Skip to main content
Hauptrubriken
CME Facharzt-Training Webinare Zeitschriften Bücher e.Medpedia Podcast
Service
Jobbörse Info & Hilfe
Fachgebiete
Anästhesiologie Allgemeinmedizin Arbeitsmedizin Augenheilkunde Chirurgie Dermatologie Gynäkologie und Geburtshilfe HNO Innere Medizin Kardiologie Neurologie Onkologie und Hämatologie Orthopädie und Unfallchirurgie Pädiatrie Pathologie Psychiatrie Radiologie Rechtsmedizin Urologie Zahnmedizin
Themen
Klimawandel und Gesundheit Neues aus dem Markt COVID-19 Praxis und Beruf Seltene Erkrankungen GOÄ & EBM Panorama
Sammlungen
Kasuistiken Algorithmen & Infografiken Blickdiagnosen Arthropedia FlapFinder (für Dermatochirurgen) Videos Kongressberichterstattung Medizin für Apothekerinnen und Apotheker Für Ärztinnen und Ärzte in Weiterbildung Für Medizinstudierende
Erweiterte Suche
Anmelden
nach oben
Child's Nervous System
Erschienen in: Child's Nervous System 9/2017

20.06.2017 | Original Paper

Cavernous malformations of central nervous system in pediatric patients: our single-centered experience in 50 patients and review of literature

verfasst von: Dattaraj Paramanand Sawarkar, Suveen Janmatti, Rajinder Kumar, Pankaj Kumar Singh, Hitesh Kumar Gurjar, Shashank Sharad Kale, Bhawani Shanker Sharma, Ashok Kumar Mahapatra

Erschienen in: Child's Nervous System | Ausgabe 9/2017

Einloggen, um Zugang zu erhalten

Abstract

Purpose

Cavernous malformations (CMs) are rare developmental cerebrovascular malformations of the central nervous system with a childhood prevalence of 0.3 to 0.53%. Our purpose was to assess the clinical features and microsurgical outcome in pediatric central nervous system (CNS) CMs.

Material and methods

We retrospectively enrolled all the CM patients admitted to our institute from 1 January 2001 to 31 December 2014. Data was analyzed for their clinical features and surgical outcome.

Results

A total of 50 patients with CMs (30 supratentorial, 14 infratentorial, and 6 spinal) with a mean age of 14 years (3–18 years, SD ±4.64) were enrolled into the study. Most of these patients (78%) were male. Size varied from 1.2 to 6 cm. Three patients had multiple CMs. Symptoms of CMs were site specific. Seizure was the most common symptom (63.3%) of CMs at supratentorial location followed by headache (46%) and neurodeficiency (26%), while all brainstem and spinal CMs presented with neurodeficiencies. History of clinically significant acute hemorrhage was present in 19.2% of supratentorial (ST) superficial CMs, 50% of ST deep CMs, 25% of cerebellar CMs, 44.4% of brainstem CMs, and 50% of spinal CMs. Forty-five CMs in 44 patients were surgically excised. Their follow-up ranged from 6 to 162 months (mean 47.2 months, SD ±53). All supratentorial CM patients showed improvement in their symptoms. Patients with preoperative seizure showed good seizure control with Engel scale I in 16 (94.1%) and Engel scale II in 1 (5.9%). In infratentorial (IT) and spinal CM patients, 92.3 and 66.7% had improvement in their neurodeficiencies, respectively. There was no mortality in our series.

Conclusion

Microsurgical excision of CNS CM results in excellent neurological outcome in pediatric patients. Early intervention is necessary in spinal CMs for better outcome.
Literatur
1.
Hoang T-A, Hasso AN (1994) Intracranial vascular malformations. Neuroimaging Clin N Am 4:823–842PubMed
2.
McCormick WF (1966) The pathology of vascular (‘arteriovenous’) malformations. J Neurosurg 24:807–816CrossRefPubMed
3.
Alexiou GA, Mpairamidis E, Sfakianos G, Prodromu N (2009) Surgical management of brain cavernomas in children. Pediatr Neurosurg 45:375–378CrossRefPubMed
4.
Rigamonti D, Hadley MN, Drayer BP (1988) Cerebral cavernous malformations: incidence and familial occurrence. N Engl J Med 319:343–347CrossRefPubMed
5.
Bergeson PS, Rekate HL, Tack ED (1992) Cerebral cavernous angiomas in the newborn. Clin Pediatr (Phila) 31:435–437CrossRef
6.
Mottolese C, Hermier M, Stan H, Jouvet A, Saint-Pierre G, Froment JC, Bret P, Lapras C (2001) Central nervous system cavernomas in the pediatric age group. Neurosurg Rev 24:55–73CrossRefPubMed
7.
Engel JJ, Van Ness PC, Rasmussen TB, Ojemann LM (1993) Outcome with respect to epileptic seizures. In: Engel JJ (ed) Surgical treatment of the epilepsies. Raven Press, New York, pp 609–621
8.
Manzano G, Green BA, Vanni S, Levi AD (2008) Contemporary management of adult intramedullary spinal tumors- pathology and neurological outcome related to surgical resection. Spinal Cord 46:540–546CrossRefPubMed
9.
Brown AP, Thompson BG, Spetzler RF (1996) The two-point method: evaluating brain stem lesions. BNI Q 12(1):20–24
10.
Kraemer DL, Awad IA (1994) Vascular malformations and epilepsy: clinical considerations and basic mechanisms. Epilepsia 35:S30–S43CrossRefPubMed
11.
Acciarri N, Galassi E, Giulioni M, Pozzati E, Grasso V, Palandri G, Badaloni F, Zucchelli M, Calbucci F (2009) Cavernous malformations of central nervous system in the pediatric age group. Pediatr Neurosurg 45:81–104CrossRefPubMed
12.
Hugelshofer M, Acciarri N, Sure U et al (2011) Effective surgical treatment of cerebral cavernous malformations: a multicenter study of 79 pediatric patients. J Neurosurg Pediatr 8:522–525CrossRefPubMed
13.
Giulioni M, Acciarri N, Padovani R, Galassi E (1995) Results of surgery in children with cerebral cavernous angiomas causing epilepsy. Br J Neurosurg 9:135–141CrossRefPubMed
14.
Mindea SA, Yang BP, Shenkar R, Bendok B, Batjer HH, Awad IA (2006) Cerebral cavernous malformations: clinical insights from genetic studies. Neurosurg Focus 21:e1PubMed
15.
Denier C, Labauge P, Brunereau L, Cavé-Riant F, Marchelli F, Arnoult M, Cecillon M, Maciazek J, Joutel A, Tournier-Lasserve E (2004) Clinical features of cerebral cavernous malformations patients with KRIT1 mutations. Ann Neurol 55(2):213e20CrossRef
16.
Ciricillo SF, Cogen PH, Edwards MS (1994) Pediatric cryptic vascular malformations: presentation, diagnosis and treatment. Pediatr Neurosurg 20:137–147CrossRefPubMed
17.
Curling OD, Kelly DL, Elster AD, Craven TE (1991) An analysis of the natural history of cavernous angiomas. J Neurosurg 75:702–708CrossRefPubMed
18.
Rigamonti D, Drayer BP, Johnson PC, Hadley MN, Zabramski J, Spetzler RF (1987) The MRI appearance of cavernous malformations (angiomas). J Neurosurg 67:518–524CrossRefPubMed
19.
Zabramski JM, Wascher TM, Spetzler RF, Johnson B, Golfinos J, Drayer B, Brown B, Rigamonti D, Brown G (1994) The natural history of familial cavernous malformations: results of an ongoing study. J Neurosurg 80:422–432CrossRefPubMed
20.
Amato MC, Madureira JF, Oliveira RS (2013) Intracranial cavernous malformations in children: a single-centered experience with 30 consecutive cases. Arq Neuropsiquiatr 71(4):220–228CrossRefPubMed
21.
Cavalheiro S, Braga FM (1999) Cavernous hemangiomas. In: Choux M, Di Rocco C, Hockley AD, Walker ML (eds) Pediatric neurosurgery. Churchill Livingstone, London, pp 691–701
22.
Bigi S, Capone Mori A, Steinlin M, Remonda L, Landolt H, Boltshauser E (2011) Cavernous malformations of the central nervous system in children: presentation, treatment and outcome of 20 cases. Eur J Paediatr Neurol 15(2):109–116. doi:10.​1016/​j.​ejpn.​2010.​09.​004 CrossRefPubMed
23.
Di Rocco C, Iannelli A, Tamburrini G (1997) Cavernous angiomas of the brain stem in children. Pediatr Neurosurg 27:92–99CrossRefPubMed
24.
Lena G, Ternier J, Paz-Paredas A, Scavarda D (2007) Central nervous system cavernomas in children (in French). Neurochirurgie 53:223–237CrossRefPubMed
25.
Scott RM (1990) Brainstem cavernous angiomas in children. Pediatr Neurosurg 16:281–286CrossRef
26.
Maraire JN, Awad IA (1995) Intracranial cavernous malformations: lesion behavior and management strategies. Neurosurgery 37:591–605CrossRefPubMed
27.
Van Lindert EJ, Tan TC, Grotenhuis JA, Wesseling P (2007) Giant cavernous hemangiomas: report of three cases. Neurosurg Rev 30:83–92CrossRefPubMed
28.
Di Rocco C, Iannelli A, Tamburrini G (1995) Surgical management of paediatric cerebral cavernomas. J Neurosurg Sci 41:343–347
29.
Sakai N, Yamada N, Nishimura Y, Shirakami S, Futamura A, Andoh T (1992) Intracranial cavernous angioma in the 1st year of life and a review of the literature. Childs Nerv Syst 8:49–52CrossRefPubMed
30.
Bertalanffy H, Gilsbach JM, Eggert HR, Seeger W (1991) Microsurgery of deep-seated cavernous angiomas: report of 26 cases. Acta Neurochir 108:91–99CrossRefPubMed
31.
Maggi G, Aliberti F, Ruggiero C, Pittore L (1997) Cerebral cavernous angiomas in critical areas. Reports of three cases in children. J Neurosurg Sci 41:353–357PubMed
32.
Nieto J, Hinojosa J, Munoz MJ, Esparza J, Ricoy R (2003) Intraventricular cavernoma in pediatric age. Childs Nerv Syst 19:60–62PubMed
33.
Di Rocco C, Iannelli A, Tamburini G (1996) Cavernomas of the central nervous system in children. A report of 22 cases. Acta Neurochir 138:1267–1274CrossRefPubMed
34.
Fortuna A, Ferrante L, Mastronardi L, Acqui M, D’Adetta R (1989) Cerebral cavernous angioma in children. Childs Nerv Syst 5:201–207CrossRefPubMed
35.
Gross BA, Du R, Popp AJ, Day AL (2010) Intramedullary spinal cord cavernous malformations. Neurosurg Focus 29(3):E14CrossRefPubMed
36.
Consales A, Piatelli G, Ravegnani M, Pavanello M, Striano P, Zoli ML, Capra V, Rossi A, Garrè ML, Calevo MG, Cama A (2010) Treatment and outcome of children with cerebral cavernomas: a survey on 32 patients. Neurol Sci 31:117–123. doi:10.​1007/​s10072-009-0157-0 CrossRefPubMed
37.
Cornelius JF, Kürten K, Fischer I, Hänggi D, Steiger HJ (2016) Quality of life after surgery for cerebral cavernoma: brainstem versus nonbrainstem location. World Neurosurg 95:315–321CrossRefPubMed
38.
Cantu C, Murillo-Bonilla L, Arauz A, Higuera J, Padilla J, Barinagarrementeria F (2005) Predictive factors for intracerebral hemorrhage in patients with cavernous angiomas. Neurol Res 27(3):314e8CrossRef
39.
Ferroli P, Sinisi M, Franzini A, Giombini S, Solero CL, Broggi G (2005) Brainstem cavernomas: long-term results of microsurgical resection in 52 patients. Neurosurgery 56:1203–1212CrossRefPubMed
40.
Fritschi JA, Reulen HJ, Spetzler RF, Zabramski JM (1994) Cavernous malformations of the brainstem. A review of 139 cases. Acta Neurochir 130:35–46CrossRefPubMed
41.
Porter RW, Detwiler PW, Spetzler RF, Lawton MT, Baskin JJ, Derksen PT, Zabramski JM (1999) Cavernous malformations of the brainstem: experience with 100 patients. J Neurosurg 90:50–58CrossRefPubMed
42.
Wang CH, Lin SM, Chen Y, Tseng SH (2003) Multiple deep-seated cavernomas in the third ventricle, hypothalamus and thalamus. Acta Neurochir 145:505–508PubMed
43.
Deutsch H, Shrivistava R, Epstein F, Jallo GI (2001) Pediatric intramedullary spinal cavernous malformations. Spine 26:427–431CrossRef
44.
Mitchell P, Hodgson TJ, Seaman S et al (2000) Stereotactic radiosurgery and the risk of haemorrhage from cavernous malformations. Br J Neurosurg 14:96–100CrossRefPubMed
45.
Bilginer B, Narin F, Hanalioglu S, Oguz KK, Soylemezoglu F, Akalan N (2014) Cavernous malformations of the central nervous system (CNS) in children: clinico-radiological features and management outcomes of 36 cases. Childs Nerv Syst 30(8):1355–1366CrossRefPubMed
46.
Chang EF, Gabriel RA, Potts MB, Berger MS, Lawton MT (2011) Supratentorial cavernous malformations in eloquent and deep locations: surgical approaches and outcomes. Clinical article. J Neurosurg 114(3):814–827CrossRefPubMed
47.
Song J, QiaoN XL, Qiu T, Li P, Wu J, Zhu W, Chen L, He KM, Zhang N, Mao Y, Zhao Y (2011) Clinical features and microsurgical treatment of pediatric patients with cerebral cavernous malformation. J Clin Neurosci 18(10):1303–1307CrossRefPubMed
48.
Shiban E, Zerr M, Huber T, Boeck-Behrends T, Wostrack M, Ringel F, Meyer B, Lehmberg J (2015) Poor diagnostic accuracy of transcranial motor and somatosensory evoked potential monitoring during brainstem cavernoma resection. Acta Neurochir 157:1963–1969CrossRefPubMed
49.
Baumann CR, Acciarri N, Bertalanffy H, Devinsky O, Elger CE, Lo Russo G, Cossu M, Sure U, Singh A, Stefan H, Hammen T, Georgiadis D, Baumgartner RW, Andermann F, Siegel AM (2007) Seizure outcome after resection of supratentorial cavernous malformations: a study of 168 patients. Epilepsia 48:559–563CrossRefPubMed
50.
Churchyard A, Khangure M, Grainger K (1992) Cerebral cavernous angioma: a potentially benign condition? Successful treatment in 16 cases. J Neurol Neurosurg Psychiatry 55:1040–1045CrossRefPubMedPubMedCentral
51.
Noto S, Fujii M, Akimura T, Imoto H, Nomura S, Kajiwara K, Kato S, Fujisawa H, Suzuki M (2005) Management of patients with cavernous angiomas presenting epileptic seizures. Surg Neurol 64:495–498CrossRefPubMed
52.
Baumann CR, Schuknecht B, Lo Russo G, Cossu M, Citterio A, Andermann F, Siegel AM (2006) Seizure outcome after resection of cavernous malformations is better when surrounding hemosiderin-stained brain also is removed. Epilepsia 47:563–566CrossRefPubMed
53.
Buckingham MJ, Crone KR, Ball WS, Berger TS (1989) Management of cerebral cavernous angiomas in children presenting with seizures. Childs Nerv Syst 5:347–349CrossRefPubMed
54.
Cohen DS, Zubay GP, Goodman RR (1995) Seizure outcome after lesionectomy for cavernous malformations. J Neurosurg 83:237–242CrossRefPubMed
55.
Ferroli P, Casazza M, Marras C, Mendola C,Franzini A, Broggi G (2006) Cerebral cavernomas and seizures: a retrospective study on 163 patients who underwent pure lesionectomy. Neurol Sci 26:390–394
56.
Hammen T, Romstöck J, Dörfler A, Kerling F, Buchfelder M, Stefan H (2007) Prediction of postoperative outcome with special respect to removal of hemosiderin fringe: a study in patients with cavernous haemangiomas associated with symptomatic epilepsy. Seizure 16:248–253
57.
Rangel-Castilla L, Spetzler RF (2015) The thalamic regions: surgical approaches to thalamic cavernous malformations, operative results, and clinical outcomes. J Neurosurg 123(3):676–685CrossRefPubMed
58.
Cantore G, Missori P, Santoro A (1999) Cavernous angiomas of the brain stem. Intra-axial anatomical pitfalls and surgical strategies. Surg Neurol 52:84–93CrossRefPubMed
59.
Zausinger S, Yousry I, Brueckmann H, Schmid-Elsaesser R, Tonn JC (2006) Cavernous malformations of the brainstem: three-dimensional-constructive interference in steady-state magnetic resonance imaging for improvement of surgical approach and clinical results. Neurosurgery 58:322–330CrossRefPubMed
60.
Abla AA, Lekovic GP, Garrett M, Wilson DA, Nakaji P, Bristol R, Spetzler RF (2010) Cavernous malformations of the brainstem presenting in childhood: surgical experience in 40 patients. Neurosurgery 67:1589–1599CrossRefPubMed
61.
Wu H, Yu T, Wang S, Zhao J, Zhao Y (2015) Surgical treatment of cerebellar cavernous malformations: a single-center experience with 58 cases. World Neurosurg 84(4):1103–1111CrossRefPubMed
62.
Knerlich-Lukoschus F, Steinbok P, Dunham C, Cochrane DD (2015) Cerebellar cavernous malformation in pediatric patients: defining clinical, neuroimaging, and therapeutic characteristics. J Neurosurg Pediatr 16(3):256–266CrossRefPubMed
63.
Bakir A, Savas A, Yilmaz E, Savas B, Erden E, Caglar S, Sener O (2006) Spinal intradural-intramedullary cavernous malformation. Case report and literature review. Pediatr Neurosurg 42:35–37CrossRefPubMed
64.
Nagib MG, O’Fallon MT (2002) Intramedullary cavernous angiomas of the spinal cord in the pediatric age group: a pediatric series. Pediatr Neurosurg 36:57–63CrossRefPubMed
65.
Scott RM, Barnes P, Kupsy W, Adelman LS (1992) Cavernous angiomas of the central nervous system in children. J Neurosurg 76:38–46CrossRefPubMed
66.
Furuya K, Sasaki T, Suzuki I, Kim P, Saito N, Kirino T (1996) Intramedullary angiographically occult vascular malformations of the spinal cord. Neurosurgery 39:1123–1132CrossRefPubMed
67.
Park SB, Jahng TA, Chung CK (2015) The clinical outcomes after complete surgical resection of intramedullary cavernous angiomas: changes in motor and sensory symptoms. Spinal Cord 47:128–133CrossRef
Metadaten
Titel
Cavernous malformations of central nervous system in pediatric patients: our single-centered experience in 50 patients and review of literature
verfasst von
Dattaraj Paramanand Sawarkar
Suveen Janmatti
Rajinder Kumar
Pankaj Kumar Singh
Hitesh Kumar Gurjar
Shashank Sharad Kale
Bhawani Shanker Sharma
Ashok Kumar Mahapatra
Publikationsdatum
20.06.2017
Verlag
Springer Berlin Heidelberg
Erschienen in
Child's Nervous System / Ausgabe 9/2017
Print ISSN: 0256-7040
Elektronische ISSN: 1433-0350
DOI
https://doi.org/10.1007/s00381-017-3429-7

Weitere Artikel der Ausgabe 9/2017

Child's Nervous System 9/2017 Zur Ausgabe

Original Paper

Exploring for the optimal structural design for the 3D-printing technology for cranial reconstruction: a biomechanical and histological study comparison of solid vs. porous structure

Original Paper

Comprehensive analysis of Iranian reports of pediatric central nervous system tumors

Letter to the Editor

Spina bifida: a prospective study from a single neurosurgical center based on the National Hospital in Niamey (Niger)

Case Report

Skull fracture with effacement of the superior sagittal sinus following drone impact: a case report

Case Report

A congenital external carotid artery-external jugular vein arteriovenous fistula was successfully treated by coil embolization (case report and literature review)

Review Paper

A comprehensive review of the foramina of Luschka: history, anatomy, embryology, and surgery

Neu im Fachgebiet Chirurgie

23.04.2024 | Ablationstherapie | Nachrichten

Wie erfolgreich ist eine Re-Ablation nach Rezidiv?

23.04.2024 | Appendizitis | Nachrichten

Hinter dieser Appendizitis steckte ein Erreger

23.04.2024 | Operationsvorbereitung | Nachrichten

Mehr Schaden als Nutzen durch präoperatives Aussetzen von GLP-1-Agonisten?

19.04.2024 | EAU 2024 | Kongressbericht | Nachrichten

Ureterstriktur: Innovative OP-Technik bewährt sich
weitere anzeigen

Update Chirurgie

Bestellen Sie unseren Fach-Newsletter und bleiben Sie gut informiert.

Newsletter bestellen

Aktuelle BDC Leitlinien-Webinare

S3-Leitlinie „Diagnostik und Therapie des Karpaltunnelsyndroms“

Karpaltunnelsyndrom BDC Leitlinien Webinare
CME: 2 Punkte

Das Karpaltunnelsyndrom ist die häufigste Kompressionsneuropathie peripherer Nerven. Obwohl die Anamnese mit dem nächtlichen Einschlafen der Hand (Brachialgia parästhetica nocturna) sehr typisch ist, ist eine klinisch-neurologische Untersuchung und Elektroneurografie in manchen Fällen auch eine Neurosonografie erforderlich. Im Anfangsstadium sind konservative Maßnahmen (Handgelenksschiene, Ergotherapie) empfehlenswert. Bei nicht Ansprechen der konservativen Therapie oder Auftreten von neurologischen Ausfällen ist eine Dekompression des N. medianus am Karpaltunnel indiziert.

Prof. Dr. med. Gregor Antoniadis
Berufsverband der Deutschen Chirurgie e.V.

S2e-Leitlinie „Distale Radiusfraktur“

Radiusfraktur BDC Leitlinien Webinare
CME: 2 Punkte

Das Webinar beschäftigt sich mit Fragen und Antworten zu Diagnostik und Klassifikation sowie Möglichkeiten des Ausschlusses von Zusatzverletzungen. Die Referenten erläutern, welche Frakturen konservativ behandelt werden können und wie. Das Webinar beantwortet die Frage nach aktuellen operativen Therapiekonzepten: Welcher Zugang, welches Osteosynthesematerial? Auf was muss bei der Nachbehandlung der distalen Radiusfraktur geachtet werden?

PD Dr. med. Oliver Pieske
Dr. med. Benjamin Meyknecht
Berufsverband der Deutschen Chirurgie e.V.

S1-Leitlinie „Empfehlungen zur Therapie der akuten Appendizitis bei Erwachsenen“

Appendizitis BDC Leitlinien Webinare
CME: 2 Punkte

Inhalte des Webinars zur S1-Leitlinie „Empfehlungen zur Therapie der akuten Appendizitis bei Erwachsenen“ sind die Darstellung des Projektes und des Erstellungswegs zur S1-Leitlinie, die Erläuterung der klinischen Relevanz der Klassifikation EAES 2015, die wissenschaftliche Begründung der wichtigsten Empfehlungen und die Darstellung stadiengerechter Therapieoptionen.

Dr. med. Mihailo Andric
Berufsverband der Deutschen Chirurgie e.V.