Evidence map›Paper›PMID 39006359›Full record

SynthesisFrontiers in endocrinology2024

The clinical characteristics and pathogenic variants of primary pigmented nodular adrenocortical disease in 210 patients: a systematic review.

Julian Sun, Lin Ding, Liping He, Hang Fu, Rui Li, Jing Feng, Jianjun Dong, Lin Liao

Abstract readSystematic Review
In one paragraph

Synthesis in Frontiers in endocrinology, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.

0numbers the graph read from it
0cells of the map it votes in
7citing papers in PubMed
–field-weighted citation impact
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

7 citing papers in PubMed.

  1. Review
  2. Article
  3. Article
  4. A Rare Case ofJCEM case reports · 2025
    Article
  5. Article
  6. Article
  7. Article
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

8 authors.

Julian Sun *School of Clinical Medicine, Shandong Second Medical University, Weifang, China.
Lin Ding *Department of Endocrinology and Metabology, Shandong Provincial Qianfoshan Hospital, Jinan, China.
Liping HeSchool of Clinical Medicine, Shandong Second Medical University, Weifang, China.
Hang FuDepartment of Endocrinology and Metabology, The First Affiliated Hospital of Shandong First Medical University & Shandong Provincial Qianfoshan Hospital, Jinan, China.
Rui LiFirst Clinical Medical College, Shandong University of Traditional Chinese Medicine, Jinan, China.
Jing FengDepartment of Endocrinology and Metabology, The First Affiliated Hospital of Shandong First Medical University & Shandong Provincial Qianfoshan Hospital, Jinan, China.
Jianjun DongDepartment of Endocrinology, Qilu Hospital of Shandong University, Jinan, China.
Lin LiaoSchool of Clinical Medicine, Shandong Second Medical University, Weifang, China.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Aims: Primary pigmented nodular adrenocortical disease (PPNAD), as a rare kind of Cushing's syndrome, is frequently misdiagnosed. To get a better understanding of the disease, we analyzed the clinical characteristics and pathogenic variants of PPNAD. Methods: Databases were searched, and the pathogenic variants and clinical manifestations of patients were summarized from the relevant articles. Results: A total of 210 patients in 86 articles were enrolled with a median age of 22 and a female-to-male ratio of 2:1. Sixty-six (31.43%) patients were combined with Carney complex (CNC) and 94.29% were combined with osteoporosis/osteopenia. Among 151 patients who underwent genetic testing, 87.42% (132/151) had pathogenic variants. Six gene mutations ( Conclusions: For young patients with Cushing's syndrome, especially female patients with spotty skin pigmentation and osteoporosis/osteopenia, PPNAD should be considered. Unilateral adrenal resection may be considered as an option for women with fertility needs. In view of the difficulty of PPNAD diagnosis, genetic testing before surgery might be a reasonable option. Patients with PPNAD with spotty skin pigmentation should consider the Systematic review registration: https://www.crd.york.ac.uk/prospero, identifier CRD42023416988.

Indexed as

Adrenal Cortex DiseasesCyclic AMP-Dependent Protein Kinase RIalpha SubunitMutationAdultCarney ComplexCushing SyndromeFemaleHumansMaleOsteoporosisYoung AdultCyclic AMP-Dependent Protein Kinase RIalpha SubunitPRKAR1A protein, humanclinical characteristicsCushing’s syndromepathogenic variantsprimary pigmented nodular adrenocortical diseasePRKAR1A

Identifiers

PMID39006359
PMCPMC11240189

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Registered trials

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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.