Abstract
Objectives
Hemangioma is a common soft tissue tumor that can be categorized mainly into cavernous, capillary, and venous subtypes. It usually develops in the head and neck and rarely in the external auditory canal (EAC).
Methods
A systematic review of patients with capillary hemangioma of the EAC was performed. A computerized systematic search was conducted in PubMed, Scopus, and Web of Science to extract relevant studies for a qualitative review.
Results
A total of 38 patients were included in the systematic review, and half of them were men. Hearing loss was the most common presenting feature (n = 19; 50%), followed by aural fullness (n = 15; 39.4%). Cavernous hemangioma was the predominant type (n = 22, 57.8%). Endaural and transcanal surgical approaches were the most frequent, with ten (26.3%) and nine (23.6%) cases, respectively. Most of the patients (n = 31; 81.5%) had no recurrence.
Conclusions
Hemangioma of the EAC may not be uncommon as healthcare professionals may expect. Hearing loss, aural fullness, and tinnitus are possible symptoms, but many patients remain asymptomatic. Surgical excision is the main effective management option, and complete resolution is the most prevalent outcome. Computed tomography assists in the diagnosis, but histopathological examination after resection is mandatory for the definitive diagnosis.
Introduction
Hemangioma is defined as a benign vascular tumor that frequently affects the head and neck region. Based on histopathological findings, hemangioma can be classified into capillary, venous, and cavernous subtypes. Capillary hemangioma is recognized from the tightly coordinated capillary-like canals, while venous and cavernous subtypes are composed of larger vascular spaces. 1 In addition, arteriovenous malformation is also considered to be a category of hemangioma. 2 Hemangioma may arise within the temporal bone, particularly in the geniculate ganglion and internal auditory meatus, and frequently arises in the temporal segment of the facial canal as well as in the hypotympanic area. 3 Hemangioma affecting the external auditory canal (EAC), with or without eardrum involvement, is also rarely reported in the literature. 4 Furthermore, it may be asymptomatic and incidentally diagnosed or suspected in patients with bloody otorrhea, hearing loss, and pulsatile tinnitus. 5 It is important to identify the site and size of capillary hemangioma to obtain an appropriate management approach. Therefore, the aim of this study was to systematically review all patients diagnosed with capillary hemangioma of the EAC in the literature to provide approximate statistics and a better understanding of reported lesions, management plans, and outcomes.
Patients and Methods
This systematic review was performed according to the recommendations of the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA; Supplementary Appendix 1). Our protocol was prepared and registered in PROSPERO (ID number
Search Strategy and Study Selection
In May 2020, a systematic computerized literature search was performed in PubMed, Scopus, and Web of Science using the following search terms: “external auditory canal” OR “external acoustic meatus” OR “external auditory meatus” AND “hemangioma.” Potentially relevant references were also searched manually. All articles reporting hemangioma in, but not limited to, the EAC were included. Studies were excluded if (i) the lesion was not in the EAC; (ii) did not provide individual data; (iii) they were reviews, book chapters, or posters; (iv) the language was not English; and (v) the full-text was unavailable. Articles were collected in one library using Endnote and then exported to an Excel file after removing duplicated references. Among the remaining reports, two authors independently screened the articles for potential eligibility using titles and abstracts.
Data Extraction and Quality Assessment
A data extraction form was prepared to blindly extract the data on study characteristics (publication year, country, year of research, and study design), patient demographics (age and sex), presenting symptoms, audiometric findings, radiological findings, lesion pathological type, surgical treatment approach, and follow-up results. Discrepancies were settled through a discussion among the authors. All included studies were assessed by at least two independent reviewers using the CARE checklist for case reports to evaluate the possible risk of bias. The reviewers then aAgreed on a quality rating among “good,” “fair,” and “poor,” as suggested by the guidelines.
Statistical Analyses
Simple descriptive statistics were performed to report variables; the mean, standard deviations, and frequencies were calculated using Microsoft Excel.
Results
Literature Search
A total of 172 articles were retrieved through a systematic search. After removing duplicates, only 130 reports remained for title/abstract screening. Of these, only 46 were potentially relevant. Finally, 35 case reports were eligible (Figure 1). Flow diagram of study selection and screening.
Basic Demographic Characteristics
The mean age of enrolled subjects (n = 38) was 46.6 ± 17.03 years. Men and women accounted for 50% of the reported population each. Hearing loss was the most common presenting feature (n = 19; 50%), followed by aural fullness (n = 15; 39.4%) and tinnitus (n = 10; 26.3%) (Figure 2). Individualized basic demographic data are presented in Table 1. Most studies were assessed with a good quality score (29 studies); however, five studies scored fair, and only one scored poor because of the lack of case report definitions in the title, keywords, or abstract; insufficient introduction; no adequate follow-up duration; or no mention of the informed consent obtained from the patient (Table 1). A detailed assessment of each domain of the CARE quality assessment tool is provided in Supplementary Appendix 2. Bar chart of reported presenting symptoms in enrolled cases. Individualized Basic Demographic Data of Included Cases. Note: M: male; F: female; EAC: external auditory canal.
Patients’ characteristics, interventions, and outcomes
Characteristics, Interventions, and Outcomes of Reported Lesions.
Blank cells have not been reported. Abbreviations: TM, tympanic membrane; ME, middle ear; EAC, external auditory canal; CHL, conductive hearing loss; Y, year; M, month; STSG, split-thickness skin graft; SNHL, sensorineural hearing loss.
Discussion
Hemangioma is a common soft tissue tumor that can be categorized into cavernous, capillary, and venous subtypes. To our knowledge, a total of 38 patients have been reported to have hemangioma of the EAC in the literature. Cavernous hemangioma accounted for more than half (57%) of the reported cases. Generally, cavernous hemangioma is most frequently reported in adults, while capillary hemangioma is most frequently reported in infants. This may be explained by the spontaneous regression of capillary lesions over time, as they develop in the dermis and more superficially than the cavernous type, which grows more deeply. 4 Capillary lesions are usually confined to the EAC. Although this was the most common finding (68.4%), and this report focused on the database search of EAC hemangiomas, tympanic membrane involvement has been reported. Two patients have had bone involvement,33,34 and one of them developed recurrence despite complete excision. 34 All reports were published from 1972 to 2020, with no sex predominance or specific and consistent presenting features. A study by Freedman et al 36 reported two men: one with otalgia, while the other with only a mass. Chen et al recently reported a boy presenting with hearing loss due to an EAC-localized capillary hemangioma. 2 As hemangioma is usually small, ranging from 5 mm to 3 cm, some patients may remain asymptomatic. A literature search yielded nine cases presenting with a mass only. A prompt diagnosis of capillary hemangioma of the EAC may be challenging, and the differential diagnosis includes carcinoma, glomus tumor, cholesteatoma, and arteriovenous malformation. 7 The otoscopic examination usually shows a small sessile or pedunculated mass present in the anterior or posterior segments of the EAC. Our search showed that computed tomography was performed in 27 (71%) of 38 patients. Pure-tone audiometry, computed tomography of the temporal bone, and magnetic resonance imaging are of great value to assess the extent of the lesion, detect bony erosion, and direct the next step of surgical management. 19 In addition, angiography may be helpful in identifying and embolizing the feeding blood vessels, particularly in large lesions with probable excessive bleeding. 19 Nonetheless, histopathology after surgical removal is essential to confirm the diagnosis and rule out any other possible pathology. Although careful observation may be sufficient for delicate asymptomatic lesions, surgical excision is the mainstay effective approach, mostly with the endaural, transcanal, or retroauricular approach.2,4,6-9 Our findings indicated that complete resolution with no recurrence is the most common outcome (81.5%), although recurrence was reported by Jackson et al. 34 Therefore, we recommend that surgeons perform further follow-ups. While the present study is the first to systematically review all cases of capillary hemangioma of the EAC, it has some limitations. First, we searched only three major databases. Second, we included only English articles, thereby possibly missing some relevant studies. Therefore, the results of the present study should be cautiously interpreted in clinical practice.
Conclusion
This short report revealed that hemangioma of the EAC may not be as uncommon as healthcare professionals may expect. The systematic search yielded 38 patients with a wide range of ages, and cavernous hemangioma was the most common type. Hearing loss, aural fullness, and tinnitus are possible presenting symptoms. Surgical excision is the most effective treatment modality, but postoperative follow-ups are recommended to avoid possible recurrence and bone erosion. Although computed tomography assists the diagnosis, histopathology following surgical resection is essential for the definitive diagnosis.
Supplemental Material
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Supplemental Material, sj-pdf-1-afs-10.1177_01455613211042449 for Clinical Approaches To External Auditory Canal Hemangiomas: A Systematic Review by Afrah Alshalan, Tawfiq Khurayzi, Majed Assiri, Abdulrahman Alsanosi and in Ear, Nose & Throat Journal
Footnotes
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The author(s) received no financial support for the research, authorship, and/or publication of this article.
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References
Supplementary Material
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