Sinus Pericranii: Systematic Review with a Case Illustration

Abstract

Sinus pericranii (SP) is a rare vascular malformation characterized by abnormal communication between intracranial and extracranial venous systems. Often asymptomatic and presenting as a soft, nonpulsatile scalp mass, SP can be mistaken for other scalp anomalies, such as scalp arteriovenous malformations (AVMs), due to overlapping clinical features. This case of a 4-year-old girl successfully treated for a giant SP is reported to illustrate the findings of a comprehensive systematic literature review. This review was conducted according to the “Preferred Reporting Items for Systematic Reviews” guidelines. Relevant studies (1985–2025) reporting patients with SP were identified from PubMed/MEDLINE databases. A total of 65 studies reporting 82 SP patients were included in this study. The median age was 8 years, ranging from 4 months to 72 years, with a male predominance of 61.0% (n = 50). No contributing factors were reported in most cases (67.1%, n = 55). However, craniosynostosis and recent trauma were reported as contributing factors to the development of SP in 7.3% (n = 6) and 18.3% (n = 15), respectively. Frontal (37.8%, n = 31), parietal (32.9%, n = 27), and occipital (13.4%, n = 11) were the most reported locations of SP. The median follow-up duration was 24 months (3–96). This is the first systematic review of the SP, where the authors highlight, by data synthesis, the clinical presentation, dural sinus involvement, management, and outcome of this rare but challenging pathology. Without management guidelines, its treatment choice is still based on the surgeon's experience. This reality calls for action for an evidence-based study.

Keywords

sinus pericranii - case illustration - systematic review - diagnosis - treatment modalities

Introduction

Sinus pericranii (SP) was first documented in the literature in 1845 by Hecker, and was later defined as a soft vascular tumor of the scalp, primarily located in the subcutaneous cranial midline, by Stromeyer in 1850.[1]

SP is a strictly vascular malformation characterized by direct abnormal communication between the intracranial and extracranial blood vessels. Of the approximately 200 reported cases, almost half are diagnosed before the second decade of life, highlighting the lesion's rarity.[2] Although the exact pathogenesis is not known, congenital predisposition and trauma are thought to be contributory factors.[3]

Clinically, this benign lesion is usually asymptomatic and appears as a nonpulsating, variable mass of soft tissue on the scalp, varying in size with changes in intracranial pressure.[4] [5]

Its rarity and similarity to other scalp malformations present a major diagnostic challenge, often resulting in misdiagnosis as scalp hemangioma, atretic cephalocele, or other vascular malformations of the scalp, particularly scalp AVM, which is a major differential diagnosis.[5] [6]

Despite the increasing number of reported cases, there is still a considerable gap in the literature on the systemic characterization of SP, including its clinical characteristics, diagnostic challenges, and optimal treatment strategies. Many reported cases provide limited and individualistic presentations and management approaches, which result in a lack of knowledge and understanding of this rare disease.

This study is the first systematic review of SP aiming to bridge the existing gap by providing a comprehensive analysis of the clinical presentation and contributing factors, along with precise recommendations for diagnosis and treatment strategies. We also report an interesting case of a giant SP that was originally diagnosed as a superficial AVM, which highlights the critical role of angiography in distinguishing these two rare conditions.

Case Presentation

Patient information: A 4-year-old girl presented with gradually increasing head mass in the mid-frontal region with a blue-brown discoloration of the adjacent frontal skin ([Fig. 1A]). The mass increases in size from birth and further increases when the patient is supine. No other symptoms or bleeding events have been reported. A prior history of head injury or infection has been denied.

Fig. 1 Case illustration images. (A) Preoperative image of the patient positioned on the operating table, with a horseshoe head holder, showing lateral and posterior views of the head with the enlarged frontoparietal subcutaneous vascular malformation. (B) 3D angio-CT demonstrating the precise location of the vascular anomaly on the skull vault. (C) Sagittal view of the angio-CT of the brain vasculature showing a midline venous anomaly, with normal brain vessels. (D) Angio-MRI showing a midline subcutaneous vascular malformation and thinning of the superior sagittal sinus. (E) DSA of the brain at the venous drainage phase showing a disruption of the venous drainage in the sagittal sinus over the frontal region with the presence of venous collectors subcutaneously (accessory SP). (F) Scalp detachment and venous pouch coagulation following waxing of the outer bone. (G) Postoperative image showing a complete collapse of the vascular pouch with preservation of skin integrity. (H) Follow-up at 1 month, indicating complete wound healing.

Clinical findings: Physical examination revealed a large 7 cm × 5 cm frontal, nonpulsating mass of the scalp, with no hair loss in this area. The patient had normal head circumference, normal motor and sensory function, and the examination of the cranial nerves was not noteworthy. Her past medical, surgical, and family histories were unimportant.

Diagnostic assessment: A brain computed tomography (CT) scan showed an extracranial, enhancing, vascular, serpiginous lesion consistent with a scalp vascular malformation. 3D angio-CT identified the exact superficial anatomical location of the mass ([Fig. 1B, C]). No bone defect was found. Magnetic resonance imaging (MRI) showed no parenchymal abnormalities other than the vascular malformation ([Fig. 1D]).

The diagnosis was originally focused on AVM in the scalp. However, digital subtraction angiography (DSA) showed that the contrast flow at the venous stage following injection into the carotid artery is very low ([Fig. 1E]). This finding suggested that this was not a typical high-flow AVM of the scalp but rather a venous malformation of the scalp consistent with parallel venous drainage of the brain into the sacrum (i.e., the SP). Based on the results of the history, physical examination, and imaging methods, especially angiography, the patient has been diagnosed with SP. The surgical resection was scheduled without preoperative embolization.

Surgical technique: The surgery was performed under general anesthesia. The patient was easily prepared for a transfusion if necessary. An incision was made in the scalp behind the entire vascular mass, and the scalp was carefully detached from the epidermal aponeurosis. Progressive cauterization of the subcutaneous vascular pouch was performed, with continuous vascular waxing of the fistula in the bone to minimize blood loss, which was especially important given the patient's young age. Despite the persistence of small blue blood vessels in the dermis, skin resection was not required. The SP was completely removed without any peri- and postoperative complications ([Fig. 1F, G]).

Follow-up and outcome: Postoperative imaging showed no residual lesion. The patient was followed up for 12 months, and no recurrence occurred. The patient was doing well, with a completely healed surgical wound ([Fig. 1H]).

Materials and Methods

Study Design

This systematic review followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. The study protocol was developed a priori but was not registered in PROSPERO.

Information Sources and Search Strategy

A comprehensive literature search of previously reported SP was conducted across PubMed and Medline databases to retrieve all relevant studies published from 1985 through January 2025.

Two authors (Y.C.H.D. and H.E.A) independently performed the search using the following MeSH terms: ((“sinus pericranii”[MeSH Terms] OR (“sinus”[All Fields] AND “pericranii”[All Fields]) OR “sinus pericranii”[All Fields]), with the following applied filters: Full text, Case Reports, Clinical Trial, Observational Study, Randomized Controlled Trial, English, Humans, resulting in 95 articles. https://pubmed.ncbi.nlm.nih.gov/?term=%28%22sinus+pericranii%22%5BMeSH+Terms%5D+OR+%28%22sinus%22%5BAll+Fields%5D+AND+%22pericranii%22%5BAll+Fields%5D%29+OR+%22sinus+pericranii%22%5BAll+Fields%5D%29+AND+%281980%3A2025%5Bpdat%5D%29&filter = simsearch3.fft&filter = pubt.casereports&filter = pubt.clinicaltrial&filter = pubt.observationalstudy&filter = pubt.randomizedcontrolledtrial&filter = lang.english&filter = hum_ani.humans&filter = years.1985-2025&sort = date

Eligibility Criteria

We included all relevant articles reporting patients affected by SP, following the previously mentioned Boolean operators and filters. Both pediatric and adult populations were eligible. Included study designs were observational studies including case reports and case series and clinical trials.

We excluded articles not dealing with SP, without full text or disaggregated data, not reporting patients' management and outcome, written in a language other than English, and duplicates.

Study Selection and Data Extraction

Selection Process

Citations were retrieved from electronic databases. No automation software was used for screening. Two reviewers independently evaluated the titles, abstracts, and full texts of all identified records to determine their relevance to the eligibility criteria. Any discrepancies between the reviewers were resolved through discussion, and a third-party reviewer was involved when the issue persisted. Subsequently, a total of 67 articles were included.

[Fig. 2] shows the PRISMA flowchart summarizing the selection process.

Fig. 2 Diagram flow chart of sinus pericranii patient selection.

Data Extraction

All 67 articles included in the quantitative analysis were systematically screened, and the following data were extracted: first author's name and year of publication, age and gender of patients, family history of SP, clinical presentation, associated malformation, lesion location, sinus involvement, lesion size, diagnosis tools, imaging findings, SP type, treatment, follow-up, and outcome ([Table 1]). We described the outcome as “complete resolution” when the authors reported that the patient was symptom-free and had no more scalp mass and as “incomplete resolution” when the patient was symptom free with some remnant of scalp mass. The SP recurrence cases were those reported to present the same form of venous malformation at the same site following a previous total resection of the lesion. A spreadsheet database was created.

Table 1 Basic characteristics of the 65 articles reporting 82 patients of sinus pericranii included after the systematic review

Name of article	Journal title	First author and year of publication	Country of origin of first author	Total number of patients
Sinus pericranii: clinical and imaging findings in two cases of spontaneous partial thrombosis	AJNR	Carpenter et al 2004	USA	2
Sinus pericranii associated with syntelencephaly: a case report	BMC Neurol	Fujino et al 2022	Japan	1
Combined treatment of surgery and sclerotherapy for sinus pericranii	Arch Craniofac Surg	Yeop Ryu et al 2019	Korea	1
Hidden connection: unusual case of vertigo as a result of sinus pericranii	Annals Ind Acad Neurol	Singh et al 2023	India	1
Sinus pericranii in a young adult with chronic headache	BMJ	Saba et al 2013	USA	1
Sinus pericranii: long-term outcome in a 10-year-old boy with a review of literature	BMJ	Goffin et al 2018	UK	1
Sinus pericranii in a neonate with the scalp hair tuft sign	BMJ	Ray et al 2021	UK	1
36-year-old man with parietal scalp mass	Brain Pathol	Cazorla et al 2016	France	1
Sinus pericranii in the setting of a posterior fossa pilocytic astrocytoma: illustrative case	J Neurosurgery Case Lessons	Jung et al 2023	Canada	1
Soft frontal swelling in a young girl: diagnostic nuances and surgical management of a rare case of sinus pericranii	Clin Case Rep (Wiley)	Mokbul et al 2024	Bangladesh	1
Sinus pericranii (parietal and occipital) with epicranial varicosities in a case of craniosynostosis	Cureus	Sharma Sr. et al 2022	India	1
Single-session percutaneous embolization with onyx and coils of sinus pericranii	Surg Neurol Int	Gatto et al 2018	Brazil	1
Sinus pericranii	Dtsch Arztebl Int	Franz Heppt et al 2020	Germany	1
Case report: a young man with frontal traumatic sinus pericranii	Front Surg	Zihao Zhang et al 2024	China	1
Endovascular transvenous embolization combined with direct function of the sinus pericranii: a case report	Interv Neuroradiol	Kessler et al 2009	France	1
Bilateral frontal sinus pericranii with an intratabular course: a case report	Interv Neuroradiol	Schenk et al 2010	Netherlands	1
Cerebrofacial venous anomalies, sinus pericranii, ocular abnormalities: a developmental delay	Interv Neuroradiol	Macit et al 2012	USA	2
Symptomatic sinus pericranii with adult onset headache: a case report with pathologic perspective	J Cerebrovasc Endovasc Neurosurg	Chung et al 2019	Republic of Korea	1
Lateral sinus pericranii with internal jugular vein communication	Radiol Case Rep	Abera et al 2023	Ethiopia	1
Sinus pericranii: a case report and literature review	Brazilian J Otorhinolaryngol	Paiva et al 2023	Brazil	1
Sinus pericranii with dominant venous outflow in the superior eyelid	Neurol Med Chir	Ito et al 2016	Japan	1
A rare and deceptive venous anomaly, sinus pericranii	J Anaesthesiol Clin Pharmacol	Rizvi et al 2015	India	1
Primary cutaneous meningioma in association with a sinus pericranii	J Am Acad Dermatol	Zeikus et al 2006	USA	1
Crouzon disease associated with sinus pericranii: a report on identical twin sisters	Childs Nerv Syst	Yasuda et al 1993	Japan	1
Sinus pericranii: color Doppler ultrasonographic findings	J Ultrasound Med	Yanik et al 2006	Türkiye	1
Sinus pericranii: from gross and neuroimaging findings to different pathophysiological changes	Child's Nerv Syst	Wen et al 2005	China	1
Sinus pericranii in the right frontal region and thrombosis	Int J Neurosci	Wang et al 2009	China	1
Chronic intermittent intracranial hypertension caused by sinus pericranii and transverse sinus stenosis: a unique case report in a child	Asian J Surg	Tang et al 2024	China	1
Characteristics of recurrent congenital sinus pericranii: case report and review of the literature	Ped Neurosurg	Tamura et al 2019	Japan	1
Giant lateral sinus pericranii: case report	J Neurosurg	Spektor et al 1998	Israel	1
Three-dimensional printing of a sinus pericranii model: technical note	Child's Nerv Syst	Simonin et al 2017	Switzerland	1
Sinus pericranii: dermatologic considerations and literature review	J Am Acad Dermatol	Sheu et al 2002	USA	2
Staged raising of a coronal flap for fronto-orbital advancement and remodeling in Saethre-Chotzen syndrome complicated by sinus pericranii	J Craniofac Surg	Shaw et al 2018	USA	1
Endovascular embolization with Onyx in the management of sinus pericranii: a case report	Neurosurg Focus	Rangel-Castilla et al 2009	USA	1
Sinus pericranii: case report	Vasc Endovasc Surg	Aburto-Murrieta et al 2011	Mexico	1
Sinus pericranii: an overview and literature review of a rare cranial venous anomaly (a review of the existing literature with case examples)	Neurosurg Rev	Akram et al 2012	UK	1
Sinus pericranii complicated by hydrocephalus: case report and literature review	Almujaiwel	Almujaiwel et al 2020	USA	1
Sinus pericranii: a case report and review of literature	Ind J Pediatr	Bhutada et al 2012	India	1
Sinus pericranii: radiological and etiopathological considerations	J Neurosurg	Bollar et al 1992	Spain	1
Clinical and imaging findings in a rare case of sinus pericranii	Child's Nerv Syst	Bouali et al 2015	Germany	1
Sinus pericranii involving the torcular sinus in a patient with hunter's syndrome and trigonocephaly: case report and review of the literature	Neurosurgery	Brisman et al 2003	USA	1
Endovascular transvenous embolization of sinus pericranii	J Neurosurg Ped	Brook et al 2009	USA	1
Recurrent sinus pericranii in a 14-year-old boy	Child's Nerv Syst	Brown et al 1987	USA	1
Sinus pericranii	Pediatr Neurosurg	Buxton et al 1999	UK	1
Sinus pericranii with a hair collar sign	Ped Dermatol	Cheraghi et al 2014	Spain	1
Sinus pericranii “don't judge a lump by its surface”	J Craniofac Surg	Shah et al 2010	UK	1
Sinus pericranii	J Craniofac Surg	David et al 1998	USA	5
Occipital sinus pericranii superseding both jugular veins: description of two rare pediatric cases	Neurosurgery	Frassanito et al 2013	Italy	2
Parietal sinus pericranii: case report and technical note	Surg Neurol	Jung et al 2000	Republic of Korea	1
Sinus pericranii: a scalp mass in a 6-month-old boy	Ped Neurosurg	Kanavaki et al 2012	Switzerland	1
Craniosynostosis in the presence of a sinus pericranii: case report	Neurosurg	Kurosu et al 1994	Japan	1
Sinus pericranii: sonographic findings	Am J Roentgenol	Luker et al 1995	USA	1
Blocked ventriculoperitoneal shunt causing raised intracranial pressure diagnosed by prominent sinus pericranii	J Clin Neurosci	Mahindu et al 2009	Australia	1
Sinus pericranii associated with craniosynostosis	J Craniofac Surg	Mitsukawa et al 2007	Japan	7
Lateral situated sinus pericranii. case report	Surg Neurol	Nishio et al 1989	Japan	1
Lateral sinus pericranii	Surg Neurol	Nozaki et al 1986	Japan	1
Sinus pericranii in the left frontal region involving the superior eyelid: a case report	J Neurol Surg	Grahovac et al 2013	Croatia	1
Sinus pericranii with unusual features: multiplicity, associated dural venous lakes and venous anomaly, and a lateral location	Acta Neurochir	Rizvi et al 2010	India	2
Spontaneous involution of two sinus pericranii – a unique case and review of the literature	J Clin Neurosci	Rozen et al 2008	Australia	1
Dynamic morphological changes of thrombosed lateral sinus pericranii revealed by serial magnetic resonance images	Child's Nerv Syst	Ryorin et al 2017	Japan	1
Sinus pericranii and myoclonic epilepsy: novel features of 3q29 microdeletion syndrome	Acta Neurologica Belgica	Samanta et al 2015	USA	1
Extremely large sinus pericranii with involvement of the torcular and associated with Crouzon's syndrome	Child's Nerv Syst	Sanders et al 2017	USA	1
Single, small, spontaneous, accessory, closed type, frontal sinus pericranii in a child: Favorable outcome with surgical excision	Neurol India	Satyarthee et al 2013	India	1
Sinus pericranii, petrosquamosal sinus and extracranial sigmoid sinus: anatomical variations to consider during a retroauricular approach	Auris Nasus Larynx	Cisneros et al 2016	Brazil	3
Sinus pericranii: demonstration using three-dimensional surface shading	J Comput Assist Tomogr	Curnes et al 2002	USA	1

Risk of Bias Assessment

The risk of bias of the included studies was assessed using the Joanna Briggs Institute (JBI) checklist for case reports.[7] For each study, two independent authors evaluated the quality of the included studies, and a third author was consulted to resolve any differences. For each case report, all eight JBI items were evaluated and scored as “Yes,” “No,” “Unclear,” or “Not applicable.” A summary of the assessment is provided in [Table 2].

Table 2 Risk of bias assessment of the included studies

Title	Author, year	Q1. Were patients' demographic characteristics clearly described?	Q2. Was the patient's history clearly described and presented as a timeline?	Q3. Was the current clinical condition of the patient on presentation clearly described?	Q4. Were diagnostic tests or assessment methods and the results clearly described?	Q5. Was the intervention(s) or treatment procedure(s) clearly described?	Q6. Was the postintervention clinical condition clearly described?	Q7. Were adverse events (harms) or unanticipated events identified and described?	Q8. Does the case report provide takeaway lessons?
Sinus pericranii: clinical and imaging findings in two cases of spontaneous partial thrombosis	Carpenter et al 2004	Yes	Yes	Yes	Yes	Yes	Yes	Unclear	Yes
Sinus pericranii associated with syntelencephaly: a case report	Fujino et al 2022	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Combined treatment of surgery and sclerotherapy for sinus pericranii	Yeop Ryu et al 2019	Yes	Yes	Yes	Yes	Yes	Yes	Unclear	Yes
Hidden connection: unusual case of vertigo as a result of sinus pericranii	Singh et al 2023	Yes	Yes	Yes	Yes	Yes	Yes	Unclear	Yes
Sinus pericranii in a young adult with chronic headache	Saba et al 2013	Yes	Yes	Yes	Yes	No	No	No	Yes
Sinus pericranii: long-term outcome in a 10-year-old boy with a review of literature	Goffin et al 2018	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii in a neonate with the scalp hair tuft sign	Ray et al 2021	Yes	Yes	Yes	Yes	NA	NA	NA	Yes
36-year-old man with parietal scalp mass	Cazorla et al 2016	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii in the setting of a posterior fossa pilocytic astrocytoma: illustrative case	Jung et al 2023	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Soft frontal swelling in a young girl: diagnostic nuances and surgical management of a rare case of sinus pericranii	Mokbul et al 2024	Yes	Yes	Yes	Yes	Yes	No	No	Yes
Sinus pericranii (parietal and occipital) with epicranial varicosities in a case of craniosynostosis	Sharma Sr. et al 2022	Yes	Yes	Yes	Yes	NA	NA	NA	Yes
Single-session percutaneous embolization with onyx and coils of sinus pericranii	Gatto et al 2018	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii	Franz Heppt et al 2020	Yes	Yes	Yes	Yes	NA	NA	NA	Unclear
Case report: a young man with frontal traumatic sinus pericranii	Zihao Zhang et al 2024	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Endovascular transvenous embolization combined with direct punction of the sinus pericranii: a case report	Kessler et al 2009	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Bilateral frontal sinus pericranii with an intratabular course: a case report	Schenk et al 2010	Yes	Unclear	Yes	Yes	NA	NA	NA	Yes
Cerebrofacial venous anomalies, sinus pericranii, ocular abnormalities: a developmental delay	Macit et al 2012	Yes	Yes	Yes	Yes	NA	NA	NA	Yes
Symptomatic sinus pericranii with adult onset headache: a case report with pathologic perspective	Chung et al 2019	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Lateral sinus pericranii with internal jugular vein communication	Abera et al 2023	Yes	Yes	Yes	Yes	Yes	No	No	Yes
Sinus pericranii: a case report and literature review	Paiva et al 2023	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii with dominant venous outflow in the superior eyelid	Ito et al 2016	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
A rare and deceptive venous anomaly, sinus pericranii	Rizvi et al 2015	Yes	Unclear	Yes	Yes	Yes	Yes	Yes	Yes
Primary cutaneous meningioma in association with a sinus pericranii	Zeikus et al 2006	Yes	Yes	Yes	Yes	Yes	No	No	Yes
Crouzon disease associated with sinus pericranii: a report on identical twin sisters	Yasuda et al 1993	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii: color Doppler ultrasonographic findings	Yanik et al 2006	Yes	Yes	Yes	Yes	NA	NA	NA	Yes
Sinus pericranii: from gross and neuroimaging findings to different pathophysiological changes	Wen et al 2005	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii in the right frontal region and thrombosis	Wang et al 2009	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Chronic intermittent intracranial hypertension caused by sinus pericranii and transverse sinus stenosis: a unique case report in a child	Tang et al 2024	Yes	Yes	Yes	Yes	NA	NA	NA	Yes
Characteristics of recurrent congenital sinus pericranii: case report and review of the literature	Tamura et al 2019	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Giant lateral sinus pericranii: case report	Spektor et al 1998	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Three-dimensional printing of a sinus pericranii model: technical note	Simonin et al 2017	Yes	Yes	Yes	Yes	NA	NA	NA	Yes
Sinus pericranii: dermatologic considerations and literature review	Sheu et al 2002	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Staged raising of a coronal flap for fronto-orbital advancement and remodeling in Saethre-Chotzen syndrome complicated by sinus pericranii	Shaw et al 2018	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Endovascular embolization with onyx in the management of sinus pericranii: a case report	Rangel-Castilla et al 2009	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii: case report	Aburto-Murrieta et al 2011	Yes	Yes	Yes	Yes	Yes	No	No	Yes
Sinus pericranii: an overview and literature review of a rare cranial venous anomaly (a review of the existing literature with case examples)	Akram et al 2012	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii complicated by hydrocephalus: case report and literature review	Almujaiwel et al 2020	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii: a case report and review of literature	Bhutada et al 2012	Yes	Yes	Yes	Yes	Yes	No	No	Yes
Sinus pericranii: radiological and etiopathological considerations	Bollar et al 1992	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Clinical and imaging findings in a rare case of sinus pericranii	Bouali et al 2015	Yes	Yes	Yes	Yes	Yes	No	No	Yes
Sinus pericranii involving the torcular sinus in a patient with hunter's syndrome and trigonocephaly: case report and review of the literature	Brisman et al 2003	Yes	Yes	Yes	Yes	NA	NA	NA	Yes
Endovascular transvenous embolization of sinus pericranii	Brook et al 2009	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Recurrent sinus pericranii in a 14-year-old boy	Brown et al 1987	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii	Buxton et al 1999	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii with a hair collar sign	Cheraghi et al 2014	Yes	Yes	Yes	Yes	No	No	No	Yes
Sinus pericranii “don't judge a lump by its surface”	Shah et al 2010	Yes	Yes	Yes	Yes	Yes	No	No	Yes
Sinus pericranii	David et al 1998	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Occipital sinus pericranii superseding both jugular veins: description of two rare pediatric cases	Frassanito et al 2013	Yes	Yes	Yes	Yes	NA	NA	NA	Yes
Parietal sinus pericranii: case report and technical note	Jung et al 2000	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii: a scalp mass in a 6-month-old boy	Kanavaki et al 2012	Yes	Yes	Yes	Yes	NA	NA	NA	Yes
Craniosynostosis in the presence of a sinus pericranii: case report	Kurosu et al 1994	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii: sonographic findings	Luker et al 1995	Yes	Yes	Yes	Yes	NA	NA	NA	Yes
Blocked ventriculoperitoneal shunt causing raised intracranial pressure diagnosed by prominent sinus pericranii	Mahindu et al 2009	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii associated with craniosynostosis	Mitsukawa et al 2007	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Lateral situated sinus pericranii: case report	Nishio et al 1989	Yes	Yes	Yes	Yes	Yes	Yes	Unclear	Yes
Lateral sinus pericranii	Nozaki et al 1986	Yes	Yes	Yes	Yes	Yes	No	Yes	Yes
Sinus pericranii in the left frontal region involving the superior eyelid: a case report	Grahovac et al 2013	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii with unusual features: multiplicity, associated dural venous lakes and venous anomaly, and a lateral location	Rizvi et al 2010	Yes	Unclear	Yes	Yes	Yes	Yes	Yes	Yes
Spontaneous involution of two sinus pericranii – a unique case and review of the literature	Rozen et al 2008	Yes	Yes	Yes	Yes	NA	NA	NA	Yes
Dynamic morphological changes of thrombosed lateral sinus pericranii revealed by serial magnetic resonance images	Ryorin et al 2017	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii and myoclonic epilepsy: novel features of 3q29 microdeletion syndrome	Samanta et al 2015	Yes	Yes	Yes	Yes	Yes	Yes	No	Yes
Extremely large sinus pericranii with involvement of the torcular and associated with Crouzon's syndrome	Sanders et al 2017	Yes	Yes	Yes	Yes	NA	NA	NA	Yes
Single, small, spontaneous, accessory, closed type, frontal sinus pericranii in a child: Favorable outcome with surgical excision	Satyarthee et al 2013	Yes	No	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii, petrosquamosal sinus and extracranial sigmoid sinus: anatomical variations to consider during a retroauricular approach	Cisneros et al 2016	Yes	Yes	Yes	Yes	Yes	Yes	Yes	Yes
Sinus pericranii: demonstration using three-dimensional surface shading	Curnes et al 2002	Yes	No	Yes	Yes	Yes	Yes	Unclear	Yes

Statistical Analysis

Statistical analysis: Descriptive statistical analysis was performed by Jamovi 2.3.18. Chi-square tests or Fisher's exact tests were used to analyze categorical variables, while t-test was used to evaluate continuous and ordinal variables. A p-value of less than 0.05 was considered to indicate statistical significance.

Results

Study Characteristics

The systematic review yielded 95 studies. After the case identification phase, we excluded 16 studies (16.8%) and 14 (14.7%) in the full-text review phase. Finally, we included 65 studies with 82 patients ([Fig. 2]). The median age was 8 years, ranging from 4 months to 72 years, with a male predominance of 61.0% (n = 50). In most cases (67.1%, n = 55), no contributing factors were reported. However, craniosynostosis and recent trauma were reported as contributing factors to the development of SP in 7.3% (n = 6) and 18.3% (n = 15), respectively. Frontal (37.8%, n = 31), parietal (32.9%, n = 27), and occipital (13.4%, n = 11) were the most reported locations of the SP ([Table 3]). [Fig. 3A] depicts the contributing factors by lesion size and the development of SP following a recent trauma (50 ± 13.8) or congenital anomalies (63.7 ± 39.2) were of the largest size. The median follow-up duration was 24 months (3–96).

Table 3 Univariate analysis of the sinus pericranii studies characteristics

Variables	Value
Age (years)	8.0 [0.3–72.0]
Sex
Male	50 (61.0)
Female	32 (39.0)
Contributing factors
Craniosynostosis	6 (7.3)
Other congenital anomalies	6 (7.3)
Recent trauma	15 (18.3)
No contributing factors	55 (67.1)
Lesion location
Frontal	31 (37.8)
Parietal	27 (32.9)
Temporal	3 (3.7)
Occipital	11 (13.4)
Fronto-parietal	4 (4.9)
Parieto-occipital	4 (4.9)
Parieto-temporal	1 (1.2)
Parieto-temporo-occipital	1 (1.2)
Sinus involvement
Superior sagittal sinus	62 (75.6)
Transverse sinus	8 (9.8)
Sigmoid sinus	1 (1.2)
Sphenoparietal sinus	3 (3.7)
Superior sagittal sinus and torcula	3 (3.7)
Transverse and sigmoid sinus	2 (2.4)
Torcula	3 (3.7)
Clinical manifestations
Chronic headache	7 (8.5)
Intracranial hypertension	19 (23.2)
Local pain	5 (6.1)
Non-pulsatile subcutaneous soft mass of the scalp	51 (62.2)
Diagnosis tools
Magnetic resonance angiography (MRA)	43 (52.4)
Digital subtraction angiography (DSA)	15 (18.3)
Ultrasound with color Doppler	8 (9.8)
Computer tomography angiography	10 (12.2)
MRA and DSA	6 (7.3)
Lesion size (mm)	45.0 [7–150]
Type of sinus pericranii
High flow	10 (12.2)
Low flow	72 (87.8)
Treatment
Conservative	22 (26.8)
Endovascular	10 (12.2)
Surgical	50 (61.0)
Follow-up duration (mo)	24.0 [3–96]
Outcome
Complete resolution	55 (67.1)
Incomplete resolution	22 (26.8)
Recurrence	5 (6.1)

Abbreviation: I-I, interval–interquartile.

Note: The data are represented as the median [I-I], or n (%).

Fig. 3 (A) Contributing factors by lesion size. CST, craniosynostosis; OCT, other congenital anomalies; RT, recent trauma; NoCF, no contributing factors. (B) Clinical presentation by lesion location. (C) Distribution of lesion size by gender.

Clinical Presentation and Lesion Location

Soft, nonpulsatile subcutaneous mass of the scalp that enlarged when lying down and reduced in size when sitting upright was the clinical presentation (62.2%, n = 51) in most reported cases. Intracranial hypertension (23.2%, n = 19) was the second most common clinical presentation for the SP. Chronic headaches (8.5%, n = 7) and local pain (6.1%, n = 5) were also reported. [Fig. 3B] highlights the clinical presentation by lesion location, and the SP revealed by a painless soft mass of the scalp were mostly frontal (80.3%), while those revealed by intracranial hypertension (33.4%), chronic headache (13.3%), and local pain (13.7%) were parietal in the majority of cases.

Sinus Involvement and Lesion Size

Following its development, the SP involved the superior sagittal sinus (75.6%; n = 62), the transverse sinus (9.8%; n = 8), the sigmoid sinus (1.2%; n = 1), the sphenoparietal sinus (3.7%; n = 3), the superior sagittal sinus and torcula (3.7%; n = 3), the transverse and sigmoid sinus (2.4%; n = 2), and the torcula (3.7%; n = 3) ([Table 3]). Moreover, [Fig. 3C] depicts the lesion size by gender, and the largest size lesions were found in females, with a median lesion size of 50 mm, ranging from 15 to 150 mm.

Treatment

[Table 4] and [Fig. 4 (A, B)] highlight the treatment options according to the lesion size and the type of SP. The smallest lesion (a mean size of 41.7 mm, 95% CI [34.7–48.8]) was reported to have been treated by surgical resection with a good outcome of complete resolution of the lesion. Endovascular and conservative treatment were the treatment options for the mean size lesion of 56.0 mm, 95% CI (32.7–79.3), and 58.8 mm, 95% CI (44.4–73.3), respectively.

Table 4 One-way ANOVA testing the mean difference among variables in terms of the type of sinus pericranii, treatment, and outcome

Variables	Age (y)	Lesion size (mm)	Follow-up (mo)	p-Value
Type sinus pericranii
High-flow	18.7 [5.9–31.5]	76.0 [45.1–106.9]	31.8 [13.4–50.2]	0.017
Low-flow	14.5 [10.2–18.8]	44.2 [38.5–50.0]	26.1 [21.7–30.5]	<0.001
Treatment
Conservative	13.6 [5.4–21.9]	58.8 [44.4–73.3]	17.4 [11.3–23.5]	<0.001
Endovascular	15.0 [2.8–27.2]	56.0 [32.7–79.3]	36.0 [11.6–60.4]	<0.001
Surgical	15.6 [10.4–20.8]	41.7 [34.7–48.8]	29.1 [24.3–33.8]	<0.001
Outcome
Complete resolution	14.7 [9.8–19.6]	49.6 [43.3–55.9]	28.2 [22.3–33.9]	<0.001
Incomplete resolution	18.2 [9.6–26.7]	47.1 [29.3–64.7]	22.9 [16.9–28.9]	<0.001
Recurrence	3.8 [0.1–7.6]	35.8 [1.4–72.9]	28.8 [3.8–53.7]	0.314

Note: Results are presented as mean difference (95% CI).

Fig. 4 (A) Treatment choice by the type of sinus pericranii. (B) Outcome by the type of sinus pericranii. (C) Sinus pericranii treatment choice by the outcome.

Outcome

It was statistically significant (p < 0.001) that the more important the lesion size is (mean: 49.6 mm; 95% CI [43.3–55.9]), the more complete resolution was achieved after surgical resection, with a very low recurrence rate of 6.1% within a follow-up mean difference of 28.8 months (95% CI [3.8–53.7]; [Table 4] and [Fig. 4C]).

Discussion

Four key findings emerged from this systematic review: (1) SP revealed by a painless soft mass of the scalp were mostly frontal (80.3%), while those revealed by intracranial hypertension (33.4%), chronic headache (13.3%), and local pain (13.7%) were parietal in the majority of cases. (2) The superior sagittal sinus was involved in most cases (75.6%; n = 62) of SP. (3) The smallest lesion, a mean size of 41.7 mm, 95% CI (34.7–48.8), was reported to have been treated by surgical resection with a good outcome of complete resolution of the lesion, while the endovascular and conservative treatments were the management options for the mean size lesion of 56.0 mm, 95% CI (32.7–79.3) and 58.8 mm, 95% CI (44.4–73.3), respectively. (4) The recurrence rate of 6.1% was within a follow-up mean difference of 28.8 months (95% CI [3.8–53.7]).

SP is a rare lesion defined as an anomalous communication between extracranial and intracranial venous systems in which blood can flow bidirectionally through enlarged vessels in the scalp. SP resembles an emissary vein in its transosseous disposition and connection to diploic drainage. However, it is distinguished by a complex network of thin-walled veins forming a varix on the external table of the skull.[8]

To our knowledge, this is the first systematic review providing a comprehensive analysis of this rare but challenging anomaly, focusing on its clinical features, contributing factors, dural sinus involvement, management, and outcomes.

A total of 65 articles, including 82 cases of SP, were examined. Our results demonstrate that this lesion can affect both pediatric populations and adults. The age range spanned from 4 months to 72 years, with a median patient age of 8 years, and a notable male predominance in 61.0% (n = 50) of cases. These demographic findings are consistent with previously reported cases.[9] [10] [11] [12] [13] [14] [15] [16] [17] [18]

Etiology

The etiology of SP is poorly understood, though congenital factors, trauma, and spontaneous formation have been implicated.[4] Interestingly, most cases (67.1%, n = 55) in our study did not report any contributing factors, highlighting the predominance of idiopathic SP. This type is thought to occur due to chronic cranial pressure caused by an enlargement of a venous aneurysm and bone destruction from cranial osteitis.[19] On the other hand, traumatic SP may result from damage to dural venous sinuses and diploic veins due to cranial injury.[4] In our study, the lesion was associated with trauma in 18.3% (n = 15). This aligns with Zhang et al and Nozaki et al who reported a history of trauma in their patients with SP.[20] [21] Meanwhile, congenital SP is thought to be associated with other congenital vascular anomalies, particularly aneurysmal internal cerebral vein, cavernous hemangioma, and systemic angioma.[4] [22] Other associated congenital abnormalities include craniosynostosis. Our study demonstrated that in 7.3% of our included cases, SP was associated with craniosynostosis. This finding aligns with the reports of Kurosu et al and Sharma Sr. et al, who each describe a case of both anomalies coexisting in patients aged 16 months and 3 years, respectively.[23] [24] Similarly, Mitsukawa et al identified a congenital combination of SP and craniosynostosis in their series of seven patients, with two patients presenting with Apert syndrome, two with Crouzon syndrome, two with oxycephaly, and one with trigonocephaly.[22] Other studies reported an association between SP and other malformations, including syntelencephaly and CHARGE syndrome.[17] [25]

Interestingly, our analysis illustrated a variation in the size of SP by etiology, with posttraumatic lesions and those associated with congenital anomalies exhibiting the largest sizes (50 ± 13.8 mm and 63.7 ± 39.2 mm), respectively, underscoring potential differences in their pathophysiological development.

Clinical Presentation and Lesion Characteristics

A soft, nonpulsatile subcutaneous scalp mass enlarging when lying down and shrinking when sitting upright emerged as the predominant clinical presentation in our study and was observed in 62.2% of the cases. This presentation was consistent across multiple studies, including that of Pavanello et al, where all 21 patients in their study exhibited the same presentation, and Mitsukawa et al, who reported a similar presentation in their series of seven patients.[3] [22] Additionally, other symptoms, including intracranial hypertension (23.2%, n = 19), chronic headaches (8.5%, n = 7), and local pain (6.1%, n = 5), were also reported.

In this review, the frontal region (37.8%, n = 31), followed by the parietal (32.9%, n = 27) and occipital (13.4%, n = 11) regions, was the most commonly affected by SP. This distribution aligns with Pavanello et al, who demonstrated that the most common locations in their case series were median or paramedian, predominantly over the parietal or frontal bone.[3] An important observation from our review is the association between SP location and clinical presentation. Frontal lesions were most often revealed by a painless soft scalp mass (80.3%), while parietal lesions were more frequently associated with intracranial hypertension (33.4%), chronic headache (13.3%), and local pain (13.7%). This is similar to Tamura et al and Tang et al, who each reported a case of parietal SP presenting with intracranial hypertension,[26] [27] and Sheu et al where two patients with parietal SP were reported to have chronic headaches.[28]

The superior sagittal sinus was the most commonly involved venous structure in our study 75.6%; n = 62) compared with other sinuses, including the transverse sinus (9.8%; n = 8) and the torcula (3.7%; n = 3). This is consistent with Mitsukawa et al, where all of their seven patients had SP involving the sagittal sinus.[22]

Diagnosis

SP can be diagnosed using various imaging techniques, including CT, MRI, and Doppler ultrasonography.[29] CT and MRI are useful for identifying any associated bone erosion.[5] Contrast-enhanced MRI and magnetic resonance angiography (MRA) can demonstrate the lesion and its drainage into the dural venous sinuses.[8] However, differentiating SP from other vascular lesions, particularly scalp AVMs can be complex. Brain MRI may not detect the transcranial vein when blood flow is minimal, and cranial CT scans without contrast typically do not show transcranial vessels.[3] DSA is the gold standard for diagnosing SP and differentiating it from other lesions. It detects the exact location, size, and course of the venous malformation and determines its flow dynamics.[3]

Our review demonstrated that most cases of SP were diagnosed using MRA (52.4%), DSA (18.3%), or both (7.3%). This is consistent with several reports, including that of Gandolfo et al, which reported the importance of DSA in classifying SP into two main patterns: “dominant SP” which serves as the primary venous drainage pathway, bypassing standard venous routes, and “accessory SP” which refers to a minor supplementary blood drainage pathway.[8]

Differential Diagnosis

Due to its rarity and similar presentation, SP can be challenging to distinguish from other scalp vascular malformations.[6] Scalp AVMs, for instance, arise due to an abnormal fistulous connection between feeding arteries and draining veins without an intervening capillary bed within the subcutaneous layers.[29] Diagnosing this high-flow vascular malformation relies primarily on CT angiography or diagnostic cerebral angiography, which helps distinguish it from SP, a lesion that communicates with the intracranial venous sinuses without involving the arterial circulation.[30] [31]

Venous cavernoma can present as a fluctuating mass that changes size with posture and can be falsely diagnosed as SP.[32] However, unlike SP, this anomaly receives blood from extracranial carotid and vertebral arteries while draining into either the extra- or intracranial venous channels. Angiography and MRI are important in differentiating the two lesions.[33]

Emissary veins are normal valveless venous channels that can enlarge due to physiological or pathological processes, including venous outflow obstruction or increased venous pressure. Although both SP dilated emissary veins connect extracranial veins to intracranial dural sinuses, SP is characterized by the presence of a subgaleal venous pouch. Clinically, dilated emissary veins may be asymptomatic or present with pulsatile tinnitus and usually lack the characteristic scalp mass with positional size variability.[34]

Cephaloceles, such as encephaloceles, can present as a bluish compressible scalp mass that can transilluminate and change in firmness due to CSF flow within the lesion. CT and MRI are the best modalities for their diagnosis.[35] [36]

Management and Outcomes

Regarding SP management, the lesion is mainly treated to resolve cosmetic problems or to prevent potential complications such as hemorrhage, infection, or traumatic air embolism.[37] The treatment rationale in the management of SP largely depends on whether the lesion is dominant or accessory. As reported by Gandolfo et al, it is contraindicated to treat dominant SP as it may lead to severe complications, including intracranial hypertension, cerebral hemorrhage, or even death. On the contrary, accessory SP is safe to treat.[8]

Treatment options include surgical resection, endovascular approaches, or combined methods.[3] The surgical method, which is considered the gold standard, involves removing the extracranial malformation and closing the emissary vein using bone wax. Outcomes are generally favorable; however, potential complications include hemorrhage due to dural sinus laceration.[37] Surgical disconnection of diploic veins has been associated with uneventful postoperative courses and decreased recurrence rates.[38] In our presented case, a stepwise cauterization of the subcutaneous vascular pouch was performed, with continuous waxing of the fistulous vessels to control blood loss.

In our systematic review, surgical resection emerged as the most effective treatment strategy in patients with a mean lesion size of 41.7 mm, 95% CI (34.7–48.8), achieving complete resolution with a very low recurrence rate of 6.1%. The results are consistent with several reports, including those of Zhang et al and Fujino et al, reinforcing the efficacy of surgical resection in patients with a lesion size of 30 and 40 mm, respectively.[20] [25]

On the other hand, endovascular and conservative treatments were generally reserved for larger lesions with a mean size of 56.0 mm, 95% CI (32.7–79.3) and 58.8 mm, 95% CI (44.4–73.3), respectively. Various studies reported good outcomes with these approaches.[37] [39] [40] [41] However, endovascular treatment was associated with necrosis of the underlying skin, and embolic complications have been reported as adverse effects in the literature.[37]

Strengths and Limitations

This is the first systematic review, supported by a significant sample size, that synthesized data from a wide pool of studies and provided valuable insights on demographics, clinical presentation, and management of this rare vascular malformation. However, we acknowledge some of the limitations, including the inherent impact of collecting evidence primarily from case reports and case series on the quality of the review. Additionally, certain critical variables, such as follow-up duration, lesion size, and outcomes, were not reported in some of the included studies.

Future Directions

Given the complexity and heterogeneity in managing this rare venous malformation, future research should focus on larger multicenter studies and the development of guidelines to standardize diagnostic protocols and management approaches. Additionally, longitudinal studies with long-term follow-ups are essential to assess posttreatment complications, recurrence, and outcomes. Further exploration of treatment modalities, including prospective comparative studies, could provide valuable insights into optimizing patient care.

Conclusion

SP is a rare and complex vascular malformation posing a significant diagnostic and therapeutic challenge due to the absence of established guidelines and existing gaps in the literature. This is the very first systematic review providing a comprehensive analysis of key patterns in presentation, etiology, and management of SP. Our findings reveal that while most cases are idiopathic, congenital predisposition and trauma play an important role in their development. Clinically, SP primarily presents as a soft, fluctuating, nonpulsatile scalp mass commonly involving the superior sagittal sinus. The diagnosis relies essentially on MRA and DSA, and treatment outcomes largely depend on the size of the lesion. Surgical resection proved most effective for smaller lesions with low recurrence rates, while endovascular and conservative modalities were often preferred for larger or complex cases. Although this review provides valuable insights, larger multicenter studies are needed to establish concise standardized diagnostic guidelines and management protocols for SP.

Conflict of Interest

None declared.

Authors' Contributions

H.E.A. contributed to conceptualization, project administration, data curation, title and abstract screening, full-text screening, methodology, writing the draft, reviewing and editing, visualization, supervision, and validation. Y.C.H.D. contributed to data curation, title and abstract screening, full-text screening, data analysis, methodology, writing the draft, reviewing and editing, visualization, supervision, and validation. M.B. contributed to project administration, data curation, reviewing and editing, visualization, supervision, and validation.

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Publication History

Article published online:
03 February 2026