Evidence map›Paper›PMID 26171129›Full record

ArticleOman medical journal2015

Dyskeratosis Congenita Without Oral Involvement: A Rare Hereditary Disease.

Fariba Iraji, Kioumars Jamshidi, Mohsen Pourazizi, Bahareh Abtahi-Naeini

Open access · diamondAbstract readCase Reports
In one paragraph

Article in Oman medical journal, 2015. 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
0.2field-weighted citation impact, top 44% 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, 12 citations in OpenAlex.

  1. Article
  2. Dyskeratosis congenita.Autopsy & case reports · 2020
    Article
  3. [Recurrent pulmonary infection and oral mucosal ulcer].Zhongguo dang dai er ke za zhi = Chinese journal of contemporary pediatrics · 2017
    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

4 authors at 1 institution in 1 country.

Fariba IrajiSkin Diseases and Leishmaniasis Research Center, Isfahan University of Medical Sciences, Isfahan, Iran.
Kioumars JamshidiSkin Diseases and Leishmaniasis Research Center, Isfahan University of Medical Sciences, Isfahan, Iran.
Mohsen PouraziziStudents' Research Committee, Semnan University of Medical Sciences, Semnan, Iran ; Students' Research Committee, Isfahan University of Medical Sciences, Isfahan, Iran.
Bahareh Abtahi-NaeiniSkin Diseases and Leishmaniasis Research Center, Isfahan University of Medical Sciences, Isfahan, Iran ; Students' Research Committee, Isfahan University of Medical Sciences, Isfahan, Iran.
Isfahan University of Medical Sciences · IR

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Dyskeratosis congenita (DC) is a rare, progressive, multi-system, inherited disorder of telomere biology, first described in 1906 as the Zinsser-Engman-Cole syndrome. The condition presents with the classic triad of nail dystrophy, reticulate skin pigmentation, and oral leukoplakia. Variable somatic abnormalities may be present; these include pulmonary, gastrointestinal, genitourinary, cerebral, and dental complications. Pancytopenia eventually develops, progressing to bone marrow failure. In our patient, the progression of DC did not follow the "classic triad" typically seen in the condition. Here we report the case of a 27-year-old male who had features consistent with this syndrome but who had a normal oral cavity associated with aplastic anemia for which longstanding nail dystrophy and reticular pigmentation were the dominant clinical features. The patient was advised to avoid smoking and severe exposure to the sun, and to return regularly for follow-up. Because all symptoms of this disorder are not necessarily seen collectively in any one patient, physicians should consider it as a probability in all patients showing signs of reticular hyperpigmentation, especially accompanied by aplastic anemia. Our report emphasizes the fact that not all components of the syndrome need to be present and in all cases of genodermatosis, pleomorphism may be expected with variable clinical manifestations. Even though many variants have been described in literature, sparing of the oral cavity as in our patient is extremely uncommon. Therefore, physicians should be aware of the mucocutaneous manifestation of DC and its diagnosis and refer the patient for better evaluation.

Indexed as

Dyskeratosis CongenitaDystrophyLeukoplakia, OralNailsSkin Pigmentation

Identifiers

PMID26171129
PMCPMC4459155
OpenAlexW865879168

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.