Evidence map›Paper›PMID 31264077›Full record

ReviewPituitary2019

Acromegaly in Carney complex.

T Cuny, T T Mac, P Romanet, H Dufour, I Morange, F Albarel, A Lagarde, F Castinetti, T Graillon, M O North and 2 more

Abstract readCase ReportsReview
PubMed Publisher
In one paragraph

Review in Pituitary, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
13citing papers in PubMed, 1 pooled it
2.7field-weighted citation impact, top 9% 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

13 citing papers in PubMed, 1 synthesis or guideline pooled it, 30 citations in OpenAlex.

  1. Guideline
  2. Familial acromegaly with a novelJCEM case reports · 2026
    Article
  3. Review
  4. Review
  5. Article
  6. Article
  7. Article
  8. Review
  9. Review
  10. Dermatological and endocrine elements in Carney complex (Review).Experimental and therapeutic medicine · 2021
    Review
  11. Review
  12. Article
  13. Genetics of Acromegaly and Gigantism.Journal of clinical medicine · 2021
    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

12 authors at 3 institutions in 1 country.

T CunyDepartment of Endocrinology, Hospital La Conception, Aix Marseille Univ, APHM, INSERM, MMG, 147 Boulevard Baille, 13005, Marseille, France. thomas.cuny@ap-hm.fr.
T T MacDepartment of Endocrinology, Hospital La Conception, Aix Marseille Univ, APHM, INSERM, MMG, 147 Boulevard Baille, 13005, Marseille, France.
P RomanetLaboratory of Molecular Biology, Hospital La Conception, Aix Marseille Univ, APHM, INSERM, MMG, Marseille, France.
H DufourDepartment of Neurosurgery, Hospital La Timone, Aix Marseille Univ, APHM, INSERM, MMG, Marseille, France.
I MorangeDepartment of Endocrinology, APHM, Hospital La Conception, Marseille, France.
F AlbarelDepartment of Endocrinology, APHM, Hospital La Conception, Marseille, France.
A LagardeLaboratory of Molecular Biology, Hospital La Conception, Aix Marseille Univ, APHM, INSERM, MMG, Marseille, France.
F CastinettiDepartment of Endocrinology, Hospital La Conception, Aix Marseille Univ, APHM, INSERM, MMG, 147 Boulevard Baille, 13005, Marseille, France.
T GraillonDepartment of Neurosurgery, Hospital La Timone, Aix Marseille Univ, APHM, INSERM, MMG, Marseille, France.
M O NorthLaboratory of Genetics and Molecular Biology, APHP, Cochin Hospital, Paris, France.
A BarlierLaboratory of Molecular Biology, Hospital La Conception, Aix Marseille Univ, APHM, INSERM, MMG, Marseille, France.
T BrueDepartment of Endocrinology, Hospital La Conception, Aix Marseille Univ, APHM, INSERM, MMG, 147 Boulevard Baille, 13005, Marseille, France.
Inserm · FRHôpital de la Conception · FRHôpital Cochin · FR

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

purposeCarney complex (CNC) is a rare autosomal dominant syndrome, characterized by mucocutaneous pigmentation, cardiac, cutaneous myxomas and endocrine overactivity. It is generally caused by inactivating mutations in the PRKAR1A (protein kinase cAMP-dependent type I regulatory subunit alpha) gene. Acromegaly is an infrequent manifestation of CNC, reportedly diagnosed in 10% of patients.

methodsWe here report the case of a patient who was concomitantly diagnosed with Carney complex, due to a new mutation in PRKAR1A ((NM_002734.3:c.80_83del, p.(Ile27Lysfs*101 in exon 2), and acromegaly. In parallel, we conducted an extensive review of published case reports of acromegaly in the setting of CNC.

resultsThe 43-year-old patient was diagnosed with an acromegaly due to a GH-secreting pituitary microadenoma resistant to somatostatin analogs. He underwent transsphenoidal surgery in our tertiary referral center, which found a pure GH-secreting adenoma. In the literature, we identified 57 cases (24 men, 33 women) of acromegaly in CNC patients. The median age at diagnosis was 28.8 ± 12 year and there were 6 cases of gigantism. Acromegaly revealed CNC in only 4 patients. 24 patients had a microadenoma and two carried pituitary hyperplasia and/or multiple adenomas, suggesting that CNC may result in a higher proportion of microadenoma as compared to non-CNC acromegaly.

conclusionsAlthough it rarely reveals CNC, acromegaly is diagnosed at a younger age in this setting, with a higher proportion of microadenomas.

Indexed as

AcromegalyAdolescentAdultCarney ComplexCyclic AMP-Dependent Protein Kinase RIalpha SubunitFemaleHumansMaleMutationYoung AdultCyclic AMP-Dependent Protein Kinase RIalpha SubunitPRKAR1A protein, humanAcromegalyCarney complexMicroadenomaMyxomaPRKAR1A

Identifiers

PMID31264077
OpenAlexW2955713669

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.