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Record W2285551727 · doi:10.1186/1710-1492-10-s2-a36

Early presentation of clinical hereditary angioedema symptoms in an infant

2014· article· en· W2285551727 on OpenAlexaffvenue
Hoang Pham, Stephanie Santucci, William H. Yang

Bibliographic record

VenueAllergy Asthma and Clinical Immunology · 2014
Typearticle
Languageen
FieldMedicine
TopicCoagulation, Bradykinin, Polyphosphates, and Angioedema
Canadian institutionsOttawa Allergy Research CorporationUniversity of Ottawa
Fundersnot available
KeywordsHereditary angioedemaMedicinePediatricsMedical diagnosisPresentation (obstetrics)Differential diagnosisHormone replacement therapy (female-to-male)DanazolQuality of life (healthcare)Intensive care medicineGynecologyDermatologySurgeryInternal medicineEndometriosisPathology

Abstract

fetched live from OpenAlex

Timely diagnosis of hereditary angioedema (HAE) is challenging in children. The barriers include lack of awareness of HAE, communication difficulties, diagnostic testing limitations, and broad differential diagnoses for symptoms of HAE. Consequently, there has been no definitive study on the age of onset of symptoms of HAE in children. This lack of awareness can result in reduced quality of life due to suboptimal treatment of symptoms, significant delay in diagnosis, and/or misdiagnosis, which can result in unnecessary tests, treatments, and procedures. Current literature suggests that the mean age of onset is in the second decade of life, which is worsened by puberty, estrogen containing contraception, or estrogen hormone replacement therapy, but symptoms can also be present under one year. Here we present a case report of an infant not previously diagnosed with clinical symptoms of HAE but born from a mother with type I HAE. Cord blood for C1-INH and C4 protein quantities were obtained at time of delivery. Parents were requested to monitor the child for symptoms and pictures were taken to document any clinically suspicious edema and/or rashes. Repeat laboratory testing was done after 1 year of age. Cord blood results show C1-INH <0.12 (0.21-0.39) g/L and C4 0.08 (0.07 – 0.30) g/L. At 9 months, the child’s mother noted slight periorbital edema, which was documented with pictures. At 14 and 18 months, the child developed a rash on her torso and arms that resembled erythema marginatum; both episodes were documented with pictures. Clinical symptoms of HAE may begin as early as 9 months without any triggers. Parents and clinicians need to be vigilant and properly diagnose HAE to optimize the quality of life for these young patients and their families. We emphasize a high index of clinical suspicion of HAE should begin even in the early infant period.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.002
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: Case report
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.002
Threshold uncertainty score0.006

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.002
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0010.000
Science and technology studies0.0010.001
Scholarly communication0.0010.001
Open science0.0000.001
Research integrity0.0010.001
Insufficient payload (model declined to judge)0.0020.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.027
GPT teacher head0.353
Teacher spread0.326 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designCase report
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations0
Published2014
Admission routes2
Has abstractyes

Explore more

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