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Hepatoerythropoietic porphyria

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Disease definition

A rare form of hepatic porphyria characterized by bullous photodermatosis.

ORPHA:95159

Classification level: Disorder

Synonym(s):
  • HEP

Prevalence: <1 / 1 000 000

Inheritance: Autosomal recessive

Age of onset: Childhood

ICD-10: E80.2

ICD-11: 5C58.1Y

OMIM: 176100

UMLS: C0162569

MeSH: D017121

GARD: 6169

Summary
Epidemiology

Fewer than a hundred cases of hepato-erythropoietic porphyria (HEP) have been described worldwide.

Clinical description

The disease begins in childhood. The main clinical signs include skin fragility, and sometimes erosive or even mutilating bullous skin lesions on sun-exposed surfaces (hands, face). Anemia and splenomegaly may be present. At bone level, delayed metacarpophalangeal growth is usually present. Hepatoerythropoietic porphyria corresponds to homozygous or heterozygous compound cases of porphyria cutanea tarda.

Etiology

The disease is due to a deficiency of uroporphyrinogen decarboxylase (UROD; the fifth enzyme in the heme biosynthesis pathway) resulting from a mutation in the UROD gene (NM_000374.5), which leads to an accumulation of porphyrins mostly in the liver. At least 30 different mutations have been described in the UROD gene, including one predominant substitution: G281D.

Diagnostic methods

Diagnosis is based on evidence of porphyrin accumulation in urine and stool with a characteristic chromatographic profile, and very high porphyrin concentrations in plasma and erythrocytes. The presence of a major (< 10%) UROD deficiency in red blood cells confirms the diagnosis. Skin biopsy is not very informative and is not recommended.

Differential diagnosis

Günther's disease is the main differential diagnosis.

Antenatal diagnosis

Antenatal diagnosis is theoretically possible, but is not proposed.

Genetic counseling

Transmission pattern is autosomal recessive. Genetic counseling should be offered to affected families to identify individuals likely to develop or transmit the disease. Genetic counselling should be offered to at-risk couples (both individuals are carriers of a heterozygous pathogenic variant) informing them that there is a 25 % risk of having an affected child at each pregnancy.

Management and treatment

Care and support mainly involves protecting the skin against light, and blood transfusions in case of anemia. Treatment with hydroxyurea and splenectomy are rarely necessary. Chloroquine and phlebotomy are of limited effectiveness in the treatment of hepato-erythropoietic porphyria.

Prognosis

The long-term prognosis is good.

Last update: March 2024 - Expert reviewer(s): Dr Neila TALBI | MetabERN*

* European Reference Network

A summary on this disease is available in Français, Logo ERN Español, Logo ERN Deutsch, Logo ERN Português, Logo ERN Nederlands Logo ERN
Detailed information

Logo ERN: produced/endorsed by ERN(s) Logo FSMR: produced/endorsed by FSMR(s)

Guidelines
Emergency guidelines
Français (2016.pdf) - Orphanet Urgences
Polski (2010.pdf) - Orphanet Urgences
English (2007.pdf) - Orphanet Urgences
Español (2007.pdf) - Orphanet Urgences
Italiano (2007.pdf) - Orphanet Urgences
Português (2007.pdf) - Orphanet Urgences
Anesthesia guidelines
English (2014) - Orphananesthesia
Čeština (2014) - Orphananesthesia
Disease review articles
Clinical genetics review
English (2022) - GeneReviews
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