Evidence map›Paper›PMID 38576642›Full record

ArticleCureus2024

Congenital Erythropoietic Porphyria: A Rare Inherited Disorder.

Porika Saikrishna, Gowrishankar Palaniswamy, Navya Pillikunte Doddareddy, Lyluma Ishfaq, Mah N Zargar, Fathima Wafa Eranhikkal, Sweta Sahu

Open access · diamondAbstract readCase Reports
In one paragraph

Article in Cureus, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.7field-weighted citation impact, top 32% 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

2 citing papers in PubMed, 3 citations in OpenAlex.

  1. Article
  2. 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

7 authors at 1 institution in 4 countries.

Porika SaikrishnaDermatology, Guntur Medical College, Guntur, IND.
Gowrishankar PalaniswamyMedicine, Saveetha Medical College and Hospital, Chennai, IND.
Navya Pillikunte DoddareddyInternal Medicine, Bangalore Medical College and Research Institute, Bengaluru, IND.
Lyluma IshfaqMedicine, Directorate of Health Services Kashmir, Srinagar, IND.
Mah N ZargarMedicine, Fatima Jinnah Medical University, Lahore, PAK.
Fathima Wafa EranhikkalDermatology, Teaching University of Geomedi, Tablisi, GEO.
Sweta SahuSurgery, Jagadguru Jayadeva Murugarajendra (JJM) Medical College, Davanagere, IND.
University of Kashmir · IN

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Congenital erythropoietic porphyria (CEP), also known as Gunther's disease, is an uncommon autosomal recessive disorder caused by a mutation in the uroporphyrinogen III synthase gene. This mutation results in reduced enzyme levels in heme synthesis and the accumulation of pathogenic porphyrin isomers, uroporphyrin I and coproporphyrin I, leading to the clinical manifestations of CEP. Typically, CEP manifests shortly after birth with severe cutaneous photosensitivity, blistering, ulceration, and scarring. Erythrodontia, acro-osteolysis, and skeletal abnormalities are frequently present in conjunction with it. It can even manifest in utero as hydrops fetalis, with pink or red diaper staining as an early diagnostic clue. In this case, we present a 17-year-old male with complaints of discharge over the left foot, blisters upon sunlight exposure, extensive mottled pigmentation, excessive facial hair, mutilated fingers, and verrucous growth over the toes. Using a Wood's lamp revealed pink fluorescence of teeth and ulcers on the foot. Laboratory investigations demonstrated anemia, leukocytopenia, thrombocytopenia, and elevated urine uroporphyrin 1 and coproporphyrin 1 levels. Current treatment approaches include sun protection to avoid further skin damage, beta-carotene to reduce oxidative stress, and blood transfusions to manage anemia. Stem cell transplantation remains the sole curative therapy for this exceedingly rare condition. This case report underscores the rarity and complexity of CEP and emphasizes the challenges in its management.

Indexed as

erythrodontiagenetic variabilityhematopoietic stem cell transplantationphotosensitivityporphyrinogens

Identifiers

PMID38576642
PMCPMC10993765
OpenAlexW4392458834

What Socratic holds

Textmetadata
LicenceCC BY
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 Socratic graph.