Cerebral cavernous angiomas: an atypical case in infancy

Lanzi, G.; Fazzi, Elisa; Orcesi, Simona; Danova, S.; Uggetti, Carla; Egitto, Maria Grazia

doi:10.1007/s003810050110

Cited by 5 publications

(4 citation statements)

References 6 publications

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“…In contrast, another series of 13 pediatric patients who received conservative management indicated satisfactory outcomes: all but one of these patients were either improved (n = 4) or stabilized (n = 8, including 6 asymptomatic patients); the other patient died of a recurrent medulloblastoma. 11 The benign clinical course with or without the regression of CMs resembled the findings in our 24 patients who received conservative management and another report, 34 in which a 10-month-old patient with a pontine CM made a complete recovery at 15 days after diagnosis and exhibited normal development during 1 year of follow-up. Heterogeneity in these results primarily reflects the distinct referral bias and significant patient demographic diversity and is partially attributed to limited follow-up duration and small sample size.…”

Section: Natural Historysupporting

confidence: 58%

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Surgical management of pediatric brainstem cavernous malformations

Li¹,

Hao²,

Tang³

et al. 2014

PED

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Object The goal of this study was to evaluate surgical outcomes of pediatric brainstem cavernous malformations (CMs) and identify the risk factors associated with postoperative full recovery and rebleeding. Methods The clinical charts and radiographs from a series of 52 pediatric patients (37 male and 15 female; mean age 12.2 years; range 1–17 years) who underwent surgery for brainstem CMs between 1996 and 2011 were reviewed. Follow-up evaluation measures were obtained retrospectively. Neurological function was evaluated using the modified Rankin Scale (mRS) score. Results The lesion locations among the 52 patients included the midbrain (n = 7, 13.5%), pons (n = 38, 73.1%), and medulla (n = 7, 13.5%). The mean duration of symptoms was 18.5 months, and the preoperative annual hemorrhage and rebleeding rates were 12.3% and 32.5% per patient-year, respectively. The mean lesion size was 2.1 cm. Gross-total resection without surgery-related death was achieved in 49 patients (94.2%). Immediate postoperative reduced neurological function was observed in 17 patients (32.7%). Surgical morbidities developed in 25 patients (48.1%) and remained in 11 patients (21.2%) after 7.9 years of follow-up. The mean mRS scores at admission, discharge after surgery, 3 and 6 months postsurgery, and recent evaluation were 2.0, 2.3, 2.0, 1.5, and 1.0, respectively. The postoperative mRS scores at 6 months (p < 0.001) and on recent evaluation (p < 0.001) were significantly lower than those at admission. Postoperative rebleeding occurred in 2 patients, and the postoperative annual rebleeding rate was 0.5% per patient-year. By the most recent evaluation, 10 patients (19.2%) had achieved full recovery and all patients were either improved (n = 32, 61.5%) or unchanged (n = 20, 38.5%). The adverse predictors for full recovery included age ≥ 12 years (HR 0.230, p = 0.021), ≥ 2 preoperative hemorrhages (HR 0.124, p = 0.048), and poor preoperative status (HR 0.197, p = 0.040). An HR < 1 predicted poor complete recoveries. The single risk factor predicting postoperative rebleeding was incomplete resection (χ2 = 4.340, p = 0.037). Conclusions Fair outcomes for pediatric brainstem CMs could be obtained through surgery, but only a few patients achieved full recovery. Thus, to minimize surgical morbidity, surgical planning must be tailored to individual patients in all cases in which an operation is warranted. Complete resection must be attempted to reduce the risk of postoperative rebleeding. The predictors associated with complete postoperative recovery were referential for determining treatment.

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Section: Natural Historysupporting

confidence: 58%

“…According to prior studies, 8,11,12,20,22,24,25,31,34,39,49 surgical candidates must be strictly evaluated and prudently selected for surgical indications. The experience and technique of the surgeon and the desires of the patient must also be considered.…”

Section: Treatment Philosophymentioning

confidence: 99%

Surgical management of pediatric brainstem cavernous malformations

Li¹,

Hao²,

Tang³

et al. 2014

PED

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“…Cavernomas are quite rare in the first year of life, and to our knowledge, only 39 cases have been reported since 1951 (Tables 1 and 2). 3,4,6–39 A few cases were detected antenatally 15,39 . The cavernoma in this study is one of the few in patients of this age group, which is familial with multiple lesions and in which the patient underwent MRI scanning.…”

Section: Discussionmentioning

confidence: 63%

“…Purely cystic lesions and lesions with peripheral oedema are also described 3 . To our knowledge, 14 of the previously studied patients in this age group underwent MRI scanning 4,21,25,26,29,32,34,36,37,39 …”

Section: Discussionmentioning

confidence: 99%

Familial cerebral cavernous haemangioma diagnosed in an infant with a rapidly growing cerebral lesion

Ng¹,

Mulyadi

Pereira³

et al. 2006

Australasian Radiology

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Cavernous haemangiomas of the central nervous system are vascular malformations best imaged by MRI. They may present at any age, but to our knowledge only 39 cases in the first year of life have previously been reported. A familial form has been described and some of the underlying genetic mutations have recently been discovered. We present the clinical features and serial MRI findings of an 8-week-old boy who presented with subacute intracranial haemorrhage followed by rapid growth of a surgically proven cavernous haemangioma, mimicking a tumour. He also developed new lesions. A strong family history of neurological disease was elucidated. A familial form of cavernous haemangioma was confirmed by identification of a KRIT 1 gene mutation and cavernous haemangiomas in the patient and other family members. We stress the importance of considering cavernous haemangiomas in the context of intracerebral haemorrhage and in the differential diagnosis of rapidly growing lesions in this age group. The family history is also important in screening for familial disease.

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