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Capillary malformation-arteriovenous malformation 1(CMAVM1)

MedGen UID:
1648501
Concept ID:
C4747394
Disease or Syndrome
Synonym: CMAVM1
Modes of inheritance:
Autosomal dominant inheritance
MedGen UID:
141047
Concept ID:
C0443147
Intellectual Product
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on one of the autosomes (i.e., the human chromosomes 1-22) in which a trait manifests in heterozygotes. In the context of medical genetics, an autosomal dominant disorder is caused when a single copy of the mutant allele is present. Males and females are affected equally, and can both transmit the disorder with a risk of 50% for each child of inheriting the mutant allele.
Not genetically inherited
MedGen UID:
988794
Concept ID:
CN307044
Finding
Source: Orphanet
clinical entity without genetic inheritance.
 
Gene (location): RASA1 (5q14.3)
 
Monarch Initiative: MONDO:0020783
OMIM®: 608354
Orphanet: ORPHA90307

Disease characteristics

Capillary malformation-arteriovenous malformation (CM-AVM) syndrome is characterized by the presence of multiple small (1-2 cm in diameter) capillary malformations mostly localized on the face and limbs. Some affected individuals also have associated arteriovenous malformations (AVMs) and/or arteriovenous fistulas (AFVs), fast-flow vascular anomalies that typically arise in the skin, muscle, bone, spine, and brain; life-threatening complications of these lesions can include bleeding, congestive heart failure, and/or neurologic consequences. Symptoms from intracranial AVMs/AVFs appear to occur early in life. Several individuals have Parkes Weber syndrome (multiple micro-AVFs associated with a cutaneous capillary stain and excessive soft-tissue and skeletal growth of an affected limb). [from GeneReviews]
Authors:
Pinar Bayrak-Toydemir  |  David A Stevenson   view full author information

Additional descriptions

From OMIM
Capillary malformation-arteriovenous malformation-1 (CMAVM1) is an autosomal dominant disorder characterized by atypical capillary malformations (CMs), often in association with fast-flow vascular malformations, including arteriovenous malformations (AVMs) and arteriovenous fistulas (AVFs), and Parkes Weber syndrome (PKWS). The CMs are usually multifocal and are surrounded by a pale halo with a central red dot; they increase in number with age. The AVMs generally occur in the brain or on the face or extremities. Intracranial AVMs include vein of Galen aneurysmal malformations (VGAMs). Parkes Weber syndrome is a specific type of CMAVM that presents with limb overgrowth, more commonly affecting one of the lower extremities (Eerola et al., 2003; Revencu et al., 2013; Johnson and Navarro, 2017). Parkes Weber syndrome is characterized by a cutaneous blush with underlying multiple micro-AVFs in association with soft-tissue and skeletal hypertrophy of the affected limb (Mulliken and Young, 1988). Genetic Heterogeneity of Capillary Malformation-Arteriovenous Malformation Also see CMAVM2 (618196), caused by mutation in the EPHB4 gene on chromosome 7q22.  http://www.omim.org/entry/608354
From MedlinePlus Genetics
Capillary malformation-arteriovenous malformation syndrome (CM-AVM) is a disorder of the vascular system, which is the body's complex network of blood vessels. The vascular system consists of arteries, which carry oxygen-rich blood from the heart to the body's various organs and tissues; veins, which carry blood back to the heart; and capillaries, which are tiny blood vessels that connect arteries and veins.

CM-AVM is characterized by capillary malformations (CMs), which are composed of enlarged capillaries that increase blood flow near the surface of the skin. These malformations look like multiple small, round, pink or red spots on the skin. In most affected individuals, capillary malformations occur on the face, arms, and legs. These spots may be visible from birth or may develop during childhood. By themselves, capillary malformations usually do not cause any health problems.

In some people with CM-AVM, capillary malformations are the only sign of the disorder. However, other affected individuals also have more serious vascular abnormalities known as arteriovenous malformations (AVMs) and arteriovenous fistulas (AVFs). AVMs and AVFs are abnormal connections between arteries, veins, and capillaries that affect blood circulation. Depending on where they occur in the body, these abnormalities can be associated with complications including abnormal bleeding, migraine headaches, seizures, and heart failure. In some cases the complications can be life-threatening. In people with CM-AVM, complications of AVMs and AVFs tend to appear in infancy or early childhood; however, some of these vascular abnormalities never cause any symptoms.

Some vascular abnormalities seen in CM-AVM are similar to those that occur in a condition called Parkes Weber syndrome. In addition to vascular abnormalities, Parkes Weber syndrome usually involves overgrowth of one limb. CM-AVM and some cases of Parkes Weber syndrome have the same genetic cause.  https://medlineplus.gov/genetics/condition/capillary-malformation-arteriovenous-malformation-syndrome

Clinical features

From HPO
Capillary hemangioma
MedGen UID:
64643
Concept ID:
C0206733
Neoplastic Process
The presence of a capillary hemangioma, which are hemangiomas with small endothelial spaces.
Arteriovenous fistula
MedGen UID:
2041
Concept ID:
C0003855
Anatomical Abnormality
An abnormal connection between an artery and vein.
Arteriovenous malformation
MedGen UID:
2042
Concept ID:
C0003857
Congenital Abnormality
An anomalous configuration of blood vessels that shunts arterial blood directly into veins without passing through the capillaries.
Capillary malformation
MedGen UID:
90955
Concept ID:
C0340803
Congenital Abnormality
A capillary malformation is a flat, sharply defined vascular stain of the skin. It may cover a large surface area or it may be scattered and appear as little islands of color. In a capillary maformation, the predominant vessels are small, slow-flow vessels (i.e., arterioles and postcapillary venules).

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVCapillary malformation-arteriovenous malformation 1

Professional guidelines

PubMed

Orme CM, Boyden LM, Choate KA, Antaya RJ, King BA
Pediatr Dermatol 2013 Jul-Aug;30(4):409-15. Epub 2013 May 13 doi: 10.1111/pde.12112. PMID: 23662773
Greene AK, Orbach DB
Clin Plast Surg 2011 Jan;38(1):95-106. doi: 10.1016/j.cps.2010.08.005. PMID: 21095475

Recent clinical studies

Etiology

Handa A, Tsujioka Y, Nishimura G, Nozaki T, Kono T, Jinzaki M, Harms T, Connolly SA, Sato TS, Sato Y
Radiographics 2024 May;44(5):e230153. doi: 10.1148/rg.230153. PMID: 38602868
Palit A, Inamadar AC
Indian J Dermatol Venereol Leprol 2022 May-Jun;88(4):452-463. doi: 10.25259/IJDVL_799_20. PMID: 35138057
Cao H, Alrejaye N, Klein OD, Goodwin AF, Oberoi S
Orthod Craniofac Res 2017 Jun;20 Suppl 1(Suppl 1):32-38. doi: 10.1111/ocr.12144. PMID: 28643916Free PMC Article
Aoki Y, Niihori T, Inoue S, Matsubara Y
J Hum Genet 2016 Jan;61(1):33-9. Epub 2015 Oct 8 doi: 10.1038/jhg.2015.114. PMID: 26446362
Rauen KA
Annu Rev Genomics Hum Genet 2013;14:355-69. Epub 2013 Jul 15 doi: 10.1146/annurev-genom-091212-153523. PMID: 23875798Free PMC Article

Diagnosis

Mahajan P, Bergstrom KL, Phung TL, Metry DW
Clin Dermatol 2022 Jul-Aug;40(4):313-321. Epub 2022 Feb 16 doi: 10.1016/j.clindermatol.2022.02.006. PMID: 35181412
Palit A, Inamadar AC
Indian J Dermatol Venereol Leprol 2022 May-Jun;88(4):452-463. doi: 10.25259/IJDVL_799_20. PMID: 35138057
Valdivielso-Ramos M, Martin-Santiago A, Azaña JM, Hernández-Nuñez A, Vera A, Perez B, Tercedor J, Feito M, Vicente A, Prat C, Lopez-Gutierrez JC, Garnacho G, Baselga E, Roe E, Palencia S, Cordero P, Moreno R, Agudo A, de la Cueva P, Torrelo A
Clin Exp Dermatol 2021 Mar;46(2):300-305. Epub 2020 Oct 20 doi: 10.1111/ced.14428. PMID: 32840927
Cao H, Alrejaye N, Klein OD, Goodwin AF, Oberoi S
Orthod Craniofac Res 2017 Jun;20 Suppl 1(Suppl 1):32-38. doi: 10.1111/ocr.12144. PMID: 28643916Free PMC Article
Greene AK, Orbach DB
Clin Plast Surg 2011 Jan;38(1):95-106. doi: 10.1016/j.cps.2010.08.005. PMID: 21095475

Therapy

Iznardo H, Roé E, Puig L, Vikula M, López-Sánchez C, Baselga E
Pediatr Dermatol 2020 Mar;37(2):342-344. Epub 2020 Jan 15 doi: 10.1111/pde.14095. PMID: 31944370
Plumptre I, Robertson F, Rennie A, James G, Syed SB
Pediatr Dermatol 2020 Jan;37(1):162-164. Epub 2019 Nov 20 doi: 10.1111/pde.14029. PMID: 31746477
Stevenson DA, Schill L, Schoyer L, Andresen BS, Bakker A, Bayrak-Toydemir P, Burkitt-Wright E, Chatfield K, Elefteriou F, Elgersma Y, Fisher MJ, Franz D, Gelb BD, Goriely A, Gripp KW, Hardan AY, Keppler-Noreuil KM, Kerr B, Korf B, Leoni C, McCormick F, Plotkin SR, Rauen KA, Reilly K, Roberts A, Sandler A, Siegel D, Walsh K, Widemann BC
Am J Med Genet A 2016 Aug;170(8):1959-66. Epub 2016 May 7 doi: 10.1002/ajmg.a.37723. PMID: 27155140Free PMC Article

Prognosis

Engel ER, Wusik K, Bright P, Vadivelu S, Taylor JM, Hammill A
J Pediatr 2024 Jan;264:113761. Epub 2023 Oct 4 doi: 10.1016/j.jpeds.2023.113761. PMID: 37797790
Say M, Tella E, Boccara O, Sauvage M, Bourrat E, Tian Y, Monfort JB, Lok C, Desierier F, Beneton N, Abasq-Thomas C, Kupfer-Bessaguet I, Mallet S, Lacour JP, Plantin P, Sigal ML, Mazereeuw-Hautier J, Mahé E; on behalf the Angio-Dermatology Group of the French Society of Dermatology, the Research Group of the French Society of Pediatric Dermatology
Ann Dermatol Venereol 2022 Mar;149(1):51-55. Epub 2021 Jul 1 doi: 10.1016/j.annder.2021.05.004. PMID: 34218940
Valdivielso-Ramos M, Martin-Santiago A, Azaña JM, Hernández-Nuñez A, Vera A, Perez B, Tercedor J, Feito M, Vicente A, Prat C, Lopez-Gutierrez JC, Garnacho G, Baselga E, Roe E, Palencia S, Cordero P, Moreno R, Agudo A, de la Cueva P, Torrelo A
Clin Exp Dermatol 2021 Mar;46(2):300-305. Epub 2020 Oct 20 doi: 10.1111/ced.14428. PMID: 32840927
Walcott BP, Smith ER, Scott RM, Orbach DB
J Neurointerv Surg 2013 Jan 1;5(1):10-4. Epub 2012 Jan 2 doi: 10.1136/neurintsurg-2011-010168. PMID: 22213836
Chee D, Phillips R, Maixner W, Southwell BR, Hutson JM
J Pediatr Surg 2010 Dec;45(12):2419-22. doi: 10.1016/j.jpedsurg.2010.08.043. PMID: 21129558

Clinical prediction guides

Engel ER, Wusik K, Bright P, Vadivelu S, Taylor JM, Hammill A
J Pediatr 2024 Jan;264:113761. Epub 2023 Oct 4 doi: 10.1016/j.jpeds.2023.113761. PMID: 37797790
El Hajjam M, Mekki A, Palmyre A, Eyries M, Soubrier F, Bourgault Villada I, Ozanne A, Carlier RY, Chinet T
J Med Genet 2021 Sep;58(9):645-647. Epub 2020 Sep 8 doi: 10.1136/jmedgenet-2019-106792. PMID: 32900839
Valdivielso-Ramos M, Martin-Santiago A, Azaña JM, Hernández-Nuñez A, Vera A, Perez B, Tercedor J, Feito M, Vicente A, Prat C, Lopez-Gutierrez JC, Garnacho G, Baselga E, Roe E, Palencia S, Cordero P, Moreno R, Agudo A, de la Cueva P, Torrelo A
Clin Exp Dermatol 2021 Mar;46(2):300-305. Epub 2020 Oct 20 doi: 10.1111/ced.14428. PMID: 32840927
Saliou G, Eyries M, Iacobucci M, Knebel JF, Waill MC, Coulet F, Ozanne A, Soubrier F
Ann Neurol 2017 Dec;82(6):972-980. Epub 2017 Dec 12 doi: 10.1002/ana.25106. PMID: 29171923
Chee D, Phillips R, Maixner W, Southwell BR, Hutson JM
J Pediatr Surg 2010 Dec;45(12):2419-22. doi: 10.1016/j.jpedsurg.2010.08.043. PMID: 21129558

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