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Record W4409590165 · doi:10.1016/j.ijscr.2025.111341

Retinal detachment in a neonate with congenital chylothorax and purpura fulminans associated with the PAK2 genetic variant: A case report

2025· article· en· W4409590165 on OpenAlexaff
Arijit Lodha, Majeeda Kamaluddeen, Stephanie A. Dotchin, Julie Lauzon, Patrick Mitchell

Bibliographic record

VenueInternational Journal of Surgery Case Reports · 2025
Typearticle
Languageen
FieldMedicine
TopicLymphatic Disorders and Treatments
Canadian institutionsUniversity of CalgaryUniversity of Alberta
Fundersnot available
KeywordsMedicinePurpura fulminansRetinal detachmentChylothoraxOphthalmologyPediatricsSurgeryRetinal

Abstract

fetched live from OpenAlex

INTRODUCTION AND IMPORTANCE: A potential relationship between bilateral retinal detachment, chylothorax, and purpura fulminans in a female neonate with a PAK2 gene variant is not commonly reported. This emphasizes the significance of early ophthalmologic assessment in neonates with congenital chylothorax. CASE PRESENTATION: A full-term female infant weighing 2.775 kg was delivered by cesarean section due to breech presentation. Prenatal imaging revealed fetal bilateral pleural effusion, suggestive of chylothorax. The neonate developed respiratory distress and purpura fulminans after birth. The absence of the red reflex in the right eye prompted a detailed ophthalmologic examination using a portable slit lamp and an indirect ophthalmoscope. The right eye revealed an ectatic pupil with posterior synechia extending from approximately 6 o'clock to 9 o'clock. Fundus examination of both eyes revealed funnel retinal detachment with multiple chronic features. A small amount of retina was draped between 4 and 7 o'clock, which may have been attached. Further ophthalmologic investigation under anesthesia, using B-scan ultrasonography and intravenous fluorescein angiography, confirmed bilateral retinal detachment. Genetic investigations revealed a PAK2 c.1115A>T, p.(Asp372Val) variant. CLINICAL DISCUSSION: In addition to presenting our case report, we reviewed other recent case reports similar to ours. Retinal detachment and bilateral pleural effusions in neonates with Knobloch syndrome have been recently reported, but without purpura fulminans. Retinal detachment in neonates can result from both congenital and acquired conditions, and requires a thoughtful approach to establish the diagnosis and provide future counseling. CONCLUSION: Bilateral retinal detachment, chylothorax, and purpura fulminans in a neonate with a PAK2 genetic variant is uncommon. This case underscores the importance of early ophthalmologic assessment and genetic testing for both neonates and their family members.

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 distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation 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 score0.139
Threshold uncertainty score0.396

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

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 head0.011
GPT teacher head0.269
Teacher spread0.258 · 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 teacher head, 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
Published2025
Admission routes1
Has abstractyes

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