scholarly journals A Report of Two Patients with Food Protein-induced Enterocolitis Mimicking Bartter Syndrome

2021 ◽  
Author(s):  
Sepideh Darougar ◽  
Arezoo Shirzani ◽  
Mahboubeh Mansouri ◽  
Masoumeh Mohkam
Keyword(s):  
Clinical Symptoms ◽  
Diagnostic Delay ◽  
Bartter Syndrome ◽  
Response To Treatment ◽  
Food Protein ◽  
Prevalent Disease ◽  
History Of ◽  
Ige Mediated ◽  
Multiple Episodes ◽  
Shed Light

Food protein-induced enterocolitis syndrome (FPIES), is a non-IgE mediated food allergy presenting in infants younger than 12 months. Diagnostic delay may occur due to overlapping clinical symptoms with several conditions. Here, we present two cases of FPIES, mistakenly diagnosed and treated as Bartter syndrome. This study aims to emphasize the several features of this syndrome that may mimic other diagnoses and sometimes leading to near-death events due to delay in the diagnosis and improper treatment. The first patient was a 30-month-old boy with multiple episodes of profuse vomiting and diarrhea within 1 hour after breastfeeding, beginning from the first month of life progressing to hypokalemia and metabolic alkalosis at the age of 5 months leading to the diagnosis of Bartter syndrome. The second patient had a history of unremitting diarrhea which had been started soon after his first breastfeeding followed by biliary vomiting on the 7th day of life. He was treated in another hospital for neonatal sepsis, however, without an appropriate response to treatment. To conclude, despite the current belief on the rarity of FPIES, it is a more prevalent disease than expected with various non-specific manifestations imitating other conditions which may result in diagnostic delay and sometimes fatalities. To shed light on the importance of the physicians’ awareness of this syndrome, these two cases are presented here as examples of FPIES imitating other disorders.

Phenotypes and natural history of food protein‐induced enterocolitis syndrome in the east Mediterranean region

2020 ◽  
Vol 41 (6) ◽  
pp. 420-427 ◽  
Author(s):  
Melike Ocak ◽  
Ayşegül Akarsu ◽  
Umit Murat Sahiner ◽  
Ozge Soyer ◽  
Bulent Enis Sekerel
Keyword(s):  
Clinical Symptoms ◽  
Emergency Department Visits ◽  
Interquartile Range ◽  
Cow’S Milk ◽  
Food Protein ◽  
Cow's Milk ◽  
Hen’S Egg ◽  
History Of ◽  
Ige Mediated ◽  
East Mediterranean Region

Background: Food protein‐induced enterocolitis syndrome (FPIES) is a rare non-IgE mediated food allergy. Objective: To delineate the differences in the spectrum of culprit foods, remission patterns, and predictors among varying cultures. Methods: We reviewed demographics, culprit foods, outcomes, and predictors in 81 children with a diagnosis of FPIES who were followed up between 2015 and 2020. Results: Eighty-one patients (55.6% boys) were enrolled, including 72 with acute FPIES and 9 with chronic FPIES. Hen’s egg was the most common culprit food (36.6%), followed by fish (26.9%), and cow’s milk (21.5%). Interestingly, cow’s milk was significantly prevalent in chronic FPIES cases (p = 0.006). The most common clinical symptoms were vomiting (100%), pallor (63.4%), and lethargy (55.9%). Emergency department visits were noted in 39 patients (41.9%), of whom 37 (39.8%) were treated with intravenous (IV) fluid. The subjects were followed up for a median (interquartile range) of 19.4 months (12.3‐41.2 months), and 26 subjects (32.1%) achieved tolerance. The median (interquartile range) age at tolerance was 2.5 years (2.1‐3.2 years). With regard to the culprit foods, hen’s egg was observed more frequently in the subjects with resolved FPIES cases (p = 0.008), whereas fish FPIES cases were high in the persistent group (p = 0.001). IgE sensitization of the culprit food was found to be an independent risk factor for the persistence of FPIES (odds ratio 4.855 [95% confidence interval, 1.131‐20.844]; p = 0.034). Conclusion: In our cohort, unlike other published series, hen’s egg and fish were the two most common culprit foods. Fish differed from other culprit foods, with significantly delayed onset and persistence, and may create a model that allows for the understanding of the disease.

scholarly journals Adults with possible food protein-induced enterocolitis syndrome with crustacean ingestion

2020 ◽  
Vol 16 (1) ◽  
Author(s):  
Daniel H. Li ◽  
Andrew Wong-Pack ◽  
Andrea Leilani Macikunas ◽  
Harold Kim
Keyword(s):  
Time Course ◽  
Immunoglobulin E ◽  
Gastrointestinal Symptoms ◽  
Case Series ◽  
Retrospective Chart Review ◽  
Food Protein ◽  
Female Predominance ◽  
Adolescents And Adults ◽  
History Of ◽  
Ige Mediated

Abstract Background Food protein-induced enterocolitis (FPIES), an entity previously thought to only affect children, has been increasingly described in adults. In this study, we report a Canadian cohort of 19 adolescents and adults with recurrent non-immunoglobulin E (IgE)-mediated gastrointestinal symptoms after crustacean ingestion, consistent with FPIES. Methods We conducted a retrospective chart review of patients in an outpatient allergy clinic from January 2005 to May 2020. Electronic records were searched using keywords for crustaceans and for symptoms consistent with FPIES. We included patients with gastrointestinal symptoms specifically to crustaceans on more than one occasion, who were 14 years or older at the time of index reaction. Exclusion criteria included symptoms suggestive of an IgE-mediated anaphylactic reaction or a likely alternative diagnosis. We identified 19 patients for our cohort who met the criteria. Results Our cohort was 68.4% female (13) and 32.6% (6) male. The average age at first reaction to crustaceans was 34 years old with a range of 14–68 years (median = 28 years; IQR = 32 years). Time from ingestion to beginning of symptoms ranged from 3 min to 6.5 h, with an average of 2.8 h (median = 2 h; IQR = 3.25 h). Duration of reaction ranged from less than a minute to over 48 h, with a mean of 9.4 h (median = 4 h; IQR = 7.75 h). Patients had 4.8 reactions on average; however, number of reactions ranged from 2 to 12.5 (median = 3, IQR = 3). All patients identified a “trigger” food in the crustacean group, and 12 subjects identified additional reactions to other seafood. Conclusions This case series will better characterize and advance our understanding of this disease entity in adults. There are key differences in the presentation of FPIES in adults compared to children, namely female predominance, difference in solid food trigger, and unpredictable time course. Future studies are needed to examine the pathophysiology and natural history of adult FPIES. Specific guidelines should be developed for the diagnosis and management in adults. Trial registration: retrospectively registered.

scholarly journals Marshall Syndrome or PFAPA (Periodic Fever, Aphthous Stomatitis, Pharyngitis, Adenopathy) Syndrome

2017 ◽  
Vol 34 (4) ◽  
pp. 222-224
Author(s):  
Most Umme Habiba Begum
Keyword(s):  
Clinical Symptoms ◽  
Periodic Fever ◽  
Cervical Lymphadenopathy ◽  
Aphthous Stomatitis ◽  
Response To Treatment ◽  
Pharyngeal Wall ◽  
H2 Blocker ◽  
Cervical Adenitis ◽  
First Case ◽  
History Of

Marshall Syndrome PFAPA (Periodic Fever, Aphthous Stomatitis, Pharyngitis, Adenitis) Syndrome is a chronic condition, typically starting 2-5 years old, in which fever occurs periodically (lasts for 3-7 days), accompanied by aphthous-like ulcers, pharyngitis and/or cervical adenitis. The patients have no clinical symptoms between episodes and it is required to exclude all other diseases before confirming the diagnosis. The dramatic response to treatment with steroid helps diagnosing PFAPA. We are presenting the case of an 8 years male patient, with the history of recurrent episodes of fever (onset at the age of two), oral ulcer and difficulty in deglutition who constantly received antibiotic therapy and or antifungal prescribed by different doctors. Clinically the patient was febrile, mildly pale, cervical lymphadenopathy, aphthous ulcers in the tongue with inflamed tonsils, covered with thick exudates and pharyngeal wall was inflamed and folliculated. There was just palpable liver. Laboratory investigations performed but it was without serological confirmation. Throat swab culture was negative. The child received steroid (Prednisolone) with favorable outcome. Subsequently, the patient presented with similar episodes of fever which disappeared within 24 hours of single dose of prednisolone. After Ranitidine (H2 blocker) prophylaxis the patient remains asymptomatic for about one and half years. With our best knowledge this is the first case report of Marshall Syndrome or PFAPA Syndrome in Bangladesh.J Bangladesh Coll Phys Surg 2016; 34(4): 222-224

scholarly journals Adults with Possible Food Protein-Induced Enterocolitis Syndrome with Crustacean Ingestion

2020 ◽  
Author(s):  
Daniel Hanzhang Li ◽  
Andrew Wong-Pack ◽  
Andrea Leilani Macikunas ◽  
Harold Kim
Keyword(s):  
Time Course ◽  
Immunoglobulin E ◽  
Gastrointestinal Symptoms ◽  
Case Series ◽  
Retrospective Chart Review ◽  
Food Protein ◽  
Female Predominance ◽  
Adolescents And Adults ◽  
History Of ◽  
Ige Mediated

Abstract Background: Food protein-induced enterocolitis (FPIES), an entity previously thought to only affect children, has been increasingly described in adults. In this study, we report a Canadian cohort of 19 adolescents and adults with recurrent non-immunoglobulin E (IgE) -mediated gastrointestinal symptoms after crustacean ingestion, consistent with FPIES.Methods: We conducted a retrospective chart review of patients in an outpatient allergy clinic from January 2005 to May 2020. Electronic records were searched using keywords for crustaceans and for symptoms consistent with FPIES. We included patients with gastrointestinal symptoms specifically to crustaceans on more than one occasion, who were 14 years or older at the time of index reaction. Exclusion criteria included symptoms suggestive of an IgE-mediated anaphylactic reaction or a likely alternative diagnosis. We identified 19 patients for our cohort who met the criteria.Results: Our cohort was 68.4% female (13) and 32.6% (6) male. The average age at first reaction to crustaceans was 34 years old with a range of 14 years to 68 years (median = 28 years; IQR = 32 years). Time from ingestion to beginning of symptoms ranged from 3 minutes to 6.5 hours, with an average of 2.8 hours (median = 2 hours; IQR = 3.25 hours). Duration of reaction ranged from less than a minute to over 48 hours, with a mean of 9.4 hours (median = 4 hours; IQR= 7.75 hours). Patients had 4.8 reactions on average; however, number of reactions ranged from 2 to 12.5 (median = 3, IQR = 3). All patients identified a “trigger” food in the crustacean group, and 12 subjects identified additional reactions to other seafood. Conclusions: We hope our case series will better characterize and advance our understanding of this disease entity in adults. There are key differences in the presentation of FPIES in adults compared to children, namely female predominance, difference in solid food trigger, and unpredictable time course. Future studies are needed to examine the pathophysiology and natural history of adult FPIES. Specific guidelines should be developed for the diagnosis and management in adults.Trial Registration: retrospectively registered

scholarly journals Cytokine Expression in CD3+ Cells in an Infant with Food Protein-Induced Enterocolitis Syndrome (FPIES): Case Report

2009 ◽  
Vol 2009 ◽  
pp. 1-4 ◽  
Author(s):  
F. Mori ◽  
S. Barni ◽  
A. Cianferoni ◽  
N. Pucci ◽  
M. de Martino ◽  
...  
Keyword(s):  
T Cell ◽  
Clinical Symptoms ◽  
Failure To Thrive ◽  
Food Challenge ◽  
Interleukin 4 ◽  
Food Protein ◽  
Double Blind ◽  
Ige Mediated ◽  
Th1 And Th2 Responses ◽  
Cell Expression

Food protein-induced enterocolitis syndrome (FPIES) is a non-IgE-mediated food allergy characterized by severe vomiting, diarrhea, and often failure to thrive in infants. Symptoms typically resolve after the triggering food-derived protein is removed from the diet and recur within few hours after the re-exposure to the causal protein. The diagnosis is based on clinical symptoms and a positive food challenge. In this study, we report a case of FPIES to rice in an 8-month-old boy. We performed a double-blind placebo-controlled food challenge (DBPCFC) to rice and we measured the intracellular T cell expression of interleukin-4 (IL-4); IL-10, and interferon (IFN-) pre-and post-challenge during an acute FPIES reaction and when tolerance to rice had been achieved. For the first time we describe an increase in T cell IL-4 and decrease in IFN- expression after a positive challenge with rice (i.e. rice triggered a FPIES attack) and an increase in T cell IL-10 expression after rice challenge 6 months later after a negative challenge (i.e., the child had acquired tolerance to rice) in an 8 month old with documented FPIES to rice. A Th2 activation associated with high IL-4 levels may contribute to the pathophysiology of the disease. On the other hand, T cell-derived IL-10 may play a role in the acquisition of immunotolerance by regulating the Th1 and Th2 responses.

scholarly journals Adults with possible food protein-induced enterocolitis syndrome with crustacean ingestion

2020 ◽  
Author(s):  
Daniel Hanzhang Li ◽  
Andrew Wong-Pack ◽  
Andrea Leilani Macikunas ◽  
Harold Kim
Keyword(s):  
Time Course ◽  
Immunoglobulin E ◽  
Gastrointestinal Symptoms ◽  
Case Series ◽  
Retrospective Chart Review ◽  
Food Protein ◽  
Female Predominance ◽  
Adolescents And Adults ◽  
History Of ◽  
Ige Mediated

Abstract Background: Food protein-induced enterocolitis (FPIES), an entity previously thought to only affect children, has been increasingly described in adults. In this study, we report a Canadian cohort of 19 adolescents and adults with recurrent non-immunoglobulin E (IgE) -mediated gastrointestinal symptoms after crustacean ingestion, consistent with FPIES. Methods: We conducted a retrospective chart review of patients in an outpatient allergy clinic from January 2005 to May 2020. Electronic records were searched using keywords for crustaceans and for symptoms consistent with FPIES. We included patients with gastrointestinal symptoms specifically to crustaceans on more than one occasion, who were 14 years or older at the time of index reaction. Exclusion criteria included symptoms suggestive of an IgE-mediated anaphylactic reaction or a likely alternative diagnosis. We identified 19 patients for our cohort who met the criteria. Results: Our cohort was 68.4% female (13) and 32.6% (6) male. The average age at first reaction to crustaceans was 34 years old with a range of 14 years to 68 years (median = 28 years; IQR = 32 years). Time from ingestion to beginning of symptoms ranged from 3 minutes to 6.5 hours, with an average of 2.8 hours (median = 2 hours; IQR = 3.25 hours). Duration of reaction ranged from less than a minute to over 48 hours, with a mean of 9.4 hours (median = 4 hours; IQR= 7.75 hours). Patients had 4.8 reactions on average; however, number of reactions ranged from 2 to 12.5 (median = 3, IQR = 3). All patients identified a “trigger” food in the crustacean group, and 12 subjects identified additional reactions to other seafood. Conclusions: This case series will better characterize and advance our understanding of this disease entity in adults. There are key differences in the presentation of FPIES in adults compared to children, namely female predominance, difference in solid food trigger, and unpredictable time course. Future studies are needed to examine the pathophysiology and natural history of adult FPIES. Specific guidelines should be developed for the diagnosis and management in adults. Trial Registration: retrospectively registered

Investigating the Dynamics of Suicidal Ideation

Crisis ◽  
2018 ◽  
Vol 39 (1) ◽  
pp. 65-69 ◽  
Author(s):  
Nina Hallensleben ◽  
Lena Spangenberg ◽  
Thomas Forkmann ◽  
Dajana Rath ◽  
Ulrich Hegerl ◽  
...  
Keyword(s):  
Suicidal Ideation ◽  
Small Sample Size ◽  
Unipolar Depression ◽  
Small Sample ◽  
History Of ◽  
Severity Of Depression ◽  
Ecological Momentary ◽  
Depressive Episodes ◽  
Ema Method ◽  
Shed Light

Abstract. Background: Although the fluctuating nature of suicidal ideation (SI) has been described previously, longitudinal studies investigating the dynamics of SI are scarce. Aim: To demonstrate the fluctuation of SI across 6 days and up to 60 measurement points using smartphone-based ecological momentary assessments (EMA). Method: Twenty inpatients with unipolar depression and current and/or lifetime suicidal ideation rated their momentary SI 10 times per day over a 6-day period. Mean squared successive difference (MSSD) was calculated as a measure of variability. Correlations of MSSD with severity of depression, number of previous depressive episodes, and history of suicidal behavior were examined. Results: Individual trajectories of SI are shown to illustrate fluctuation. MSSD values ranged from 0.2 to 21.7. No significant correlations of MSSD with several clinical parameters were found, but there are hints of associations between fluctuation of SI and severity of depression and suicidality. Limitations: Main limitation of this study is the small sample size leading to low power and probably missing potential effects. Further research with larger samples is necessary to shed light on the dynamics of SI. Conclusion: The results illustrate the dynamic nature and the diversity of trajectories of SI across 6 days in psychiatric inpatients with unipolar depression. Prediction of the fluctuation of SI might be of high clinical relevance. Further research using EMA and sophisticated analyses with larger samples is necessary to shed light on the dynamics of SI.

Component resolved diagnostics in peanut sensitized children with and without a history of clinical reaction

2020 ◽  
Vol 41 (5) ◽  
pp. 336-340
Author(s):  
Yasmin Hamzavi Abedi ◽  
Cristina P. Sison ◽  
Punita Ponda
Keyword(s):  
Retrospective Chart Review ◽  
Clinical History ◽  
Specific Ige ◽  
Exact Test ◽  
Ara H 1 ◽  
Sige Level ◽  
History Of ◽  
Laboratory Procedures ◽  
Ige Mediated ◽  
The Relationship

Background: Serum Peanut-specific-IgE (PN-sIgE) and peanut-component-resolved-diagnostics (CRD) are often ordered simultaneously in the evaluation for peanut allergy. Results often guide the plans for peanut oral challenge. However, the clinical utility of CRD at different total PN-sIgE levels is unclear. A commonly used predefined CRD Ara h2 cutoff value in the literature predicting probability of peanut challenge outcomes is 0.35kUA/L. Objective: To examine the utility of CRD in patients with and without a history of clinical reactivity to peanut (PN). Methods: This was a retrospective chart review of 196 children with PN-sIgE and CRD testing, of which, 98 patients had a clinical history of an IgE-mediated reaction when exposed to PN and 98 did not. The Fisher's exact test was used to assess the relationship between CRD and PN-sIgE at different cutoff levels, McNemar test and Gwet’s approach (AC1 statistic) were used to examine agreement between CRD and PN-sIgE, and logistic regression was used to assess differences in the findings between patients with and without reaction history. Results: Ara h 1, 2, 3, or 9 (ARAH) levels ≤0.35 kUA/L were significantly associated with PN-sIgE levels <2 kUA/L rather than ≥2 kUA/L (p < 0.0001). When the ARAH threshold was increased to 1 kUA/L and 2 kUA/L, these thresholds were still significantly associated with PN-sIgE levels of <2, <5, and <14 kUA/L. These findings were not significantly different in patients with and without a history of clinical reactivity. Conclusion: ARAH values correlated with PN-sIgE. Regardless of clinical history, ARAH levels are unlikely to be below 0.35, 1, or 2 kUA/L if the PN-sIgE level is >2 kUA/L. Thus, if possible, practitioners should consider PN-sIgE rather than automatically ordering CRD with PN-sIgE every time. Laboratory procedures that allow automatically and reflexively adding CRD when the PN-sIgE level is ≤5 kUA/L can be helpful. However, further studies are needed in subjects with challenge-proven PN allergy.

Editorial Introduction

2020 ◽  
Vol 4 (1) ◽  
pp. 1
Author(s):  
Editors of the JIOWS
Keyword(s):  
Nineteenth Century ◽  
Indian Ocean ◽  
Western Indian Ocean ◽  
Late Nineteenth Century ◽  
Fourth Volume ◽  
History Of ◽  
The Indian Ocean ◽  
Complex Relationships ◽  
Indian Ocean World ◽  
Shed Light

The editors are proud to present the first issue of the fourth volume of the Journal of Indian Ocean World Studies. This issue contains three articles, by James Francis Warren (Murdoch University), Kelsey McFaul (University of California, Santa Cruz), and Marek Pawelczak (University of Warsaw), respectively. Warren’s and McFaul’s articles take different approaches to the growing body of work that discusses pirates in the Indian Ocean World, past and present. Warren’s article is historical, exploring the life and times of Julano Taupan in the nineteenth-century Philippines. He invites us to question the meaning of the word ‘pirate’ and the several ways in which Taupan’s life has been interpreted by different European colonists and by anti-colonial movements from the mid-nineteenth century to the present day. McFaul’s article, meanwhile, takes a literary approach to discuss the much more recent phenomenon of Somali Piracy, which reached its apex in the last decade. Its contribution is to analyse the works of authors based in the region, challenging paradigms that have mostly been developed from analysis of works written in the West. Finally, Pawelczak’s article is a legal history of British jurisdiction in mid-late nineteenth-century Zanzibar. It examines one of the facets that underpinned European influence in the western Indian Ocean World before the establishment of colonial rule. In sum, this issue uses two key threads to shed light on the complex relationships between European and other Western powers and the Indian Ocean World.

Same Address, Different Doors: Post-Heritage Deconstruction of the Heritage Household in Upstairs Downstairs in the 1970s and 2010s

2020 ◽  
Author(s):  
William Abbiss
Keyword(s):  
Final Section ◽  
Social Structures ◽  
The Third ◽  
The Past ◽  
War Narrative ◽  
Critical History ◽  
The Social ◽  
Fourth Series ◽  
History Of ◽  
Multiple Episodes

This article offers a ‘post-heritage’ reading of both iterations of Upstairs Downstairs: the LondonWeekend Television (LWT) series (1971–5) and its shortlived BBC revival (2010–12). Identifying elements of subversion and subjectivity allows scholarship on the LWT series to be reassessed, recognising occasions where it challenges rather than supports the social structures of the depicted Edwardian past. The BBC series also incorporates the post-heritage element of self-consciousness, acknowledging the parallel between its narrative and the production’s attempts to recreate the success of its 1970s predecessor. The article’s first section assesses the critical history of the LWT series, identifying areas that are open to further study or revised readings. The second section analyses the serialised war narrative of the fourth series of LWT’s Upstairs, Downstairs (1974), revealing its exploration of female identity across multiple episodes and challenging the notion that the series became more male and upstairs dominated as it progressed. The third section considers the BBC series’ revised concept, identifying the shifts in its main characters’ positions in society that allow the series’ narrative to question the past it evokes. This will be briefly contrasted with the heritage stability of Downton Abbey (ITV, 2010–15). The final section considers the household of 165 Eaton Place’s function as a studio space, which the BBC series self-consciously adopts in order to evoke the aesthetics of prior period dramas. The article concludes by suggesting that the barriers to recreating the past established in the BBC series’ narrative also contributed to its failure to match the success of its earlier iteration.

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