Endoplasmic reticulum stress in autoimmune diseases: Can altered protein quality control and/or unfolded protein response contribute to autoimmunity? A critical review on Sjögren's syndrome

Publication date: Available online 8 June 2018
Source:Autoimmunity Reviews
Author(s): María-José Barrera, Sergio Aguilera, Isabel Castro, Sergio González, Patricia Carvajal, Claudio Molina, Marcela A. Hermoso, María-Julieta González
For many years, researchers in the field of autoimmunity have focused on the role of the immune components in the etiopathogenesis of autoimmune diseases. However, some studies have demonstrated the importance of target tissues in their pathogenesis and the breach of immune tolerance. The immune system as well as target tissue cells (plasmatic, β-pancreatic, fibroblast-like synoviocytes, thyroid follicular and epithelial cells of the lachrymal glands, salivary glands, intestine, bronchioles and renal tubules) share the characteristic of secretory cells with an extended endoplasmic reticulum (ER). The function of these cells depends considerably on a normal ER function and calcium homeostasis, so they can produce and secrete their main components, which include glycoproteins involved in antigenic presentation such as major histocompatibility complex (MHC) class I and II. All these proteins are synthesized and modified in the ER, and for this reason disturbances in the normal functions of this organelle such as protein folding, protein quality control, calcium homeostasis and redox balance, promote accumulation of unfolded or misfolded proteins, a condition known as ER stress. Autoimmune diseases are characterized by inflammation, which has been associated with an ER stress condition. Interestingly, patients with these diseases contain circulating auto-antibodies against chaperone proteins (such as Calnexin and GRP94), thus affecting the folding and assembly of MHC class I and II glycoproteins and their loading with peptide.The main purpose of this article is to review the involvement of the protein quality control and unfolded protein response (UPR) in the ER protein homeostasis (proteostasis) and their alterations in autoimmune diseases. In addition, we describe the interaction between ER stress and inflammation and evidences are shown of how autoimmune diseases are associated with an ER stress condition, with a special emphasis on the second most prevalent autoimmune rheumatic disease, Sjögren's syndrome.



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Autosomic dominant familial Behçet disease and haploinsufficiency A20: A review of the literature

Publication date: Available online 8 June 2018
Source:Autoimmunity Reviews
Author(s): Berteau Florian, Rouviere Bénédicte, Delluc Aurélien, Nau Alice, Le Berre Rozenn, Sarrabay Guillaume, Touitou Isabelle, Claire de Moreuil
IntroductionBehçet disease (BD) is a systemic vasculitis involving vessels from any size with various clinical features. Most BD cases are multifactorial and associated with the HLA B51 antigen. In rare and severe early onset cases, dominant Mendelian transmission has been linked to mutations in the TNFAIP3 gene encoding A20. Herein, we propose a systematic review of the literature about the haploinsufficiency A20 (HA20) published cases.Systematic reviewOur review of the 45 cases of HA20 from literature highlights the similarities and the differences between this genetic auto-inflammatory disease and classical BD. HA20 looks like BD if we consider recurrent oral (87%) and genital (67%) ulcers, arthralgia or arthritis (42%), skin involvement (53%) such as erythema nodosum or abdominal symptoms (60%) such as abdominal pain, digestive ulcers or diarrhea. However, HA20 differs from classical BD because its geographical distribution is ubiquitous, sex ratio is inversed (one man for two women), first symptoms occur in early childhood (median age = 5.5 years; interquartile range: 1–10) instead of adulthood, recurrent fever is common (62%) unlike classical BD, HLA B51 antigen is uncommon and abdominal symptoms are over-represented compared to classical BD. In addition, response to colchicine in HA20 is inconstant (24%) unlike classical BD.Discussion/conclusionHigh fever flares and digestive involvement starting in early childhood seem to be hallmarks of HA20 clinical features. Response to colchicine is unpredictable and biotherapies like anti-TNFα and anti IL1 appear to be treatments of choice, like for other auto-inflammatory diseases. Prospective description of larger cohort of HA20 cases is needed to understand better when this disease must be looked for and how to treat these patients.



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Re: “Seasonal Changes in Serum Thyrotropin Concentrations Observed from Big Data Obtained During Six Consecutive Years from 2010 to 2015 at a Single Hospital in Japan” by Yoshihara et al. (Thyroid 2018;28:429–436)

Thyroid, Ahead of Print.


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Editorial Board

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Author Index

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What is blocking early peanut introduction?

Peanut allergy affects up to 2% to 4% of US children,1,2 and prevalence appears to be increasing.3 Most (approximately 75–80%) retain peanut allergy and risk for anaphylaxis into adulthood.4 Recognizing the burden of peanut allergy, professional societies advised dietary interventions in infants and young children to lower risk of developing peanut allergy. Based on a changing body of evidence during the past 2 decades, guidelines have shifted from delayed introduction of allergenic foods to early and sustained ingestion of peanut.

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Information for Authors

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Atopic dermatitis

In the current issue of the Annals, Bridgman et al1 provide an overview on the multidimensional burden of atopic dermatitis (AD). It is clear from this perspective that patients with AD are affected in multiple ways, leading to impaired quality of life and social functioning, low self-esteem, more sick days, reduced physical activity, and academic and work impairment. These effects lead to disruption not only of the patients' lives but that of their families, with increased feelings of helplessness, stress, and low general life satisfaction.

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Information for Readers

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Table of Contents

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The many faces of eczema and its treatments

Atopic dermatitis (AD), contact dermatitis, and irritant dermatitis share many similarities, and are often tied together in a Gordian knot of etiopathogenesis and therapeutic considerations. The pruritus, eczematous morphology, and waxing-waning presentations can be clinically indistinguishable, and as more of the scientific underpinnings are elucidated, connections in their complex pathophysiology continue to be found.1,2 Differentiating between these entities, especially when more than 1 may be present simultaneously, can be a daunting task.

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Paul Klenerman's The Immune System, A Very Short Introduction, is aptly titled and yet quite useful....

Paul Klenerman's The Immune System, A Very Short Introduction, is aptly titled and yet quite useful. Klenerman skillfully explains and summarizes an intricate and complex system into a short read that can certainly be completed in an afternoon. This brief book presents a helicopter view of the immune system, or perhaps more accurately a seagull's view, as it occasionally dives down deeper to take a closer look between arching overviews. A broad range of readers will be able to glean some utility from the book, because it offers important concepts in a fun, informative, and concise read.

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Galectin-7 promotes proliferation and Th1/2 cells polarization toward Th1 in activated CD4+ T cells by inhibiting The TGFβ/Smad3 pathway

Publication date: September 2018
Source:Molecular Immunology, Volume 101
Author(s): Zhenlong Luo, Yudong Ji, Dean Tian, Yong Zhang, Sheng Chang, Chao Yang, Hongmin Zhou, Zhonghua Klaus Chen
Galectin-7 (Gal-7) has been associated with cell proliferation and apoptosis. It is known that Gal-7 antagonises TGFβ-mediated effects in hepatocytes by interacting with Smad3. Previously, we have demonstrated that Gal-7 is related to CD4+ T cells responses; nevertheless, its effect and functional mechanism on CD4+ T cells responses remain unclear. The murine CD4+ T cells were respectively cultured with Gal-7, anti-CD3/CD28 mAbs, or with anti-CD3/CD28 mAbs & Gal-7. The effects of Gal-7 on proliferation and the phenotypic changes in CD4+ T cells were assessed by flow cytometry. The cytokines from CD4+ T cells were analysed by quantitative real-time PCR. Subcellular localisation and expression of Smad3 were determined by immunofluorescence staining and Western blot, respectively. Gal-7 enhanced the proliferation of activated CD4+ T cells in a dose- and β-galactoside-dependent manner. Additionally, Gal-7 treatment did not change the ratio of Th2 cells in activated CD4+ T cells, while it increased the ratio of Th1 cells. Gal-7 also induced activated CD4+ T cells to produce a higher level of IFN-γ and TNF-α and a lower level of IL-10. Moreover, Gal-7 treatment significantly accelerated nuclear export of Smad3 in activated CD4+ T cells. These results revealed a novel role of Gal-7 in promoting proliferation and Th1/2 cells polarization toward Th1 in activated CD4+ T cells by inhibiting the TGFβ/Smad3 pathway.



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Information for Readers

Publication date: June 2018
Source:Annals of Allergy, Asthma & Immunology, Volume 120, Issue 6





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Table of Contents

Publication date: June 2018
Source:Annals of Allergy, Asthma & Immunology, Volume 120, Issue 6





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Information for Authors

Publication date: June 2018
Source:Annals of Allergy, Asthma & Immunology, Volume 120, Issue 6





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Clinical phenotypes of bronchial hyperresponsiveness in school-aged children

Publication date: Available online 8 June 2018
Source:Annals of Allergy, Asthma & Immunology
Author(s): Eun Lee, Young-Ho Kim, Hyun-Ju Cho, Ji-Sun Yoon, Sungsu Jung, Song-I Yang, Hyung Young Kim, Ji-Won Kwon, Ju-Hee Seo, Hyo Bin Kim, So Yeon Lee, Soo-Jong Hong
BackgroundBronchial hyperresponsiveness (BHR), one of the key features of asthma, shows a diverse natural course in school-aged children, but, studies on BHR phenotypes are lacking.ObjectiveWe classified BHR phenotypes according to onset age and persistence in children and investigated the characteristics and factors associated with each phenotype in a longitudinal study.MethodsWe analyzed 1,305 elementary school children from the Children's HEalth and Environmental Research (CHEER) study, a 4-year prospective follow-up study with 2-year intervals starting at a mean age of 7 years. Total serum IgE levels and blood eosinophils (%) were measured, and allergy work-up including methacholine challenge tests with ISSAC questionnaire were performed at each survey.ResultsWe classified the four BHR phenotypes as non-BHR (n=942, 72.2%), early-onset transient BHR (n=201, 15.4%), late-onset BHR (n=87, 6.7%), and early-onset persistent BHR (n=75, 5.7%). Early-onset persistent BHR is characterized by an increased eosinophils (%), total serum IgE level, sensitization rate, a decreased lung function and an increased risk of newly diagnosed asthma during follow-up (aOR, 3.89; 95% CI, 1.70-8.88). The two early-onset phenotypes were associated with peripheral airway dysfunction. The late-onset BHR phenotype related with increased risks of AR symptoms at baseline and later sensitization against inhalant allergens.ConclusionThe early-onset persistent BHR phenotype in school-aged children is associated with high atopic burden and increased risk of newly diagnosed asthma, whereas the late-onset BHR phenotype related with later sensitization and AR symptoms. Diverse BHR phenotypes in children have specific characteristics that require targeted follow-ups.



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Editorial Board

Publication date: June 2018
Source:Annals of Allergy, Asthma & Immunology, Volume 120, Issue 6





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The many faces of eczema and its treatments

Publication date: June 2018
Source:Annals of Allergy, Asthma & Immunology, Volume 120, Issue 6
Author(s): Peter A. Lio




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Author Index

Publication date: June 2018
Source:Annals of Allergy, Asthma & Immunology, Volume 120, Issue 6





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Atopic dermatitis

Publication date: June 2018
Source:Annals of Allergy, Asthma & Immunology, Volume 120, Issue 6
Author(s): Donald Y.M. Leung




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What is blocking early peanut introduction?

Publication date: June 2018
Source:Annals of Allergy, Asthma & Immunology, Volume 120, Issue 6
Author(s): Elizabeth Feuille, Anna Nowak-Węgrzyn




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Paul Klenerman. The Immune System, A Very Short Introduction. United Kingdom: Oxford University Press, 2018. Soft copy, 144 pages, $11.95. ISBN: 019875390X; 978-0198753902.

Publication date: June 2018
Source:Annals of Allergy, Asthma & Immunology, Volume 120, Issue 6
Author(s): Stephen LeBlanc




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Crosstalk between type I and II interferons in regulation of myeloid cell responses during bacterial infection

William J Crisler | Laurel L Lenz

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Fibrotomy with diode laser (980 nm) and habit correlation in oral submucous fibrosis: a report of 30 cases

Abstract

To evaluate the efficacy of laser fibrotomy in patients with oral submucous fibrosis (OSMF) and also to correlate the habit variables causing OSMF. Thirty patients diagnosed clinically and histopathologically with OSMF were included in the study. Laser fibrotomy was done under local anesthesia using Zolar diode laser (980 nm frequency) at 2-W power in contact mode with fibro-optic cutting tip. The patients were advised to practice mouth-opening exercises rigorously, and topical corticosteroid was given for burning sensation and pain for 6 months. Pre- and post-treatment comparison of mouth opening, burning sensation, tongue protrusion, and cheek flexibility was analyzed after a follow-up period of 9 months. After a follow-up period of 9 months from the surgical intervention, the mean value of difference in pre- and post-treatment of VAS score, mouth opening, and tongue protrusion was found to be 3.3 ± 2.1, 7.7 ± 5.1, 1.6 ± 2.7, and 0.38 ± 0.61 respectively. Using the Wilcoxon signed rank test, all the above difference in mean was found to be significant with p value < 0.05. With above suggested significant results, lasers can provide an alternative and better means for surgical fibrotomy, relieving trismus in moderate OSMF as they are minimally invasive, cause less hemorrhage, and have faster healing and minimal surgical site scaring and relapse.

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Validity of Chronic Oral Mucosal Diseases Questionnaire in oral submucous fibrosis

Abstract

Objective

To validate the use of Chronic Oral Mucosal Diseases Questionnaire in oral submucous fibrosis.

Materials and methods

Hundred consecutive oral submucous fibrosis (OSF) patients were requested to respond to Visual Analogue Scale (VAS) score and complete the Oral Health Impact Profile 14 (OHIP 14) and COMDQ. Spearman's rho correlation was used to assess the relationship between VAS and COMDQ score and COMDQ and OHIP 14 score. The reliability of the instrument was assessed using Cronbach's alpha analysis. Discriminant analysis was used to assess if COMDQ significantly differentiated between OSF population with and without any prior treatment done as well as OSF with mouth opening less than and more than or equal to 25 mm.

Results

The Spearman's correlation coefficient showed a high significance between the different domains of COMDQ and OHIP 14 and COMDQ and VAS. This study demonstrated a good convergent validity between the overall COMDQ and OHIP 14 scores (0.88).The overall Cronbach's alpha value was found to be 0.75 indicating an acceptable internal consistency reliability of COMDQ. The tool was unable to differentiate between patients with mouth opening between less than and more than or equal to 25 mm and patients with and without prior treatment.

Conclusion

These results provide evidence that COMDQ can be used for OSF patients.

Clinical relevance

COMDQ could be used as a valid and reliable tool for further clinical OSF research.

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Minimal Clinically Important Differences for Measures of Treatment Efficacy in Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis

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Platelet rich plasma with microneedling and trichloroacetic acid peel for treatment of striae distensae

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Biofunctionalization of synthetic bone substitutes with angiogenic stem cells: Influence on regeneration of critical-size bone defects in an in vivo murine model

The aim of this study was to investigate the influence of human bone marrow−derived endothelial progenitor cells (EPC) on vascularization and bone regeneration in combination with a bone-substitute material (BSM) in a critical-size bone defect in a murine model. Critical-size bone defects were performed and the defects were filled according to the group membership.

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Dental consequences of pterygomaxillary dysjunction during fronto-facial monobloc advancement in children with internal distraction for Crouzon syndrome

Crouzon syndrome is a syndromic faciocraniosynostosis that can be associated with severe fronto-facial retrusion leading to major functional impairments: extreme exorbitism may be vision-threatening and severe respiration impairment can be life-threatening. The procedure of choice for the primary correction of this retrusion is fronto-facial monobloc advancement (FFMBA) with internal or external distraction. FFMBA involves pterygomaxillary dysjunction (PMD), using either a superior or an intra-oral approach.

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Bilateral dermoid ovarian torsion in a young woman: a case report

Of all patients undergoing emergency surgery for acute pelvic pain, approximately 2.7% of cases are caused by ovarian torsion. We report a rare occurrence of bilateral ovarian torsion in a young woman.

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Voice Changes in the Elderly

The impact of aging is as inevitable in the larynx as on all biologic systems. The muscles of larynx have the potential to atrophy, the elastin fibers of lamina propria thin with age, and mucous production diminishes. As a result, vocal folds fail to approximate appropriately and the stress on once-robust vocal folds increases. These changes present as poor voice quality, vocal tension, tremor, and altered fundamental frequency. Rather than consider presbyphonia as an immutable diagnosis, we must see it as an opportunity to elevate our standard of care and set goals to work for therapeutic improvement of voice quality.

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Rhinoplasty for South East Asian Nose

The South East Asian nose is usually small with voluminous thick skin, low dorsum, wide and hanging ala, bulbous tip, and retracted premaxilla. The surgical approach of rhinoplasty for these types of noses includes the following: dorsal augmentation, counterrotation and projection of the tip, and correction of hanging ala and alar flare/base. A usual ideal rhinoplasty outcome of a South East Asian nose should be a nose that fits the face with good function and has all the aesthetic landmarks achieved: natural looking dorsum, supratip break, tip, subtip break, columellar show, good alar-columellar relationship, improved premaxilla, improved nostril, and improved alar flare.

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Updating nasalisation: From concept to technique and results

Publication date: Available online 7 June 2018
Source:European Annals of Otorhinolaryngology, Head and Neck Diseases
Author(s): R. Jankowski, C. Rumeau, D.T. Nguyen, P. Gallet
Nasalisation is a surgical technique proposed for the treatment of nasal polyposis in 1995. The technique as initially described was based on large opening of the maxillary, sphenoidal and frontal ostia and resection of the middle turbinates, with the aim of resecting the mucosa of the lateral masses of the ethmoid as completely and safely as possible. Recent findings on the evolution and development of the nose and sinuses and sinus physiology allow both the concept and the technique of nasalisation to be updated. According to evo-devo theory, the ethmoid is not a paranasal sinus but the skull-base bone housing the olfactory mucosa. In humans, the olfactory mucosa can be distinguished as functional in the upper recess of both olfactory clefts and vestigial in the rest of the ethmoid bone. Nasal polyposis presents clinically as a specific disease of the vestigial olfactory mucosa of the human ethmoid (and not as a particular kind of chronic rhinosinusitis). The aim of surgery for nasal polyposis is thus maximal resection of the vestigial olfactory mucosa, conserving olfactory function as long as possible. By the production, storage and bolus release of nitric oxide thanks to the sphincter function of the paranasal (maxillary, sphenoidal and frontal) sinus ostia, the paranasal sinuses perform a respiratory function that should be conserved as much as possible. The nasalisation technique has been modified in order for the vestigial olfactory mucosa to be completely resected while sparing the sinus ostia. Middle turbinate surgery still needs to be evaluated: resection is a step toward maximal removal of vestigial olfactory mucosa, while conservation could ensure olfactory cleft integrity. Thus, updating the concept of nasalisation enables the nasalisation technique to be integrated in the evo-devo concept of rhinology.



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Nasal septal abscess caused by anaerobic bacteria of oral flora

Publication date: Available online 7 June 2018
Source:Auris Nasus Larynx
Author(s): Yukiyoshi Hyo, Hisaki Fukushima, Tamotsu Harada, Hirotaka Hara
Although nasal septal abscess (NSA) was formerly common, it has become rare since the development of antibiotics. NSA, if left untreated, can lead to intracranial complications such as meningitis and eventually result in saddle-nose deformity. NSA often occurs after injury, and indigenous skin bacteria such as Staphylococcus aureus are frequently detected. We treated a patient who had injured the upper alveolus in a fall on the stairs and developed NSA two weeks later. Anaerobic bacteria, including Veillonella parvula and Peptostreptococcus sp., were detected. Symptoms were relieved by needle and incisional drainage. Our patient represents a very rare case of NSA in terms of the cause of onset and the detected bacteria. Early drainage can result in good outcomes.



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Use of a folded extended vertical lower trapezius island myocutaneous flap to repair large pharyngocutaneous fistulae developing after salvage total laryngectomy

Publication date: Available online 8 June 2018
Source:International Journal of Oral and Maxillofacial Surgery
Author(s): Z.-q. Huang, B. Zhou, W.-l. Chen, J.-l. Zhong, Y. Wang
The purpose of this study was to evaluate the outcomes of surgery involving the use of folded trapezius flaps to repair large pharyngocutaneous fistulae (PCFs) developing after salvage total laryngectomy. Folded extended vertical lower trapezius island myocutaneous flaps (TIMFs) were created to repair large PCFs that developed after salvage total laryngectomy in eight patients. The maximum fistula dimension was 4.5×3.0cm and the minimum was 2.0×1.8cm. The skin paddle of the extended vertical TIMF ranged from 5cm to 9cm in width and 10cm to 23cm in length. The inner lining ranged from 3cm×3cm to 6cm×6cm in dimension and the outer lining from 6cm×5cm to 16cm×9cm. The folded flaps provided both inner mucosa and outer lining. All flaps survived. No fistula recurrence or stricture developed. After 6–24months of follow-up, six patients showed no evidence of disease, one was alive with disease, and one had died of local recurrence at 20months. The folded flap was very reliable and is well-suited for repairing large PCFs, even early large PCFs and those featuring carotid artery rupture.



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Change in the posterior airway after mandibular distraction osteogenesis in patients with ankylosis of the temporomandibular joint: a retrospective study

Publication date: Available online 7 June 2018
Source:British Journal of Oral and Maxillofacial Surgery
Author(s): R.Y. Bi, X.T. Luo, N. Jiang, S.S. Zhu, Y.F. Li
Mandibular distraction osteogenesis (DO) has been shown to lead to considerable improvement in obstruction of the posterior airway space in patients with ankylosis of the temporomandibular joint (TMJ), and our objective was to find out if we could confirm these findings. Seventeen patients had spiral computed tomographic (CT) scans before and after DO. After treatment, the overall posterior airway space was enlarged in all three sections of the airway (oropharyngeal, glossopharyngeal, and laryngeal). We then compared rates of change in the airway among the sections using 2-dimensional and 3-dimensional assessments, and found that the rate of change in 3-dimensional assessment of volume was significantly higher than that in the 2-dimensional (62% compared with 34%). We also found that the higher 3-dimensional rate of change came from changes in the oropharyngeal and glossopharyngeal sections, while there was no significant difference between the 2- and 3-dimensional rates of change in the laryngeal section. Because the laryngeal section had the most robust enlargement after DO in both the overall area of the posterior airway space (increased by 54%) and volume (increased by 73%), we concluded that 3-dimensional assessments were more sensitive to smaller changes in the airway space during the operation. This suggests that 3-dimensional assessments are preferable in the prediction and evaluation of the effects of DO on the posterior airway space.



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Long-term quality of life measured by the University of Washington QoL questionnaire (version 4) in patients with oral cancer treated with or without reconstruction with a microvascular free flap

Publication date: Available online 7 June 2018
Source:British Journal of Oral and Maxillofacial Surgery
Author(s): J. Yue, S. Zhuo, H. Zhang, X. Liu, W. Zhang
We used the University of Washington Quality of Life (UW-QoL) questionnaire (version 4) to assess the long-term quality of life (QoL) of patients with oral squamous cell carcinoma treated with or without reconstruction with a microvascular free flap, and all patients with T2–T4 oral squamous cell carcinoma (SCC) treated in this way were eligible for the study. A total of 139 patients' personal details, medical history, and QoL scores were collected and analysed. The mean (SD) overall QoL score was 73.09 (14.8) for patients with T2–T4 oral SCC. The mean (SD) global QoL scores of patients who had had reconstructions was 75.68 (13.85) and of patients who had not 71.00 (15.34) (t=1.864, df=137, p=0.064). Univariate and multivariate analyses indicated that site of tumour, T stage, and the need for postoperative radiotherapy had significant effects on the global QoL scores. Among patients with T2 oral SCC there was no significant difference between patients who did and did not have reconstructions. However, there were significant differences among patients with T3/T4 SCC in scores for appearance, recreation, mood, anxiety, chewing, swallowing, and speech depending on whether they did or did not have reconstructions. There were significant differences in the domains recreation, chewing, speech, and taste, depending on the primary site of the tumour. We conclude that reconstruction with a microvascular free flap had a beneficial effect on the treatment of SCC and improved the QoL of patients with oral SCC.



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An incredibly dilated Wirsung mimicking a sero-cystic neoplasm of the pancreatic head

Abstract

A diabetic patient who at a routine abdominal ultrasounds was found to have a very dilated pancreatic duct. Computed tomography (CT) scan diagnosed a sero-cystic lesion of the pancreatic head. Gastroduodenoscopy discovered a duodenal hyperemic area, which was sampled. Biopsy demonstrated intramucosal vascular emboli from a neuroendocrine carcinoma positive for Chromogranin A and Somatostatin and negative for Gastrin. Cholangio-magnetic resonance imaging revealed that the sero-cystic lesion found at CT, was being mimicked by the enormously dilated pancreatic duct but suggested the possibility of an intraductal or ampullar neoplasm. Blood and urine tests were not helpful and an octreoscan was negative. The patient underwent surgery. Direct exploration confirmed the severe pancreatic duct dilation and a cephalic lesion requiring a pancreatoduodenectomy. Histology confirmed a neuroendocrine tumor infiltrating the duodenum. We conclude that despite modern sophisticated imaging and endoscopic techniques, the evaluation of bilio-pancreatic region can be challenging and can reserve surgical surprises.

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Heterotopic ossification following use of i-Factor for spinal fusion in Mucopolysaccharidosis 1: a case report

Abstract

Mucopolysaccharidosis is a rare group of genetic disorder which results in a complex of anomalies involving various systems. In Mucopolysaccharidosis 1 progressive thoracolumbar kyphosis is a common presentation which can result in instability and neurological deficit. Posterior spinal surgery is performed to correct deformity and obtain spinal fusion. Peptide enhanced bone graft substitute (i-Factor^TM) is relatively a new component with proven efficacy to obtain early spinal fusion. An 8-year-old child with progressive high lumbar kyphosis due to Mucopolysaccharidosis 1 was admitted for Posterior spinal fusion with i-Factor bone graft substitute. Postoperatively patient had serous discharge from the wound which settled without intervention. A month after the surgery spinal radiographs revealed heterotopic ossification at the distal end of spinal construct in the paraspinal region. Patient remained asymptomatic and clinically well.

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Thinking outside the rectum: a unique approach to the retrieval of gluteal foreign bodies

Abstract

Penetrating gluteal wounds with retained foreign bodies are relatively uncommon and therefore there is no agreed-upon method of extraction. Retrieval of such objects can be difficult to achieve due to imperfect anatomy and a lack of clear planes across which the objects traverse, sometimes requiring novel techniques for foreign body retrieval. We saw a 37-year-old male with a 2-month history of a draining abscess on his right buttock. CT and manual probing of the wound demonstrated a 5-cm tract with a possible foreign body within. We took the patient to the operating room for exploration of the tract under general anesthesia, allowing for palpation of the foreign object as well as digital rectal exam, which identified an object passing posterior to the rectum. A cystoscope was used to widen the tract and allow for better visualization. Grasping forceps were inserted into the cystoscope and used to capture/retrieve the object.

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The evaluation of pretreatment neutrophil–lymphocyte ratio and derived neutrophil–lymphocyte ratio in patients with laryngeal neoplasms

Publication date: Available online 8 June 2018
Source:Brazilian Journal of Otorhinolaryngology
Author(s): Gorkem Eskiizmir, Uzdan Uz, Ece Onur, Beyhan Ozyurt, Gizem Karaca Cikrikci, Nevin Sahin, Arzu Oran, Onur Celik
IntroductionSystemic inflammatory biomarkers are promising predictive and prognostic factors for solid cancers. The neutrophil–lymphocyte ratio and derived neutrophil–lymphocyte ratio are used to predict inflammation and used as biomarker in several malignancies.ObjectiveThe purpose of this study was to demonstrate the diagnostic, predictive and prognostic role of neutrophil–lymphocyte ratio and derived neutrophil–lymphocyte ratio in patients with laryngeal neoplasms.MethodsA retrospective study was conducted on medical records involving 229 patients with benign, premalignant and malignant laryngeal neoplasms between 2002 and 2015. The diagnostic, predictive and prognostic role of neutrophil–lymphocyte ratio and derived neutrophil–lymphocyte ratio were evaluated using uni– and multivariate analysis.ResultsThe neutrophil–lymphocyte ratio and derived neutrophil–lymphocyte ratio were not statistically different between patients with benign, premalignant and malignant laryngeal neoplasms. Both neutrophil–lymphocyte ratio and derived neutrophil–lymphocyte ratio were predictive factors for stage, lymph node metastasis, and distant metastasis. Patients with high neutrophil–lymphocyte ratio value (≥4) had a poor prognosis when compared with patients with low neutrophil–lymphocyte ratio value (5 year, Overall Survival: 69.0% vs. 31.1%, p<0.001; 5 year, disease free survival: 70.0% vs. 32.7%, p˂0.001; 5 year, locoregional recurrence free survival: 69.7% vs. 32.0%, p<0.001). Furthermore, neutrophil–lymphocyte ratio was an independent prognostic factor for 5 year: Overall survival (HR=2.396; 95% CI 1.408–4.077; p=0.001), Disease free survival (HR=2.246; 95% CI 1.322–3.816; p=0.006) and locoregional recurrence free survival (HR=2.210; 95% CI 1.301–3.753; p=0.003).ConclusionPretreatment neutrophil–lymphocyte ratio is a useful and reliable predictive and prognostic biomarker for patients with laryngeal carcinoma.



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Subcutaneous emphysema to the head and neck: an unlikely traumatic origin

Subcutaneous emphysema to the neck represents a rare entity mainly derived from iatrogenic and traumatic origin. We report a case of a subcutaneous emphysema resulted from an intraoral injury aiming to emphasise the significance of precise medical history to identify an unlikely mechanism. A 40-year-old female patient was presented with subcutaneous emphysema extending from the region underneath the left eye up to the submandibular area of the neck. The patient complained of painful, swollen neck. The patient mentioned that the symptoms began after an intraoral injury accidentally caused by the use of a high-pressure water jet device meant for car wash. The patient was managed conservatively and was discharged after 2 days. This case highlights the urgency for clinical suspicion for unlikely mechanisms. Moreover, the manufacturer companies should alert and give official warning for the contingency of injury due to incorrect or careless usage.

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Terminal deoxynucleotidyl transferase (TdT)-negative T-cell lymphoblastic lymphoma with loss of the T-cell lineage-specific marker CD3 at relapse: a rare entity with an aggressive outcome

Terminal deoxynucleotidyl transferase (TdT)-negative T-cell lymphoblastic lymphoma is a variant of T-cell lymphoblastic lymphoma/T-cell lymphoblastic leukaemia. TdT is a marker of immaturity expressed in 90%–95% cases of lymphoblastic lymphoma and useful in differentiating it from other mature lymphomas/leukaemias. It has been associated with poorer response to chemotherapy and a more aggressive outcome. Here we present a case of TdT-negative T-cell lymphoblastic lymphoma in a 28-year-old man who presented with superior vena cava syndrome. The patient was treated with hyper-cyclophosphamide,vincristine, Adriamycin, dexamethasone (CVAD), however unfortunately suffered a relapse 1 year later. A unique feature of our case was that on relapse, the patient lost expression of the T-cell lineage-specific marker CD3, which has previously not been reported in association with TdT-negative T-cell lymphoblastic lymphoma. The patient failed to respond to chemotherapy on his relapse and died.

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Histiocytic necrotising lymphadenitis identical to Kikuchi-Fujimoto disease in CNS lupus

Description 

A 36-year-old woman presented after a first generalised tonic–clonic seizure. On clinical examination, prominent cervical and axillary lymphadenopathy was noted. Brain MRI showed diffuse, partially enhancing, hyperintense lesions (figure 1). An axillary lymph node biopsy revealed typical findings of histiocytic necrotising lymphadenitis, compatible with Kikuchi-Fujimoto disease (figure 2).1 Laboratory studies showed positive antinuclear antibodies, low complement levels and pancytopenia. In the cerebrospinal fluid (CSF), lymphocytic pleocytosis (34/μL), elevated lactate (3.3 mmol/L) and protein (104 mg/dL) levels were noted. Investigations for lymphoma were negative. Furthermore, negative serological and PCR findings for Epstein-Barr virus, cytomegalovirus and HIV were found. Interferon-gamma release assay and PCR for tuberculosis, along with serological investigations for toxoplasmosis and syphilis, were negative. The cytological examination of the CSF revealed activated lymphocytes and plasma cells. CSF and blood cultures remained negative. On diagnostic evidence of pericarditis and a positive history for photosensitivity and...

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Bilateral ocular paralysis in an ocular emergency

Description Case description

A 70-year-old female patient was brought to the ocular emergency department with the chief complaints of bilateral severe ptosis and total external ophthalmoplegia (L>R) for the past 1 week (figure 1). There was no history of any significant medical or surgical intervention. On evaluation, there was a 6 mm ptosis in the right eye and complete ptosis in the left eye along with an axial proptosis of 20 mm in the right eye and 21 mm in the left eye as measured using a Hertel exophthalmometer. Both eyes had complete restriction of extraocular movements in all gazes (figure 1) along with mildly dilated pupils with a sluggish reaction to light. Conjunctiva showed severe chemosis in both eyes (L>R). The anterior segment was slightly shallow on both sides with nuclear sclerosis of grade I to II. Intraocular pressures were 18 and 20 mm Hg in the right and...

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Novel psychoactive substances: overdose of 3-fluorophenmetrazine (3-FPM) and etizolam in a 33-year-old man

Though illegal in the UK, in many countries novel psychoactive substances are quasi-legal synthetic compounds that are widely available online under the guise of research chemicals. These substances are relatively cheap and are often undetectable in standard drug screens. Nearly 200 such compounds are introduced yearly, and little is usually known about their metabolism or physiological effects. Consequently, managing patients in overdose situations on largely unknown substances usually involves supportive care, however anticipating and managing atypical side effects are challenging in the absence of knowledge of these compounds. In this report, we discuss our encounter with a 33-year-old unconscious man presenting with coingestion of a novel stimulant 3-fluorophenmetrazine with a rarely used benzodiazepine etizolam. This patient developed seizure-like activity and delayed widespread T-wave inversions, both of which ultimately resolved without sequelae.

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Chronic calcified subdural haematoma found after presentation of symptomatic glioma

Description 

A patient with neurofibromatosis 1 and childhood ventriculoperitoneal (VP) shunt insertion for hydrocephalus presented with 4 weeks of short-term and working memory impairment. Apart from right-hand tremor, he denied headache, visual changes, weakness and dyscoordination. A CT head showed a right-sided subdural lesion with 22 mm midline shift. The MRI head showed a right inferior-frontal enhancing mass suspicious of high-grade glioma and right extensively calcified subdural extra-axial collection, with mixed heterogeneous predominantly T1-hyperintense contents (figure 1A).

Figure 1

(A) Preoperative MRI shows a right inferior frontal mass suspicious of high-grade glioma and the extra-axial collection with mixed heterogeneous predominantly T1-hyperintense contents, and extensive calcification. (B) Postoperative MRI showing more than 90% debulking of the enhancing tumour (glioblastoma, WHO grade IV) and the right subdural evacuation.

He underwent right fronto-temporo-parietal craniotomy with chronic calcified subdural haematoma evacuation, right frontal mass resection plus duraplasty (figure...

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Acyclovir-induced thrombocytopaenia in a patient with SLE

Acyclovir has been used in the treatment of herpes simplex and varicella zoster viral infections for over 30 years. The side effects of oral treatment at standard doses are rare and include headache, diarrhoea, dizziness and malaise. We report a patient with a new diagnosis of systemic lupus erythematosus (SLE) who developed thrombocytopaenia within days on a therapeutic dose with acyclovir. Prompt discontinuation of acyclovir and treatment with intravenous immunoglobulin resulted in reversal of the above potentially serious complication. Therefore a high index of suspicion should be exercised in patients with SLE who require treatment with acyclovir for herpes viral infections. In these patients regular platelet count measurement should be considered while on treatment with the above antiviral agent.

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Symptomatic hyperthyroidism in metastatic testicular mixed germ cell tumour

This is a case of a young 34-year-old man who had symptomatic hyperthyroidism from an extremely high serum beta HCG level in the setting of a metastatic stage IIIC mixed germ cell tumour. The patient's thyroid function normalised with treatment of the cancer as the beta HCG levels declined.

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Orbital cysticercosis

A young male patient presented to our ocular emergency department with chief complaints of progressive pain, redness, diplopia and a right-sided face turn. Ocular examination revealed severely restricted extraocular movements along with retinal folds in the left eye. Initial orbital ultrasound and CT findings were equivocal; however, serology favoured an infective cause. Considering the endemicity of the disease and equivocal investigation findings, a diagnosis of orbital cysticercosis with an atypical presentation was made. The patient was managed medically with a combination of oral albendazole and steroids over a period of 6 weeks to achieve optimal results.

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Babesiosis as a cause of false-positive HIV serology

This is a case of a 71-year-old homosexual man who presented with a 4-day history of fever, weakness and headaches, near syncope, nausea and poor oral intake. The patient denied recent travel or sick contacts but had significant tick bites in the last 4 weeks. A peripheral blood smear showed 0.5% parasitaemia with signet ring appearance organisms consistent with Babesia microti. Serology testing for HIV 1 and 2 by ELISA and western blot were positive. Treatment for Babesia was started and the patient improved. Repeat serology testing for HIV was negative. To the best of our knowledge, this is the first case of false-positive HIV serology that is associated with active babesiosis. In this case, the positive HIV serology turned negative after successful treatment of babesiosis.

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Salvage therapy for acute severe ulcerative colitis during pregnancy

Ulcerative colitis (UC) is a chronic inflammatory bowel disease with an age of onset that affects young people during the peak of their reproductive years. Management of flares of disease during pregnancy can be complex and there are few case reports of pregnant women with acute severe ulcerative colitis (ASUC). We present the case of a 31-year-old pregnant woman who at 16 weeks gestation developed ASUC in the context of primary non-response to infliximab therapy. She subsequently underwent an emergency laparoscopic colectomy after failing to respond to hydrocortisone and cyclosporine salvage therapy. Her pregnancy was further complicated by HELLP (Haemolysis, Elevated liver enzymes and Low Platelets) syndrome resulting in premature delivery at 27 weeks gestation. This case highlights the management issues involved in ASUC during pregnancy and the assessment of disease activity, use of salvage therapies, and provides a framework to approach this complex medical emergency.

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Obstructive hydrocephalus caused by colloid cyst presenting as a schizophrenia-like psychosis

This report presents a young woman with psychotic symptoms that were highly suspicious of schizophrenia. Previous attempts of antidepressant therapy of mood symptoms had been ineffective. However, she also presented with discrete signs of an underlying neurological condition, and as a part of the diagnostic workup, neuroimaging revealed ventriculomegaly. She was diagnosed with obstructive hydrocephalus and experienced full remission of psychotic symptoms after neurosurgical intervention with a ventriculoperitoneal shunt. This atypical presentation of obstructive hydrocephalus underlines the importance of neuroimaging in psychiatric differential diagnostics. Otherwise, this young woman had been diagnosed with schizophrenia, instead of organic delusional disorder secondary to a neurological condition, with possible irreversible damage to brain tissue as a consequence.

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Concomitant sarcoidosis and papillary thyroid cancer with severe hypercalcaemia as the main symptom

A 65-year-old Caucasian man was admitted to our hospital due to incidental finding of plasma ionised calcium=1.83 mmol/L. During the previous months, he had felt more tired and had experienced an unintended weight loss of 6–8 kg. A CT scan revealed an inhomogeneous thyroid gland and enlarged lymph nodes along the cervical vessels and in the mediastinum. The patient underwent total thyroidectomy and, at the same time, two lymph nodes were removed. Histological examination of the thyroid gland revealed papillary thyroid carcinoma (PTC), T1aN0M0, and histological examination of the lymph nodes showed non-caseating granulomatous inflammation. After further investigation, a diagnosis of sarcoidosis (SA) was made.

Coexistence of SA and PTC may yield a diagnostic challenge as both diseases might involve the lymph nodes of the neck. Furthermore, the case illustrates the importance of close follow-up of even moderate hypercalcaemia, since it may evolve into a hypercalcaemic crisis.

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Functional septorhinoplasty in the pediatric and adolescent patient

Publication date: August 2018
Source:International Journal of Pediatric Otorhinolaryngology, Volume 111
Author(s): Jennifer C. Fuller, Patricia A. Levesque, Robin W. Lindsay
ObjectivesTo describe pediatric and adolescent patients undergoing functional septorhinoplasty and to analyze both subjective and objective outcomes.MethodsRetrospective review of prospective cohort study of patients ≤18 years old undergoing functional septorhinoplasty between 2013 and 2016 at a tertiary care center. Patient demographics, nasal exam, procedure, and pre- and postoperative nasal obstruction symptom evaluation (NOSE) score, EuroQOL 5-dimension (EQ5D), and peak nasal inspiratory flow (PNIF) scores were analyzed.Results39 patients, 48.7% male, mean age 15.9 years (range 7–18), with nasal obstruction underwent functional septorhinoplasty with mean follow up of 8.5 months. Patients reported a history of allergies (46.5%), nasal fracture (59.0%), and previous nasal surgery (25.6%). Most common exam findings included internal nasal valve narrowing (92.3%), superior/dorsal septal deviation (74.4%), external nasal valve narrowing (43.6%), caudal septal deviation (35.9%), and a narrow middle vault (33.3%). Septal cartilage grafts were placed in 79.5% of patients and PDS plate was used in 28.2%. Most common procedures included spreader grafts (84.6%), columellar strut graft (30.8%), and swinging door (23.1%). Of patients with both baseline and postoperative scores, at last follow up NOSE scores (SD) decreased from 59.0 (23.7) to 21.2 (8.8) (n = 26, p < 0.001), EQ5D VAS scores increased from 76.2 (17.7) to 85.8 (13.5), (n = 19, p = 0.056), and PNIF scores increased from 66.2 (25.3) to 90.8 (46) L/min, (n = 13, p < 0.01); all mean differences met the minimal clinically important difference for each score. 2 patients underwent revision surgery and there was one complication of a nasal abscess.ConclusionsFunctional septorhinoplasty is safe and effective in select pediatric and adolescent patients with significant nasal obstruction and results in significant improvements in both subjective and objective outcomes measures.



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Clinical significance of spontaneous nystagmus in pediatric patients

Publication date: August 2018
Source:International Journal of Pediatric Otorhinolaryngology, Volume 111
Author(s): Guangwei Zhou, Cassandra Goutos, Sophie Lipson, Jacob Brodsky
ObjectiveTo analyze spontaneous nystagmus recorded in pediatric patients and determine its diagnostic value.Materials and methodsRetrospective review of 86 patients (mean age = 13.9 ± 4.7 years) with definitive spontaneous nystagmus recorded by videonystagmography (VNG). Spontaneous nystagmus was categorized and analyzed based on its characteristics. Correlations between spontaneous nystagmus and established clinical diagnoses were explored.ResultsAmong the patients seen in a period of three years at our pediatric vestibular clinic, 15.4% (86 out of 560) were found to have spontaneous nystagmus and half of them had horizontal nystagmus with a fixed-direction. The majority of cases with spontaneous nystagmus (77%, 66/86) were present without fixation only, while the presence of spontaneous nystagmus with and without fixation was found in 16 patients (19%). Most patients with spontaneous nystagmus without fixation only (68%) were diagnosed with a peripheral vestibular disorder (e.g., vestibular neuritis) or vestibular migraine. In contrast, vertical nystagmus was found in 15 patients, most of whom had central vestibular disorders. Other rare forms of nystagmus or abnormal eye movements, such as pendular nystagmus, ocular oscillation or flutter were recorded in only four patients.ConclusionsSpontaneous nystagmus in children and young adults with possible vestibular impairments may vary in clinical presentation. Identification of its characteristics may help to make an accurate clinical diagnosis. While spontaneous nystagmus associated with peripheral vestibular disorders can be readily recognized, spontaneous nystagmus in pediatric patients with vestibular migraine appears to be more complex and variable in form.



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A 3D Analysis of Mandibular Plating Strategies in Oromandibular Reconstruction.

Condition:   Head and Neck Cancer
Intervention:   Device: 3D mandibular plate
Sponsor:   University Health Network, Toronto
Recruiting

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Evaluation of Ocoxin®-Viusid® in Advanced Stomach Cancer and Gastric Esophagogastric Junction

Conditions:   Stomach Neoplasm;   Gastrointestinal Neoplasms;   Digestive System Neoplasm;   Esophageal Neoplasms;   Head and Neck Neoplasms;   Gastrointestinal Disease;   Digestive System Disease;   Esophageal Diseases;   Stomach Diseases;   Esophagogastric Junction Disorder
Intervention:   Dietary Supplement: Ocoxin-Viusid®
Sponsor:   Catalysis SL
Not yet recruiting

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Chronic obstructive pulmonary disease subpopulations and phenotyping

Publication date: June 2018
Source:Journal of Allergy and Clinical Immunology, Volume 141, Issue 6
Author(s): Leopoldo N. Segal, Fernando J. Martinez
Information for Category 1 CME CreditCredit can now be obtained, free for a limited time, by reading the review articles in this issue. Please note the following instructions.Method of Physician Participation in Learning Process: The core material for these activities can be read in this issue of the Journal or online at the JACI Web site: www.jacionline.org. The accompanying tests may only be submitted online at www.jacionline.org. Fax or other copies will not be accepted.Date of Original Release: June 2018. Credit may be obtained for these courses until May 31, 2019.Copyright Statement: Copyright © 2018-2019. All rights reserved.Overall Purpose/Goal: To provide excellent reviews on key aspects of allergic disease to those who research, treat, or manage allergic disease.Target Audience: Physicians and researchers within the field of allergic disease.Accreditation/Provider Statements and Credit Designation: The American Academy of Allergy, Asthma & Immunology (AAAAI) is accredited by the Accreditation Council for Continuing Medical Education (ACCME) to provide continuing medical education for physicians. The AAAAI designates this journal-based CME activity for a maximum of 1.00 AMA PRA Category 1 Credit™. Physicians should claim only the credit commensurate with the extent of their participation in the activity.List of Design Committee Members: Leopoldo N. Segal, MD, MS, and Fernando J. Martinez, MD, MS (authors); Zuhair K. Ballas, MD (editor)Disclosure of Significant Relationships with Relevant CommercialCompanies/Organizations: L. N. Segal has received grants from the National Institutes of Health and personal fees from Advanced Inhalation Therapies. F. J. Martinez has received a grant from the National Heart, Lung, and Blood Institute; has received personal fees from Continuing Education, Forest Laboratories, GlaxoSmithKline, Nycomed/Takeda, AstraZeneca, Boehringer Ingelheim, Bellerophon (formerly Ikaria), Genentech, Novartis, Pearl, Roche, Sunovion, Theravance, CME Incite, the Annenberg Center for Health Sciences at Eisenhower, Integritas, InThought, the National Association for Continuing Education, Paradigm Medical Communications, PeerVoice, UpToDate, Haymarket Communications, the Western Society of Allergy and Immunology, Proterixbio, Unity Biotechnology, ConCert Pharmaceuticals, Lucid, Methodist Hospital, Columbia University, Prime Healthcare, WebMD, the PeerView Network, the California Society of Allergy and Immunology, Chiesi, and the Puerto Rico Thoracic Society and is on the COPD advisory board for Janssen. Z. K. Ballas (editor) disclosed no relevant financial relationships.Activity Objectives:1. To understand the different phenotypes of chronic obstructive pulmonary disease (COPD), including comorbidities, clinical course, and treatment implications.2. To understand the pathogenesis of COPD.3. To understand the characteristics of COPD exacerbations.Recognition of Commercial Support: This CME activity has not received external commercial support.List of CME Exam Authors: Ryan Israelsen, MD, Katherine McCormack, MD, Hannah Duffey, MD, Allison Hicks, MD, Joseph Spahn, MD, and Maureen Egan, MDDisclosure of Significant Relationships with Relevant CommercialCompanies/Organizations: The exam authors disclosed no relevant financial relationships.The diagnosis and treatment of chronic obstructive pulmonary disease (COPD) has been based largely on a one-size-fits-all approach. Diagnosis of COPD is based on meeting the physiologic criteria of fixed obstruction in forced expiratory flows and treatment focus on symptomatic relief, with limited effect on overall prognosis. However, patients with COPD have distinct features that determine very different evolutions of the disease. In this review we highlight distinct subgroups of COPD characterized by unique pathophysiologic derangements, response to treatment, and disease progression. It is likely that identification of subgroups of COPD will lead to discovery of much needed disease-modifying therapeutic approaches. We argue that a precision approach that integrates multiple dimensions (clinical, physiologic, imaging, and endotyping) is needed to move the field forward in the treatment of this disease.



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Chronic obstructive pulmonary disease subpopulations and phenotyping

Publication date: June 2018
Source:Journal of Allergy and Clinical Immunology, Volume 141, Issue 6





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Of pleiotropy and trajectories: Does the TGF-β pathway link childhood asthma and chronic obstructive pulmonary disease?

Publication date: June 2018
Source:Journal of Allergy and Clinical Immunology, Volume 141, Issue 6
Author(s): Avery DeVries, Donata Vercelli
The study of developmental trajectories is where epigenetics truly shines. The "epi" in epigenetics captures the fact that although epigenetic processes also preside over the maintenance and termination of gene expression, the unfolding and remodeling of chromatin architecture are especially critical to prepare genes for regulated transcription. These properties imply being on a path, a trajectory to events that will occur later thanks to epigenetic programming. Thus epigenetics is about timed and timely events. In this article we discuss epigenetic and genetic evidence from several independent studies of asthma, chronic obstructive pulmonary disease, and lung function, which converge to highlight a potential role of the TGF-β gene pathway in these processes. These results raise the possibility that at least in a subset of subjects, these conditions might be functionally connected in ways that need to be further defined but that likely reflect the uniquely pleiotropic nature of TGF-β pathway genes, particularly their ability to control both lung development and immune responses essential for regulation and inflammation. Further characterization of this pathway in longitudinally phenotyped populations might unmask novel trajectories to lung disease that begin in utero and unfold into old age.



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The Editors' Choice

Publication date: June 2018
Source:Journal of Allergy and Clinical Immunology, Volume 141, Issue 6
Author(s): Zuhair K. Ballas




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News Beyond Our Pages

Publication date: June 2018
Source:Journal of Allergy and Clinical Immunology, Volume 141, Issue 6
Author(s): Marc E. Rothenberg, Jean Bousquet




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Is 9 more than 2 also in allergic airway inflammation?

Publication date: June 2018
Source:Journal of Allergy and Clinical Immunology, Volume 141, Issue 6
Author(s): Holger Garn




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Biological exacerbation clusters demonstrate asthma and chronic obstructive pulmonary disease overlap with distinct mediator and microbiome profiles

Publication date: June 2018
Source:Journal of Allergy and Clinical Immunology, Volume 141, Issue 6
Author(s): Michael A. Ghebre, Pee Hwee Pang, Sarah Diver, Dhananjay Desai, Mona Bafadhel, Koirobi Haldar, Tatiana Kebadze, Suzanne Cohen, Paul Newbold, Laura Rapley, Joanne Woods, Paul Rugman, Ian D. Pavord, Sebastian L. Johnston, Michael Barer, Richard D. May, Christopher E. Brightling
BackgroundExacerbations of asthma and chronic obstructive pulmonary disease (COPD) are heterogeneous.ObjectiveWe sought to investigate the sputum cellular, mediator, and microbiome profiles of both asthma and COPD exacerbations.MethodsPatients with severe asthma or moderate-to-severe COPD were recruited prospectively to a single center. Sputum mediators were available in 32 asthmatic patients and 73 patients with COPD assessed at exacerbation. Biologic clusters were determined by using factor and cluster analyses on a panel of sputum mediators. Patterns of clinical parameters, sputum mediators, and microbiome communities were assessed across the identified clusters.ResultsThe asthmatic patients and patients with COPD had different clinical characteristics and inflammatory profiles but similar microbial ecology. Three exacerbation biologic clusters were identified. Cluster 1 was COPD predominant, with 27 patients with COPD and 7 asthmatic patients exhibiting increased blood and sputum neutrophil counts, proinflammatory mediators (IL-1β, IL-6, IL-6 receptor, TNF-α, TNF receptors 1 and 2, and vascular endothelial growth factor), and proportions of the bacterial phylum Proteobacteria. Cluster 2 had 10 asthmatic patients and 17 patients with COPD with increased blood and sputum eosinophil counts, type 2 mediators (IL-5, IL-13, CCL13, CCL17, and CCL26), and proportions of the bacterial phylum Bacteroidetes. Cluster 3 had 15 asthmatic patients and 29 patients with COPD with increased type 1 mediators (CXCL10, CXCL11, and IFN-γ) and proportions of the phyla Actinobacteria and Firmicutes.ConclusionsA biologic clustering approach revealed 3 subgroups of asthma and COPD exacerbations, each with different percentages of patients with overlapping asthma and COPD. The sputum mediator and microbiome profiles were distinct between clusters.

Graphical abstract

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Blood eosinophil count thresholds and exacerbations in patients with chronic obstructive pulmonary disease

Publication date: June 2018
Source:Journal of Allergy and Clinical Immunology, Volume 141, Issue 6
Author(s): Jeong H. Yun, Andrew Lamb, Robert Chase, Dave Singh, Margaret M. Parker, Aabida Saferali, Jørgen Vestbo, Ruth Tal-Singer, Peter J. Castaldi, Edwin K. Silverman, Craig P. Hersh
BackgroundEosinophilic airway inflammation in patients with chronic obstructive pulmonary disease (COPD) is associated with exacerbations and responsivity to steroids, suggesting potential shared mechanisms with eosinophilic asthma. However, there is no consistent blood eosinophil count that has been used to define the increased exacerbation risk.ObjectiveWe sought to investigate blood eosinophil counts associated with exacerbation risk in patients with COPD.MethodsBlood eosinophil counts and exacerbation risk were analyzed in patients with moderate-to-severe COPD by using 2 independent studies of former and current smokers with longitudinal data. The Genetic Epidemiology of COPD (COPDGene) study was analyzed for discovery (n = 1,553), and the Evaluation of COPD Longitudinally to Identify Predictive Surrogate Endpoints (ECLIPSE) study was analyzed for validation (n = 1,895). A subset of the ECLIPSE study subjects were used to assess the stability of blood eosinophil counts over time.ResultsCOPD exacerbation risk increased with higher eosinophil counts. An eosinophil count threshold of 300 cells/μL or greater showed adjusted incidence rate ratios for exacerbations of 1.32 in the COPDGene study (95% CI, 1.10-1.63). The cutoff of 300 cells/μL or greater was validated for prospective risk of exacerbation in the ECLIPSE study, with adjusted incidence rate ratios of 1.22 (95% CI, 1.06-1.41) using 3-year follow-up data. Stratified analysis confirmed that the increased exacerbation risk associated with an eosinophil count of 300 cells/μL or greater was driven by subjects with a history of frequent exacerbations in both the COPDGene and ECLIPSE studies.ConclusionsPatients with moderate-to-severe COPD and blood eosinophil counts of 300 cells/μL or greater had an increased risk exacerbations in the COPDGene study, which was prospectively validated in the ECLIPSE study.



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License Suspended for Doctor Who Danced, Rapped in Surgeries

An Atlanta dermatologist used videos of the surgeries for marketing. Now at least seven women have filed suit against her. A bride-to-be who sought a cosmetic procedure is now on a feeding tube.
Medscape Medical News

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Announcements

Dear Colleagues,

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EACMFS Awards

The Council of EACMFS wishes to ensure that all members of the Association are aware of the current awards and prizes that are available. These are designed to provide educational support and also allow the opportunity for trainees and those who have recently achieved specialist status to visit units outside their own departments.

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Antifungal efficacy of herbs

Publication date: Available online 7 June 2018
Source:Journal of Oral Biology and Craniofacial Research
Author(s): FAHAD. M. SAMADI, SHAISTA SUHAIL, MANJARI SONAM, NEETA SHARMA, SHRUTI SINGH, SUSHIL GUPTA, ASHWINI DOBHAL, HARSHA PRADHAN




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Long-term follow-up of inactive occlusal caries lesions: 4-5-year results

Abstract

Objective

To assess the clinical behavior of inactive caries lesion on the occlusal sites of permanent molars over 4–5 years and to estimate the risk for progression of caries-inactive sites compared with sound ones.

Methods

Clinical examinations were conducted at baseline (n = 258) and after 4–5 years and included the recording of dental plaque and dental caries at the occlusal surfaces and the eruption stage of each permanent molar.

Results

One hudred ninety-three schoolchildren were followed (response rate of 74.8%), totalizing 1152 teeth. Of the children, 30.6% (n = 59) presented at least one molar containing an active lesion, filling, or that had been extracted; according to the activity criterion, inactive lesions presented around a twofold increased risk for caries progression than sound surfaces (OR = 2.34 95%CI = 1.51–3.62). Thirteen percent (n = 25) of the children presented at least one molar progressing to dentine cavity, filling, or extraction; according to the severity criterion, inactive caries lesions presented a significantly higher risk for progression when compared with sound surfaces (OR = 2.69, 95% CI = 1.50–4.83).

Conclusion

The vast majority of lesions (85–90%) identified as inactive enamel caries at baseline did not progress over 4–5 years. Despite this fact, it was possible to detect an increased risk for caries progression in caries-inactive occlusal sites compared with the sound ones.

Clinical relevance

Considering the low progression rates, inactive caries lesions do not need a specific caries-controlling treatment and should be monitored longitudinally in the same manner as sound surfaces.

https://ift.tt/2sKMHrE

Remineralization capacity of carious and non-carious white spot lesions: clinical evaluation using ICDAS and SS-OCT

Abstract

Objectives

To assess the remineralization capacity of carious, non-carious, and combined white spot lesions (WSLs) using the ICDAS and SS-OCT.

Materials and methods

This clinical trial was based on a quasi-experimental design. Forty-two healthy subjects (median age 26.6 years), who visited university hospital and had at least one WSL with an ICDAS score of 2 or 1, were recruited. The subjects chewed a non-blind sugar-free gum containing bioavailable calcium and fluoride for 3 months. The remineralization capacities of carious and non-carious 121 WSLs were assessed using ICDAS by two calibrated non-blind examiners and optical boundary depth (BD) by SS-OCT at a monthly recall. The outcome variables, transitions of ICDAS score, mean BD, and mean BD recovery rate (RR%), were statistically analyzed using the chi-square test, two way-repeated measures ANOVA, and Wilcoxon rank sum test, respectively (alpha = 0.05).

Results

Based on the visual inspection, OCT images at the baseline, 72 WSLs were purely carious, 20 were non-carious (developmental) lesions, while 29 were combined (carious-developmental). The responses of WSLs over time showed to be highly variable. There was a significant difference in transitions of ICDAS scores after 3 months between carious and non-carious WSLs (p < 0.05) and non-carious and combined WSLs (p < 0.05). Carious and combined WSLs underwent significant changes in the mean BD between baseline (161.8 ± 56.8 μm) and 2 months (130.7 ± 57.4 μm) or 3 months (119.1 ± 57.5 μm) (p < 0.05), while there was no significant difference between baseline (132.2 ± 26.2 μm) and 2 months (122.8 ± 24.1 μm) or 3 months (119.8 ± 22.6 μm) in non-carious WSLs (p > 0.05). There was a significant difference in mean RR% after 2 and 3 months between carious and non-carious WSLs (p < 0.05).

Conclusions

The remineralization capacity of WSL was variable among the cases and subjects, and depended on the WSLs history, etiology (carious, non-carious, or combined lesion) and structure (histological pattern).

Clinical relevance

Carious WSLs showed the highest remineralization potential.

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The impact of premature birth on dental maturation in the permanent dentition

Abstract

Objectives

To evaluate tooth development and calculate dental maturity score in prematurely born children and to compare the findings with full-term born children.

Material and methods

Nine-year-old preterm children were selected from the Swedish Medical Birth Register. One group consisted of 36 extremely preterm children (born before week 29), and the other included 38 very preterm children (born during weeks 29 to 32). Panoramic radiography was performed on each child and the preterm children were compared with 42 full-term born children. Five observers independently assessed the tooth development stages for all teeth in the left mandible (31–37) on the panoramic radiographs according to the method described by Demirjian et al. (Hum Biol 45:211–227, 1973). Data from tooth development stages were compiled and converted into a dental maturity score for each group. Kappa values were calculated for intra- and inter-observer agreement.

Results

When the different development stages for each individual tooth were compared, all observers presented a significant delay in the maturity of tooth 37 for the extremely preterm group (p ≤ 0.002). The extremely preterm group had a significantly lower dental maturity score than the full-term group, as assessed by each observer (p ≤ 0.006). Kappa values for inter-observer agreement varied between 0.31 and 0.71 depending on tooth and intra-observer agreement was between 0.16 and 1.0.

Conclusions

At age 9, the extremely preterm children had a general delay in tooth development.

Clinical relevance

The increased survival rate of extremely preterm babies adds a new group of children to society. Dental clinicians should be aware that the delay in tooth development could impact the timing of orthodontic diagnostics and potential treatment.

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Massive thrombus trapped within an atrial septal defect

Description 

A 39-year-old morbidly obese man with a medical history of tobacco abuse and paroxysmal atrial fibrillation (Afib) on rivaroxaban presented to the emergency department with acute onset of shortness of breath and severe pleuritic chest pain. His vital signs were significant for blood pressure of 100/60 mm Hg, heart rate of 110 per minute, respiratory rate of 30 per minute and oxygen saturation of 80% on a non-rebreather mask. Physical examination was unremarkable, except for increased work of breathing. Arterial blood gas (ABG) showed pH 7.49, Pa_CO2 30 mm Hg and Pa_O2 86 mm Hg. Laboratory tests were significant for elevated troponin at 2.35 ng/mL and B-type natriuretic peptide at 309 pg/mL. ECG showed sinus tachycardia with new onset incomplete right bundle block and T-wave inversion in the right precordial and the inferior leads (figure 1A). A chest CT revealed extensive bilateral pulmonary embolism (PE), involving both main pulmonary arteries and suspected mass in the right...

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Magnetic sphincter augmentation is an effective option for refractory duodeno-gastro-oesophageal reflux following Billroth II gastrectomy

Bile reflux into the gastric stump and then into the oesophagus is a common event after distal gastrectomy and Billroth II reconstruction. In addition to typical symptoms of nausea, epigastric pain and bile vomiting, acid reflux can also occur in patients with concomitant hiatus hernia and lower oesophageal sphincter incompetency. Diverting the bile away from the oesophagus by conversion into a Roux-en-Y anastomosis or by completion gastrectomy and Roux-en-Y esophagojejunostomy have so far represented the mainstay of treatment. We report the first case of magnetic sphincter augmentation to relieve refractory reflux symptoms after Billroth II gastrectomy. The procedure was performed through a laparoscopic approach and proved very effective at 1-year follow-up.

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Big heart

Description

A 66-year-old female patient with a history of persistent atrial fibrillation and severe mitral stenosis secondary to rheumatic heart disease for which she underwent mitral valve replacement 25 years ago presented with progression of her baseline dyspnoea. On presentation, she had stable vital signs; neck examination revealed bilateral congested neck veins with prominent systolic venous pulsations. Chest and heart auscultation revealed a well-heard mechanical click, a pansystolic murmur heard over the tricuspid area and diminished air entry over the right lung base.

ECG revealed atrial fibrillation. Laboratory work-up showed that she was adequately anticoagulated but otherwise unremarkable; chest X-ray showed significant cardiomegaly, tracheal shift to the right and obliteration of the right basal lung field (figure 1).

Figure 1

Chest X-ray showing significant cardiomegaly, tracheal shift to the right and obliteration of the right basal lung field.

Echocardiography showed a...

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Meningovascular syphilis causing recurrent stroke and diagnostic difficulties: a scourge from the past

We describe a case of a 49-year-old man who presented with recurrent strokes in the left middle cerebral artery territory, manifesting with dysphasia, higher cognitive deficits, motor deficits and subsequent infarcts in the right middle cerebral and anterior cerebral artery territories, manifesting with seizures, behavioural and social issues. A key issue of the case was the diagnostic difficulty faced by the physicians. 'Meningovascular syphilis' was subsequently confirmed and appropriate treatment was given but there was subsequent relapse with worsening of the symptoms. Multiple specialists were involved in the management, namely stroke team, neurologists, psychiatrists, infectious disease and multidisciplinary therapy teams. This case highlights the need to be vigilant to the resurgence of syphilis, a scourge from the past, as a cause of stroke, especially in individuals who have had exposure to affected people. It is easy to miss the diagnosis and mistake it for other conditions unless a detailed history is taken and appropriate investigations are conducted, with a low threshold for diagnosis.

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Uterocutaneous fistula as the primary presentation of a gynaecological malignancy

We report a case of high-grade gynaecological carcinoma presenting as a uterocutaneous fistula. A 59-year-old woman presented with a discharging abdominal wall wound. Imaging confirmed a large solid pelvic mass forming a sinus tract with the anterior abdominal wall. The tract tunnelled through a previous caesarean section scar. Biopsy indicated a high-grade gynaecological carcinoma, with features suggestive of endometrioid adenocarcinoma. The patient underwent two cycles of chemotherapy. Despite this, the mass increased in size and was complicated by abdominal wall wound breakdown. Chemotherapy was ceased. Surgical and palliative options are under consideration.

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Dangerous mistake: an accidental caffeine overdose

Caffeine (1,3,7-trimethylxanthine) is a natural product commonly presented in food's composition, beverages and medicinal products. Generally, it is thought to be safe under normal dosage, yet it can be fatal in case of severe intoxication. We report a case of a healthy 32-year-old woman who went to the local emergency department (ED) 30 min after ingesting, accidentally, 5000 mg of anhydrous caffeine for a preworkout supplement. At the ED, she presented an episode of presyncope followed by agitation. ECG showed polymorphic broad complex QRS tachycardia and arterial blood gas revealed metabolic acidaemia with severe hypokalemia. The dysrhythmia was successfully treated with intravenous propranolol. Acid–base and hydroelectrolytic disorders were also corrected. A persistent sinus tachycardia was observed in the first 2 days in the ward and 5 days later she was discharged asymptomatic with internal medicine follow-up.

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Gallbladder volvulus with segmental right liver lobe hypoplasia/atrophy: a preoperative diagnostic dilemma

Gallbladder volvulus (GBV) due to rotation of the gall bladder (GB) around its own mesentery is a rare surgical emergency and often identified intraoperatively. Typically, cholecystitis is the initial clinical diagnosis, but a high index of suspicion on imaging can alert the physician for the possibility of GBV requiring urgent surgical intervention. We describe a case of a young female patient with hypoplasia/atrophy of the posterior segment of the right liver lobe and a GB with no hepatic attachments but only mesenteric pedicle. She presented with first episode of sudden-onset, severe right subcostal pain. The ultrasonogram and magnetic resonance cholangiopancreatogram findings were suggestive of GBV. She underwent laparoscopic exploration that confirmed GBV of a free-floating GB with a thrombosed cystic artery. The GB was detorted, and cholecystectomy was performed. She had an uneventful postoperative course and was discharged with no complications. Histopathological examination showed intramural haematoma of the GB with wall necrosis.

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Systolic heart failure in a patient with primary aldosteronism

A 53-year-old African man with a 25-year history of uncontrolled hypertension and systolic heart failure presented with an acute congestive heart failure exacerbation. He was found to have severe hypokalaemia, so additional testing was performed, and primary aldosteronism was confirmed. CT scan showed a 1.2x2.4 cm well-defined, homogeneous adenoma of the left adrenal gland. Adrenal vein sampling confirmed unilateral primary aldosteronism with lateralisation to the left adrenal gland. The patient was started on spironolactone and was referred to surgery for laparoscopic left adrenalectomy. Primary aldosteronism is associated with high cardiovascular morbidity and mortality due to activation of cardiac mineralocorticoid receptors. Studies suggest these negative effects can be reduced with early intervention and treatment. This case highlights the importance of investigating secondary causes of hypertension in young patients and the potential adverse cardiac effects of primary aldosteronism when it goes undiagnosed and untreated for years.

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Segmental fracture of the scaphoid

We present a rare case of segmental fracture of the scaphoid. This case highlights the importance of using appropriate imaging modalities to assess the presence and morphology of fractures.

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Neuropsychological assessment as an objective tool to monitor treatment response in anti-N-methyl-D-aspartate receptor encephalitis

We report a 1-year follow-up of a young woman with anti-N-methyl-D-aspartate receptor encephalitis. Management of autoimmune encephalitis remains challenging as objective and clinically relevant biomarkers are sought, which allow for the monitoring of treatment response. While further investigation is required, we believe that this case highlights the importance of following a comprehensive neuropsychological profile as a clinically relevant biomarker to guide therapeutic decision-making. By relying on the neuropsychological assessment of the patient, treatment with more toxic medications was avoided and her antiepileptic drug regimen was simplified.

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Duplication cyst of the pylorus: a rare cause of gastric outlet obstruction

Alimentary tract duplications are a rare congenital malformation. They can present with varied symptoms owing to the locality of the duplication, along the gastrointestinal tract. Out of these duplications, the ones along the pylorus are the most rare. These are usually only diagnosed intraoperatively, as it is not a common differential on imaging due to its rarity. In lieu of the literature currently available, pyloric duplication cyst can present anytime from 1 week of age to 5 years, with some cases being detected antenatally due to the prevalence of regular antenatal scanning. Surgery remains the main stay of treatment with the goal of complete excision of the cyst and complete removal of the cyst mucosal lining. We report the case of a 5-year-old girl, which to our knowledge is the first ever reported case from Karachi, Pakistan.

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Phaeochromocytoma found on cardiovascular magnetic resonance in a patient presenting with acute myocarditis: an unusual association

Myocarditis is inflammation of the cardiac muscle. The symptoms, signs and basic investigation findings can mimic that of myocardial infarction. The most common cause is infection (most commonly viral). Cardiovascular magnetic resonance (CMR) is the gold standard non-invasive diagnostic test for potential acute myocarditis as it allows assessment of myocardial oedema and scar. A man aged 25 years was admitted with chest pain, dizziness, headache, palpitations and sweating. His troponin was mildly positive. A CMR was performed which showed mild myocarditis and a right suprarenal mass which was confirmed to be a phaeochromocytoma based on biochemistry and a dedicated imaging workup. Phaeochromocytoma can lead to cardiac involvement in the form of left ventricular dysfunction, or catecholamine-induced myocarditis.

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Editorial board

Publication date: June–July 2018
Source:Annales de Dermatologie et de Vénéréologie, Volume 145, Issues 6–7





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Effets paradoxaux des biothérapies — Une dénomination erronée ?

Publication date: June–July 2018
Source:Annales de Dermatologie et de Vénéréologie, Volume 145, Issues 6–7
Author(s): D. Jullien




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Le phénomène de la langue verte, ou le diagnostic par Google

Publication date: June–July 2018
Source:Annales de Dermatologie et de Vénéréologie, Volume 145, Issues 6–7
Author(s): C. Godillot, A. Laprie, C. Eid, J.-C. Fricain, S. Boulinguez, E. Casassa, E. Vigarios, V. Sibaud
IntroductionLa consommation régulière de cannabis peut s'associer à certaines toxicités endobuccales, souvent mal connues du clinicien : xérostomie, caries, maladie parodontale, leucoedème, hyperplasie gingivale, colonisation à Candida albicans, leucoplasie ou gingivite…ObservationNous rapportons ici l'apparition d'une coloration verte de la langue chez un patient, consécutive à l'inhalation intensive de marijuana.DiscussionCette complication n'a qu'exceptionnellement été rapportée dans la littérature médicale. Paradoxalement, elle est clairement identifiée sur différents moteurs de recherche sur internet, notamment Google.BackgroundRegular cannabis use may be associated with several oral changes not usually identified by dermatologists: xerostomia, increased risk of caries, periodontitis, leukoedema, gingival hyperplasia, and higher prevalence and density of Candida albicans, leukoplakia or gingivitis.Patients and methodsWe report herein the appearance of a characteristic green tongue in a patient following intensive marijuana inhalation.DiscussionThis complication has rarely been reported in the medical literature. Paradoxically, it is clearly described in different Internet search engines, particularly Google.



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Lymphome T cutané et systémique traité avec succès par greffe haplo-identique

Publication date: June–July 2018
Source:Annales de Dermatologie et de Vénéréologie, Volume 145, Issues 6–7
Author(s): M. Méchineaud, M. Mercier, Y. Le Corre, A. Croué, N. Ifrah, L. Martin
IntroductionNous rapportons un cas de lymphome T cutané et systémique ayant résisté au traitement conventionnel et dont l'évolution a conduit à réaliser une greffe de moelle haplo-identique.ObservationUne femme de 53 ans consultait pour un érythème infiltré du visage, des lésions kératosiques du tronc et des adénopathies centimétriques axillaires et inguinales. Le diagnostic de syndrome de Sézary (SS), forme leucémique d'un lymphome T cutané épidermotrope, était retenu. Après 3 ans de traitement par méthotrexate, la patiente développait un SS transformé avec atteinte viscérale. Le fort risque de rechute et l'absence de donneur HLA-compatible amenait à la réalisation d'une greffe haplo-identique de moelle osseuse. La patiente était en rémission complète à 2 ans et demi. L'évolution était cependant marquée par l'apparition, un an après la greffe, d'une éruption lichénoïde blaschko-distribuée dont l'aspect histologique était compatible avec une réaction du greffon contre l'hôte (GVH) chronique ; elle était traitée efficacement par bétaméthasone topique.DiscussionNous rapportons, à notre connaissance, la première greffe haplo-identique réalisée pour un lymphome cutané T transformé et systémique. Ce traitement pourrait désormais être une option pour les patients nécessitant une allogreffe mais n'ayant pas de donneur compatible. Les lésions lichénoïdes blaschko-distribuées attribuées à une GVH chronique pourraient être la conséquence d'une perte d'immunotolérance vis-à-vis de clones embryologiques anormaux, provoquant ainsi une réaction inflammatoire médiée par les lymphocytes T.BackgroundHerein, we report a case of systemic cutaneous T-cell lymphoma refractory to standard therapy, the course of which resulted in haplo-identical bone marrow grafting.Patients and methodsA 53-year-old woman consulted for facial erythema with infiltration, keratotic lesions on the trunk, and adenopathies measuring around 1cm on the axilla and inguinal folds. A diagnosis was made of Sézary syndrome (SS), a leukaemic form of epidermotropic cutaneous T-cell lymphoma. After three years of treatment with methotrexate, the patient developed transformed SS with visceral involvement. Given the high risk of relapse and the absence of an HLA-compatible donor, haploidentical bone marrow grafting was performed. The patient was still in complete remission two and a half years later. The disease course was nevertheless marked by the emergence one year after grafting of a Blaschko-distributed lichenoid eruption having histological features consistent with chronic graft-versus-host disease (GVHD); treatment with topical betamethasone proved efficacious.DiscussionTo our knowledge, this is the first reported case of haploidentical grafting for systemic and transformed cutaneous T-cell lymphoma. This approach could henceforth represent a therapeutic option for patients requiring an allograft in the absence of compatible donors. The Blaschko-distributed lichenoid lesions attributed to chronic GVHD could be the result of reduced immune tolerance to abnormal embryological clones leading to a T-lymphocyte-mediated inflammatory reaction.



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Ichtyoses congénitales : les biothérapies aussi ?

Publication date: June–July 2018
Source:Annales de Dermatologie et de Vénéréologie, Volume 145, Issues 6–7
Author(s): O. Dereure




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Calendrier

Publication date: June–July 2018
Source:Annales de Dermatologie et de Vénéréologie, Volume 145, Issues 6–7





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A mixed methods examination of distress and person-centred experience of head and neck lymphoedema

Publication date: August 2018
Source:Oral Oncology, Volume 83
Author(s): Jodie L. Nixon, Amanda E. Pigott, Bena Cartmill, Jane Turner, Jennifer Fleming, Sandro V. Porceddu
PurposeThis study aimed to examine the course and nature of distress and quality of life (QoL) during and after head and neck lymphoedema (HNL) treatment in people who developed HNL following treatment for head and neck cancer (HNC).MethodsThis study (n = 10) used a mixed method explanatory design to explore distress associated with HNL. Component 1 used a prospective repeated measures design to examine distress during a 22-week HNL program. Component 2 used a qualitative interview approach to understand the patient experience of distress after completion of HNL treatment.ResultsDuring the HNL program distress associated with HNL significantly reduced from baseline to week 6 (p = 0.015), and baseline to week 22 (p = 0.007). There were no significant differences in QoL, body image or fear of cancer progression over time. Self-reported presence of HNL significantly reduced from baseline to week 6 (p = 0.02), week 6 to week 22 (p = 0.026), and from baseline to week 22 (p = 0.001).Qualitative interviews using thematic analysis following HNL treatment, revealed 6 major themes associated with the experiences of distress related to HNL – psychological impact; physical appearance and pattern/timing; experience of receiving treatment; day to day impact; supports that helped manage distress; and adjustment to a new normal.ConclusionsThis study found that distress associated with cancer treatment-related HNL may reduce with the delivery of a HNL program. Interview data following completion of the HNL treatment identified several themes related to HNL and its relationship with distress, function and day-to-day life.Clinical Trial registration: HREC/12/QPAH/295.



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