The Use of Targeted Monoclonal Antibodies in the Treatment of ABPA—A Case Series

Allergic bronchopulmonary aspergillosis (ABPA) is a pulmonary disorder occurring in response to Aspergillus fumigatus that can complicate the course of asthma and cystic fibrosis. Here we present a case of acute ABPA without central bronchiectasis, a case of chronic active ABPA with central bronchiectasis, and a case of severe relapsing ABPA with central bronchiectasis. All three were initially treated with corticosteroids and antifungal agents but had an incomplete response. These patients were then treated with anti-IgE therapy with omalizumab before being switched to the anti-IL5R agent benralizumab. They responded well to both agents. These case reports highlight the potential role of omalizumab and benralizumab in the treatment of ABPA, but further studies are required to evaluate the effectiveness of these medications. Longer follow-up periods and objective measurements of the impact of treatment are necessary.

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Potential role of the cellular allergen stimulation test (CAST) in diagnosis of allergic bronchopulmonary aspergillosis (ABPA) in cystic fibrosis

Pediatric Pulmonology ◽

10.1002/ppul.20572 ◽

2007 ◽

Vol 42 (4) ◽

pp. 314-318 ◽

Cited By ~ 11

Author(s):

Stephanie Ringer ◽

Uta-Christina Hipler ◽

Peter Elsner ◽

Felix Zintl ◽

Jochen Mainz

Keyword(s):

Cystic Fibrosis ◽

Potential Role ◽

Allergic Bronchopulmonary Aspergillosis ◽

Stimulation Test

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Beyond the Guidelines: Treatment of Allergic Bronchopulmonary Aspergillosis in Cystic Fibrosis

Annals of Pharmacotherapy ◽

10.1177/10600280211022065 ◽

2021 ◽

pp. 106002802110220

Author(s):

Madeline G. Tompkins ◽

Rebecca Pettit

Keyword(s):

Cystic Fibrosis ◽

Clinical Practice ◽

Alternative Treatment ◽

Case Reports ◽

Allergic Bronchopulmonary Aspergillosis ◽

High Risk Patient ◽

Case Series ◽

Care Plan ◽

Treatment Modalities ◽

Treatment Approaches

Objective: To review the available literature addressing alternative allergic bronchopulmonary aspergillosis (ABPA) treatment options for patients with cystic fibrosis (CF). Data Sources: A literature search of PubMed was performed (January 2002 to April 2021) using the following search terms: allergic bronchopulmonary aspergillosis, aspergillus-related lung disease, cystic fibrosis. Manufacturer prescribing information, clinical practice guidelines, and data from ClinicalTrials.gov were incorporated in the reviewed data. Study Selection and Data Extraction: Relevant English-language studies or those conducted in humans were considered for inclusion. Data Synthesis: Available literature for alternative ABPA treatments in CF is lacking randomized controlled trials, but there is considerable support in case reports and case series describing the benefits in pediatric and adult patients. Recent literature has begun to explore the place in therapy for novel, corticosteroid-sparing treatment approaches. The alternative therapies summarized in this review all resulted in clinical improvement and subsequent discontinuation or dose reductions of oral corticosteroids, with minimal reported adverse drug effects. Relevance to Patient Care and Clinical Practice: Although corticosteroids are the cornerstone of ABPA management, the toxicities can be significant limitations in an already high-risk patient population. Patients may fail or become intolerant to guideline-recommended therapies and require alternative treatment approaches. Conclusions: Alternative treatment modalities for ABPA in patients with CF, including azole antifungals, pulsed intravenous glucocorticoids, omalizumab, mepolizumab, and inhaled amphotericin, appear to be efficacious and well tolerated. Pharmacological properties including route of administration, storage and stability, beyond use dating, and adverse effects of the various treatment modalities must be considered when selecting a practical care plan for patients.

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Use of monoclonal antibodies for allergic bronchopulmonary aspergillosis in patients with asthma and cystic fibrosis: literature review

Therapeutic Advances in Respiratory Disease ◽

10.1177/1753466620961648 ◽

2020 ◽

Vol 14 ◽

pp. 175346662096164 ◽

Cited By ~ 1

Author(s):

Isabel C. Eraso ◽

Saveria Sangiovanni ◽

Eliana I. Morales ◽

Liliana Fernández-Trujillo

Keyword(s):

Cystic Fibrosis ◽

Literature Review ◽

Case Reports ◽

Scientific Evidence ◽

Allergic Bronchopulmonary Aspergillosis ◽

Case Series ◽

Lung Damage ◽

Biological Drugs ◽

Significant Clinical Improvement ◽

Sparing Effect

Allergic bronchopulmonary aspergillosis (ABPA) is a hypersensitivity reaction (HR) mediated by antigens to Aspergillus fumigatus. It is estimated that 2–15% of patients with cystic fibrosis (CF) and between 1% and 5% of asthmatics develop ABPA, affecting approximately 4.8 million people worldwide. The goals of treatment are controlling inflammation, reducing the number of exacerbations and limiting the progression of lung damage. Systemic steroids are therefore used as the mainstay therapy, along with antifungal medications. However, many patients do not respond or develop side effects to treatment. In this scenario, biological drugs such as Omalizumab, Mepolizumab, Benralizumab and Dupilumab have been implemented in clinical practice, even though there is a lack of scientific evidence to support their use. We performed a literature review of the studies carried out which analyzed biologics for the management of ABPA in adult populations with asthma and CF. To our knowledge this is the first literature review that included all biologics. We included a total of 32 studies, all but one were descriptive studies, and the vast majority evaluated the use of Omalizumab. Biologics appeared to have more benefit for patients with ABPA and asthma than CF, specifically at decreasing the frequency of acute exacerbations and by having a steroid-sparing effect. Although a decrease in serum IgE level is considered a measure of therapy success, values may not decline as expected in the context of a significant clinical improvement, highlighting the importance of measuring patient-oriented outcomes. As evidence comes mainly from case series and case reports, randomized controlled trials are needed to evaluate further the safety and efficacy of biologics in ABPA. The reviews of this paper are available via the supplemental material section.

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Pneumonia-related pneumatoceles in infants: CT assessment and image-guided treatment

BJR|case reports ◽

10.1259/bjrcr.20210191 ◽

2021 ◽

Author(s):

Elisa Mercanzin ◽

Pietro Andrea Bonaffini ◽

Antonino Barletta ◽

Francesco Stanco ◽

Clarissa Valle ◽

...

Keyword(s):

Percutaneous Drainage ◽

Potential Role ◽

Scientific Literature ◽

Case Series ◽

Consensus Guidelines ◽

Successful Management ◽

Image Guided ◽

Life Threatening

Lung pneumatoceles represent a potential life-threatening complication of pneumonia in infants, especially when they do not spontaneously reabsorb. In this category of patients, scientific literature lacks and no consensus guidelines for management have been proposed. Imaging plays a key role in the diagnosis, characterization, and follow-up of pneumatoceles. Image-guided percutaneous drainage can be considered a safe and effective treatment in children, although it is not widely recognized in newborns and infants. The aim of this case series review is to describe the main CT features of complicated or persistent pneumatoceles in infants and to highlight the potential role of image-guided percutaneous drainage as an effective approach for their treatment. Successful management of four infants affected by pneumonia-related pneumatoceles with percutaneous drainage is presented.

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Mechanical thrombectomy in pediatric stroke: systematic review, individual patient data meta-analysis, and case series

Journal of Neurosurgery Pediatrics ◽

10.3171/2019.5.peds19126 ◽

2019 ◽

Vol 24 (5) ◽

pp. 558-571 ◽

Cited By ~ 3

Author(s):

Kartik Bhatia ◽

Hans Kortman ◽

Christopher Blair ◽

Geoffrey Parker ◽

David Brunacci ◽

...

Keyword(s):

Systematic Review ◽

Ischemic Stroke ◽

Acute Ischemic Stroke ◽

Mechanical Thrombectomy ◽

Case Reports ◽

Meta Analysis ◽

Case Series ◽

Neurological Outcomes

OBJECTIVEThe role of mechanical thrombectomy in pediatric acute ischemic stroke is uncertain, despite extensive evidence of benefit in adults. The existing literature consists of several recent small single-arm cohort studies, as well as multiple prior small case series and case reports. Published reports of pediatric cases have increased markedly since 2015, after the publication of the positive trials in adults. The recent AHA/ASA Scientific Statement on this issue was informed predominantly by pre-2015 case reports and identified several knowledge gaps, including how young a child may undergo thrombectomy. A repeat systematic review and meta-analysis is warranted to help guide therapeutic decisions and address gaps in knowledge.METHODSUsing PRISMA-IPD guidelines, the authors performed a systematic review of the literature from 1999 to April 2019 and individual patient data meta-analysis, with 2 independent reviewers. An additional series of 3 cases in adolescent males from one of the authors’ centers was also included. The primary outcomes were the rate of good long-term (mRS score 0–2 at final follow-up) and short-term (reduction in NIHSS score by ≥ 8 points or NIHSS score 0–1 at up to 24 hours post-thrombectomy) neurological outcomes following mechanical thrombectomy for acute ischemic stroke in patients < 18 years of age. The secondary outcome was the rate of successful angiographic recanalization (mTICI score 2b/3).RESULTSThe authors’ review yielded 113 cases of mechanical thrombectomy in 110 pediatric patients. Although complete follow-up data are not available for all patients, 87 of 96 (90.6%) had good long-term neurological outcomes (mRS score 0–2), 55 of 79 (69.6%) had good short-term neurological outcomes, and 86 of 98 (87.8%) had successful angiographic recanalization (mTICI score 2b/3). Death occurred in 2 patients and symptomatic intracranial hemorrhage in 1 patient. Sixteen published thrombectomy cases were identified in children < 5 years of age.CONCLUSIONSMechanical thrombectomy may be considered for acute ischemic stroke due to large vessel occlusion (ICA terminus, M1, basilar artery) in patients aged 1–18 years (Level C evidence; Class IIb recommendation). The existing evidence base is likely affected by selection and publication bias. A prospective multinational registry is recommended as the next investigative step.

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Neurological symptoms, manifestations, and complications associated with severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) and coronavirus disease 19 (COVID-19)

Journal of Neurology ◽

10.1007/s00415-021-10406-y ◽

2021 ◽

Author(s):

Biyan Nathanael Harapan ◽

Hyeon Joo Yoo

Keyword(s):

Severe Acute Respiratory Syndrome ◽

Case Reports ◽

Case Series ◽

Altered Mental Status ◽

Neurological Complications ◽

Neurological Symptoms ◽

Neurological Manifestations ◽

Fisher Syndrome ◽

Novel Coronavirus ◽

The Impact

AbstractSevere acute respiratory syndrome coronavirus 2 (SARS-CoV-2), a novel coronavirus, is responsible for the outbreak of coronavirus disease 19 (COVID-19) and was first identified in Wuhan, China in December 2019. It is evident that the COVID-19 pandemic has become a challenging world issue. Although most COVID-19 patients primarily develop respiratory symptoms, an increasing number of neurological symptoms and manifestations associated with COVID-19 have been observed. In this narrative review, we elaborate on proposed neurotropic mechanisms and various neurological symptoms, manifestations, and complications of COVID-19 reported in the present literature. For this purpose, a review of all current published literature (studies, case reports, case series, reviews, editorials, and other articles) was conducted and neurological sequelae of COVID-19 were summarized. Essential and common neurological symptoms including gustatory and olfactory dysfunctions, myalgia, headache, altered mental status, confusion, delirium, and dizziness are presented separately in sections. Moreover, neurological manifestations and complications that are of great concern such as stroke, cerebral (sinus) venous thrombosis, seizures, meningoencephalitis, Guillain–Barré syndrome, Miller Fisher syndrome, acute myelitis, and posterior reversible encephalopathy syndrome (PRES) are also addressed systematically. Future studies that examine the impact of neurological symptoms and manifestations on the course of the disease are needed to further clarify and assess the link between neurological complications and the clinical outcome of patients with COVID-19. To limit long-term consequences, it is crucial that healthcare professionals can early detect possible neurological symptoms and are well versed in the increasingly common neurological manifestations and complications of COVID-19.

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Salvage therapy of bleeding esophageal tumor by fully covered self-expandable metallic stent: A case report

SAGE Open Medical Case Reports ◽

10.1177/2050313x21997198 ◽

2021 ◽

Vol 9 ◽

pp. 2050313X2199719

Author(s):

Sundus Bilal ◽

Saad Muhammad Saeed ◽

Muhammad Zeeshan Siddique ◽

Muhammad Saqib ◽

Shafqat Mehmood ◽

...

Keyword(s):

Case Reports ◽

Esophageal Tumor ◽

Metallic Stent ◽

Diverse Group ◽

Expandable Metallic Stent ◽

Lost To Follow Up ◽

Expandable Metallic Stents ◽

Tamponade Effect

The role of self-expandable metallic stents is gradually evolving for a diverse group of benign and malignant gastrointestinal tract problems, with luminal obstruction being by far the most common. Although its role in refractory variceal bleeding is well established, it has rarely been tried for tumor-related bleeding, with only a few case reports in this regard. We share our experience of successfully controlling esophageal tumor–related bleeding with the use of a fully covered self-expandable metallic stent. A 58-year-old woman with irresectable distal esophageal cancer, presented with hematemesis. Esophago-gastro-duodenoscopy revealed an obstructing esophageal tumor with diffuse oozing of blood. This was unamenable to local injection of adrenaline and hemospray; therefore, a temporary self-expandable metallic stent was parked to create a tamponade effect. This successfully stopped bleeding and the patient remained asymptomatic till discharge. However, she was lost to follow-up, and therefore, the stent was removed after a period of 5 months instead of 2 weeks.

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Role of Radiotherapy in Recurrent Intra-Abdominal Yolk Sac Tumor

Case Reports in Oncology ◽

10.1159/000517022 ◽

2021 ◽

pp. 1010-1018

Author(s):

Marhendra Satria Utama ◽

Andi Kurniadi ◽

A.A. Citra Yunda Prahastiwi ◽

Antony A. Adibrata

Keyword(s):

Case Reports ◽

Yolk Sac ◽

Yolk Sac Tumor ◽

Appropriate Treatment ◽

Curative Radiotherapy ◽

Chief Complaints ◽

Abdominal Mri ◽

Malignant Germ Cell Tumor

Yolk sac tumor (YST) is a rare malignant germ cell tumor with no appropriate treatment strategy to date. However, patients are treated on a case-to-case basis as per various case reports that have been published. Here, we present a case of 27-year-old female patient who presented to us with chief complaints of severe abdominal pain associated with leucorrhea. She previously had a similar pain episode, which was then evaluated by a multidisciplinary team. She was diagnosed with YST. After that, she underwent 6 cycles of chemotherapy, but there was no improvement. Then the medical oncologist referred her to performed radiotherapy. Then, the radiation oncologist decided to give her curative radiotherapy of 3D-CRT. After completing her sessions, she felt better and clinically improving. After that, she was discharged and scheduled a follow-up visit for first evaluation. At her follow-up visit, she was feeling well, and we decided to have an abdominal MRI.

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Pallidal stimulation as treatment for camptocormia in Parkinson’s disease

npj Parkinson s Disease ◽

10.1038/s41531-020-00151-w ◽

2021 ◽

Vol 7 (1) ◽

Author(s):

Yijie Lai ◽

Yunhai Song ◽

Daoqing Su ◽

Linbin Wang ◽

Chencheng Zhang ◽

...

Keyword(s):

Parkinson’S Disease ◽

Parkinson's Disease ◽

Case Reports ◽

Structural Connectivity ◽

Video Recordings ◽

Lead Placement ◽

Pallidal Stimulation ◽

The Impact ◽

Deep Brain

AbstractCamptocormia is a common and often debilitating postural deformity in Parkinson’s disease (PD). Few treatments are currently effective. Deep brain stimulation (DBS) of the globus pallidus internus (GPi) shows potential in treating camptocormia, but evidence remains limited to case reports. We herein investigate the effect of GPi-DBS for treating camptocormia in a retrospective PD cohort. Thirty-six consecutive PD patients who underwent GPi-DBS were reviewed. The total and upper camptocormia angles (TCC and UCC angles) derived from video recordings of patients who received GPi-DBS were used to compare camptocormia alterations. Correlation analysis was performed to identify factors associated with the postoperative improvements. DBS lead placement and the impact of stimulation were analyzed using Lead-DBS software. Eleven patients manifested pre-surgical camptocormia: seven had lower camptocormia (TCC angles ≥ 30°; TCC-camptocormia), three had upper camptocormia (UCC angles ≥ 45°; UCC-camptocormia), and one had both. Mean follow-up time was 7.3 ± 3.3 months. GPi-DBS improved TCC-camptocormia by 40.4% (angles from 39.1° ± 10.1° to 23.3° ± 8.1°, p = 0.017) and UCC-camptocormia by 22.8% (angles from 50.5° ± 2.6° to 39.0° ± 6.7°, p = 0.012). Improvement in TCC angle was positively associated with pre-surgical TCC angles, levodopa responsiveness of the TCC angle, and structural connectivity from volume of tissue activated to somatosensory cortex. Greater improvement in UCC angles was seen in patients with larger pre-surgical UCC angles. Our study demonstrates potential effectiveness of GPi-DBS for treating camptocormia in PD patients. Future controlled studies with larger numbers of patients with PD-related camptocormia should extend our findings.

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A Comprehensive Review Evaluating the Impact of Protein Source (Vegetarian vs. Meat Based) in Hepatic Encephalopathy

Nutrients ◽

10.3390/nu13020370 ◽

2021 ◽

Vol 13 (2) ◽

pp. 370

Author(s):

Umair Iqbal ◽

Ravirajsinh N. Jadeja ◽

Harshit S. Khara ◽

Sandeep Khurana

Keyword(s):

Hepatic Encephalopathy ◽

Protein Intake ◽

Alternative Therapy ◽

Case Series ◽

Current Evidence ◽

Branch Chain Amino ◽

The Impact ◽

Protein Diets

Hepatic encephalopathy (HE) is a common neurological consequence in patients with cirrhosis and has a healthcare burden of USD 5370 to 50,120 per patient annually. HE significantly hampers the quality of life and is a major cause of morbidity and mortality. Patients with cirrhosis are at a high risk for protein-calorie malnutrition due to altered metabolism. Current evidence has changed the old belief of protein restriction in patients with cirrhosis and now 1.2 to 1.5 g/kg/day protein intake is recommended. Case series and studies with small numbers of participants showed that a vegetarian protein diet decreases the symptoms of HE when compared to a meat-based diet, but the evidence is limited and requires further larger randomized controlled trials. However, vegetable or milk-based protein diets are good substitutes for patients averse to meat intake. Branch chain amino acids (BCAA) (leucine, isoleucine and valine) have also been shown to be effective in alleviating symptoms of HE and are recommended as an alternative therapy in patients with cirrhosis for the treatment of HE. In this review, we provide an overview of current literature evaluating the role of protein intake in the management of HE in cirrhosis.

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