Evidence map›Paper›PMID 18855117›Full record

ArticleJournal of inherited metabolic disease2008

Detection of treatable neonatal liver disease by expanded newborn screening.

R J Mackay, D Bratkovic, R Couper, G P Davidson, R Fahy, J M Fletcher, E Ranieri

Abstract readCase Reports
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In one paragraph

Article in Journal of inherited metabolic disease, 2008. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
0.2field-weighted citation impact, top 47% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

0 citing papers in PubMed, 4 citations in OpenAlex.

No citing paper in PubMed yet.

4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

7 authors at 2 institutions in 2 countries.

R J MackayDepartment of Genetic Medicine, Women's and Children's Hospital, Adelaide, South Australia, Australia. richard.mackay@cdhb.govt.nz
D Bratkovic
R Couper
G P Davidson
R Fahy
J M Fletcher
E Ranieri
Women's and Children's Hospital · AUAlice Springs Hospital · AU

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Two neonates were identified at age 48 h by expanded newborn screening, with abnormal methionine and tyrosine concentrations, which were confirmed on repeat samples. Evidence of previously unsuspected liver disease was found at recall, and there was radiological and biochemical evidence of severe liver disease with hepatic synthetic failure. After inborn errors of metabolism (IEMs) were excluded, both were considered to have neonatal haemochromatosis, on the basis of raised ferritin, iron saturation, and very high α-fetoprotein and confirmed by a mildly hyperferritinaemic sibling in the first case, and raised ferritin and iron saturation in the second. However, it was not feasible to obtain tissue confirmation as the requirement for early therapy precluded biopsy. The babies were treated with antioxidants and iron-chelating agents, and the coagulopathy and hypoalbuminaemia were corrected. Both made a complete recovery and remain well after follow-up. Newborn screening programmes could consider advising clinicians, when tyrosine and methionine values are elevated, that once IEMs are excluded liver disease from other causes must be sought. Neonatal haemochromatosis is an example of one such disease that is potentially treatable.

Indexed as

Neonatal Screeningalpha-FetoproteinsAmino Acid Metabolism, Inborn ErrorsAntioxidantsBiomarkersBlood Coagulation DisordersDiagnosis, DifferentialFerritinsHemochromatosisHumansHypoalbuminemiaInfant, NewbornIronIron Chelating AgentsLiverMaleAFP protein, humanalpha-FetoproteinsAntioxidantsBiomarkersFerritinsIronIron Chelating AgentsMethionineTyrosine

Identifiers

PMID18855117
OpenAlexW2080725134

What OpenQuestion holds

Textmetadata
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.