A case of multiple arteriovenous malformations and diffuse venous abnormalities with facial port-wine stain

Fukushima, Takashi; Hamano, Kenzo; Shin, Kenji; Kawashima, Koichiro; Fujiwara, J.; Anno, Izumi

doi:10.1007/bf00571122

Cited by 14 publications

(3 citation statements)

References 17 publications

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“…Other congenital syndromes associated with port‐wine stains include the Klippel‐Trenaunay‐Weber syndrome with the associated hypertrophy of skeletal tissue, 12 as well as the Cobb syndrome, which may be a sign of underlying arteriovenous malformation of the spinal cord 13,14 . It is also not uncommon to see a port‐wine stain overlying an arteriovenous malformation, arterial malformation, or venous malformation, 15,16 therefore requiring the physician to look beyond the skin for any underlying problem.…”

Section: Percent Of Patients Showing Thickening or Hypertrophy Of Pormentioning

confidence: 99%

The Medical Necessity of Evaluation and Treatment of Port‐Wine Stains

Geronemus

Ashinoff

1991

The Journal of Dermatologic Surgery and Oncology

168

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New lasers and improved laser delivery systems have allowed for the safe and effective treatment of port-wine stains in patients of all ages. The satisfactory results obtained by laser treatment have increased the number of patients seeking consultation regarding their birthmarks. It is imperative that physicians recognize the various medical syndromes and problems associated with port-wine stains. A review of 415 patients with facial port-wine stains has revealed hypertrophy and/or nodularity in 65% of patients by the fifth decade of life, which increases significantly the morbidity of these lesions. It is believed that laser treatment will minimize the medical and psychologic complications that result from the natural evolution of port-wine stains.

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Section: Percent Of Patients Showing Thickening or Hypertrophy Of Pormentioning

confidence: 99%

The Medical Necessity of Evaluation and Treatment of Port‐Wine Stains

Geronemus

Ashinoff

1991

The Journal of Dermatologic Surgery and Oncology

168

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“…Two other case reports demonstrated SWS variants that were associated with multiple AVMs as well as venous sinus occlusion, but neither case showed progressive brain edema as noted in our case. 7,13 All reported cases of SWS associated with AVM occurred in infants, but the underlying mechanism for such an association remains unknown. Perhaps the failed brain vessel differentiation connected with cases of SWS that are associated with an AVM occurs at a different stage than what is accepted for classic cases of SWS.…”

Section: Discussionmentioning

confidence: 99%

Sturge-Weber syndrome associated with arteriovenous malformation in a patient presenting with progressive brain edema and cyst formation

Nishino¹,

Ito²,

Sorimachi³

et al. 2010

PED

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Sturge-Weber syndrome (SWS) is a neurocutaneous disorder presenting with a facial port-wine stain, along with an occipital leptomeningeal angiomatosis that is typically located ipsilateral to the stain. In this paper, the authors present a rare case of SWS associated with an arteriovenous malformation (AVM) instead of an angiomatosis in the ipsilateral occipital lobe. While the patient was in the care of the authors, the AVM progressively enlarged, and was accompanied by progressive stenoocclusive changes of the venous system. The resulting brain edema finally brought about a serious neurological condition 13 years after the initial diagnosis. Transarterial embolization and medical treatments decreased the edema. Subsequently, however, a large intraparenchymal cyst appeared, aggravating the patient's motor weakness. Aspiration of the cyst ameliorated these symptoms. The analysis of the fluid from the cyst revealed that it contained a very high concentration of protein. Although there is no proven pathogenic mechanism to explain these protein concentrations and the enlargement of the AVM, the authors hypothesize that the progressive edema resulted from a synergic augmentation of the inflow from the AVM and the progressive obstruction of venous drainage that is a hallmark of SWS. The formation of the cyst probably resulted from the blood vessel hyperpermeability that is inherent to SWS.

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“…Of these, 669 were duplicates and 2,348 were unique citations. This search yielded 12 citations meeting the criteria for inclusion and contained reports of 21 patients [13,14,15,16,17,18,19,20,21,22,23,24]. Reports of 2 additional patients were found in 2 references from the included papers [25,26].…”

Section: Resultsmentioning

confidence: 99%

Hemorrhage Risk in Pediatric Patients with Multiple Intracranial Arteriovenous Malformations

et al. 2016

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Background/Aims: This study aims to characterize the clinical features, treatment strategies, and annual hemorrhage incidence rate of pediatric patients with multiple arteriovenous malformations (MAVM). Methods: The PubMed and EMBASE databases and the arteriovenous malformations (AVM) database at the Johns Hopkins Hospital were searched for reports of pediatric patients (under 21 years of age) with MAVM. Data related to demographics, clinical features, management, and treatment outcomes were analyzed using descriptive statistics. Twenty-four pediatric patients met the inclusion criteria. Results: The annual hemorrhage incidence rate was 2.1%. The most common presenting features were neurological deficit (38%) and hemorrhage (21%). Treatment with embolization has become the most frequently used modality. In patients undergoing staged treatment of MAVM, hemorrhage of an untreated nidus (n = 1), visualization of a new nidus (n = 2), or disappearance of a draining vein (n = 1) occurred. Conclusion: The annual hemorrhage incidence rate for pediatric patients with MAVM approaches the upper range of previously reported hemorrhage rates for solitary AVM. A staged approach to treating MAVM requires close follow-up as changes to the remaining nidi may occur during the latency period. Limitations of this study include its small sample size and reporting bias.

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A case of multiple arteriovenous malformations and diffuse venous abnormalities with facial port-wine stain

Cited by 14 publications

References 17 publications

The Medical Necessity of Evaluation and Treatment of Port‐Wine Stains

The Medical Necessity of Evaluation and Treatment of Port‐Wine Stains

Sturge-Weber syndrome associated with arteriovenous malformation in a patient presenting with progressive brain edema and cyst formation

Hemorrhage Risk in Pediatric Patients with Multiple Intracranial Arteriovenous Malformations

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