 vaccines in China and the United States, vaccine efficacy emerged as the most important driver for the American public, whereas the cost of vaccination had the greatest impact on the Chinese public. This difference was likely due to the relatively stable pandemic situation in China at the time and the lower perceived risk of COVID-19. As a result, the Chinese population was more price sensitive and reluctant to pay for vaccination [ 31 , 37 , 41 ].
For the other category, several different attributes were highlighted, depending on the specific population or situation. When people perceive the threat of a disease, their desire to be vaccinated becomes more urgent. In a study among health care workers in China, participants’ expectations about the future development of COVID-19 had a greater impact on their decision to be vaccinated than their perceived risk of infection or actual case rates, which may have been influenced by their previous experience with seasonal influenza vaccination [ 30 ]. The mortality rate of COVID-19 was considered the most influential factor in the uptake of COVID-19 booster shots in Vietnam. This study was conducted during a pandemic wave in Vietnam, which may have led to an increased perception of public health risks and a greater inclination toward COVID-19 vaccination [ 55 ]. To achieve herd immunity, government authorities can implement policies of incentives and penalties for vaccination to encourage population-wide uptake. A study conducted in the Netherlands revealed that respondents particularly disliked policies that penalized those who were not vaccinated, such as mandatory testing at their own expense if they were not vaccinated [ 44 ]. Instead, they favored policies that rewarded vaccination, such as giving vaccinated individuals additional privileges through a vaccination passport. This finding is consistent with a study in Hong Kong, which found that quarantine-free travel was considered the most important motivator among university students and staff, given their frequent engagement in international travel [ 33 ].
The source of vaccine information also influences vaccine decision-making [ 30 ]. Variation in the sender of vaccination appointment invitation via SMS text messaging and recommenders may potentially influence the public’s willingness to vaccinate against a disease [ 30 , 46 , 73 ]. Furthermore, the acceptance of vaccines was observed to change as the firsthand information about vaccine side effects and effectiveness was provided by friends and family in India [ 26 ].
In HICs, COVID-19 mortality risk was the second most important attribute after effectiveness, as respondents in all 6 high-income European countries from a study of public preferences for COVID-19 vaccine distribution prioritized candidates with higher mortality risks [ 63 ]. However, individuals from LMICs appeared to be more concerned about vaccine safety than those from HICs. This may be related to greater confidence in vaccine safety in HICs due to the earlier initiation and higher rates of COVID-19 vaccination [ 85 ]. In contrast, in some LMICs, vaccine safety was reported as the main reason influencing the willingness to vaccinate due to the rapid development of the COVID-19 vaccines [ 26 , 43 , 47 , 59 , 68 , 69 , 74 , 88 ].
Interestingly, the preference for COVID-19 vaccines may also have changed as the pandemic progressed [ 63 ]. Similarly, effectiveness remained the most important attribute in all periods, possibly due to the continuing severity of the pandemic and the fear of the possible emergence of new coronavirus strains [ 43 ]. Before the pandemic wave, the information on vaccine effectiveness was limited [ 26 ], but people still considered vaccine effectiveness to be the most important driver of vaccination. However, during the pandemic, the public’s perception of the health risk increased. As vaccines were introduced and used, people seemed to become more concerned about the duration of vaccine protection and preferred a longer vaccine protection [ 11 , 50 ]. After the pandemic wave, as the pandemic situation gradually stabilized, cost, combined with their perception of the risk of susceptibility, became more important in their preferences. However, despite this shift, most of the public still believed that people who are at higher risk of infection or death should be vaccinated first [ 63 ].
Our study had several limitations. First, not all studies used the same attributes and levels, which limited our ability to perform a quantitative synthesis and directly compare the estimates of model parameters. Instead, we qualitatively synthesized and summarized the range of attributes that may be useful in the formative stage of attribute selection in future DCE surveys investigating the preference for COVID-19 vaccine. Second, although DCEs have been shown to be a valid method for eliciting preferences, the experiment may not represent real market choices but rather hypothetical scenarios with plausible and realistic attributes. However, it offers opportunities to evaluate vaccines that are not yet available in the market or to specific population [ 68 ]. Third, the commonly used classification of outcome, cost, and process was used in order to better explain the public’s preference for vaccine attributes. However, several attributes could not be properly classified, and a fourth category (ie, other attributes) had to be added [ 19 ]. Meanwhile, the variety of attributes included may make it difficult to appropriately name and interpret this category as a whole. Fifth, the PREFS checklist is limited to 5 questions and fails to elicit several criteria that should be reported in DCE studies. Also, it does not provide sufficient tools to assess the biases in a DCE, such as selection bias and nonresponse bias [ 79 , 89 ]. Finally, although there was no specific theoretical framework to structure our qualitative analysis from the 4 identified categories, our classification was based on previous studies [ 18 , 19 , 82 , 90 , 91 ] and our own findings. This synthesis led us to categorize attributes into 4 main classes, providing a clear structure for analyzing and presenting participants’ vaccine preferences and making it easier to compare their preferences across different studies.
In conclusion, this systematic review synthesized the global evidence on preferences for COVID-19 vaccines using the DCE methodology. Vaccine effectiveness and safety were found to be the main drivers for COVID-19 vaccination, highlighting the importance of global collaboration to improve vaccine effectiveness and minimize side effects, as well as the importance of communicating this vaccine-related information to the public to maximize the uptake of COVID-19 vaccines. The subgroup analyses emphasized the importance of differences in vaccine preference of specific populations and time periods in optimizing the acceptance of COVID-19 vaccines. These findings may serve as valuable insights for government agencies involved in the social mobilization process for COVID-19 vaccination. However, the response to the pandemic is a continuous learning process [ 92 ]. It is crucial for policy makers to consider preference evidence when designing policies to promote vaccination.
The authors have not received a specific grant for this research from any funding agency in the public, commercial, or not-for-profit sectors.
All data relevant to the study are included in the article or uploaded as supplemental information. Data sets of this study are available upon reasonable request to the corresponding author.
YH, SF, and YZ are joint first authors. HJ conceived the study and its methodology. YH, SF, and YZ designed, refined, and implemented the search strategy; screened articles for inclusion; and extracted and curated the data. All authors contributed to the interpretation of the results. YH, SF, and YZ wrote the initial draft of the manuscript. HJ and HW critically reviewed the manuscript. HJ supervised the study design and provided overall guidance. All authors approved the final draft of the manuscript. HJ had full access to all the data used in this study, and all authors had final responsibility for the decision to submit for publication.
None declared.
PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) 2020 checklist.
Search strategies.
Attributes included in each category.
The detailed distribution of the study period across countries.
Preference for COVID-19 vaccines among high-income countries and low- and middle-income countries (n=53).
Preference for COVID-19 vaccines in the different study periods (n=53).
Assessment of 47 included studies quality using the Purpose, Respondents, Explanation, Findings, and Significance checklist.
discrete choice experiment |
high-income country |
low- and middle-income country |
Purpose, Respondents, Explanation, Findings, and Significance |
Preferred Reporting Items for Systematic Reviews and Meta-Analyses |
Edited by A Mavragani; submitted 19.01.24; peer-reviewed by T Ricks, I Saha; comments to author 11.04.24; revised version received 01.05.24; accepted 26.05.24; published 29.07.24.
©Yiting Huang, Shuaixin Feng, Yuyan Zhao, Haode Wang, Hongbo Jiang. Originally published in JMIR Public Health and Surveillance (https://publichealth.jmir.org), 29.07.2024.
This is an open-access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work, first published in JMIR Public Health and Surveillance, is properly cited. The complete bibliographic information, a link to the original publication on https://publichealth.jmir.org, as well as this copyright and license information must be included.
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Idiopathic mesenteric phlebosclerosis (IMP) is an extremely rare disease with an unclear pathogenesis and risk factors. The clinical manifestations of IMP are mostly non-specific, mainly consisting of digestive symptoms such as abdominal pain, bloating and diarrhea. The diagnosis of IMP mainly relies on abdominal computed tomography (CT) and colonoscopy. Pathological changes associated with IMP often involve fibrous degeneration of the venous wall, which results in the thickening of the colonic wall and longitudinal calcification of the mesenteric arteries. Currently, there is no standard treatment protocol for IMP, and nonsurgical treatment is the mainstay of most medical centers. In this study, we reported a case of a 55-year-old female patient with IMP whose main clinical presentation was recurrent abdominal pain. The patient's initial diagnosis was considered an incomplete intestinal obstruction and received non-surgical treatments; however, the efficacy of the treatment was unsatisfactory. After completing abdominal CT and colonoscopy, we excluded common diseases of the digestive system (e.g., tumors, Crohn's disease), and finally considered that this patient had a high likelihood of IMP. This patient eventually underwent laparoscopic enlarged right hemicolectomy due to recurrent symptoms and poor outcomes of non-surgical treatment. Postoperative pathological results confirmed the diagnosis of IMP. During the follow-up period, the patient recovered well without recurrence of IMP. Furthermore, we have reviewed the literature related to IMP and summarized the etiology, risk factors, diagnostic methods, treatment options and prognosis of IMP.
Keywords: case report; digestive disease; idiopathic mesenteric phlebosclerosis; laparoscopic surgery; literature review.
Copyright © 2024 Liu, Tong, Shi, Zeng, Luo, Yang, Cai, Wang, Luo and Wang.
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Toshihiro ide.
1 Division of Neurology, Department of Internal Medicine, Faculty of Medicine, Saga University, Saga Japan
2 Department of Neurosurgery, Faculty of Medicine, Saga University, Saga Japan
Shinichi aishima.
3 Department of Pathology and Microbiology, Faculty of Medicine, Saga University, Saga Japan
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Accurately identifying fulminant demyelinating diseases is important for sudden onset of asymmetric cerebral white matter lesions with mass effect. Initially, immunotherapy should be administered; however, surgical intervention should be performed with poor response to medical management and evident signs of cerebral herniation.
A case of fulminant demyelinating disease of the central nervous system that required decompressive craniectomy 8 days after symptom onset is presented. The patient recovered without sequelae after a combination of neurosurgery and immunotherapy with steroids and has remained relapse‐free for 4 years.
Fulminant demyelinating disease is a rare group of inflammatory demyelinating disorders that rapidly progress to disability within days to weeks, eventually requiring hospitalization and aggressive treatment for acute attacks. 1 The present patient developed brain herniation within 8 days after symptom onset, but recovered without neurological sequelae after decompressive craniectomy and immunotherapy with steroids. Nevertheless, there have been few case reports of decompressive craniectomy for fulminant demyelinating disease, and the present case report aimed to clarify the clinical characteristics of such cases along with a review of the literature.
A 63‐year‐old woman with no history of prior viral infections or vaccinations developed a headache in mid‐October 2018. On the following day, she became lightheaded and developed nausea, and she visited a local general hospital. On medical examination, her consciousness was clear, and she had no paralysis. However, she had diplopia and ataxia of both upper limbs. Magnetic resonance imaging (MRI) of the brain showed T2 high‐signal intensity lesions around the dentate nuclei of bilateral cerebellar hemispheres to the cerebellar peduncles, the right ventral side of the medulla oblongata, and the tegmentum (Figure 1A ). On the 5th day, a mild disturbance of consciousness and paralysis of the left upper limb appeared, and MRI of the brain showed a new lesion in the right temporal lobe (Figure 1B ). On the 6th day, she was urgently transferred to our hospital for further examination and treatment.
Brain MRI. Fluid attenuated inversion recovery (FLAIR) images show T2 high‐signal intensity lesions around the dentate nuclei, cerebellar peduncles, the right ventral side of the medulla oblongata, and tegmentum (Day 3, A). A new lesion is detected in the right temporal lobe (Day 5, B). The lesions expand rapidly, and extensive T2 high‐signal intensity lesions are observed in the bilateral cerebellar hemispheres, brainstem, bilateral thalami, right basal ganglia, right posterior limb of the internal capsule, and subcortical cerebral hemispheres. The abnormal signal area also extends to the left cingulate gyrus via the body of the corpus callosum. The lesion has a mass effect; the right ventricle is slightly deformed, and the median structure is deviated to the left (Day 6, C). The lesion has enlarged further, and the brain swelling is aggravated (Day 8, D). The brain swelling has improved, and the lesion has shrunk (4 years after onset, E).
On admission, her temperature was 36.8°C, pulse was 63 beats/min, blood pressure was 146/88 mmHg, respiratory rate was 17 breaths/min, and general physical examination showed no abnormalities of note. On neurological examination, mild impairment of consciousness (Glasgow Coma Scale (GCS) E4V4M6), left hemiparesis (MMT 3), and ataxia of the extremities were evident. Meningeal stiffness was also observed. Blood tests showed mild inflammatory findings, with white blood cell count of 11,100 cells/μL and C‐reactive protein (CRP) of 1.26 mg/dL, but liver function, renal function, electrolytes, and coagulation function were all normal. Serum rheumatoid factor, anti‐nuclear antibody, anti‐SS‐A antibody, anti‐SS‐B antibody, myeloperoxidase‐anti‐neutrophil cytoplasmic antibody (MPO‐ANCA), serine proteinase3‐anti‐neutrophil cytoplasmic antibody (PR3‐ANCA), and other autoimmune markers were all negative. Anti‐aquaporin4‐antibody (anti‐AQP4 antibody) was negative, measured by both an enzyme‐linked immunosorbent assay (ELISA) and a cell‐based assay (CBA). Anti‐MOG antibody was also negative by a CBA. A cerebrospinal fluid (CSF) examination showed a normal initial pressure of 11 cmH 2 0 and a mildly elevated cell count with mononuclear cell predominance (50 cells/μL). CSF protein, β2‐microglobulin, and IgG were elevated at 96 mg/dL, 1.71 μg/mL, and 10.7 mg/dL, respectively. Myelin basic protein (MBP) was markedly elevated at 33,000 pg/mL. The IgG index was 0.64. There were no atypical cells on CSF cytology. The interleukin 6 (IL‐6) level in the CSF was markedly elevated at 1410 pg/mL. The electrocardiogram and chest X‐ray showed no abnormalities. MRI of the brain showed extensive T2 high‐signal intensity lesions in the bilateral cerebellar hemispheres, brainstem, bilateral thalami, right basal ganglia, right posterior limb of the internal capsule, and subcortical cerebral hemispheres. The abnormal signal area also extended to the left cingulate gyrus via the body of the corpus callosum. The lesion had a mass effect, with the right ventricle slightly deformed and the median structures deviated to the left (Figure 1C ). A faint contrast effect was seen at the margins of the bilateral frontal lobe lesions. No lesion was found on spinal cord MRI.
The rapid appearance of the white matter‐dominant lesions required a distinction between an inflammatory demyelinating disease of the central nervous system (CNS) and drug‐induced, toxic, or metabolic encephalopathy. However, the medical history and laboratory findings of the patient did not support the latter etiology. Furthermore, if the bilateral cerebellar hemispheres, brainstem, and bilateral thalami were involved, the asymmetric nature of cerebral white matter lesions with a pronounced mass effect and the marked increase in MBP in the CSF strongly suggested fulminant inflammatory demyelinating disease of the CNS. Therefore, steroid pulse therapy (methylprednisolone 1 g/day for 5 days) was started on the 6th day, but the disturbance of consciousness gradually worsened despite the initiation of steroids, and a follow‐up MRI showed further worsening of the brain swelling (Figure 1D ). On the 8th day, her level of consciousness decreased to GCS E1V2M4, and she appeared to have developed brain herniation; emergency decompressive craniectomy was performed on the same day. At the time of craniotomy, a brain biopsy from the right temporal lobe was performed for diagnosis. The brain biopsy specimens showed small haemorrhagic foci in the cerebral white matter (Figure 2A ) and infiltration of CD68‐positive foamy macrophages around the perivascular area (Figure 2B,C ). Klüver–Barrera staining showed demyelinating plaques around the perivascular area (Figure 2D ). These pathological findings were consistent with acute demyelinating encephalomyelitis (ADEM). After the decompressive craniectomy, immunotherapy with steroids was continued.
Brain biopsy specimens obtained from the right temporal lobe. Small haemorrhagic foci are scattered in the cerebral white matter (A). The white matter has been infiltrated with foamy macrophages (B). CD68 immunohistochemical staining is positive for histiocytes in the cerebral white matter (C). Demyelinating plaques with decreased density and staining of myelinated sheaths are seen on Klüver–Barrera staining (D). These findings suggest demyelinating disease. (A, B, hematoxylin and eosin staining, scale bar = 100 μm; C, CD68 immunostaining, scale bar = 100 μm; D, Klüver–Barrera staining, scale bar = 100 μm).
The patient required mechanical ventilation, but she was extubated on the 20th day, and she was able to follow instructions. After extubation, she was able to speak without aphasia. Following the steroid pulse therapy, oral prednisolone 50 mg/day was started, which was maintained for 2 weeks and then gradually decreased by 5 mg/week. The left hemiplegia recovered gradually, and gait training was started on the 36th day of the disease; she was able to walk on her own within 2 months from disease onset. Cranioplasty was performed on the 42nd day after improvement of brain swelling and herniation on brain MRI. After tapering, prednisolone was maintained at 10 mg/day. The left hemiplegia recovered completely, and she was discharged home on the 122nd day. For 4 years after discharge, she has been clinically and radiologically free of recurrence (Figure 1E ).
Fulminant inflammatory demyelinating disease of the CNS is a rare group of diseases in which immune‐mediated destruction of myelin occurs, but the clinical course, histopathology, and imaging findings differ from those of classical multiple sclerosis (MS). They include ADEM and its variants, acute haemorrhagic leukoencephalitis (AHL or Hurst disease), severe relapses of MS, variants of MS (tumefactive MS, Marburg variant, Baló's concentric sclerosis, myelinoclastic diffuse sclerosis), and neuromyelitis optica (NMO)‐spectrum disorders. 1 In the present case, since the patient met the diagnostic criteria for ADEM and had no recurrence for 4 years, she was diagnosed with monophasic ADEM as a clinical subtype of ADEM. 2 ADEM is an immune‐mediated demyelinating disease of the CNS that commonly affects children and young adults. 3 The radiological features of ADEM include multifocal and diffuse white matter lesions with bilateral thalamic and bilateral basal ganglia involvement, which often affects the cerebellum and the brainstem. 1 The imaging findings in our case were also compatible with ADEM. The hyperacute form of ADEM accounts for 2% of cases and is associated with rapid progression of symptoms, malignant brain edema, and a high mortality rate. 4
The interesting feature of this case is that, although the patient presented with rapid cerebral herniation due to fulminant demyelination, she recovered with no neurological sequelae after treatment by a combination of decompressive craniectomy and immunotherapy. There are a few reports of cases requiring decompressive craniectomy for autoimmune diseases of the CNS, including demyelinating diseases, primary angiitis of the CNS, 5 and neuropsychiatric systemic lupus erythematosus. 6 The reports of fulminant demyelinating disease of the CNS requiring decompressive craniectomy are summarized in Table 1 . So far, 17 cases have been reported (including the present case), with 8 cases of ADEM, 7 , 8 , 9 , 10 , 11 , 12 , 13 4 cases of AHL, 14 , 15 , 16 , 17 3 cases of TDLs, 18 , 19 , 20 and 2 cases of the Marburg variant of MS. 21 , 22 The age of onset ranged widely from 1 to 63 years, but 16 of 17 cases were in their 50s or younger, and 4 were children. The age of onset was the oldest in the present case. In addition, the incidence was higher in females (12 of 17 cases). Prior infections were known in 9 cases, and most of them were respiratory tract infections such as upper respiratory tract infection and pneumonia (including 2 cases of mycoplasma infection). In addition, ADEM and AHL were often accompanied by prior infection (9 of 12 cases). Steroids were the most commonly used immunotherapy in combination with decompressive craniectomy (all 16 cases with information available). Intravenous methylprednisolone (IVMP) was the most common, with 8 cases. Intravenous immunoglobulin (IVIG) and plasma apheresis therapy were also used in some cases. The outcome was complete recovery in 5 cases, neurological sequelae in 10 cases, and death in 1 case. Of the 16 cases—including the present case and the cases with information available—most (14/16) initially underwent immunotherapy. For the remaining cases, only one underwent immunotherapy after decompressive craniectomy, whereas another underwent both treatments simultaneously. In cases where immunotherapy was administered first, surgical intervention was subsequently performed if the response to medical management was inadequate, specifically in cases with (1) deteriorating consciousness, (2) increased intracranial pressure, and (3) worsening radiological findings. Previous reviews on fulminant demyelinating diseases emphasized that immunotherapy should be the first line of treatment, with decompressive craniectomy considered thereafter if the response to medical management was poor. 1 Therefore, immunotherapy with steroids should be initially administered in fulminant demyelinating diseases aiming to reduce inflammation and edema, although decompressive craniectomy should be performed in cases that present signs of cerebral herniation.
Clinical characteristics of reported cases of decompressive craniectomy for fulminant demyelinating disease.
No. | Reference | Age (y)/sex | Diagnosis | Preceding infection | Initial neurological symptoms | Duration | Immunotherapy | Clinical outcome |
---|---|---|---|---|---|---|---|---|
1 | Current case | 63/F | ADEM | No | Headache, ataxia | 8 days | IVMP, PSL | Recovered |
2 | 7 | 34/F | ADEM | No | Headache | 11 days | IVMP, PSL, IVIG | Mild hemiparesis |
3 | 8 | 51/F | ADEM | RTI | Headache, spatial disorientation | – | IVMP | Ambulatory |
4 | 9 | 38/F | ADEM | RTI | Headache, hemiparesis | 2 days | DEX, IVMP, PSL | Recovered |
5 | 10 | 41/M | ADEM | RTI | Dysarthria | 5 days | IVMP, IVIG | mRS score 3 |
6 | 11 | 18‐month‐old/F | ADEM | Febrile rash | Left hemibody seizures with fluctuating consciousness | 5 days | Corticotherapy, IVIG | Discrete hemiparesis |
7 | 12 | 32/F | ADEM | RTI | Hemiparesis | Few hours | Steroids | Death |
8 | 13 | 37/F | ADEM | RTI | Headache | 4 days | DEX | Recovered |
9 | 14 | 17/M | AHL | MP infection | Difficulties in walking | – | Steroids | Paraplegia |
10 | 15 | 31/M | AHL | MP infection | Right arm weakness | 6 days | Steroids, PE | Recovered |
11 | 16 | 7/F | AHL | No | Headache | 4 days | IVMP, PSL | Demonstrate impulsivity |
12 | 17 | 25/F | AHL | RTI | Headache | 3 weeks | IVMP, PE | Able to walk with a cane |
13 | 18 | 6/M | TDLs | No | Behavioral change, altered gait | 1 month | DEX, IVMP, PSL | Recovered |
14 | 19 | 50/F | TDLs | No | Partial seizures, dysdiadochokinesis | 2 weeks | Steroids | Very mild dysphasia and slight paresis of the right arm |
15 | 20 | 53/F | TDLs | – | – | Few hours | – | – |
16 | 21 | 38/M | Marburg variant of MS | No | Right lower extremity palsy | – | DEX | Alive |
17 | 22 | 31/F | Marburg variant of MS | No | Consciousness deterioration, hemiparesis | 7 days | DEX, PP, IVIG | mRS score 2 |
Abbreviations: ADEM, acute disseminated encephalomyelitis; AHL, acute haemorrhagic leukoencephalitis; DEX, dexamethasone; IVIG, intravenous immunoglobulin; IVMP, intravenous methylprednisolone; MP, Mycoplasma pneumoniae ; mRS, modified Rankin Scale; MS, multiple sclerosis; PE, plasma exchange; PP, plasmapheresis; PSL, prednisolone; RTI, respiratory tract infection; TDLs, tumefactive demyelination lesions.
Although the cytokine profile in the CSF was not investigated in any of these reports, the present case was characterized by a markedly elevated IL‐6 level in the CSF. IL‐6 is a pleiotropic cytokine present during inflammation. In inflammatory diseases of the CNS, elevated levels of IL‐6 in CSF have been reported in NMO, 23 ADEM, 24 AHL, 25 and progressive leukoencephalitis after SARS‐CoV‐2 infection. 26 The clinical efficacy of satralizumab, a humanized monoclonal antibody targeting the IL‐6 receptor, has already been demonstrated in NMO in a randomized, controlled trial, 27 and tocilizumab, another humanized monoclonal antibody targeting the IL‐6 receptor, has been reported to be effective in AHL in childhood. 28 Although decompressive craniectomy and immunotherapy with steroids were effective in the present case, it may be important to evaluate IL‐6 levels in the CSF, and administration of IL‐6 receptor inhibitors may be one treatment option for the clinical management of fulminant demyelinating disease, considering that mortality and serious disability may accompany the disease. It is important to continue to enhance our knowledge of IL‐6 in CNS inflammatory diseases.
In summary, a case of a fulminant demyelinating disease of the CNS that required decompressive craniectomy for rapid development of cerebral herniation was presented. It is important to perform neurosurgery with immunotherapy within a reasonable period of time because fatal cerebral herniation can develop even in autoimmune diseases of the CNS within a week of onset. In addition, the IL‐6 level in the CSF was significantly elevated in this case. It is important to investigate the cytokine/chemokine profile in the CSF of cases of fulminant demyelination.
Toshihiro Ide: Conceptualization; investigation; writing – original draft; writing – review and editing. Ryo Ebashi: Supervision. Makoto Eriguchi: Supervision. Shinichi Aishima: Supervision. Tatsuya Abe: Supervision. Hideo Hara: Writing – review and editing.
This case report did not receive any specific grant from funding agencies in the public, commercial, or not‐for‐profit sectors.
None declared.
Written, informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.
Ide T, Ebashi R, Eriguchi M, Aishima S, Abe T, Hara H. Fulminant demyelinating disease of the central nervous system effectively treated with a combination of decompressive craniectomy and immunotherapy: A case report and literature review . Clin Case Rep . 2024; 12 :e9059. doi: 10.1002/ccr3.9059 [ CrossRef ] [ Google Scholar ]
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Chatgpt could help clinicians more effectively review medical literature by prioritizing and summarizing research abstracts from journals related to their specialties..
University of Kansas Medical Center researchers have demonstrated that ChatGPT could help clinicians keep up with ever-growing medical knowledge by prioritizing and summarizing journal abstracts.
The research team emphasized that global clinical knowledge is expanding at a pace that makes it difficult for clinicians to stay abreast of new medical literature and practice guidelines.
"There are about a million new articles added to PubMed every year," explained Daniel Parente, MD, Ph.D., assistant professor of family medicine and community health at the University of Kansas Medical Center, in a press release. "Even if you're a physician restricting your focus to your field, it can still be many thousands of articles you might think about reading."
The study authors further noted that on top of sifting through the literature to find articles relevant to their fields, clinicians must then review each article. Article abstracts can help streamline this process, but reviewing these -- many of which are around 300 words -- can be time-consuming, as well.
This study shows us that these tools already have some ability to help us review the literature a little bit faster, as well as figure out where we need to focus our attention. Daniel Parente, MD, Ph.D. Assistant professor of family medicine and community health, University of Kansas Medical Center
Given recent developments in AI technology, the researchers set out to investigate whether a large language model ( LLM ) could be used by clinicians to systematically review medical literature.
The team selected ChatGPT-3.5 and tasked the tool with summarizing 140 peer-reviewed abstracts from 14 journals. To assess the LLM's performance, human physicians were asked to rate the quality, accuracy and bias of the ChatGPT-generated summaries.
From there, the researchers compared how well both ChatGPT and the clinicians could rate the relevance of each journal and abstract to particular medical specialties.
The analysis revealed that the LLM's summaries were on average 70% shorter than the original abstracts, reducing the length from 2,438 to 739 characters. The human raters determined that ChatGPT's summaries were also generally high-quality and accurate, with low bias.
However, the LLM was found to have hallucinated in four of the 140 cases, and there were 20 instances of minor inaccuracies found. Despite the presence of these inaccuracies, it was established that they did not change the meanings of the original abstracts.
ChatGPT was less successful at identifying relevance. The LLM performed similarly to the clinicians when classifying whether an entire journal was relevant to a given specialty, but the model fell short when asked to do the same with individual articles.
"We asked the human (physician) raters to say, is this relevant to primary care or internal medicine or surgery? And then we compared to ChatGPT's relevance ratings, and we found that at least the ChatGPT-3.5 model is not quite ready to do that yet. It works well at identifying if a journal is relevant to primary care, but it's not great for identifying if an article is relevant to primary care," Parente noted.
These findings led the researchers to conclude that the use of ChatGPT in healthcare could help family physicians streamline their literature review process, and the team designed software for this purpose during the study. However, the authors underscored that critical medical decisions should still be made based on thorough evaluations of full-text research and clinical guidelines.
The researchers also indicated that as new versions of ChatGPT are released, they are likely to get better at determining the relevance of scientific articles.
"This study shows us that these tools already have some ability to help us review the literature a little bit faster, as well as figure out where we need to focus our attention," said Parente. "And it seems very likely that future versions of these technologies that are smarter and more capable will only enhance that."
Shania Kennedy has been covering news related to health IT and analytics since 2022.
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Examples of literature reviews. Step 1 - Search for relevant literature. Step 2 - Evaluate and select sources. Step 3 - Identify themes, debates, and gaps. Step 4 - Outline your literature review's structure. Step 5 - Write your literature review.
A literature review is a document or section of a document that collects key sources on a topic and discusses those sources in conversation with each other (also called synthesis ). The lit review is an important genre in many disciplines, not just literature (i.e., the study of works of literature such as novels and plays).
A literature review is a critical analysis and synthesis of existing research on a particular topic. It provides an overview of the current state of knowledge, identifies gaps, and highlights key findings in the literature. 1 The purpose of a literature review is to situate your own research within the context of existing scholarship ...
What kinds of literature reviews are written? Narrative review: The purpose of this type of review is to describe the current state of the research on a specific topic/research and to offer a critical analysis of the literature reviewed. Studies are grouped by research/theoretical categories, and themes and trends, strengths and weakness, and gaps are identified.
A literature review is a review and synthesis of existing research on a topic or research question. A literature review is meant to analyze the scholarly literature, make connections across writings and identify strengths, weaknesses, trends, and missing conversations. A literature review should address different aspects of a topic as it ...
A literature review is a survey of scholarly sources on a specific topic. It provides an overview of current knowledge, allowing you to identify relevant theories, methods, and gaps in the existing research. There are five key steps to writing a literature review: Search for relevant literature. Evaluate sources.
A literature review is an overview of the previously published works on a topic. The term can refer to a full scholarly paper or a section of a scholarly work such as a book, or an article. Either way, a literature review is supposed to provide the researcher /author and the audiences with a general image of the existing knowledge on the topic ...
The purpose of a literature review. The four main objectives of a literature review are:. Studying the references of your research area; Summarizing the main arguments; Identifying current gaps, stances, and issues; Presenting all of the above in a text; Ultimately, the main goal of a literature review is to provide the researcher with sufficient knowledge about the topic in question so that ...
A literature review is an integrated analysis-- not just a summary-- of scholarly writings and other relevant evidence related directly to your research question.That is, it represents a synthesis of the evidence that provides background information on your topic and shows a association between the evidence and your research question.
A literature review is a written work that: Compiles significant research published on a topic by accredited scholars and researchers; Surveys scholarly articles, books, dissertations, conference proceedings, and other sources; Examines contrasting perspectives, theoretical approaches, methodologies, findings, results, conclusions.
A literature review can be just a simple summary of the sources, but it usually has an organizational pattern and combines both summary and synthesis. A summary is a recap of the important information of the source, but a synthesis is a re-organization, or a reshuffling, of that information. It might give a new interpretation of old material or ...
Mapping the gap. The purpose of the literature review section of a manuscript is not to report what is known about your topic. The purpose is to identify what remains unknown—what academic writing scholar Janet Giltrow has called the 'knowledge deficit'—thus establishing the need for your research study [].In an earlier Writer's Craft instalment, the Problem-Gap-Hook heuristic was ...
Most literature reviews are embedded in articles, books, and dissertations. In most research articles, there are set as a specific section, usually titled, "literature review", so they are hard to miss.But, sometimes, they are part of the narrative of the introduction of a book or article. This section is easily recognized since the author is engaging with other academics and experts by ...
Steps to Completing a Literature Review. Find. Conduct searches for relevant information. Evaluate. Critically review your sources. Summarize. Determine the most important and relevant information from each source, theories, findings, etc. Synthesize. Create a synthesis matrix to find connections between resources, and ensure your sources ...
The word "literature review" can refer to two related things that are part of the broader literature review process. The first is the task of reviewing the literature - i.e. sourcing and reading through the existing research relating to your research topic. The second is the actual chapter that you write up in your dissertation, thesis or ...
A formal literature review is an evidence-based, in-depth analysis of a subject. There are many reasons for writing one and these will influence the length and style of your review, but in essence a literature review is a critical appraisal of the current collective knowledge on a subject. Rather than just being an exhaustive list of all that ...
WRITING A TARGETED LITERATURE REVIEW a targeted literature review is NOT: ¡ a sophisticated evaluation of the entire literature or literatures related to your topic ¡ a set of thinly connected summaries of important related works haphazardly selected from many subfields a targeted literature review IS: ¡ a carefully curated set of sources from a small number of subfield literatures
the literature review journey, this chapter is designed to help you understand the process and skills involved in navigating the literature and reaching your ultimate destination. Learning Outcomes By the end of this chapter you should be able to: • explain what a literature review is.
Literature reviews are in great demand in most scientific fields. Their need stems from the ever-increasing output of scientific publications .For example, compared to 1991, in 2008 three, eight, and forty times more papers were indexed in Web of Science on malaria, obesity, and biodiversity, respectively .Given such mountains of papers, scientists cannot be expected to examine in detail every ...
at each of these in turn.IntroductionThe first part of any literature review is a way of inviting your read. into the topic and orientating them. A good introduction tells the reader what the review is about - its s. pe—and what you are going to cover. It may also specifically tell you.
Steps for Conducting a Lit Review; Finding "The Literature" Organizing/Writing; APA Style This link opens in a new window; Chicago: Notes Bibliography This link opens in a new window; MLA Style This link opens in a new window; Sample Literature Reviews. Sample Lit Reviews from Communication Arts; Have an exemplary literature review? Get Help!
A literature review is a survey of scholarly sources that provides an overview of a particular topic. Literature reviews are a collection of the most relevant and significant publications regarding that topic in order to provide a comprehensive look at what has been said on the topic and by whom. The basic components of a literature review include:
Review the most influential work around any topic by area, genre & time. Paper * Patent Grant Clinical-Trial. Web. Expert · Past Year Past 5 Years ALL ·. LLM. Expand Tweak. Try: style transfer · covid vaccine · more | research copilot · academic writing · review by venue.
Literature Review Schizophrenia Symptomatology and Heterogeneity Schizophrenia disorder (SZ) is a serious mental illness that affects approximately 1% of the population; characterised by: (1) an extreme disintegration with reality in form of delusions and hallucinations (i.e., positive symptoms); (2) a number of negative symptoms (e.g ...
Often enough the grandeur, audacity and nuttiness of the opening ceremony shone through onscreen, but commentary, cutaways and commercials fragmented the TV experience, our critic writes.
Background: Vaccination can be viewed as comprising the most important defensive barriers to protect susceptible groups from infection. However, vaccine hesitancy for COVID-19 is widespread worldwide. Objective: We aimed to systematically review studies eliciting the COVID-19 vaccine preference using discrete choice experiments. Methods: A literature search was conducted in PubMed, Embase, Web ...
Postoperative pathological results confirmed the diagnosis of IMP. During the follow-up period, the patient recovered well without recurrence of IMP. Furthermore, we have reviewed the literature related to IMP and summarized the etiology, risk factors, diagnostic methods, treatment options and prognosis of IMP.
Book review. First published online July 31, 2024. Book Review: Linguistics and English Literature: An Introduction (Cambridge Introductions to the English Language) ... Adamson H. D., Linguistics and English Literature: An Introduction (Cambridge Introductions to the English Language), Cambridge: Cambridge University Press, 2019; xviii + 348 ...
Ide T, Ebashi R, Eriguchi M, Aishima S, Abe T, Hara H. Fulminant demyelinating disease of the central nervous system effectively treated with a combination of decompressive craniectomy and immunotherapy: A case report and literature review. Clin Case Rep. 2024; 12:e9059. doi: 10.1002/ccr3.9059 [Google Scholar]
Given recent developments in AI technology, the researchers set out to investigate whether a large language model could be used by clinicians to systematically review medical literature.The team selected ChatGPT-3.5 and tasked the tool with summarizing 140 peer-reviewed abstracts from 14 journals. To assess the LLM's performance, human physicians were asked to rate the quality, accuracy and ...