Death from cerebrovascular infarction in a patient with PHACES syndrome

Espuñes, S. Prieto; Santos‐Juanes, Jorge; Villanueva, A. Medina; Concha-Torre, Andrés; Galán, Corsino Rey; Río, José Sánchez del

doi:10.1016/j.jaad.2003.07.028

Cited by 11 publications

(3 citation statements)

References 8 publications

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“…We are aware of at least six other possible reports in patients with known cerebrovascular anomalies who developed the acute onset of seizures, hemiparesis, aphasia, and/or progressive neurologic deficits, including one of our cases (NW‐1) [Billson and Gillam, 1984; Pascual‐Castroviejo et al, 1995; Frieden et al, 1996; Aeby et al, 2003; Luo et al, 2003; Bhattacharya et al, 2004; however, incomplete information prevents definitive diagnosis in these patients. Similarly, two other reports describe cerebral infarction in PHACE patients following surgery, though it cannot be determined in either case whether this was a complication of PHACE, surgery, or both [Slavotinek et al, 2002; Espunes et al, 2004. The diagnosis of pediatric AIS, especially during infancy, is probably often missed due to subtle and/or transient manifestations.…”

Section: Discussionmentioning

confidence: 98%

The pyrolysis behaviors of polyimide foam derived from 3,3′,4,4′‐benzophenone tetracarboxylic dianhydride/4,4′‐oxydianiline

Shen

Zhan

Wang

et al. 2009

J of Applied Polymer Sci

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ABSTRACT:The thermal stability and pyrolysis behaviors of polyimide (PI) foam derived from 3,3 0 ,4,4 0 -benzophenone tetracarboxylic dianhydride (BTDA)/4,4 0 -oxydianiline (4,4 0 -ODA) in air and in nitrogen were studied. The decomposition products of PI foam were analyzed by thermogravimetry-Fourier transform infrared spectroscopy (TG-FTIR). Several integral and differential methods reported in the literatures were used in decomposition kinetics analysis of PI foam. The results indicated that the PI foam was easier to decompose in air than in nitrogen, with $ 55% residue remaining in nitrogen versus zero in air at 800 o C. The main pyrolysis products were CO 2 , CO, and H 2 O in air and CO 2 , CO, H 2 O, and small organic molecules in nitrogen. The different dynamic methods gave similar results that the apparent activation energies, pre-exponential factors, and reaction orders were higher in nitrogen than those in air.

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Section: Discussionmentioning

confidence: 98%

The pyrolysis behaviors of polyimide foam derived from 3,3′,4,4′‐benzophenone tetracarboxylic dianhydride/4,4′‐oxydianiline

Shen

Zhan

Wang

et al. 2009

J of Applied Polymer Sci

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“…We are aware of at least six other possible reports in patients with known cerebrovascular anomalies who developed the acute onset of seizures, hemiparesis, aphasia, and/or progressive neurologic deficits, including one of our cases (NW-1) [Billson and Gillam, 1984;Pascual-Castroviejo et al, 1995;Frieden et al, 1996;Aeby et al, 2003;Luo et al, 2003;Bhattacharya et al, 2004]; however, incomplete information prevents definitive diagnosis in these patients. Similarly, two other reports describe cerebral infarction in PHACE patients following surgery, though it cannot be determined in either case whether this was a complication of PHACE, surgery, or both [Slavotinek et al, 2002;Espunes et al, 2004]. The diagnosis of pediatric AIS, especially during infancy, is probably often missed due to subtle and/or transient manifestations.…”

Section: Cerebral and Cerebrovascular Anomaliesmentioning

confidence: 97%

A prospective study of PHACE syndrome in infantile hemangiomas: Demographic features, clinical findings, and complications

Metry

Haggstrom

Drolet

et al. 2006

American J of Med Genetics Pt A

274

210

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PHACE (OMIM no. 606519) is a neurocutaneous syndrome that refers to the association of large, plaque-like, "segmental" hemangiomas of the face, with one or more of the following anomalies: posterior fossa brain malformations, arterial cerebrovascular anomalies, cardiovascular anomalies, eye anomalies, and ventral developmental defects, specifically sternal defects and/or supraumbilical raphe. The etiology and pathogenesis of PHACE is unknown, and potential risk factors for the syndrome have not been systematically studied. The purpose of this study was thus to determine (1) the incidence of PHACE and associated anomalies among a large cohort of hemangioma patients, (2) whether certain demographic, prenatal or perinatal risk factors predispose infants to this syndrome, and (3) whether the cutaneous distribution of the hemangioma can be correlated to the types of anomalies present. We undertook a prospective, cohort study of 1,096 children with hemangiomas, 25 of whom met criteria for PHACE. These 25 patients represented 20% of infants with segmental facial hemangiomas. Compared to previous reports, our PHACE patients had a higher incidence of cerebrovascular and cardiovascular anomalies. Two developed acute arterial ischemic stroke during infancy, while two with cardiovascular anomalies showed documented evidence of normalization, suggesting that both progressive and regressive vascular phenomena may occur in this syndrome. Correlation to the anatomic location of the hemangioma appears to be helpful in determining which structural abnormalities might be present. A comparison of demographic and perinatal data between our PHACE cases and the hemangioma cohort overall showed no major differences, except a trend for PHACE infants to be of slighter higher gestational age and born to slightly older mothers. Eighty-eight percent were female, a finding which has been noted in multiple other reports. Further research is needed to determine possible etiologies, optimal evaluation, and outcomes.

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“…Several of the neurologic symptoms described in PHACES patients such as seizures, hemiparesis, aphasia and/ or progressive neurologic deficits may result from unrecognized pediatric AIS [4,[20][21][22][23][24]. Two other reports describe cerebral infarction in PHACES patients following surgery, though it cannot be determined in either case whether this was a complication of PHACES, surgery, or both [25,26]. Similarly, unrecognized AIS may also explain the nonspecific neuroimaging findings reported, in particular cerebral hypoplasia, cortical dysplasia and frontal lobe calcification.…”

Section: Arterial Cerebrovascular Anomaliesmentioning

confidence: 99%