04 April 2022: Articles
Management of an Extensive Intraparotid and Auricular Arteriovenous Malformation
Rare disease
Sze Ying Yee E* , Shuyi Guo E , Chi Long Ho EDOI: 10.12659/AJCR.935337
Am J Case Rep 2022; 23:e935337
Table 2. Summary of the reported cases of arteriovenous malformation of the auricle in the literature.
| Authors | Nr pt | Age (y), gender | Lesion site | Symptoms, duration (Schobinger clinical stage) | Key Imaging modalities | Main feeding arteries | Treatment | Follow up (FU) duration; outcome |
|---|---|---|---|---|---|---|---|---|
| Ramadass T (2000) []6 | 1 | 25 y, M | R auricle | Swelling and bleeding 7 y (Stage III) | Angio: enlarged and tortuous vessels | PAA, OA | Staged surgery: Exc+STSG >> Ear elevation | 4 m; lost to FU after last surgery |
| Pham TH (2001) []7 | 1 | 41 y, M | L auricle | Swelling, PT, intermittent bleeding, 6 y (Stage III) | Angio: diffuse network of shunts | PAA, STA, OA | Embo >> Exc+STSG | 2 y; no recurrence |
| Wu JK (2005) []8 | 41 | 26 y (range 1–55) | Auricle and extraauricular involvement (Retroauricular: 46.3%, Neck: 22%, None: 22%) | PT, bleeding, pain, bruit/thrill (Stage II and III) | Angio and MRI | PAA, STA, OA | Observation (nr: 12, 29.3%), Embo (nr: 9, 21.9%), Exc+Embo (nr: 20, 48.8%) | 5y FU in 20 pts with amputation: Controlled (n: 16) Improved (n: 3) Persistent (n: 1) |
| Meher R (2008) []9 | 2 | pt 1: 16 y, M; pt 2: 22y, F. | pt 1: L auricle; pt 2: R auricle. | pt 1: Intermittent pain; pt 2: swelling, PT, bleeding 10 y (both Stage III) | pt 1: DS: Multiple dilated anechoic areas; pt 2: MRA: dilated serpiginous structures. | pt 2: PAA, STA | Both had Exc+STSG | NA |
| Saxena SK (2008) []10 | 1 | 21 y, F | R auricle | Swelling+PT (Stage II) | Angio: diffuse shunts with STA | STA | Failed Embo >> Exc+PAL | 3 y, no recurrence |
| Wu HJ (2008) []11 | 1 | 20 y, M | L auricle | PT 7m (Stage II) | MRI: abnormal signal voiding intensity of the mass | PAA, STA | Failed Embo >> Exc | 2 y, no recurrence |
| Whitty LA (2009) []12 | 1 | 15 y, M | L auricle | Swelling, bruit+intermittent pain 2 y (Stage III) | NA | NA | Exc | 1m, no recurrence |
| Zheng LZ (2009) []13 | 17 | 25.4 y (range 3–47), 11 M, 6 F | Auricle | Ear swelling, redness, thrill/ bruit, ulceration, hemorrhage and infection (majority in Stage II and III). All were present at birth | Angio contralateral | PAA, STA, OA, FA, MA, branches of ICA and ECA | Embo (nr: 17, 100%); prior PAL, incomplete resection, and/or Embo (nr: 10, 58.8%) | 3–4 m; AVMs were devascularized 100% (nr: 3, 17.6%), ≥50% (nr: 11, 64.8%), |
| Jin YB (2009) []14 | 8 | 31.5y (range 10–59), 5 M, 3 F | Auricle | Bleeding, ulceration, disfiguring (Stages I, II and III) | MRI | NA | Embo (nr: 8, 100%); prior PAL (nr: 1, 12.5%). | 12.6 m (range, 5–27 m). AVMs were devascularized 100% (nr: 6, 75%), ≥50% (nr: 2, 25%) |
| Prasad KC (2011) []15 | 1 | 45 y, M | Auricle | Swelling, 2–3 y (Stage II) | Angio: AVM and PAA aneurysm | PAA | PAL+Exc | 2 y, no recurrence |
| Goel A (2011) []16 | 1 | 22 y, F | R auricle | Swelling, pulsatile tinnitus bleeding, 4 y (Stage III) | DS: AVM | NA | Exc+STSG | NA |
| Meena BK (2013) []17 | 1 | 21 y, F | L auricle | Swelling, pulsatile tinnitus bleeding 1 y (Stage III) | DS, CTA: Enlarged serpiginous structures | PAA, STA | Exc | NA |
| Dixit SG (2013) []18 | 1 | 21 y, M | Auricle | Swelling since birth (Stage II) | MRA: Tortuous vessel | PAA | Embo | NA |
| Anesti K (2014) []5 | 1 | 34 y, M | L auricle and L parotid gland | Progressive enlargement, bleeding, pain, skin tightness and pulsations at night (Stage III) | MRI: AVM of the left ECA supplying the scalp and the left ear | STA, OA | Embo >> Exc+MCF closure | No recurrence |
| In’t Veld M (2016) []19 | 1 | 30 y, M | L auricle | Pain, redness, pulsatile swelling >10 y (Stage III) | MRI: Vessels with prominent flow voids | PAA, STA | Embo | 2 y; no recurrence |
| Kim SH (2017) []20 | 1 | 60 y, M | R auricle | Swelling+bleeding with PT 3 y (Stage III) | CTA: Inner vascular tangles | PAA | Embo >> Exc | 3 m; no recurrence |
| Ishikawa K (2021) []21 | 1 | 46 y, M | Auricle | Swelling/macrotia (Stage III) | Angio | NA | Embo >> scleroth >> Exc | 6 m; no recurrence |
| Yee & Ho (2022) [current study] | 1 | 40 y, F | L parotid+ L auricle+ extra-auricular | Swelling+palpable thrills, 6 m (Stage II) | MRI: Serpiginous vessels with “honeycomb” flow voids | PAA, STA | Embo >> Exc+MCF closure | 3.5 y; no recurrence |
| Angio – angiography; AVM – arteriovenous malformation; BWPC – bone wax packing and curettage; CTA – computed tomography angiography; CECT – contrast enhanced CT; Embo – embolization; DS – Doppler sonography; ECA – external carotid artery; Exc – excision; FA – facial artery; LA – lingual artery; IAA – inferior alveolar artery; ICA – internal carotid artery; MA – maxillary artery; MCF – musculocutaneous flap; MRI – magnetic resonance imaging; MRA – magnetic resonance angiography; NA – no available information; nr – number(s); OA ,– occipital artery; PAA – posterior auricular artery; PAL – proximal artery ligation; PT – pulsatile tinnitus; Scleroth – sclerotherapy; STA – superficial temporal artery; STSG – Split-Thickness Skin Graft; Symbols >> – followed by; + – and; F – female; M – male; m – month(s); pt – patient; R – right; L – left; y – year(s). | ||||||||