Raras
Buscar doenças, sintomas, genes...
Estomatocitose
ORPHA:98365CID-10 · D58.8CID-11 · 3A10.YDOENÇA RARA
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Introdução

O que você precisa saber de cara

📋

Estomatocitose hereditária descreve uma série de condições humanas herdadas, predominantemente autossômicas dominantes, que afetam os glóbulos vermelhos e criam a aparência de uma área de palidez central em forma de fenda (estomatócito) entre os eritrócitos no esfregaço de sangue periférico. As membranas celulares dos eritrócitos podem "vazar" anormalmente íons de sódio e/ou potássio, causando anormalidades no volume celular. A estomatocitose hereditária deve ser distinguida de causas adquiridas de estomatocitose, incluindo toxicidade por dilantina e alcoolismo, bem como de artefatos do processo de preparo dos esfregaços de sangue periférico.

Publicações científicas
209 artigos
Último publicado: 2026 Mar 19
Medicamentos
1 registrados
SENICAPOC

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SENICAPOC
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SUS: Sem cobertura SUSScore: 0%
CID-10: D58.8
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Sinais e sintomas

O que aparece no corpo e com que frequência cada sintoma acontece

Partes do corpo afetadas

🩸
Sangue
13 sintomas
🧠
Neurológico
13 sintomas
🫃
Digestivo
6 sintomas
🦴
Ossos e articulações
5 sintomas
👁️
Olhos
5 sintomas
📏
Crescimento
4 sintomas

+ 55 sintomas em outras categorias

Características mais comuns

Baixa estatura
Intolerância ao exercício
Palidez anêmica
Anemia hemolítica episódica
Hipertensão
Acidose tubular renal
111sintomas
Sem dados (111)

Os sintomas variam de pessoa para pessoa. Abaixo estão as 111 características clínicas mais associadas, ordenadas por frequência.

Baixa estaturaShort stature
Intolerância ao exercícioExercise intolerance
Palidez anêmicaAnemic pallor
Anemia hemolítica episódicaEpisodic hemolytic anemia
HipertensãoHypertension

Linha do tempo da pesquisa

Publicações por ano — veja quando o interesse científico cresceu
Anos de pesquisa1desde 2026
Total histórico209PubMed
Últimos 10 anos85publicações
Pico201812 papers
Linha do tempo
2026Hoje · 2026📈 2018Ano de pico🧪 2020Primeiro ensaio clínico
Publicações por ano (últimos 10 anos)

Encontrou um erro ou informação desatualizada? Sugira uma correção →

Genética e causas

O que está alterado no DNA e como passa nas famílias

Genes associados

8 genes identificados com associação a esta condição.

RHAGAmmonium transporter Rh type ADisease-causing germline mutation(s) inTolerante
FUNÇÃO

Component of the ankyrin-1 complex, a multiprotein complex involved in the stability and shape of the erythrocyte membrane (PubMed:35835865). Heterotrimer with RHCE (RHAG)2(RHCE), that transports ammonium and its related derivative methylammonium, in both neutral and ionic forms, across the erythrocyte membrane (PubMed:11062476, PubMed:11861637, PubMed:15572441, PubMed:15856280, PubMed:19273840, PubMed:21849667, PubMed:22012326, PubMed:24077989, PubMed:26354748). The transport of NH4(+) is elect

LOCALIZAÇÃO

Membrane

VIAS BIOLÓGICAS (3)
Erythrocytes take up oxygen and release carbon dioxideErythrocytes take up carbon dioxide and release oxygenRhesus glycoproteins mediate ammonium transport
MECANISMO DE DOENÇA

Regulator type Rh-null hemolytic anemia

Form of chronic hemolytic anemia in which the red blood cells have a stomatocytosis and spherocytosis morphology, an increased osmotic fragility, an altered ion transport system, and abnormal membrane phospholipid organization.

EXPRESSÃO TECIDUAL(Baixa expressão)
Sangue
2.6 TPM
Nervo tibial
1.7 TPM
Coração - Átrio
1.7 TPM
Cervix Ectocervix
1.5 TPM
Cervix Endocervix
1.1 TPM
OUTRAS DOENÇAS (2)
overhydrated hereditary stomatocytosisRh deficiency syndrome
HGNC:10006UniProt:Q02094
RHDBlood group Rh(D) polypeptideCandidate gene tested inAltamente restrito
FUNÇÃO

May be part of an oligomeric complex which is likely to have a transport or channel function in the erythrocyte membrane

LOCALIZAÇÃO

Cell membrane

VIAS BIOLÓGICAS (1)
Rhesus blood group biosynthesis
MECANISMO DE DOENÇA

Hemolytic disease of fetus and newborn, RH-induced

A disease that occurs in pregnancies in which mothers who lack the D antigen (RhD) of the Rh blood group have been exposed to the RhD-positive red cells of the fetus. The resulting maternal autoantibodies cross the placenta and destroy fetal red cells.

EXPRESSÃO TECIDUAL(Baixa expressão)
Glândula salivar
2.3 TPM
Sangue
2.3 TPM
Baço
1.2 TPM
Cervix Endocervix
0.9 TPM
Ovário
0.9 TPM
OUTRAS DOENÇAS (2)
Rh deficiency syndromehemolytic disease of fetus and newborn, RH-induced
HGNC:10009UniProt:Q02161
RHCEBlood group Rh(CE) polypeptideCandidate gene tested inTolerante
FUNÇÃO

Component of the ankyrin-1 complex, a multiprotein complex involved in the stability and shape of the erythrocyte membrane (PubMed:35835865). Mediates the primary membrane attachment site for ANK1 when associated with RHAG (PubMed:35835865). May participate in the ammonium and carbon dioxide transport through the heterotrimer form (Probable)

LOCALIZAÇÃO

Membrane

VIAS BIOLÓGICAS (1)
Rhesus blood group biosynthesis
MECANISMO DE DOENÇA

Rh-null, amorph type

An autosomal recessive condition characterized by red blood cells that lack all Rh antigens, have increased osmotic fragility, diminished lifespan, and show changes in morphology resulting in stomatocytosis. Rh-null individuals have mild to moderate hemolytic anemia. They are at risk of having adverse reactions in response to transfusion or pregnancy, because they may produce antibodies against several of the Rh antigens.

EXPRESSÃO TECIDUAL(Baixa expressão)
Pituitária
4.3 TPM
Testículo
4.1 TPM
Glândula salivar
2.6 TPM
Sangue
2.3 TPM
Brain Spinal cord cervical c-1
1.9 TPM
OUTRAS DOENÇAS (1)
Rh deficiency syndrome
HGNC:10008UniProt:P18577
KCNN4Intermediate conductance calcium-activated potassium channel protein 4Disease-causing germline mutation(s) inTolerante
FUNÇÃO

Intermediate conductance calcium-activated potassium channel that mediates the voltage-independent transmembrane transfer of potassium across the cell membrane through a constitutive interaction with calmodulin which binds the intracellular calcium allowing its opening (PubMed:10026195, PubMed:10961988, PubMed:11425865, PubMed:15831468, PubMed:17157250, PubMed:18796614, PubMed:26148990, PubMed:9326665, PubMed:9380751, PubMed:9407042). The current is characterized by a voltage-independent activat

LOCALIZAÇÃO

Cell membraneCell projection, ruffle membrane

VIAS BIOLÓGICAS (1)
Ca2+ activated K+ channels
MECANISMO DE DOENÇA

Dehydrated hereditary stomatocytosis 2

An autosomal dominant hemolytic anemia characterized by primary erythrocyte dehydration. Erythrocytes exhibit decreased total cation and potassium content that are not accompanied by a proportional net gain of sodium and water. Affected individuals typically manifest mild to moderate compensated hemolytic anemia, with an increased erythrocyte mean corpuscular hemoglobin concentration and a decreased osmotic fragility, both of which reflect cellular dehydration. Their red cells exhibit a panel of various shape abnormalities such as elliptocytes, hemighosts, schizocytes, and very rare stomatocytic cells. Complications such as splenomegaly and cholelithiasis, resulting from increased red cell trapping in the spleen and elevated bilirubin levels, respectively, may occur.

EXPRESSÃO TECIDUAL(Ubíquo)
Glândula salivar
115.7 TPM
Linfócitos
53.8 TPM
Skin Sun Exposed Lower leg
34.3 TPM
Próstata
31.7 TPM
Baço
25.2 TPM
OUTRAS DOENÇAS (3)
dehydrated hereditary stomatocytosis 2cystic fibrosisdehydrated hereditary stomatocytosis
HGNC:6293UniProt:O15554
SLC2A1Solute carrier family 2, facilitated glucose transporter member 1Disease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Facilitative glucose transporter, which is responsible for constitutive or basal glucose uptake (PubMed:10227690, PubMed:10954735, PubMed:18245775, PubMed:19449892, PubMed:25982116, PubMed:27078104, PubMed:32860739). Has a very broad substrate specificity; can transport a wide range of aldoses including both pentoses and hexoses (PubMed:18245775, PubMed:19449892). Most important energy carrier of the brain: present at the blood-brain barrier and assures the energy-independent, facilitative trans

LOCALIZAÇÃO

Cell membraneMelanosomePhotoreceptor inner segment

VIAS BIOLÓGICAS (3)
Vitamin C (ascorbate) metabolismCellular hexose transportRegulation of insulin secretion
MECANISMO DE DOENÇA

GLUT1 deficiency syndrome 1

A neurologic disorder showing wide phenotypic variability. The most severe 'classic' phenotype comprises infantile-onset epileptic encephalopathy associated with delayed development, acquired microcephaly, motor incoordination, and spasticity. Onset of seizures, usually characterized by apneic episodes, staring spells, and episodic eye movements, occurs within the first 4 months of life. Other paroxysmal findings include intermittent ataxia, confusion, lethargy, sleep disturbance, and headache. Varying degrees of cognitive impairment can occur, ranging from learning disabilities to severe intellectual disability.

EXPRESSÃO TECIDUAL(Ubíquo)
Nervo tibial
703.8 TPM
Skin Not Sun Exposed Suprapubic
272.6 TPM
Skin Sun Exposed Lower leg
265.8 TPM
Vagina
176.1 TPM
Esôfago - Mucosa
154.8 TPM
OUTRAS DOENÇAS (8)
childhood onset GLUT1 deficiency syndrome 2hereditary cryohydrocytosis with reduced stomatindystonia 9encephalopathy due to GLUT1 deficiency
HGNC:11005UniProt:P11166
PIEZO1Piezo-type mechanosensitive ion channel component 1Disease-causing germline mutation(s) inTolerante
FUNÇÃO

Pore-forming subunit of the mechanosensitive non-specific cation Piezo channel required for rapidly adapting mechanically activated (MA) currents and has a key role in sensing touch and tactile pain (PubMed:23479567, PubMed:23695678, PubMed:25955826, PubMed:37590348). Piezo channels are homotrimeric three-blade propeller-shaped structures that utilize a cap-motion and plug-and-latch mechanism to gate their ion-conducting pathways (PubMed:37590348). Generates currents characterized by a linear cu

LOCALIZAÇÃO

Endoplasmic reticulum membraneEndoplasmic reticulum-Golgi intermediate compartment membraneCell membraneCell projection, lamellipodium membrane

VIAS BIOLÓGICAS (3)
Turbulent (oscillatory, disturbed) flow shear stress activates signaling by PIEZO1 and integrins in endothelial cellsHigh laminar flow shear stress activates signaling by PIEZO1 and PECAM1:CDH5:KDR in endothelial cellsMechanical load activates signaling by PIEZO1 and integrins in osteocytes
MECANISMO DE DOENÇA

Dehydrated hereditary stomatocytosis 1 with or without pseudohyperkalemia and/or perinatal edema

An autosomal dominant hemolytic anemia characterized by primary erythrocyte dehydration. DHS erythrocytes exhibit decreased total cation and potassium content that are not accompanied by a proportional net gain of sodium and water. DHS patients typically exhibit mild to moderate compensated hemolytic anemia, with an increased erythrocyte mean corpuscular hemoglobin concentration and a decreased osmotic fragility, both of which reflect cellular dehydration. Patients may also show perinatal edema and pseudohyperkalemia due to loss of potassium from red cells stored at room temperature. A minor proportion of red cells appear as stomatocytes on blood films. Complications such as splenomegaly and cholelithiasis, resulting from increased red cell trapping in the spleen and elevated bilirubin levels, respectively, may occur. The course of DHS is frequently associated with iron overload, which may lead to hepatosiderosis.

EXPRESSÃO TECIDUAL(Ubíquo)
Cólon sigmoide
119.2 TPM
Pulmão
101.5 TPM
Fibroblastos
94.8 TPM
Bladder
86.0 TPM
Útero
82.6 TPM
OUTRAS DOENÇAS (3)
dehydrated hereditary stomatocytosis with or without pseudohyperkalemia and/or perinatal edemalymphatic malformation 6dehydrated hereditary stomatocytosis
HGNC:28993UniProt:Q92508
ABCB6ATP-binding cassette sub-family B member 6Disease-causing germline mutation(s) inTolerante
FUNÇÃO

ATP-dependent transporter that catalyzes the transport of a broad-spectrum of porphyrins from the cytoplasm to the extracellular space through the plasma membrane or into the vesicle lumen (PubMed:17661442, PubMed:23792964, PubMed:27507172, PubMed:33007128). May also function as an ATP-dependent importer of porphyrins from the cytoplasm into the mitochondria, in turn may participate in the de novo heme biosynthesis regulation and in the coordination of heme and iron homeostasis during phenylhydr

LOCALIZAÇÃO

Cell membraneMitochondrion outer membraneEndoplasmic reticulum membraneGolgi apparatus membraneEndosome membraneLysosome membraneLate endosome membraneEarly endosome membraneSecreted, extracellular exosomeMitochondrionEndosome, multivesicular body membraneMelanosome membrane

VIAS BIOLÓGICAS (1)
Mitochondrial ABC transporters
MECANISMO DE DOENÇA

Microphthalmia/Coloboma 7

A disorder of eye formation, ranging from small size of a single eye to complete bilateral absence of ocular tissues. Ocular abnormalities like opacities of the cornea and lens, scaring of the retina and choroid, and other abnormalities may also be present. Ocular colobomas are a set of malformations resulting from abnormal morphogenesis of the optic cup and stalk, and the fusion of the fetal fissure (optic fissure).

OUTRAS DOENÇAS (12)
microphthalmia, isolated, with coloboma 7familial pseudohyperkalemiadyschromatosis universalis hereditaria 3obsolete blood group, langereis system
HGNC:47UniProt:Q9NP58
SLC4A1Band 3 anion transport proteinDisease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Functions both as a transporter that mediates electroneutral anion exchange across the cell membrane and as a structural protein (PubMed:10926824, PubMed:14734552, PubMed:1538405, PubMed:16227998, PubMed:20151848, PubMed:24121512, PubMed:28387307, PubMed:35835865). Component of the ankyrin-1 complex of the erythrocyte membrane; required for normal flexibility and stability of the erythrocyte membrane and for normal erythrocyte shape via the interactions of its cytoplasmic domain with cytoskeleta

LOCALIZAÇÃO

Cell membraneBasolateral cell membrane

VIAS BIOLÓGICAS (3)
Bicarbonate transportersErythrocytes take up carbon dioxide and release oxygenErythrocytes take up oxygen and release carbon dioxide
MECANISMO DE DOENÇA

Ovalocytosis, Southeast Asian

An autosomal dominant hematologic disorder characterized by ovalocytic erythrocytes that are rigid and exhibit reduced expression of many erythrocyte antigens. Clinical manifestations include mild hemolysis, intermittent jaundice and gallstones. However, the disorder is most often asymptomatic.

EXPRESSÃO TECIDUAL(Tecido-específico)
Sangue
78.9 TPM
Rim - Medula
27.8 TPM
Rim - Córtex
26.2 TPM
Baço
4.2 TPM
Cerebelo
1.2 TPM
OUTRAS DOENÇAS (11)
autosomal dominant distal renal tubular acidosissoutheast Asian ovalocytosisrenal tubular acidosis, distal, 4, with hemolytic anemiaobsolete blood group, diego system
HGNC:11027UniProt:P02730

Medicamentos e terapias

SENICAPOCPhase 1

Mecanismo: Intermediate conductance calcium-activated potassium channel protein 4 blocker

Ver mais no OpenTargets

Variantes genéticas (ClinVar)

54 variantes patogênicas registradas no ClinVar.

🧬 RHAG: NM_000324.3(RHAG):c.194T>G (p.Phe65Cys) ()
🧬 RHAG: NM_000324.3(RHAG):c.1153_1154del (p.Leu385fs) ()
🧬 RHAG: NM_000324.3(RHAG):c.1163G>A (p.Trp388Ter) ()
🧬 RHAG: NM_000324.3(RHAG):c.212del (p.Phe71fs) ()
🧬 RHAG: NM_000324.3(RHAG):c.1078A>G (p.Met360Val) ()
Ver todas no ClinVar

Classificação de variantes (ClinVar)

Distribuição de 191 variantes classificadas pelo ClinVar.

10
181
Patogênica (5.2%)
VUS (94.8%)
VARIANTES MAIS SIGNIFICATIVAS
PIEZO1: NM_001142864.4(PIEZO1):c.7170dup (p.Arg2391fs) [Likely pathogenic]
HSALR1: NM_001142864.4(PIEZO1):c.790T>C (p.Tyr264His) [Uncertain significance]
PIEZO1: NM_001142864.4(PIEZO1):c.6329G>A (p.Arg2110Gln) [Uncertain significance]
KCNN4: NM_002250.3(KCNN4):c.158C>T (p.Ser53Leu) [Uncertain significance]
KCNN4: NM_002250.3(KCNN4):c.1264C>A (p.Pro422Thr) [Uncertain significance]

Diagnóstico

Os sinais que médicos procuram e os exames que confirmam

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Tratamento e manejo

Remédios, cuidados de apoio e o que precisa acompanhar

Pipeline de tratamentos
Pipeline regulatório — de medicamentos já aprovados a drogas em pesquisa exploratória.
2Fase 21
1Fase 11
·Pré-clínico1
Medicamentos catalogadosEnsaios clínicos· 1 medicamento · 2 ensaios
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Onde tratar no SUS

Hospitais de referência no Brasil e o protocolo oficial do SUS (PCDT)

🇧🇷 Atendimento SUS — Estomatocitose

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Selecione um estado ou use sua localização para ver resultados.

Dados de DATASUS/CNES, SBGM, ABNeuro e Ministério da Saúde. Sempre confirme a disponibilidade diretamente com o estabelecimento.

Pesquisa ativa

Ensaios clínicos abertos e novidades científicas recentes

Pesquisa e ensaios clínicos

2 ensaios clínicos encontrados.

Distribuição por fase
Ver todos no ClinicalTrials.gov
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Publicações mais relevantes

Timeline de publicações
85 papers (10 anos)
#1

The Significance of the Piezo1-Mediated Mechanotransduction Pathway in Normal Morphogenesis.

DNA and cell biology2026 Feb 05

Mechanical forces are fundamental drivers of morphogenesis, yet the molecular mechanisms that convert these physical cues into transcriptional responses remain incompletely understood. This review synthesizes current evidence identifying the mechanosensitive ion channel Piezo1 as a master regulator of developmental processes. The structural and biophysical principles underlying Piezo1 function are highlighted, focusing on its trimeric architecture and force-from-lipids gating mechanism that directly couples membrane tension to Ca2+ influx. Its spatiotemporal expression during embryogenesis is reviewed, and the downstream pathways it activates are examined, including mitogen-activated protein kinase (MAPK) and yes-associated protein/transcriptional co-activator with PDZ-binding moti (YAP/TAZ), alongside crucial crosstalk with canonical morphogen signaling cascades such as Notch, Wntwingless/integrated signaling pathway (Wnt)/beta-catenin (β-catenin), and bone morphogenetic protein/transforming growth factor-beta (BMP/TGF-β). Functional studies across diverse model systems demonstrate that Piezo1 orchestrates conserved morphogenetic events, including vascular and lymphatic patterning, neurogenesis, epithelial morphogenesis, myoblast fusion, and osteogenesis. Human genetic data further underscore its nonredundant role, linking gain-of-function mutations to dehydrated hereditary stomatocytosis and loss-of-function mutations to primary lymphatic dysplasia. Collectively, these findings establish Piezo1 as an essential integrator of mechanical and biochemical signals, central to tissue patterning and organ formation. The review concludes by emphasizing Piezo1's therapeutic potential in regenerative medicine and developmental disorders, while also underscoring the challenges of targeting such a broadly influential mechanosensor.

#2

Clinical clues to recognizing hereditary dehydrated stomatocytosis (DHSt) in children.

Archivos argentinos de pediatria2026 Mar 19

Dehydrated hereditary stomatocytosis (DHSt) is a heterogeneous group of rare hemolytic disorders with autosomal dominant inheritance. A series of 20 cases demonstrates that early identification of DHSt can prevent unnecessary interventions and improve the management of anemia, iron overload, and other complications in pediatric and adult patients. The presence of elevated mean corpuscular hemoglobin concentration (MCHC) with resistant erythrocytes suggested a possible association with variants in PIEZO1. Patients with KCNN4 variants showed no clear signs of erythrocyte dehydration, but, as with PIEZO1, macrocytosis, hemolytic anemia, and iron overload were common manifestations. Las estomatocitosis hereditarias deshidratadas (DHSt) constituyen un grupo heterogéneo de trastornos hemolíticos raros con herencia autosómica dominante. Una serie de 20 casos revela cómo la identificación precoz de las DHSt puede evitar intervenciones innecesarias y mejorar el manejo de la anemia, la sobrecarga de hierro y otras complicaciones en pacientes pediátricos y adultos. La presencia de una concentración de hemoglobina corpuscular media (CHCM) elevada con eritrocitos resistentes sugirió una posible asociación con variantes en PIEZO1. Los pacientes con variantes en KCNN4 no mostraron signos claros de deshidratación eritrocitaria, pero, al igual que en PIEZO1, la macrocitosis, la anemia hemolítica y la sobrecarga de hierro fueron manifestaciones frecuentes.

#3

Additive effect of multiple genetic variants in SEC23B and PIEZO1 on iron metabolism dyshomeostasis in hereditary anemias.

HemaSphere2026 Jan

Hereditary anemias encompass a genetically heterogeneous spectrum of disorders, often involving multi-locus inheritance, which can complicate clinical management and worsen disease severity. This study investigates the impact of the co-inheritance of SEC23B loss-of-function pathogenic variants, which lead to congenital dyserythropoietic anemia type II (CDA II), and PIEZO1 gain-of-function pathogenic variants, associated with dehydrated hereditary stomatocytosis type I (DHS1), on hematological parameters and iron metabolism. Among 583 patients with suspected hereditary anemia, 13 were found to carry both SEC23B and PIEZO1 variants, leading to a dual diagnosis of CDA II and DHS1. Compared to those with isolated CDA II, these patients exhibited a significantly higher absolute reticulocyte count and bone marrow responsiveness index, alongside an increased prevalence of elevated ferritin levels. Functional studies in Hep3B human hepatoma cells confirmed that SEC23B knockdown combined with PIEZO1 gain-of-function led to marked ferritin accumulation and reduced hepcidin expression, driven by altered BMP/SMAD signaling and ERK1/2 MAPK pathway. These findings demonstrate how multi-locus inheritance can modify disease severity, particularly by exacerbating iron overload. Our results underscore the clinical relevance of comprehensive genetic testing for enhanced risk stratification and personalized management of hereditary anemias.

#4

A Rare Combination of High-Affinity Hemoglobin, Non-Transfusion-Dependent Thalassemia (Αlpha-Triplication and Codon 39 Mutation), and Hereditary Stomatocytosis.

American journal of hematology2026 Apr
#5

The evolving landscape of hereditary stomatocytosis.

Blood2025 Jun 26

Hereditary stomatocytosis represents a heterogeneous group of inherited erythrocyte membrane defects characterized by hemolytic anemia of variable degree, with alterations in cellular salt and water, ranging from dehydration to overhydration, and the presence of stomatocytes on peripheral blood smear. This condition encompasses various subtypes, each with distinct clinical and genetic features. The pathophysiology underlying these conditions involves altered red blood cell membrane properties, leading to impaired deformability and alterations in cation permeability and volume, causing increased susceptibility to hemolysis. Advancements in genetic testing have enabled the identification of some causative genes in the last years, such as PIEZO1, KCNN4, and ABCB6. These genetic discoveries have facilitated a deeper understanding of the molecular mechanisms underlying the pathogenesis and have paved the way for improved diagnostic accuracy and genetic counseling. This review provides an overview of the clinical presentation, pathophysiology, molecular genetics, diagnosis, and management strategies of hereditary stomatocytosis, highlighting recent advancements in the field of dehydrated hereditary stomatocytosis (DHS), or hereditary xerocytosis, and hepatic iron overload. This latter is directly associated with the physiological role of PIEZO1, the causative gene of DHS, at hepatic and macrophagic levels. Particularly, gain-of-function mutations in PIEZO1 account for a pleiotropic syndrome characterized by different phenotypes depending on the expression of PIEZO1 in multiple cells and tissues.

Publicações recentes

Ver todas no PubMed

📚 EuropePMC105 artigos no totalmostrando 85

2026

Clinical clues to recognizing hereditary dehydrated stomatocytosis (DHSt) in children.

Archivos argentinos de pediatria
2026

Additive effect of multiple genetic variants in SEC23B and PIEZO1 on iron metabolism dyshomeostasis in hereditary anemias.

HemaSphere
2026

The Significance of the Piezo1-Mediated Mechanotransduction Pathway in Normal Morphogenesis.

DNA and cell biology
2026

A Rare Combination of High-Affinity Hemoglobin, Non-Transfusion-Dependent Thalassemia (Αlpha-Triplication and Codon 39 Mutation), and Hereditary Stomatocytosis.

American journal of hematology
2025

[Dehydrated hereditary stomatocytosis in 23 cases: a single-center retrospective cohort study from Peking Union Medical College Hospital (2018-2024)].

Zhonghua xue ye xue za zhi = Zhonghua xueyexue zazhi
2025

Clinical and Genetic Analysis of Dehydrated Hereditary Stomatocytosis: A Case Report.

Clinical case reports
2025

Diagnosis of Dehydrated Hereditary Stomatocytosis in a 60-Year-Old Female Patient.

Cureus
2025

Concurrent Case of Glucose-6-Phosphate Deficiency and Dehydrated Hereditary Stomatocytosis in a 4-Month-old Boy.

Journal of pediatric hematology/oncology
2025

Novel compound heterozygous PIEZO1 variants in dehydrated hereditary stomatocytosis initially suspected as myelodysplastic syndromes: a case report.

Frontiers in oncology
2025

A Chinese pediatric patient with thalassemia traits and compound heterozygous mutations in the PIEZO1 gene suspected of having dehydrated hereditary stomatocytosis.

Hematology (Amsterdam, Netherlands)
2025

Dehydrated Hereditary Stomatocytosis (DHS): A Rare Inherited Hemolytic Disorder With Unusual Hypochromic Microcytic Anemia.

Cureus
2025

The evolving landscape of hereditary stomatocytosis.

Blood
2025

Dehydrated Hereditary Stomatocytosis in a Very Preterm Twin.

Pediatric blood & cancer
2025

Myelodysplastic syndrome with ring chromosomes in a case of dehydrated hereditary stomatocytosis 1 (DHS1).

International journal of hematology
2024

Hereditary Hemolytic Anemia Due to PIEZO1 Red Blood Cell Membrane Defect.

Hemoglobin
2025

RAS signaling pathway is essential in regulating PIEZO1-mediated hepatic iron overload in dehydrated hereditary stomatocytosis.

American journal of hematology
2025

Hereditary stomatocytosis in the general population: A genetically based prevalence estimate from a 109 039 individual Danish cohort.

American journal of hematology
2024

Modifiable lifestyle factors influencing psychiatric disorders mediated by plasma proteins: A systemic Mendelian randomization study.

Journal of affective disorders
2023

Transient presence of stomatocytes: A clue to the diagnosis of overhydrated hereditary stomatocytosis in a child with beta-thalassemia.

Journal of clinical laboratory analysis
2023

The cation-leaky hereditary stomatocytosis syndromes: A tale of six proteins.

British journal of haematology
2023

Dehydrated hereditary stomatocytosis masquerading as primary haemochromatosis: a diagnostic challenge.

Pathology
2023

Sequence Similarity among Structural Repeats in the Piezo Family of Mechanosensitive Ion Channels.

Microbial physiology
2023

A critical role for altered red cell cation permeability in pathogenesis of sickle cell disease and other haemolytic anaemias.

British journal of haematology
2023

Overhydrated hereditary stomatocytosis: A rare cause of familiar persistent macrocytosis due to SLC4A1 variants.

Pediatric blood & cancer
2023

Juvenile Hemochromatosis With Non-transfused Hemolytic Anemia Caused by a De Novo PIEZO1 Gene Mutation.

Journal of pediatric hematology/oncology
2023

Variant spectrum of PIEZO1 and KCNN4 in Japanese patients with dehydrated hereditary stomatocytosis.

Human genome variation
2023

Proteome alterations in erythrocytes with PIEZO1 gain-of-function mutations.

Blood advances
2022

Structural and mutational studies suggest key residues to determine whether stomatin SPFH domains form dimers or trimers.

Biochemistry and biophysics reports
2022

A Gardos channelopathy associated with nonimmune hydrops and fetal loss.

Clinical genetics
2022

Mechanosensitive Pannexin 1 Activity Is Modulated by Stomatin in Human Red Blood Cells.

International journal of molecular sciences
2022

New KCNN4 Variants Associated With Anemia: Stomatocytosis Without Erythrocyte Dehydration.

Frontiers in physiology
2021

Diagnosing dehydrated hereditary stomatocytosis due to a KCNN4 Gardos channel mutation: understanding challenges through study of a multi-generational family.

EJHaem
2022

Global PIEZO1 Gain-of-Function Mutation Causes Cardiac Hypertrophy and Fibrosis in Mice.

Cells
2022

Reticulocyte Maturation and Variant Red Blood Cells.

Frontiers in physiology
2021

Confounding factors in the diagnosis and clinical course of rare congenital hemolytic anemias.

Orphanet journal of rare diseases
2022

PIEZO1-gene gain-of-function mutations with lower limb lymphedema onset in an adult: Clinical, scintigraphic, and noncontrast magnetic resonance lymphography findings.

American journal of medical genetics. Part A
2021

[Diagnosis of congenital hemolytic anemia by comprehensive gene analysis: significance and limitations].

[Rinsho ketsueki] The Japanese journal of clinical hematology
2021

Complex Modes of Inheritance in Hereditary Red Blood Cell Disorders: A Case Series Study of 155 Patients.

Genes
2021

Characterisation of Asp669Tyr Piezo1 cation channel activity in red blood cells: an unexpected phenotype.

British journal of haematology
2021

Trends in Piezo Channel Research Over the Past Decade: A Bibliometric Analysis.

Frontiers in pharmacology
2021

PIEZO1 mutation: a rare aetiology for fetal ascites.

BMJ case reports
2020

Targeted Next Generation Sequencing (NGS) to Diagnose Hereditary Hemolytic Anemias.

International journal of hematology-oncology and stem cell research
2020

A novel PIEZO1 mutation in a patient with dehydrated hereditary stomatocytosis: a case report and a brief review of literature.

Italian journal of pediatrics
2020

[Pathogenesis and diagnosis of hereditary stomatocytosis].

Zhonghua xue ye xue za zhi = Zhonghua xueyexue zazhi
2020

Low HbA1c With Normal Hemoglobin in a Diabetes Patient Caused by PIEZO1 Gene Variant: A Case Report.

Frontiers in endocrinology
2020

Inactive dimeric structure of the protease domain of stomatin operon partner protein.

Acta crystallographica. Section D, Structural biology
2020

Expression of South East Asian Ovalocytic Band 3 Disrupts Erythroblast Cytokinesis and Reticulocyte Maturation.

Frontiers in physiology
2020

RBCs prevent rapid PIEZO1 inactivation and expose slow deactivation as a mechanism of dehydrated hereditary stomatocytosis.

Blood
2020

[A case of hereditary stomatocytosis with Gilbert syndrome and secondary hemochromatosis].

Zhonghua nei ke za zhi
2020

Gain-of-function mutations in PIEZO1 directly impair hepatic iron metabolism via the inhibition of the BMP/SMADs pathway.

American journal of hematology
2019

Advances in understanding the pathogenesis of red cell membrane disorders.

British journal of haematology
2019

[Hereditary stomatocytosis with PIEZO1 gene mutations: report of five cases and literature review].

Zhonghua xue ye xue za zhi = Zhonghua xueyexue zazhi
2019

Dehydrated hereditary stomatocytosis: clinical perspectives.

Journal of blood medicine
2019

[Red cell membrane disorders and thalassemia].

[Rinsho ketsueki] The Japanese journal of clinical hematology
2019

PIEZO1 Hypomorphic Variants in Congenital Lymphatic Dysplasia Cause Shape and Hydration Alterations of Red Blood Cells.

Frontiers in physiology
2019

Fluorescence microscopy of piezo1 in droplet hydrogel bilayers.

Channels (Austin, Tex.)
2019

Dietary fatty acids fine-tune Piezo1 mechanical response.

Nature communications
2018

Hereditary stomatocytosis: an unusual cause of severe neonatal jaundice.

Singapore medical journal
2018

Genotype-phenotype correlation and risk stratification in a cohort of 123 hereditary stomatocytosis patients.

American journal of hematology
2018

Inherited or acquired modifiers of iron status may dramatically affect the phenotype in dehydrated hereditary stomatocytosis.

European journal of haematology
2019

Dehydrated Hereditary Stomatocytosis Presenting as Severe Perinatal Ascites and Cholestasis.

Journal of pediatric gastroenterology and nutrition
2018

Targeted next generation sequencing for the diagnosis of patients with rare congenital anemias.

European journal of haematology
2018

Dehydrated hereditary stomatocytosis causing fetal hydrops and perinatal ascites.

British journal of haematology
2018

Use of Laser Assisted Optical Rotational Cell Analyzer (LoRRca MaxSis) in the Diagnosis of RBC Membrane Disorders, Enzyme Defects, and Congenital Dyserythropoietic Anemias: A Monocentric Study on 202 Patients.

Frontiers in physiology
2018

The Molecular Basis for Altered Cation Permeability in Hereditary Stomatocytic Human Red Blood Cells.

Frontiers in physiology
2018

Dehydrated hereditary stomatocytosis: Prenatal management of ascites and pleural effusions.

Taiwanese journal of obstetrics & gynecology
2018

Human phenotypes caused by PIEZO1 mutations; one gene, two overlapping phenotypes?

The Journal of physiology
2018

Importance of the Average Glucose Level and Estimated Glycated Hemoglobin in a Diabetic Patient with Hereditary Hemolytic Anemia and Liver Cirrhosis.

Internal medicine (Tokyo, Japan)
2018

PIEZO1-R1864H rare variant accounts for a genetic phenotype-modifier role in dehydrated hereditary stomatocytosis.

Haematologica
2018

Hereditary stomatocytosis: An underdiagnosed condition.

American journal of hematology
2017

[A Review of 7 Cases of Laparoscopic Cholecystectomy for Pediatric Cholecystolithiasis].

Journal of UOEH
2017

[Clinical features of hereditary stomatocytosis: 12 cases report and literatures review].

Zhonghua xue ye xue za zhi = Zhonghua xueyexue zazhi
2017

'Gardos Channelopathy': a variant of hereditary Stomatocytosis with complex molecular regulation.

Scientific reports
2017

Red cell membrane disorders.

International journal of laboratory hematology
2016

New insights on hereditary erythrocyte membrane defects.

Haematologica
2016

A hypothesis of target cell formation in sickle cell disease.

Medical hypotheses
2016

Red blood cell-derived microparticles: An overview.

Blood cells, molecules & diseases
2016

Band 3, the human red cell chloride/bicarbonate anion exchanger (AE1, SLC4A1), in a structural context.

Biochimica et biophysica acta
2016

PIEZO1 gene mutation in a Japanese family with hereditary high phosphatidylcholine hemolytic anemia and hemochromatosis-induced diabetes mellitus.

International journal of hematology
2016

Dehydrated hereditary stomatocytosis.

American journal of hematology
2015

Impaired PIEZO1 function in patients with a novel autosomal recessive congenital lymphatic dysplasia.

Nature communications
2015

Novel mutations in PIEZO1 cause an autosomal recessive generalized lymphatic dysplasia with non-immune hydrops fetalis.

Nature communications
2015

Novel Gardos channel mutations linked to dehydrated hereditary stomatocytosis (xerocytosis).

American journal of hematology
2015

Dehydrated hereditary stomatocytosis masquerading as MDS.

Blood
2015

ICSH guidelines for the laboratory diagnosis of nonimmune hereditary red cell membrane disorders.

International journal of laboratory hematology
Ver todos os 105 no EuropePMC

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Doenças relacionadas

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Referências e fontes

Bases de dados externas citadas neste artigo

Publicações científicas

Artigos indexados no PubMed ligados a esta doença no grafo RarasNet — título, periódico e PMID direto da fonte, sem intermediação de IA.

  1. The Significance of the Piezo1-Mediated Mechanotransduction Pathway in Normal Morphogenesis.
    DNA and cell biology· 2026· PMID 41645043mais citado
  2. Clinical clues to recognizing hereditary dehydrated stomatocytosis (DHSt) in children.
    Archivos argentinos de pediatria· 2026· PMID 41842708mais citado
  3. Additive effect of multiple genetic variants in SEC23B and PIEZO1 on iron metabolism dyshomeostasis in hereditary anemias.
    HemaSphere· 2026· PMID 41657939mais citado
  4. A Rare Combination of High-Affinity Hemoglobin, Non-Transfusion-Dependent Thalassemia (Αlpha-Triplication and Codon 39 Mutation), and Hereditary Stomatocytosis.
    American journal of hematology· 2026· PMID 41641815mais citado
  5. The evolving landscape of hereditary stomatocytosis.
    Blood· 2025· PMID 40233304mais citado
  6. [Dehydrated hereditary stomatocytosis in 23 cases: a single-center retrospective cohort study from Peking Union Medical College Hospital (2018-2024)].
    Zhonghua Xue Ye Xue Za Zhi· 2025· PMID 41407458recente

Bases de dados e fontes oficiais

Identificadores e referências canônicas usadas para montar este verbete.

  1. ORPHA:98365(Orphanet)
  2. MONDO:0020102(MONDO)
  3. GARD:19456(GARD (NIH))
  4. Variantes catalogadas(ClinVar)
  5. Busca completa no PubMed(PubMed)
  6. Q3973817(Wikidata)

Dados compilados pelo RarasNet a partir de fontes abertas (Orphanet, OMIM, MONDO, PubMed/EuropePMC, ClinicalTrials.gov, DATASUS, PCDT/MS). Este conteúdo é informativo e não substitui avaliação médica.

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Estomatocitose
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Estomatocitose

ORPHA:98365 · MONDO:0020102
CID-10
D58.8 · Outras anemias hemolíticas hereditárias especificadas
CID-11
Medicamentos
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MedGen
UMLS
C1262483
EuropePMC
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