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Resistant Chronic Spontaneous Urticaria - A Case Series Narrative Review of Treatment Options. 耐药慢性自发性荨麻疹-治疗方案的病例系列叙述回顾。
IF 2.2 Q3 Medicine Pub Date : 2022-01-01 DOI: 10.1177/21526575221144951
Sujoy Khan, Charu Chopra, Alaistair Mitchell, Alla Nakonechna, Patrick Yong, Mohammed Yousuf Karim

Background: Chronic spontaneous urticaria (CSU) can be extremely debilitating to the patient and challenging for the treating clinician. The National Institute of Health and Clinical Excellence (NICE) in the United Kingdom (UK) recommendation of omalizumab for patients who fail to respond to high-dose anti-histamines has improved treatment options and quality of life. However, there is still lack of clear guidelines for treatment of patients resistant to standard and anti-IgE therapies.

Methods: We discuss the therapeutic strategies employed among nine extremely resistant CSU cases and the heterogeneity between guidelines from different societies.

Results: Patients with anti-histamine-resistant urticaria either remained on omalizumab or started on immunosuppressive drugs (dapsone or ciclosporin) when they stopped responding to omalizumab. We used clinical assessment, skin biopsies (when available) and previous published reports to consider dapsone (for predominantly neutrophilic infiltration), or ciclosporin at doses between 2 and 4 mg/kg/day. One patient with ciclosporin-resistant urticaria responded to mycophenolate mofetil. Two patients remain on long-term omalizumab due to its relative safety and efficacy including 1 patient with underlying antibody deficiency where omalizumab was preferred over risks of using immunosuppressive medications.

Conclusions: These case studies bring to light the real-world difficulties in managing patients with resistant CSU and the need for generating the evidence base on alternative therapeutic options such as synergistic use of biologics and immunosuppressive drugs.

背景:慢性自发性荨麻疹(CSU)对患者来说非常虚弱,对治疗临床医生来说也是一个挑战。英国国家健康与临床卓越研究所(NICE)推荐对大剂量抗组胺药无效的患者使用omalizumab,改善了治疗选择和生活质量。然而,对于治疗对标准和抗ige治疗有耐药性的患者,仍然缺乏明确的指导方针。方法:我们讨论了9例极耐药CSU病例的治疗策略以及不同社会指南之间的异质性。结果:抗组胺耐药荨麻疹患者在对奥玛珠单抗无效时,要么继续使用奥玛珠单抗,要么开始使用免疫抑制药物(氨苯砜或环孢素)。我们使用临床评估、皮肤活检(如有)和先前发表的报告来考虑氨苯砜(主要用于嗜中性粒细胞浸润)或环孢素,剂量在2至4mg /kg/天之间。1例环孢素耐药荨麻疹患者对霉酚酸酯有反应。由于omalizumab的相对安全性和有效性,两名患者仍然长期服用omalizumab,其中包括1名潜在抗体缺乏的患者,其中omalizumab优于使用免疫抑制药物的风险。结论:这些病例研究揭示了现实世界中管理耐药CSU患者的困难,以及为替代治疗方案(如生物制剂和免疫抑制药物的协同使用)提供证据的必要性。
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引用次数: 2
Smell and Taste in Children with Covid-19. Covid-19患儿的嗅觉和味觉
IF 2.2 Q3 Medicine Pub Date : 2022-01-01 DOI: 10.1177/21526575221144950
Piero Giuseppe Meliante, Alessia Marcellino, Roger Altomari, Alessia Testa, Andrea Gallo, Massimo Ralli, Marco de Vincentiis, Riccardo Lubrano

Objectives: To assess the frequency of loss of smell and taste in children during Covid-19 infection and their prevalence along with other symptoms, as well as the recovery of chemosensory function once healed.

Methods: To evaluate symptoms during infection, we adapted the Scandinavian adaptation of the Multi-Clinic Smell and Taste Questionnaire and the modified Monel-Jefferson questionnaire. For smell analysis we used Odor Identification (OI) and two variants of the Odor Discrimination (OD) test, and we compared the results with those of a control group.

Results: We enrolled nine patients in our experimental group and nine in our control group. Fever was the most frequent symptom (55% of cases), followed by anosmia and ageusia (44% of cases), muscle pain and asthenia (22% of cases) and diarrhea, abdominal pain, cough, and headache (11% of cases). In 11% of cases, olfactory symptoms were the only manifestation of the disease. There was no statistically significant difference in OI test and OD tests between the two groups (Children healed from Covid-19 and Control Group).

Conclusion: Loss of smell and taste are the second most common symptoms of pediatric Covid-19, and they should always be tested because they can be the only manifestations of infection. Olfactory function in Covid-19 children decreases with increasing age and improves with the passage of time after illness.

目的:评估儿童在Covid-19感染期间嗅觉和味觉丧失的频率及其伴随其他症状的流行情况,以及治疗后化学感觉功能的恢复情况。方法:采用斯堪的纳维亚版多临床嗅觉味觉问卷和改良的Monel-Jefferson问卷对感染期间的症状进行评估。对于气味分析,我们使用气味识别(OI)和两种变体的气味辨别(OD)测试,并将结果与对照组的结果进行比较。结果:实验组9例,对照组9例。发烧是最常见的症状(55%的病例),其次是嗅觉丧失和衰老(44%的病例),肌肉疼痛和乏力(22%的病例)以及腹泻、腹痛、咳嗽和头痛(11%的病例)。在11%的病例中,嗅觉症状是该疾病的唯一表现。两组(新冠肺炎患儿和对照组)OI测试和OD测试差异无统计学意义。结论:嗅觉和味觉丧失是儿童Covid-19的第二大常见症状,由于它们可能是感染的唯一表现,因此应始终进行检测。新冠肺炎患儿的嗅觉功能随着年龄的增长而下降,随着患病后时间的推移而改善。
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引用次数: 1
Orbital Complications of Acute Sinusitis in Pediatric Patients: Management of Chandler III Patients 儿童急性鼻窦炎的眼眶并发症:Chandler III患者的处理
IF 2.2 Q3 Medicine Pub Date : 2022-01-01 DOI: 10.1177/21526575221097311
Mohamad Z. Saltagi, Cyrus C. Rabbani, Kunal S. Patel, Todd J Wannemuehler, Rao V. Chundury, Elisa A. Illing, J. Ting
Background Surgery is often avoided in the setting of pediatric orbital complications from acute sinusitis unless necessitated by alarming ophthalmological signs. Criteria for surgical intervention are not well-defined. Objective We aim to review our experiences, management practices and patient outcomes over a ten-year period for Chandler III patients. Methods A retrospective review was performed from January 1, 2007 through December 31, 2016 of patients treated for orbital symptoms secondary to acute sinusitis at a free-standing tertiary-care pediatric hospital. Results Of the 186 patients reviewed, 42 Chandler III patients were included. Average age was 82.6 months (SD 50.6) with a slight male predominance (M to F, 1.8 to 1). 27 patients (64.3%) underwent intervention including endoscopic sinus surgery (ESS) with or without orbitotomy. Late surgical intervention (>48hrs from admission) demonstrated significant increase in overall length of stay (LOS) when compared with early surgical intervention and/or medical management (median, 6.9 vs 3.6 vs 3.7 days; p < 0.01). Postoperative LOS was also higher in the late surgery group compared with patients who had surgery within 48 hours of admission, but this did not reach statistical significance [median, 3.8 vs 2.8 days, p= 0.12]. There was no significant difference in overall abscess volume between patients who underwent intervention and those who did not (1019 mm3 vs 805 mm3, p = 0.5), but abscess width ≥ 1.2 cm was associated with higher rates of intervention. An alarming extraocular exam was the most common factor associated with surgical intervention. Conclusion Pediatric subperiosteal orbital abscess may prompt surgical intervention by ESS. An alarming ophthalmologic exam should prompt consideration of early intervention, which may lead to decreased overall and post-operative length of hospital stay. Level of Evidence 4 Meeting Information American Rhinologic Society, Fall National Meeting. Chicago, IL, USA. September 8–9, 2017.
背景:急性鼻窦炎引起的儿童眼眶并发症通常避免手术,除非有警示的眼科症状。手术干预的标准没有明确定义。我们的目的是回顾我们的经验,管理实践和病人的结果在十年期间钱德勒III型患者。方法回顾性分析2007年1月1日至2016年12月31日在某独立三级儿科医院因急性鼻窦炎继发眼眶症状就诊的患儿。结果186例患者中,42例为钱德勒III型患者。平均年龄82.6个月(SD 50.6),男性稍占优势(M对F, 1.8对1)。27例(64.3%)患者接受了包括内窥镜鼻窦手术(ESS)在内的干预,合并或不合并眼眶切开。与早期手术干预和/或医疗管理相比,晚期手术干预(入院后48小时)显示总住院时间(LOS)显著增加(中位数,6.9天vs 3.6天vs 3.7天;p < 0.01)。手术时间较晚的患者术后LOS也高于入院48小时内手术的患者,但差异无统计学意义[中位数,3.8 vs 2.8天,p= 0.12]。在接受干预的患者和未接受干预的患者之间,总脓肿体积没有显著差异(1019 mm3 vs 805 mm3, p = 0.5),但脓肿宽度≥1.2 cm与更高的干预率相关。令人担忧的眼外检查是与手术干预相关的最常见因素。结论小儿骨膜下眶内脓肿可行ESS手术干预。一个令人担忧的眼科检查应该提示考虑早期干预,这可能导致减少总体和术后住院时间。证据级别4会议信息美国鼻科学学会秋季全国会议。芝加哥,伊利诺伊州,美国。2017年9月8日至9日。
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引用次数: 3
COVID-19 Infection in Patients with Humoral Immunodeficiency: A Case Series and Literature Review 体液免疫缺陷患者的COVID-19感染:病例系列和文献综述
IF 2.2 Q3 Medicine Pub Date : 2022-01-01 DOI: 10.1177/21526575221096044
Maaz Jalil, Julianne Pietras, S. N. Ahmed, P. Daniels, R. Hostoffer
Background The coronavirus 2019 disease (COVID-19) has infected many individuals worldwide and continues to pose a significant threat to those with weakened immune systems. The data evaluating the clinical outcomes of patients with humoral immunodeficiencies that contract COVID-19 is limited and conflicting. Objective To describe the clinical outcomes of COVID-19 infections in patients with primary humoral immunodeficiency and compare results to current literature. Methods We conducted a retrospective cohort review on 15 patients with a humoral immunodeficiency defined as Common Variable Immunodeficiency, Specific Antibody Deficiency, or unspecified hypogammaglobulinemia, who contracted COVID-19. Severity scores were determined to evaluate the clinical outcomes of these patients. Results Of our 15-patient cohort, 33% of individuals with a humoral immunodeficiency infected with COVID-19 had moderate to severe disease, requiring hospitalization or resulting in death. COVID-19 mortality rate was found to be 7%. All 5 of our patients with severe COVID-19 infection had at least 1 comorbidity or risk factor. Conclusion Within our cohort of humoral immunodeficient patients infected with COVID-19, we found a higher rate of moderate to severe COVID-19 infection and worse clinical outcomes, particularly in patients with comorbidities or risk factors.
2019冠状病毒病(COVID-19)已经感染了全球许多人,并继续对免疫系统较弱的人构成重大威胁。评估体液免疫缺陷患者感染COVID-19的临床结果的数据有限且相互矛盾。目的了解COVID-19感染原发性体液免疫缺陷患者的临床结局,并与现有文献进行比较。方法:我们对15例感染COVID-19的体液免疫缺陷(定义为常见可变免疫缺陷、特异性抗体缺陷或未明确的低丙种球蛋白血症)患者进行了回顾性队列研究。确定严重程度评分来评估这些患者的临床结果。在我们的15例患者队列中,33%的体液免疫缺陷感染COVID-19的个体患有中度至重度疾病,需要住院治疗或导致死亡。新冠肺炎死亡率为7%。所有5例严重COVID-19感染患者均至少有1种合并症或危险因素。在我们的体液免疫缺陷患者感染COVID-19队列中,我们发现中度至重度COVID-19感染率更高,临床结果更差,特别是在有合并症或危险因素的患者中。
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引用次数: 4
Choanal Atresia Repair With Stents and Flaps: A Systematic Review Article. 用支架和皮瓣修复后肛门闭锁:一篇系统综述文章。
IF 2.2 Q3 Medicine Pub Date : 2021-12-17 eCollection Date: 2021-01-01 DOI: 10.1177/21526567211058052
Hemail M Alsubaie, Wedyan H Almosa, Ali S Al-Qahtani, Osama Margalani

Background: Choanal Atresia is an obliteration by an atretic plate of the posterior choanae due to a failure of the bucconasal membrane to rupture. The insertion of stents post choanal atresia repair is familiar. Still, there are limited studies in the literature that specify the time to remove it, the best materials, and the effectiveness of each kind.

Objective: Our study aims to compare different types of choanal atresia stents and flaps and the outcome of different kinds of stents and flaps that use post-operatively. Also, to assess the risk of restenosis post-operatively with varying types of Stent and flaps.

Methods: A systematic review via databases for different types of stents and flaps used in choanal atresia, including 31 studies with precise technique, variety of stents, restenosis rates, and risk factors (unilaterality, the component of the atretic plate). And about 10 different approaches to flaps reconstruction were mentioned.

Results: According to the data, we observed a successful rate of choanal atresia repair by using the ordinary ETT post-operatively ranged from 28 to 94.2%, which could be explained due to many factors. Post-operative Instructions on care and suctioning provided a good impact. Other types of stents found in case series like steroid eluting stents, Nelaton catheters, Silastic stents, or modified ETT have a promising future during 26 to 39 weeks follow-up. Still, they need further studies with randomization and more data. Flaps with different approaches and techniques showed promising results and fewer complication rates with or without stents, also now have been used in practice providing suitable alternatives for stents.

Conclusion: The original types of choanal atresia stents were shown to have a wide variety of results, while innovative materials of stents showed promising results, however, in relatively small case studies. Flaps were now used in practice giving other choices for stents with fewer complications, better healing, and new choana formation.

背景:后鼻孔闭锁是由于鼻鼻膜破裂失败导致后鼻孔闭锁钢板闭塞。后肛门闭锁修补后置入支架是常见的。尽管如此,文献中仍然有有限的研究详细说明了去除它的时间,最佳材料以及每种材料的有效性。目的:比较不同类型的后肛门闭锁支架及皮瓣的术后应用效果。此外,评估不同类型的支架和皮瓣术后再狭窄的风险。方法:通过数据库对用于后肛门闭锁的不同类型支架和皮瓣进行系统回顾,包括31项采用精确技术、支架种类、再狭窄率和危险因素(单侧、锁板组成)的研究。我们提到了10种不同的皮瓣重建方法。结果:根据资料,我们观察到术后使用普通ETT修复后肛门闭锁的成功率在28 ~ 94.2%之间,这可能是由多种因素引起的。术后护理和吸痰指导提供了良好的效果。在26 - 39周的随访中,其他类型的支架如类固醇洗脱支架、内拉顿导管、硅橡胶支架或改良ETT有很好的应用前景。不过,他们还需要进一步的随机化研究和更多的数据。不同的方法和技术的皮瓣显示出良好的结果和较少的并发症发生率,有或没有支架,现在也已用于实践中提供合适的替代支架。结论:原始类型的后肛门闭锁支架具有广泛的效果,而创新材料的支架具有良好的效果,但在相对较小的病例研究中。皮瓣现在在实践中使用,为支架提供了其他选择,并发症更少,愈合更好,新血管形成。
{"title":"Choanal Atresia Repair With Stents and Flaps: A Systematic Review Article.","authors":"Hemail M Alsubaie,&nbsp;Wedyan H Almosa,&nbsp;Ali S Al-Qahtani,&nbsp;Osama Margalani","doi":"10.1177/21526567211058052","DOIUrl":"https://doi.org/10.1177/21526567211058052","url":null,"abstract":"<p><strong>Background: </strong>Choanal Atresia is an obliteration by an atretic plate of the posterior choanae due to a failure of the bucconasal membrane to rupture. The insertion of stents post choanal atresia repair is familiar. Still, there are limited studies in the literature that specify the time to remove it, the best materials, and the effectiveness of each kind.</p><p><strong>Objective: </strong>Our study aims to compare different types of choanal atresia stents and flaps and the outcome of different kinds of stents and flaps that use post-operatively. Also, to assess the risk of restenosis post-operatively with varying types of Stent and flaps.</p><p><strong>Methods: </strong>A systematic review via databases for different types of stents and flaps used in choanal atresia, including 31 studies with precise technique, variety of stents, restenosis rates, and risk factors (unilaterality, the component of the atretic plate). And about 10 different approaches to flaps reconstruction were mentioned.</p><p><strong>Results: </strong>According to the data, we observed a successful rate of choanal atresia repair by using the ordinary ETT post-operatively ranged from 28 to 94.2%, which could be explained due to many factors. Post-operative Instructions on care and suctioning provided a good impact. Other types of stents found in case series like steroid eluting stents, Nelaton catheters, Silastic stents, or modified ETT have a promising future during 26 to 39 weeks follow-up. Still, they need further studies with randomization and more data. Flaps with different approaches and techniques showed promising results and fewer complication rates with or without stents, also now have been used in practice providing suitable alternatives for stents.</p><p><strong>Conclusion: </strong>The original types of choanal atresia stents were shown to have a wide variety of results, while innovative materials of stents showed promising results, however, in relatively small case studies. Flaps were now used in practice giving other choices for stents with fewer complications, better healing, and new choana formation.</p>","PeriodicalId":45192,"journal":{"name":"Allergy & Rhinology","volume":null,"pages":null},"PeriodicalIF":2.2,"publicationDate":"2021-12-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/ec/40/10.1177_21526567211058052.PMC8842470.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39627522","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 2
Successful Anti-SARS-CoV-2 Spike Protein Antibody Response to Vaccination in MAGT1 Deficiency. MAGT1缺乏症成功接种抗sars - cov -2刺突蛋白抗体
IF 2.2 Q3 Medicine Pub Date : 2021-11-24 eCollection Date: 2021-01-01 DOI: 10.1177/21526567211056239
Maaz Jalil, Marija Rowane, Jayanth Rajan, Robert Hostoffer

Background: Novel messenger RNA vaccines against severe acute respiratory syndrome coronavirus (SARS-CoV-2) have been vital in resolving the coronavirus disease-2019 (COVID-19) pandemic. Detection of neutralizing antibodies (NAbs) against the SARS-CoV-2 spike protein (S) confirms immunogenicity with high sensitivity and specificity. Few recent studies with primary and secondary immunodeficient cohorts present adequate or reduced antibody response. We describe the first reported successful response to anti-SARS-CoV-2 S antibody post-vaccination in magnesium transporter 1 (MAGT1) gene deficiency, more commonly recognized as x-linked immunodeficiency with magnesium defect, Epstein-Barr Virus infection, and neoplasia (XMEN).

Case presentation: We present a 30-year-old male with selective anti-polysaccharide antibody deficiency, peripheral blood CD5  +  /CD19  +  B-cell predominance (97%), MAGT1 mutation, and reduced CD16  +  CD56  +  natural killer- and/or CD8  +  T-cell receptor, Group 2, Member D expression. His initial immunological evaluation revealed all seronegative post-vaccination antibody titers but clinically adequate response to protein antigens tetanus and diphtheria anti-toxoids.COVID-19 vaccination and associated serology antibody testing was recommended at this office visit. Anti-SARS-CoV-2 immunoglobulin (Ig)M and IgG antibodies before and after the first BNT162b2 mRNA COVID-19 vaccine doses, as well as nucleocapsid antibody, were negative. S protein total antibody was reactive after the second dose.

Discussion: Robust immunological sequelae post-COVID-19 vaccination in the general population are well-documented in the recent literature. Few studies have evaluated COVID-19 vaccination antibody response in immunodeficient patients. The majority positive anti-S antibody detection in most primary immunodeficient (PID) patients among the few studies in the literature, such as the present case, support the safety and efficacy of mRNA COVID-19 vaccination in immunodeficient patients, although larger scale studies are needed.

Conclusion: We demonstrate successful vaccination in the PID MAGT1 deficiency in this first reported case of reactive anti-S antibody post-COVID-19 vaccination.

背景:针对严重急性呼吸综合征冠状病毒(SARS-CoV-2)的新型信使RNA疫苗在解决冠状病毒病-2019 (COVID-19)大流行中至关重要。检测针对SARS-CoV-2刺突蛋白(S)的中和抗体(nab)证实具有高灵敏度和特异性的免疫原性。最近对原发性和继发性免疫缺陷队列的研究很少显示足够的或降低的抗体反应。我们描述了首次报道的镁转运蛋白1 (MAGT1)基因缺陷疫苗接种后抗sars - cov - 2s抗体的成功应答,镁转运蛋白1 (MAGT1)基因缺陷更常被认为是x连锁免疫缺陷伴镁缺陷、eb病毒感染和肿瘤(XMEN)。病例介绍:我们报告了一名30岁男性,患有选择性抗多糖抗体缺乏,外周血CD5 + /CD19 + b细胞优势(97%),MAGT1突变,CD16 + CD56 +自然杀伤细胞和/或CD8 + t细胞受体减少,2组,成员D表达。他最初的免疫学评估显示,所有疫苗接种后抗体滴度均为血清阴性,但临床对蛋白质抗原破伤风和白喉抗类毒素有足够的反应。在该办公室就诊时,建议进行COVID-19疫苗接种和相关血清学抗体检测。第一次BNT162b2 mRNA COVID-19疫苗剂量前后抗sars - cov -2免疫球蛋白(Ig)M和IgG抗体以及核衣壳抗体均为阴性。第二次给药后S蛋白总抗体出现反应。讨论:最近的文献充分记录了普通人群接种covid -19疫苗后的强烈免疫后遗症。很少有研究评估免疫缺陷患者的COVID-19疫苗抗体反应。文献中为数不多的研究,如本病例,在大多数原发性免疫缺陷(PID)患者中,大多数抗s抗体检测阳性,支持免疫缺陷患者接种mRNA COVID-19疫苗的安全性和有效性,尽管需要更大规模的研究。结论:在首次报道的covid -19疫苗接种后反应性抗s抗体病例中,我们证明了在PID MAGT1缺陷中成功接种疫苗。
{"title":"Successful Anti-SARS-CoV-2 Spike Protein Antibody Response to Vaccination in <i>MAGT1</i> Deficiency.","authors":"Maaz Jalil,&nbsp;Marija Rowane,&nbsp;Jayanth Rajan,&nbsp;Robert Hostoffer","doi":"10.1177/21526567211056239","DOIUrl":"https://doi.org/10.1177/21526567211056239","url":null,"abstract":"<p><strong>Background: </strong>Novel messenger RNA vaccines against severe acute respiratory syndrome coronavirus (SARS-CoV-2) have been vital in resolving the coronavirus disease-2019 (COVID-19) pandemic. Detection of neutralizing antibodies (NAbs) against the SARS-CoV-2 spike protein (S) confirms immunogenicity with high sensitivity and specificity. Few recent studies with primary and secondary immunodeficient cohorts present adequate or reduced antibody response. We describe the first reported successful response to anti-SARS-CoV-2 S antibody post-vaccination in magnesium transporter 1 (MAGT1) gene deficiency, more commonly recognized as x-linked immunodeficiency with magnesium defect, Epstein-Barr Virus infection, and neoplasia (XMEN).</p><p><strong>Case presentation: </strong>We present a 30-year-old male with selective anti-polysaccharide antibody deficiency, peripheral blood CD5  +  /CD19  +  B-cell predominance (97%), MAGT1 mutation, and reduced CD16  +  CD56  +  natural killer- and/or CD8  +  T-cell receptor, Group 2, Member D expression. His initial immunological evaluation revealed all seronegative post-vaccination antibody titers but clinically adequate response to protein antigens tetanus and diphtheria anti-toxoids.COVID-19 vaccination and associated serology antibody testing was recommended at this office visit. Anti-SARS-CoV-2 immunoglobulin (Ig)M and IgG antibodies before and after the first BNT162b2 mRNA COVID-19 vaccine doses, as well as nucleocapsid antibody, were negative. S protein total antibody was reactive after the second dose.</p><p><strong>Discussion: </strong>Robust immunological sequelae post-COVID-19 vaccination in the general population are well-documented in the recent literature. Few studies have evaluated COVID-19 vaccination antibody response in immunodeficient patients. The majority positive anti-S antibody detection in most primary immunodeficient (PID) patients among the few studies in the literature, such as the present case, support the safety and efficacy of mRNA COVID-19 vaccination in immunodeficient patients, although larger scale studies are needed.</p><p><strong>Conclusion: </strong>We demonstrate successful vaccination in the PID MAGT1 deficiency in this first reported case of reactive anti-S antibody post-COVID-19 vaccination.</p>","PeriodicalId":45192,"journal":{"name":"Allergy & Rhinology","volume":null,"pages":null},"PeriodicalIF":2.2,"publicationDate":"2021-11-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/01/c9/10.1177_21526567211056239.PMC8640323.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39784232","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 1
Olfactory-Specific Quality of Life Outcomes after Endoscopic Endonasal Surgery of the Sella. 鼻内窥镜手术后嗅觉特异性生活质量结果。
IF 2.2 Q3 Medicine Pub Date : 2021-10-29 eCollection Date: 2021-01-01 DOI: 10.1177/21526567211045041
Milap D Raikundalia, Ryan J Huang, Lyndon Chan, Tracy Truong, Maragatha Kuchibhatla, James Merchant, Ralph Abi Hachem, Patrick J Codd, Ali R Zomorodi, Jordan I Teitelbaum, Bradley J Goldstein, David W Jang

Objective: To assess olfactory outcomes as measured by an olfactory-specific quality of life (QOL) questionnaire in patients undergoing EESBS for sellar lesions.

Design: Retrospective case series.

Setting: Tertiary academic medical center.

Participants: In total, 36 patients undergoing EESBS for lesions limited to the sella were evaluated.

Main outcome measures: The following were performed before and three months after surgery: 22-Item Sinonasal Outcomes Test (SNOT-22), University of Pennsylvania Smell Identification Test (UPSIT), and the Assessment of Self-reported Olfactory Functioning (ASOF), which has three domains: subjective olfactory capability scale (SOC), smell-related problems (SRP), and olfactory-related quality of life (ORQ).

Results: Median age at surgery was 52.5 years, with a median tumor size of 1.8 cm (range: 0.2 to 3.9 cm). Pre- and postoperative median scores were 35 [34, 36.2] and 34.5 [32, 36] for UPSIT, 21 [7.5, 33.5] and 21.5 [6.8, 35.7] for SNOT-22, 10 [9, 10] and 9 [8, 10] for ASOF-SOC, 5 [4.8, 5] and 4.5 [4, 5] for ASOF-SRP, and 5 [5, 5] and 5 [4.5, 5] for ASOF-ORQ. There was no significant change in the two of the three domains of the ASOF. Correlation between ASOF and UPSIT scores were weak. Older age and larger tumor size were associated with worsened olfaction after surgery.

Conclusions: Patients did not experience significant changes in olfactory-specific QOL three months after EESBS, as measured by two domains of the ASOF. The ASOF may serve as a useful adjunctive tool for assessing olfaction after surgery. The lack of correlation between UPSIT and ASOF suggests the need for more research in subjective olfactory-related quality of life after surgery.

目的:通过嗅觉特异性生活质量(QOL)问卷评估鞍区病变患者接受EESBS治疗的嗅觉结果。设计:回顾性病例系列。环境:三级学术医疗中心。参与者:总共评估了36例接受EESBS治疗仅限于鞍区病变的患者。主要观察指标:术前和术后3个月分别进行22项鼻腔结局测试(SNOT-22)、宾夕法尼亚大学嗅觉识别测试(UPSIT)和自我报告嗅觉功能评估(ASOF),包括主观嗅觉能力量表(SOC)、嗅觉相关问题(SRP)和嗅觉相关生活质量(ORQ)三个领域。结果:手术时中位年龄为52.5岁,中位肿瘤大小为1.8 cm(范围:0.2 ~ 3.9 cm)。UPSIT的术前和术后中位评分分别为35分[34,36.2]和34.5分[32,36],SNOT-22的中位评分为21分[7.5,33.5]和21.5分[6.8,35.7],ASOF-SOC的中位评分为10分[9,10]和9分[8,10],ASOF-SRP的中位评分为5分[4.8,5]和4.5分[4,5],ASOF-ORQ的中位评分为5分[5,5]和5分[4.5,5]。在ASOF的三个领域中,这两个领域没有明显的变化。ASOF与UPSIT评分相关性较弱。年龄越大和肿瘤大小越大与术后嗅觉恶化有关。结论:通过ASOF的两个域测量,EESBS后3个月患者的嗅觉特异性生活质量没有显著变化。ASOF可以作为评估术后嗅觉的有用辅助工具。UPSIT和ASOF之间缺乏相关性,这表明需要对术后主观嗅觉相关的生活质量进行更多的研究。
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引用次数: 4
The Successful Vaccination of an IVIgG Naive CVID Patient with an mRNA COVID-19 Vaccine. mRNA - COVID-19疫苗成功接种一名IVIgG初治CVID患者
IF 2.2 Q3 Medicine Pub Date : 2021-10-06 eCollection Date: 2021-01-01 DOI: 10.1177/21526567211049744
Maaz Jalil, John M Abraham, Robert Hostoffer

Introduction: Different subtypes of vaccines have been developed to help protect populations from COVID-19. Currently, three vaccines have been authorized by the United States Food and Drug Administration for emergency use to combat the COVID-19 pandemic. With COVID-19 vaccination rates increasing, it is important to know whether immunodeficient patients have the capacity to mount an immune response with the available vaccines.

Case report: A 78-year-old female with Common Variable Immunodeficiency and anti-IgA antibodies who is naïve to IVIgG treatment responded positively to a COVID-19 mRNA vaccine. Successful seroconversion was proved by having positive COVID-19 spike protein IgG antibodies weeks after the vaccination. Her recent IgG, IgA, and IgM levels were all significantly reduced. Previously, she had no response to the polysaccharide pneumococcal vaccine, but did maintain titers afterTdap vaccination.

Discussion: Immunodeficient patients are a susceptible population during a pandemic. Unfortunately, there is a paucity of research on the infectivity, vaccination, and outcome of these patients during the COVID-19 outbreak. Our patient with CVID was able to respond to protein/toxoid vaccines, but did not respond to polysaccharide pneumococcal vaccine. After inoculation with an mRNA COVID-19 vaccine she was able to create COVID-19 spike protein IgG antibodies.

Conclusion: We present a case of successful vaccination to COVID-19 by an mRNA vaccine in an IVIgG naïve CVID patient.

已经开发了不同亚型的疫苗,以帮助保护人群免受COVID-19的侵害。目前,美国食品和药物管理局已批准三种疫苗紧急使用,以应对COVID-19大流行。随着COVID-19疫苗接种率的提高,了解免疫缺陷患者是否有能力利用现有疫苗产生免疫反应非常重要。病例报告:一名78岁女性,患有常见可变免疫缺陷和抗iga抗体,对IVIgG治疗naïve,对COVID-19 mRNA疫苗反应积极。在接种疫苗数周后,检测到COVID-19刺突蛋白IgG抗体阳性,证明血清转化成功。近期IgG、IgA、IgM水平均明显降低。以前,她对多糖肺炎球菌疫苗没有反应,但在接种tdap疫苗后确实维持了滴度。讨论:免疫缺陷患者是大流行期间的易感人群。不幸的是,在2019冠状病毒病暴发期间,缺乏对这些患者的传染性、疫苗接种和结果的研究。本例CVID患者对蛋白/类毒素疫苗有应答,但对多糖肺炎球菌疫苗无应答。接种mRNA COVID-19疫苗后,她能够产生COVID-19刺突蛋白IgG抗体。结论:我们报告了一例IVIgG naïve CVID患者通过mRNA疫苗成功接种COVID-19的病例。
{"title":"The Successful Vaccination of an IVIgG Naive CVID Patient with an mRNA COVID-19 Vaccine.","authors":"Maaz Jalil,&nbsp;John M Abraham,&nbsp;Robert Hostoffer","doi":"10.1177/21526567211049744","DOIUrl":"https://doi.org/10.1177/21526567211049744","url":null,"abstract":"<p><strong>Introduction: </strong>Different subtypes of vaccines have been developed to help protect populations from COVID-19. Currently, three vaccines have been authorized by the United States Food and Drug Administration for emergency use to combat the COVID-19 pandemic. With COVID-19 vaccination rates increasing, it is important to know whether immunodeficient patients have the capacity to mount an immune response with the available vaccines.</p><p><strong>Case report: </strong>A 78-year-old female with Common Variable Immunodeficiency and anti-IgA antibodies who is naïve to IVIgG treatment responded positively to a COVID-19 mRNA vaccine. Successful seroconversion was proved by having positive COVID-19 spike protein IgG antibodies weeks after the vaccination. Her recent IgG, IgA, and IgM levels were all significantly reduced. Previously, she had no response to the polysaccharide pneumococcal vaccine, but did maintain titers afterTdap vaccination.</p><p><strong>Discussion: </strong>Immunodeficient patients are a susceptible population during a pandemic. Unfortunately, there is a paucity of research on the infectivity, vaccination, and outcome of these patients during the COVID-19 outbreak. Our patient with CVID was able to respond to protein/toxoid vaccines, but did not respond to polysaccharide pneumococcal vaccine. After inoculation with an mRNA COVID-19 vaccine she was able to create COVID-19 spike protein IgG antibodies.</p><p><strong>Conclusion: </strong>We present a case of successful vaccination to COVID-19 by an mRNA vaccine in an IVIgG naïve CVID patient.</p>","PeriodicalId":45192,"journal":{"name":"Allergy & Rhinology","volume":null,"pages":null},"PeriodicalIF":2.2,"publicationDate":"2021-10-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/f7/0e/10.1177_21526567211049744.PMC8514739.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39526108","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 1
Qualitative Olfactory Disorders: Patient Experiences and Self-Management. 定性嗅觉障碍:患者经验和自我管理。
IF 2.2 Q3 Medicine Pub Date : 2021-09-22 eCollection Date: 2021-01-01 DOI: 10.1177/21526567211004251
Carl Philpott, Joanne Dixon, Duncan Boak

Background: Qualitative olfactory disorders in the form of parosmia and phantosmia are very subjective and cannot be measured at present. They pose an unpleasant experience for patients and a therapeutic challenge for clinicians.

Objective: This study aimed to characterise the specific experiences of patients affected by the qualitative symptoms of parosmia and phantosmia including both triggers for symptoms and self-help measures they have tried.

Methods: A cross-sectional survey questionnaire was developed with the input of patient experts within the charity Fifth Sense. The survey was then open online for 3 months to charity members complaining of qualitative symptoms. The survey captured the frequency and impact of symptoms and self-management undertaken. Reflective feedback was also captured from a patient workshop.

Results: There were 100 participants; 61% female, age range 13-88. Common self-reported aetiology included sinonasal disease (17%), idiopathic (33%) and post-viral olfactory loss (26%) and post-traumatic olfactory loss (23%). Parosmia was reported as a daily symptom in 67% compared to 31% for phantosmia; 36% complained of suffering with both symptoms. Only 4% of respondents reported having received any successful treatment for their qualitative symptoms and 58% reported having received no treatment whatsoever. Olfactory training was the most common self-management method reported.

Conclusion: This study illustrates that qualitative disturbances remain problematic for those who experience them due to the duration of symptoms, the relative lack of experience or knowledge amongst medical professionals and the lack of therapeutic options. In future, consideration needs to be given to adaptation and coping strategies to help patients deal with these symptoms.

背景:以嗅觉缺失和幻觉为形式的定性嗅觉障碍是非常主观的,目前还无法测量。它们给患者带来了不愉快的体验,对临床医生来说也是治疗上的挑战。目的:本研究的目的是表征的具体经验的患者影响的质性症状和幻影包括触发症状和自救措施,他们已经尝试。方法:在慈善机构第五感的病人专家的投入下,制定了一份横断面调查问卷。该调查随后在网上进行,为期3个月,向抱怨定性症状的慈善机构成员开放。调查记录了症状出现的频率和影响以及所进行的自我管理。反思性反馈也从患者研讨会中获得。结果:共100人;61%为女性,年龄13-88岁。常见的自我报告病因包括鼻窦疾病(17%)、特发性(33%)、病毒后嗅觉丧失(26%)和创伤后嗅觉丧失(23%)。67%的人报告说,缺失症是一种日常症状,而幻影症的这一比例为31%;36%的人抱怨患有这两种症状。只有4%的答复者报告其定性症状得到了任何成功的治疗,58%的答复者报告没有得到任何治疗。嗅觉训练是最常见的自我管理方法。结论:这项研究表明,由于症状持续时间长,医疗专业人员相对缺乏经验或知识,以及缺乏治疗选择,对那些经历过定性障碍的人来说仍然是一个问题。未来,需要考虑适应和应对策略,以帮助患者处理这些症状。
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引用次数: 10
Idiopathic Anaphylaxis: A Diagnosis of Exclusion. 特发性过敏反应:排除诊断。
IF 2.2 Q3 Medicine Pub Date : 2021-09-13 eCollection Date: 2021-01-01 DOI: 10.1177/21526567211041925
Anjeanette Mendez, Barry J Pelz

We report the case of a 67-year-old female with hypertension and rheumatoid arthritis who had 5 unprovoked episodes of anaphylaxis in an 18-month period of time. We review idiopathic anaphylaxis, including its definition, diagnostic work-up, and differential diagnosis.

我们报告的情况下,一个67岁的女性高血压和类风湿关节炎谁有5无因发作的过敏反应在18个月的时间。我们回顾特发性过敏反应,包括其定义,诊断检查和鉴别诊断。
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引用次数: 0
期刊
Allergy & Rhinology
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