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[PMID]: 27366306
[Au] Autor:Sathyanarayanan R; Suresh V; Therese Thomas BA
[Ad] Address:Department of Oral and Maxillofacial Surgery, Indira Gandhi Institute of Dental Sciences, Puducherry, India.
[Ti] Title:Polymorphous Low-Grade Adenocarcinoma of the Palate: A Rare Case Report.
[So] Source:Iran J Cancer Prev;9(1):e3447, 2016 Feb.
[Is] ISSN:2008-2398
[Cp] Country of publication:Iran
[La] Language:eng
[Ab] Abstract:INTRODUCTION: Polymorphous low-grade adenocarcinoma (PLGA) has been known as a comparatively less aggressive malignant tumor that predominantly occurs in the minor salivary glands. It has presented as a painless, slow growing tumor in the oral cavity. It has been reported to occur rarely in the palate and the prognosis of this lesion is far better than adenocarcinoma as the regional metastasis would be very minimal. A clear difference between the biologic behaviors of adenocarcinoma and PLGA has been reported in literature. CASE PRESENTATION: A 63-year-old female reported at our institute with a swelling in relation to her upper left back region of her jaw for three months, gradual in onset and associated with pain. After clinical and radiological examination, the differential diagnosis were consolidated abscess or minor salivary gland tumor. An incisional biopsy was done and the lesion was diagnosed as PLGA. The lesion was treated by wide excision and reconstruction with immediate obturator. The post-operative follow up showed no evidence of recurrence and the healing was satisfactory. CONCLUSIONS: The site of predilection of PLGA is more in favor of palate (49 - 77.8%) followed by either upper lip or buccal mucosa (7.4 - 13.4%). There are sporadic reports of metastasis sometimes even transformation to a high grade adenocarcinoma, sometimes ending in mortality. Cervical lymph node metastasis is rare with reported incidence of 5 - 15% and is more commonly seen in recurrent tumor than the initial diseases. Extra palatal PLGAs present with significant papillary growth or arising from ventral surface of tongue frequently metastasize to cervical lymph nodes. Distant metastasis is very rare with an incidence of 7.5% and the site involved is the lung which is attributed to the inadequate control of the disease.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1607
[Da] Date of entry for processing:160701
[St] Status:PubMed-not-MEDLINE
[do] DOI:10.17795/ijcp-3447

  2 / 5399 MEDLINE  
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[PMID]: 27228997
[Au] Autor:Gisler V; Müller S; Müller L; Jörg-Walther L; Sendi P
[Ad] Address:Department of Infectious Diseases, Bern University Hospital, University of Bern, Bern, Switzerland....
[Ti] Title:Acute Angioedema Triggered by Daptomycin.
[So] Source:Infect Dis Ther;5(2):201-5, 2016 Jun.
[Is] ISSN:2193-8229
[Cp] Country of publication:New Zealand
[La] Language:eng
[Ab] Abstract:INTRODUCTION: Daptomycin is a cyclic lipopeptide antibiotic, frequently administered for Staphylococcus aureus bloodstream infections. Numerous studies have shown that daptomycin is relatively safe and well tolerated. Serious adverse events possibly related to this antimicrobial compound are rare. We report a case of acute angioedema triggered by daptomycin. CASE REPORT: A 60-year-old woman with S. aureus bacteremia without identified source was treated intravenously with high-dose beta-lactams at our institution. Because S. aureus bacteremia persisted on day 6, and in parallel, acute kidney injury developed, antimicrobial treatment was switched to a combination therapy with daptomycin and ceftriaxone. Shortly after completion of the first daptomycin administration, the patient developed lip and tongue swelling and dyspnea. Acute angioedema was clinically evident. Antibiotic therapy was switched to vancomycin, and the further clinical course was favorable. An intradermal test showed a significant wheal diameter for daptomycin, but negative results for ceftriaxone. CONCLUSION: The association with daptomycin in this case is either probable or certain. Clinicians should be aware that daptomycin can cause immediate-type hypersensitivity reactions, including acute angioedema, even upon first administration.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1607
[Da] Date of entry for processing:160701
[St] Status:PubMed-not-MEDLINE
[do] DOI:10.1007/s40121-016-0111-4

  3 / 5399 MEDLINE  
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[PMID]: 27295845
[Au] Autor:Safi MF; Wright-Harp W; Lucker JR; Payne JC; Harris O
[Ti] Title:A REVIEW OF ELECTRICAL STIMULATION AND ITS EFFECT ON LINGUAL, LABIAL AND BUCCAL MUSCLE STRENGTH.
[So] Source:Int J Orofacial Myology;40:12-29, 2014 Nov.
[Is] ISSN:0735-0120
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:BACKGROUND: Lingual, labial and buccal weakness (LLBW) is a widespread consequence of several neurological insults. LLBW impact on oral motor functions such as speech production and swallowing is well documented in the literature. Therefore, it is important for the speech-language pathologists to have access to evidence-based approaches for treatment. Thus, it is imperative that the speech-language pathology field search for effective treatment approaches and explore new treatment modalities that can improve therapy outcomes. One relatively new modality in this field is neuromuscular electrical stimulation (NMES). AIMS: The purpose of this paper is fivefold: (a) to provide an overview of the general effects of NMES on skeletal muscles; (b) to review the effect of NMES on orofacial musculature evaluating the potential appropriateness of NMES for use in strengthening lingual, labial and buccal muscles; (c) to identify future directions for research with consideration of its potential role in improving speech intelligibility and the oral preparatory phase of swallowing in patients with oral motor weakness; (d) to provide a brief anatomic and physiologic bases of LLBW; (e) to provide background information for orofacial myologists who may encounter individuals with LLBW. MAIN CONTRIBUTION: NMES is a modality that is commonly used in physical therapy and occupational therapy fields that assists in treating several motor and sensory muscular disorders including muscular weakness. The literature reviewed demonstrate that very limited data related to the use of NMES on orofacial muscles exist despite the fact that these muscles can be easily accessed by electrical stimulation from the surface. CONCLUSIONS: This review of the research using electrical stimulation of muscles highlights the need for experimental treatment studies that investigate the effect of NMES on orofacial weakness.
[Mh] MeSH terms primary: Electric Stimulation Therapy/methods
Lip Diseases/therapy
Mouth Diseases/therapy
Muscle Weakness/therapy
Tongue Diseases/therapy
[Mh] MeSH terms secundary: Deglutition/physiology
Facial Muscles/physiopathology
Humans
Muscle Strength/physiology
Neuromuscular Diseases/therapy
Speech Intelligibility/physiology
[Pt] Publication type:JOURNAL ARTICLE; REVIEW
[Em] Entry month:1606
[Js] Journal subset:D
[Da] Date of entry for processing:160614
[St] Status:MEDLINE

  4 / 5399 MEDLINE  
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[PMID]: 25847463
[Au] Autor:Bodner L; Manor E; Joshua BZ; Shaco-Levy R
[Ad] Address:Department of Oral and Maxillofacial Surgery, Soroka Medical Center, Ben-Gurion University of the Negev, Beer-Sheva, Israel....
[Ti] Title:Oral Mucoceles in Children--Analysis of 56 New Cases.
[So] Source:Pediatr Dermatol;32(5):647-50, 2015 Sep-Oct.
[Is] ISSN:1525-1470
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:BACKGROUND: Mucoceles are common cystic lesions of the oral mucosa. Extravasation mucoceles (EMs) are mainly found in the lower lip of young patients, whereas retention mucoceles (RMs) are usually located in the cheek or palate of older patients. This study was undertaken to more fully characterize the clinicopathologic features of mucoceles in pediatric patients. METHODS: The records of 56 pediatric patients with mucoceles were included in the study. Age, sex, history of trauma, intraoral site, size, and method of treatment were evaluated. Histopathologically the lesions were classified as being EMs or RMs. RESULTS: The age range was 1.5 to 16 years (mean age 11.2 yrs). Of the 56 patients, 24 (43%) were males and 32 (57%) were females, with a male:female ratio of 1:1.33. A history of trauma was recorded in 32 (57%) patients of the lower lip. The intraoral sites were the lower lip (38 [68%]), tongue (10 [18%]), and floor of the mouth (8 [14%]). Of the 56 patients, 44 (79%) were EM and 12 (21%) were ranulas. No RMs were found. Mucoceles ranged from 0.3 to 3.8 cm in diameter (mean 0.9 cm). The treatment of EMs was surgical excision. Cryosurgey, electrosurgery, and carbon dioxide laser were also used. CONCLUSION: In contrast to adults, where EM and RM types can be found, among children all cases are of the EM type. The disparate site and age incidences of EMs and RMs of the oral mucosa suggest that these two types are not related and possibly have a different etiopathogenesis.
[Mh] MeSH terms primary: Mouth Diseases/pathology
Mouth Mucosa/pathology
Mucocele/pathology
[Mh] MeSH terms secundary: Adolescent
Child
Child, Preschool
Female
Humans
Infant
Male
Mucocele/therapy
Retrospective Studies
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1606
[Js] Journal subset:IM
[Da] Date of entry for processing:150911
[St] Status:MEDLINE
[do] DOI:10.1111/pde.12535

  5 / 5399 MEDLINE  
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[PMID]: 26228106
[Au] Autor:Suzuki E; Izumi Y; Chiba Y; Horikawa R; Matsubara Y; Tanaka M; Ogata T; Fukami M; Naiki Y
[Ad] Address:Department of Molecular Endocrinology, National Research Institute for Child Health and Development, Tokyo, Japan.
[Ti] Title:Loss-of-Function SOX10 Mutation in a Patient with Kallmann Syndrome, Hearing Loss, and Iris Hypopigmentation.
[So] Source:Horm Res Paediatr;84(3):212-6, 2015.
[Is] ISSN:1663-2826
[Cp] Country of publication:Switzerland
[La] Language:eng
[Ab] Abstract:BACKGROUND: Kallmann syndrome (KS) is a clinically and genetically heterogeneous disorder consisting of hypogonadotropic hypogonadism and anosmia. KS is occasionally associated with deafness. Recently, mutations in SOX10, a well-known causative gene of Waardenburg syndrome (WS) characterized by deafness, skin/hair/iris hypopigmentation, Hirschsprung disease, and neurological defects, have been identified in a few patients with KS and deafness. However, the current understanding of the clinical consequences of SOX10 mutations remains fragmentary. CASE REPORT: A Japanese male patient presented with sensory deafness, blue irises, and anosmia, but no hair/skin hypopigmentation, Hirschsprung disease, or neurological abnormalities. He showed no pubertal sex development at 15.1 years of age. Blood examinations revealed low levels of FSH and testosterone. RESULTS: Molecular analysis detected a de novo p.Leu145Pro mutation in SOX10, which has previously been reported in a patient with WS and Hirschsprung disease. The mutation was predicted to be probably damaging. The mutant protein barely exerted in vitro transactivating activity. CONCLUSIONS: These results highlight the significance of SOX10 haploinsufficiency as a genetic cause of KS with deafness. Importantly, our data imply that the same SOX10 mutations can underlie both typical WS and KS with deafness without skin/hair hypopigmentation, Hirschsprung disease, or neurological defects.
[Mh] MeSH terms primary: Hearing Loss/genetics
Iris Diseases/genetics
Kallmann Syndrome/genetics
SOXE Transcription Factors/genetics
[Mh] MeSH terms secundary: Child
Exons/genetics
Follicle Stimulating Hormone/blood
Hearing Loss/complications
Humans
Hypopigmentation/complications
Hypopigmentation/genetics
Iris Diseases/complications
Kallmann Syndrome/complications
Male
Mutation, Missense
Sexual Maturation
Testosterone/blood
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE; RESEARCH SUPPORT, NON-U.S. GOV'T
[Nm] Name of substance:0 (SOX10 protein, human); 0 (SOXE Transcription Factors); 3XMK78S47O (Testosterone); 9002-68-0 (Follicle Stimulating Hormone)
[Em] Entry month:1606
[Js] Journal subset:IM
[Da] Date of entry for processing:150924
[St] Status:MEDLINE
[do] DOI:10.1159/000436965

  6 / 5399 MEDLINE  
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[PMID]: 26190394
[Au] Autor:Bénateau H; Traoré H; Chatellier A; Caillot A; Ambroise B; Veyssière A
[Ad] Address:Service de chirurgie maxillofaciale et plastique, centre hospitalier universitaire de Caen, avenue de la Côte-de-Nacre, 14000 Caen, France; Laboratoire EA 4652 microenvironnement cellulaire et pathologie, université de Caen-Basse-Normandie, avenue de la Côte-de-Nacre, 14032 Caen cedex 5, France....
[Ti] Title:Prise en charge de l'enfant en mission humanitaire maxillofaciale. [Child care management in maxillofacial humanitarian mission].
[So] Source:Rev Stomatol Chir Maxillofac Chir Orale;116(4):250-60, 2015 Sep.
[Is] ISSN:2213-6541
[Cp] Country of publication:France
[La] Language:fre
[Ab] Abstract:Our practice in a humanitarian (or crisis) context differs from what we experience in daily practice. There are several reasons for this. First, the diseases encountered are sometimes unfamiliar, such as sequelae of noma, or the presentation of familiar diseases may be unusual, such as facial malformations seen at a late stage. Secondly, these missions take place in developing countries, and consequently, evaluation and anticipation of possible malnutrition should be considered, especially because facial diseases themselves may be responsible for nutritional problems. Lastly, conditions are often difficult, occurring in an unusual environment, and we sometimes have to face communication and equipment problems. The goal of our work, based on a 15-year experience (in Bamako and Mopti with the Association "Santé et Développement", and in Ouagadougou with the organization "Les enfants du noma") and the analysis of literature, is to point out these features and maybe to be helpful to others.
[Mh] MeSH terms primary: Altruism
Child Care/methods
Oral Surgical Procedures
[Mh] MeSH terms secundary: Adolescent
Ankylosis/epidemiology
Ankylosis/surgery
Arteriovenous Malformations/epidemiology
Arteriovenous Malformations/surgery
Bone Neoplasms/epidemiology
Bone Neoplasms/surgery
Burns/complications
Burns/epidemiology
Burns/surgery
Child
Child, Preschool
Cleft Lip/epidemiology
Cleft Lip/surgery
Cleft Palate/epidemiology
Cleft Palate/surgery
Female
Fibroma, Ossifying/epidemiology
Fibroma, Ossifying/surgery
Humans
Infant
Jaw Neoplasms/epidemiology
Jaw Neoplasms/surgery
Lip Diseases/congenital
Lip Diseases/epidemiology
Lip Diseases/surgery
Male
Mali/epidemiology
Maxillary Artery/abnormalities
Maxillary Artery/surgery
Noma/complications
Noma/epidemiology
Noma/pathology
Noma/surgery
Oral Surgical Procedures/statistics & numerical data
Retrospective Studies
[Pt] Publication type:ENGLISH ABSTRACT; JOURNAL ARTICLE
[Em] Entry month:1606
[Js] Journal subset:D; IM
[Da] Date of entry for processing:150910
[St] Status:MEDLINE

  7 / 5399 MEDLINE  
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[PMID]: 27346122
[Au] Autor:Peng WJ; Zhang H; Li YX; Li CY; Yan WR
[Ad] Address:Department of Epidemiology and Statistics, School of Public Health, Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430030, China.
[Ti] Title:[Log-linear model used in the hybrid design of case-parents triad/control-mother dyad].
[So] Source:Zhonghua Liu Xing Bing Xue Za Zhi;37(6):886-90, 2016 Jun 10.
[Is] ISSN:0254-6450
[Cp] Country of publication:China
[La] Language:chi
[Ab] Abstract:This study introduced the application of a log-linear model in the hybrid design of case-parents triad/control-mother dyad. Data related to the association between cleft lip with palate (CLP) and methylenetetrahydrofolate reductase (MTHFR) gene A1298C diversity was analyzed. Log-linear model based on likelihood ratio tests (LRTs) was used to analyze the relationships between mother, offspring genotypes and CLP. Data from our study noticed that children of mothers carrying the CC genotype presented a lower risk of CLP, comparing with the children of mothers carrying the AA genotype, with S2=0.45 (95%CI: 0.26-0.79). Offspring that carrying the AC genotype presented a lower risk of CLP, comparing with the offspring that carrying the AA genotype, with R1=0.69 (95% CI: 0.48-0.97). However, no other types of relationships were found. The power of hybrid design was greater than the case-parents study (0.86>0.78). MTHFR A1298C polymorphism seemed to have played an important role in the etiology on both cleft lip and palate. Data from the hybrid design and the log-linear model could help researchers to explore the effects of genotypes from both mothers and the offspring. This study design would present stronger power than the regular case-parents studies thus suitable for studies on the etiology of diseases in early lives, as birth defects.
[Pt] Publication type:ENGLISH ABSTRACT; JOURNAL ARTICLE
[Em] Entry month:1606
[Js] Journal subset:IM
[St] Status:In-Data-Review
[do] DOI:10.3760/cma.j.issn.0254-6450.2016.06.029

  8 / 5399 MEDLINE  
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[PMID]: 25896166
[Au] Autor:van Zyl-Smit RN; Naidoo J; Wainwright H; Said-Hartley Q; Davids M; Goodman H; Rogers S; Dheda K
[Ad] Address:Division of Pulmonology & UCT Lung Institute, Department of Medicine, Lung Infection and Immunity Unit, University of Cape Town, Cape Town, South Africa. richard.vanzyl-smit@uct.ac.za....
[Ti] Title:HIV associated Lymphocytic Interstitial Pneumonia: a clinical, histological and radiographic study from an HIV endemic resource-poor setting.
[So] Source:BMC Pulm Med;15:38, 2015.
[Is] ISSN:1471-2466
[Cp] Country of publication:England
[La] Language:eng
[Ab] Abstract:BACKGROUND: There is a paucity of clinical and histopathological data about HIV-associated lymphocytic interstitial pneumonitis (LIP) in adults from HIV endemic settings. The role of Ebstein-Barr virus (EBV) in the pathogenesis remains unclear. METHODS: We reviewed the clinical, radiographic and histopathological features of suspected adult LIP cases at the Groote Schuur Hospital, Cape Town South Africa, over a 6 year period. Archived tissue sections were stained for CD3, CD4, CD8, CD20 and LMP-1 antigen (an EBV marker). RESULTS: 42 cases of suspected LIP(100% HIV-infected) were identified. 75% of patients were empirically treated for TB prior to being referred to the chest service for further investigation. Tissue samples were obtained using trans-bronchial biopsy. 13/42 were classified as definite LIP (lymphocytic infiltrate with no alternative diagnosis), 19/42 probable LIP (lymphocytic infiltrate but evidence of anthracosis or fibrosis) and 10 as non-LIP (alternative histological diagnosis). Those with definite LIP were predominantly young females (85%) with a median CD4 count of 194 (IQR 119-359). Clinical or radiological features had poor predictive value for LIP. Histologically, the lymphocytic infiltrate comprised mainly B cells and CD8 T cells. The frequency of positive EBV LMP-1 antigen staining was similar in definite and non- LIP patients [(2/13 (15%) vs. 3/10 (30%); p = 0.52]. CONCLUSIONS: In a HIV endemic setting adult HIV-associated LIP occurs predominantly in young women. The diagnosis can often be made on transbronchial biopsy and is characterized by a predominant CD8 T cell infiltrate. No association with EBV antigen was found.
[Mh] MeSH terms primary: CD8-Positive T-Lymphocytes/immunology
Endemic Diseases
HIV Infections/epidemiology
Lung Diseases, Interstitial/immunology
[Mh] MeSH terms secundary: Adult
Antigens, CD20/immunology
Antigens, CD3/immunology
Antigens, CD4/immunology
Antigens, CD8/immunology
Female
HIV Infections/complications
Health Resources
Humans
Lung Diseases, Interstitial/etiology
Lung Diseases, Interstitial/physiopathology
Male
Middle Aged
South Africa/epidemiology
Viral Matrix Proteins/metabolism
Young Adult
[Pt] Publication type:JOURNAL ARTICLE
[Nm] Name of substance:0 (Antigens, CD20); 0 (Antigens, CD3); 0 (Antigens, CD4); 0 (Antigens, CD8); 0 (EBV-associated membrane antigen, Epstein-Barr virus); 0 (Viral Matrix Proteins)
[Em] Entry month:1606
[Cu] Class update date: 150512
[Lr] Last revision date:150512
[Js] Journal subset:IM
[Da] Date of entry for processing:150512
[St] Status:MEDLINE
[do] DOI:10.1186/s12890-015-0030-2

  9 / 5399 MEDLINE  
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[PMID]: 27236852
[Au] Autor:Ramos Macías Á; Borkoski-Barreiro SA; Falcón González JC; Ramos de Miguel Á
[Ad] Address:Hearing Loss Unit, Otorhinolaryngology, Head and Neck Department, Complejo Hospitalario Universitario Insular Materno Infantil, Av. Marítima del Sur s/n, Las Palmas of Gran Canaria 35016, Spain. Electronic address: aramos.gcc@gmail.com....
[Ti] Title:AHL, SSD and bimodal CI results in children.
[So] Source:Eur Ann Otorhinolaryngol Head Neck Dis;133 Suppl 1:S15-20, 2016 Jun.
[Is] ISSN:1879-730X
[Cp] Country of publication:France
[La] Language:eng
[Ab] Abstract:OBJECTIVE: This article aims to demonstrate that children with asymmetric hearing loss (AHL), specifically those with single side deafness (SSD condition) and pure SSD, with substantial hearing (>70% normal speech discrimination) in the ear with hearing aid (HA), obtain clinically relevant speech perception benefit after cochlear implantation in the contralateral side. RESULTS: Retrospective study of two groups: (1) 36 AHL children with bimodal hearing and (2) SSD children with SSD. They had follow-ups of 24 and 12 months duration, respectively. The AHL children where implanted on one ear and depending on the contralateral ear condition, they were divided into two groups: children who reach a disyllabic speech test score lower than 50% in quiet at 65dB-SPL without lip reading (27) and children with test score higher than 70% (9). The second group consisted on 2 children implanted to obtain SSD condition, as preliminary data, with 1 year of follow-up. They showed benefits of binaural stimulation. CONCLUSION: Children in a bimodal situation, with substantial hearing (>70% speech discrimination) in the ear with HA, obtain clinically relevant speech perception benefit after cochlear implantation in the contralateral side.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1606
[Js] Journal subset:IM
[St] Status:In-Data-Review

  10 / 5399 MEDLINE  
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[PMID]: 26859621
[Au] Autor:Jones K; Pitceathly RD; Rose MR; McGowan S; Hill M; Badrising UA; Hughes T
[Ad] Address:Department of Neurology, King's College Hospital NHS Foundation Trust, 9th floor Ruskin Wing, Denmark Hill, London, UK, SE5 9RS.
[Ti] Title:Interventions for dysphagia in long-term, progressive muscle disease.
[So] Source:Cochrane Database Syst Rev;2:CD004303, 2016.
[Is] ISSN:1469-493X
[Cp] Country of publication:England
[La] Language:eng
[Ab] Abstract:BACKGROUND: Normal swallowing function is divided into oral, pharyngeal, and oesophageal phases. The anatomy and physiology of the oral cavity facilitates an oral preparatory phase of swallowing, in which food and liquid are pushed towards the pharynx by the tongue. During pharyngeal and oesophageal phases of swallowing, food and liquid are moved from the pharynx to the stomach via the oesophagus. Our understanding of swallowing function in health and disease has informed our understanding of how muscle weakness can disrupt swallowing in people with muscle disease. As a common complication of long-term, progressive muscle disease, there is a clear need to evaluate the current interventions for managing swallowing difficulties (dysphagia). This is an update of a review first published in 2004. OBJECTIVES: To assess the effects of interventions for dysphagia in people with long-term, progressive muscle disease. SEARCH METHODS: On 11 January 2016, we searched the Cochrane Neuromuscular Specialised Register, the Cochrane Central Register of Controlled Trials (CENTRAL), MEDLINE, EMBASE, AMED, LILACS, and CINAHL. We checked references in the identified trials for additional randomised and quasi-randomised controlled trials. We also searched ClinicalTrials.gov and the World Health Organization International Clinical Trials Registry Platform on 12 January 2016 for ongoing or completed but unpublished clinical trials. SELECTION CRITERIA: We included randomised and quasi-randomised controlled trials that assessed the effect of interventions for managing dysphagia in adults and children with long-term, progressive muscle disease, compared to other interventions, placebo, no intervention, or standard care. Quasi-randomised controlled trials are trials that used a quasi-random method of allocation, such as date of birth, alternation, or case record number. Review authors previously excluded trials involving people with muscle conditions of a known inflammatory or toxic aetiology. In this review update, we decided to include trials of people with sporadic inclusion body myositis (IBM) on the basis that it presents as a long-term, progressive muscle disease with uncertain degenerative and inflammatory aetiology and is typically refractory to treatment. DATA COLLECTION AND ANALYSIS: We applied standard Cochrane methodological procedures. MAIN RESULTS: There were no randomised controlled trials (RCTs) that reported results in terms of the review's primary outcome of interest, weight gain or maintenance. However, we identified one RCT that assessed the effect of intravenous immunoglobulin on swallowing function in people with IBM. The trial authors did not specify the number of study participants who had dysphagia. There was also incomplete reporting of findings from videofluoroscopic investigations, which was one of the review's secondary outcome measures. The study did report reductions in the time taken to swallow, as measured using ultrasound. No serious adverse events occurred during the study, although data for the follow-up period were lacking. It was also unclear whether the non-serious adverse events reported occurred in the treatment group or the placebo group. We assessed this study as having a high risk of bias and uncertain confidence intervals for the review outcomes, which limited the overall quality of the evidence. Using GRADE criteria, we downgraded the quality of the evidence from this RCT to 'low' for efficacy in treating dysphagia, due to limitations in study design and implementation, and indirectness in terms of the population and outcome measures. Similarly, we assessed the quality of the evidence for adverse events as 'low'. From our search for RCTs, we identified two other non-randomised studies, which reported the effects of long-term intravenous immunoglobulin therapy in adults with IBM and lip-strengthening exercises in children with myotonic dystrophy type 1. Headaches affected two participants treated with long-term intravenous immunoglobulin therapy, who received a tailored dose reduction; there were no adverse events associated with lip-strengthening exercises. Both non-randomised studies identified improved outcomes for some participants following the intervention, but neither study specified the number of participants with dysphagia or demonstrated any group-level treatment effect for swallowing function using the outcomes prespecified in this review. AUTHORS' CONCLUSIONS: There is insufficient and low-quality RCT evidence to determine the effect of interventions for dysphagia in long-term, progressive muscle disease. Clinically relevant effects of intravenous immunoglobulin for dysphagia in inclusion body myositis can neither be confirmed or excluded using the evidence presented in this review. Standardised, validated, and reliable outcome measures are needed to assess dysphagia and any possible treatment effect. Clinically meaningful outcomes for dysphagia may require a shift in focus from measures of impairment to disability associated with oral feeding difficulties.
[Mh] MeSH terms primary: Deglutition Disorders/drug therapy
Immunoglobulins, Intravenous/therapeutic use
Immunologic Factors/therapeutic use
Muscular Diseases/complications
Muscular Diseases/drug therapy
[Mh] MeSH terms secundary: Adult
Child
Chronic Disease
Deglutition
Deglutition Disorders/etiology
Humans
Myositis, Inclusion Body/complications
Myositis, Inclusion Body/drug therapy
Randomized Controlled Trials as Topic
[Pt] Publication type:JOURNAL ARTICLE; RESEARCH SUPPORT, NON-U.S. GOV'T; REVIEW
[Nm] Name of substance:0 (Immunoglobulins, Intravenous); 0 (Immunologic Factors)
[Em] Entry month:1606
[Js] Journal subset:IM
[Da] Date of entry for processing:160301
[St] Status:MEDLINE
[do] DOI:10.1002/14651858.CD004303.pub4


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