Evidence map›Paper›PMID 35450414›Full record

ArticleJournal of the Endocrine Society2022

Primary Adrenal Insufficiency in a Boy with Type I Diabetes: The Importance of Considering X-linked Adrenoleukodystrophy.

Rebecca E Wiersma, Ashish O Gupta, Troy C Lund, Kyriakie Sarafoglou, Elizabeth I Pierpont, Paul J Orchard, Bradley S Miller

Open access · goldAbstract readCase Reports
In one paragraph

Article in Journal of the Endocrine Society, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
0.6field-weighted citation impact, top 33% 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

3 citing papers in PubMed, 4 citations in OpenAlex.

  1. Article
  2. Review
  3. Addison's Disease: Diagnosis and Management Strategies.International journal of general medicine · 2023
    Review
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 1 country.

Rebecca E WiersmaUniversity of Minnesota Pediatric Residency Program, Minneapolis, MN 55454, USA.ORCID https://orcid.org/0000-0002-9937-8091
Ashish O GuptaDivision of Pediatric Blood and Marrow Transplantation, University of Minnesota, Minneapolis, MN 55454, USA.
Troy C LundDivision of Pediatric Blood and Marrow Transplantation, University of Minnesota, Minneapolis, MN 55454, USA.
Kyriakie SarafoglouDivision of Endocrinology, Department of Pediatrics, University of Minnesota Medical School, Minneapolis, MN 55454, USA.ORCID https://orcid.org/0000-0002-5741-3629
Elizabeth I PierpontDivision of Clinical Behavioral Neuroscience, Department of Pediatrics, University of Minnesota, Minneapolis, MN 55454, USA.
Paul J OrchardDivision of Pediatric Blood and Marrow Transplantation, University of Minnesota, Minneapolis, MN 55454, USA.
Bradley S MillerDivision of Endocrinology, Department of Pediatrics, University of Minnesota Medical School, Minneapolis, MN 55454, USA.
University of Minnesota · USUniversity of Minnesota Medical Center · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Primary adrenal insufficiency (PAI) is often the first clinical sign of X-linked adrenoleukodystrophy (X-ALD), a rare genetic disorder that can present with various clinical phenotypes. A subset of boys with X-ALD develop cerebral ALD (cALD), characterized by progressive central demyelination, neurocognitive decline, and ultimately death. Timely intervention with hematopoietic cell transplant (HCT) can be a life-saving therapy by stopping progression of cerebral disease. We report the case of an 11-year-old boy with type 1 diabetes mellitus who presented with PAI, growth delay, and symptoms of attention deficit hyperactivity disorder. Given his history of T1DM, his PAI was presumed to be autoimmune and he was started on hydrocortisone and fludrocortisone. Eleven months later brain magnetic resonance imaging revealed white matter hyperintensity consistent with advanced cALD. The degree of disease progression at the time of diagnosis rendered the patient ineligible for transplant and he has continued to experience progressive neurologic decline. Initial symptoms of cALD are often subtle but should not be overlooked, as early identification of X-ALD is critical to allow early intervention with lifesaving HCT. PAI typically presents prior to the onset of neurologic symptoms. All boys who present with PAI should undergo workup for X-ALD with plasma very long chain fatty acid testing, even in the setting of underlying autoimmune disease.

Indexed as

autoimmune polyglandular syndromecerebral ALDprimary adrenal insufficiencytype I diabetesX-linked adrenoleukodystrophy

Identifiers

PMID35450414
PMCPMC9017996
OpenAlexW4223440442

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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.