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Record W2116427204 · doi:10.1212/wnl.0b013e31826356ad

Pearls & Oy-sters: Maternally inherited diabetes and deafness presenting with ptosis and macular pattern dystrophy

2012· article· en· W2116427204 on OpenAlexaff
Olufunmilola A. Ogun, Claire A. Sheldon, Jason J.S. Barton

Bibliographic record

VenueNeurology · 2012
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicMitochondrial Function and Pathology
Canadian institutionsSt. Paul's Hospital
Fundersnot available
KeywordsOphthalmoparesisPtosisMedicineOphthalmologyMacular dystrophyOptic neuropathyDiabetes mellitusNystagmusAudiologyRetinalEndocrinologyOptic nerve

Abstract

fetched live from OpenAlex

Ptosis can occur with a variety of mitochondrial syndromes, not just chronic progressive external ophthalmoplegia.• Macular pattern dystrophy is highly characteristic of maternally inherited diabetes and deafness (MIDD), and funduscopy should be performed on all subjects with personal or family history of diabetes and deafness. OY-STER• Only 15% of patients with MIDD have diabetes at diagnosis, and diabetes may start at ages ranging from the teens to the fifth decade.Mitochondrial disorders can cause an array of neuroophthalmologic manifestations such as ptosis, external ophthalmoparesis, nystagmus, pigmentary retinopathy, and optic neuropathy. 1 The m.3243AϾG point mutation in the mitochondrial genome, an alanine-toguanine transition at position 3243 of mitochondrial DNA, in the MT-TL1 gene that encodes the tRNALeu (UUR), commonly presents as an encephalopathy accompanied by lactic acidosis and stroke-like episodes (MELAS).Many mutation carriers, however, have a wide range of neurologic, cardiac, endocrine, gastrointestinal, and psychiatric symptoms, as well as other mitochondrial syndromes.2 Another syndrome also associated with this mutation is maternally inherited diabetes and deafness (MIDD).3 Ptosis and ophthalmoparesis are not generally seen in MIDD, 4 with only 1 report of 2 patients with ptosis in this condition.5 We present another patient with ptosis associated with MIDD and the m.3243AϾG mutation, in whom the key diagnostic feature was the macular pattern dystrophy typical of MIDD.CASE REPORT A 54-year-old woman had right ptosis since her early teens (figure).This was stable for decades but began to worsen at age 40.Surgical correction was attempted twice but ptosis recurred.From the Departments of Ophthalmology and Visual Sciences (O.O., C.S., J.J.S.B.) and Medicine (Neurology) (O.O., J.J.

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 score0.006
Threshold uncertainty score0.022

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.002
Meta-epidemiology (narrow)0.0020.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0020.001
Science and technology studies0.0010.001
Scholarly communication0.0010.001
Open science0.0010.001
Research integrity0.0020.001
Insufficient payload (model declined to judge)0.0060.001

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 head0.010
GPT teacher head0.217
Teacher spread0.207 · 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

Citations12
Published2012
Admission routes1
Has abstractyes

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