scalp_cirsoid_aneurysm

Scalp cirsoid aneurysm

They are also referred to as aneurysma serpentinum, aneurysm racemosum, or plexiform angioma.

Cirsoid aneurysm of the scalp is one of the rarest occurrences in neurosurgery. It is an aneurysmal tumor formed by arteriovenous fistula of the arteries and veins of the scalp 1)

They can be easily misdiagnosed and mistreated.

Historically their unusual portly appearance led to various synonyms being coined for the entity 2).

Epidemiology

Scalp AVM (SAVM) is a rare condition 3) 4) and infrequently encountered by the neurosurgeon 5).

Classification

Yokouchi et al . classified scalp arteriovenous fistulae into three types: Type A: has a single fistulous connection from the proximal portion of the artery, Type B: has a single fistulous connection but from multiple distal portions of the arteries and Type C: where there are multiple fistulae constituting the plexiform feeding type.

Sometimes the drainage can be intracranial with an associated cerebral arteriovenous malformationYokouchi et al . classified scalp arteriovenous fistulae into three types: Type A: has a single fistulous connection from the proximal portion of the artery, Type B: has a single fistulous connection but from multiple distal portions of the arteries and Type C: where there are multiple fistulae constituting the plexiform feeding type.

Sometimes the drainage can be intracranial with an associated cerebral arteriovenous malformation

Etiology

Its origin can be congenital or traumatic.

Congenital arteriovenous malformations (AVMs) of scalp are rare. They are usually not symptomatic at birth and are often misdiagnosed as haemangiomas. To date, only two cases of symptomatic neonatal scalp AVM have been reported in literature. Pathophysiology of congenital AVM is not completely understood but genetic and acquired causes are implicated. Diagnosis and management are often difficult and require multidisciplinary approach. Hussain et al. report a rare case of symptomatic congenital scalp AVM in a 10-day-old neonate who was successfully managed 6).

Clinical

The clinical picture presents with complaints of increased scalp, scalp disfigurement, pain and neurological symptoms.

They can present a subcutaneous scalp lump or a large, pulsatile mass with a propensity to skin erosion and massive haemorrhage 7) 8).

The symptoms associated with cirsoid aneurysm of scalp vary according to the size of the fistula. Common clinical manifestations include loud bruit, pulsatile scalp mass, headache, and tinnitus. If left untreated, there is an increased risk of developing life-threatening complications such as aneurysmal hemorrhage or scalp necrosis 9) 10) 11).

Diagnosis

Since cerebral blood flow may decrease depending on the progression of the lesion, the cerebral blood flow should be evaluated. Considering the treatment modalities depending on the lesion can provide treatment with less recurrence and higher patient satisfaction 12).

Treatment

Scalp cirsoid aneurysm treatment.

Outcome

Even after complete surgical resection, a case of recurrence after 18 years has been reported 13), which is why regular follow-up is advised.

Systematic Review

Sofela et al. conducted a PUBMED, SCOPUS, OVID SP, SciELO, and INFORMA search using the keywords; “cirsoid,” “aneurysm,” “arteriovenous,” “malformation,” “scalp,” “vascular,” and “fistula.” They identified 74 pertinent papers, reporting 242 cases in addition to our reported index case.

Median age at presentation was 25 yr (range 1-72 yr); male to female ratio was 2.5:1. The most common symptoms were a pulsatile mass (94% of patients), headaches (25%), and tinnitus (20%). The median duration of symptoms was 3 yr (6 d to 31 yr), with 60.2% occurring spontaneously, 32.23% traumatic, and the rest iatrogenic. A total of 58.5% of cases were managed with surgical excision only, 21.6% with endovascular embolization only, and 14.5% with a combination of both methods. The complication rate observed in the endovascular embolization treatment cohort (55.8%) was significantly higher than that observed in the surgical excision only cohort (9.9%) (P < .00001) and in the combined therapy cohort (0%) (P < .00001). There is a low recurrence rate after treatment irrespective of modality: surgical excision only (6.3%), endovascular embolization only (8.3%), and combined therapy (0%).

Scalp cirsoid aneurysms are associated with good prognoses when recognized and managed appropriately. They suggested combining surgery with endovascular embolization as the optimum treatment modality 14).

Case series

Scalp cirsoid aneurysm case series.

Case reports

Scalp cirsoid aneurysm case reports.

Unclassified

4: Heiferman DM, Syed HR, Li D, Rothstein BD, Shaibani A, Tomita T. Resection of an Embolized Cirsoid Aneurysm With Intracranial Venous Drainage: 2-Dimensional Operative Video. Oper Neurosurg (Hagerstown). 2019 Mar 1;16(3):E94. doi: 10.1093/ons/opy303. PubMed PMID: 30295882.

5: Albuquerque Sousa LH, Maranha Gatto LA, Demartini Junior Z, Koppe GL. Scalp Cirsoid Aneurysm: An Updated Systematic Literature Review and an Illustrative Case Report. World Neurosurg. 2018 Nov;119:416-427. doi: 10.1016/j.wneu.2018.08.098. Epub 2018 Aug 24. Review. PubMed PMID: 30149169.

6: Li D, Heiferman DM, Rothstein BD, Syed HR, Shaibani A, Tomita T. Scalp Arteriovenous Malformation (Cirsoid Aneurysm) in Adolescence: Report of 2 Cases and Review of the Literature. World Neurosurg. 2018 Aug;116:e1042-e1046. doi: 10.1016/j.wneu.2018.05.161. Epub 2018 Jun 1. Review. PubMed PMID: 29864562.

7: Varala S, Arakkal GK, Malkud S, Narayana B. Cirsoid aneurysm of scalp. Indian J Dermatol Venereol Leprol. 2018 Jan-Feb;84(1):57-58. doi: 10.4103/ijdvl.IJDVL_270_17. PubMed PMID: 29176252.

8: Gangadharaswamy SB, Maulyavantham Nagaraj N, Pai BS. Surgical management of scalp arteriovenous malformations using a novel surgical technique-Case series. Int J Surg Case Rep. 2017;37:250-253. doi: 10.1016/j.ijscr.2017.06.057. Epub 2017 Jul 8. PubMed PMID: 28715722; PubMed Central PMCID: PMC5514622.

9: Munakomi S, Bhattarai B, Cherian I. Conquering the odds: Cirsoid aneurysm with holocranial feeders-staged embolization, excision and grafting. Asian J Neurosurg. 2015 Jul-Sep;10(3):259-61. doi: 10.4103/1793-5482.161167. PubMed PMID: 26396624; PubMed Central PMCID: PMC4553749.

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11: Goel V, Verma AK, Singh S, Puri SK. Cirsoid aneurysm of scalp: demonstration on CT angiography (CTA). BMJ Case Rep. 2013 Dec 11;2013. pii: bcr2013202459. doi: 10.1136/bcr-2013-202459. PubMed PMID: 24336588; PubMed Central PMCID: PMC3863086.

12: Chowdhury FH, Haque MR, Kawsar KA, Sarker MH, Momtazul Haque AF. Surgical management of scalp arterio-venous malformation and scalp venous malformation: An experience of eleven cases. Indian J Plast Surg. 2013 Jan;46(1):98-107. doi: 10.4103/0970-0358.113723. PubMed PMID: 23960313; PubMed Central PMCID: PMC3745130.

13: Saade C, Wilkinson M, Parker G, Dubenec S, Brennan P. Multidetector computed tomography in the evaluation of cirsoid aneurysm of the scalp–a manifestation of trauma. Clin Imaging. 2013 May-Jun;37(3):558-60. doi: 10.1016/j.clinimag.2012.07.001. Epub 2012 Aug 13. PubMed PMID: 23601772.

14: El Shazly AA, Saoud KM. Results of surgical excision of cirsoid aneurysm of the scalp without preoperative interventions. Asian J Neurosurg. 2012 Oct;7(4):191-6. doi: 10.4103/1793-5482.106651. PubMed PMID: 23559986; PubMed Central PMCID: PMC3613641.

15: Kumar A, Ahuja CK, Khandelwal N, Bakshi JB. Cirsoid aneurysm of the right pre-auricular region: an unusual cause of tinnitus managed by endovascular glue embolisation. J Laryngol Otol. 2012 Sep;126(9):923-7. doi: 10.1017/S0022215112001466. Epub 2012 Jul 5. PubMed PMID: 22874530.

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21: Muthukumar N, Rajagopal V, Manoharan AV, Durairaj N. Surgical management of cirsoid aneurysms. Acta Neurochir (Wien). 2002 Apr;144(4):349-56. PubMed PMID: 12021881.

22: Hendrix LE, Meyer GA, Erickson SJ. Cirsoid aneurysm treatment by percutaneous injection of sodium tetradecyl sulfate. Surg Neurol. 1996 Dec;46(6):557-60; discussion 560-1. PubMed PMID: 8956889.

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25: Heilman CB, Kwan ES, Klucznik RP, Cohen AR. Elimination of a cirsoid aneurysm of the scalp by direct percutaneous embolization with thrombogenic coils. Case report. J Neurosurg. 1990 Aug;73(2):296-300. PubMed PMID: 2366088.

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31: Balsys R, Cross R. Multiple aneurysm formation as a complication of interventive angiography. Radiology. 1978 Jan;126(1):91-2. PubMed PMID: 619442.

32: Bretto P, Pegoraro M, Ponzio F. [Recurrent cirsoid aneurysm of the scalp]. Minerva Chir. 1977 Nov 30;32(22):1409-12. Italian. PubMed PMID: 600430.

33: Chatterji P, Purohit GN, Soni NK. Cirsoid aneurysm of the auricle and adjoining scalp. J Laryngol Otol. 1977 Nov;91(11):997-1002. PubMed PMID: 591785.

34: Mohanty S, Rao CJ. A large cirsoid aneurysm of the scalp associated with epilepsy. J Neurol Neurosurg Psychiatry. 1976 Sep;39(9):835-6. PubMed PMID: 993804; PubMed Central PMCID: PMC492470.

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37: Coulthard R. Cirsoid aneurysm of the scalp. Nurs Mirror Midwives J. 1966 Apr 22;122(4):63-4. PubMed PMID: 5176504.

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39: OLDFIELD MC, ADDISON NV. Cirsoid aneurysms of the scalp. Br Med J. 1962 Jul 7;2(5296):23-4. PubMed PMID: 14481706; PubMed Central PMCID: PMC1925556.

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41: GASPARINI FILHO S, MAYALL RC. [On a case of cirsoid aneurysm of the frontal region]. Angiologia. 1961 Nov-Dec;13:327-31. Spanish. PubMed PMID: 13897170.

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45: GONI MORENO I. [Cirsoid aneurysm of the scalp at 6 years of being operated]. Bol Trab Acad Argent Cir. 1949 Sep 7;33(19):587. Spanish. PubMed PMID: 15398981.

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47: ELKIN DC. Cirsoid aneurysm of the scalp. Ann Surg. 1946 Apr;123:591-600. PubMed PMID: 21024588.

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49: ELKIN DC. Cirsoid aneurysm of the scalp; report of four cases. Trans South Surg Assoc. 1946;57:122-31. PubMed PMID: 20283384.

50: Clunie T, Aberd CB. CIRSOID ANEURYSM OF THE SCALP. Br Med J. 1936 Dec 12;2(3962):1183-6. PubMed PMID: 20780323; PubMed Central PMCID: PMC2459002.

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