Venous malformations (VMs) of the head and neck region are a common condition that
occurs due to abnormal vascular morphogenesis. Typically, VMs are soft and compressible.
The common symptoms include low self-esteem due to an unsightly appearance, pain because
of thrombosis, and rarely, palpable phleboliths. Phleboliths result from the stagnation
of blood in these low-flow malformations, causing a thrombus and progressive lamellar
fibrosis, followed by mineralization. We present a case of a VM on the cheek with
multiple large palpable phleboliths.
Our 21-year-old patient presented with facial asymmetry and irregularity on the left
side of the face since childhood ([Fig. 1]). He was diagnosed with a large VM of the left side cheek in the masticator space,
buccal space, infra-temporal fossa, and left parotid and submandibular spaces with
multiple phleboliths ([Fig. 2]).
Fig. 1. Preoperative image with multiple large phleboliths (black arrows) and a solitary
venous malformation on the upper lip (red arrow).
Fig. 2. Magnetic resonance imaging (from 2019).
The optimal treatment for VM would be complete excision of the lesion to avoid any
recurrence [1]. However, in this patient, excision could have resulted in a deformity worse than
the malformation. The accepted first-line treatment of problematic VMs is sclerotherapy
[2], but a palpable phlebolith remains unaffected by sclerotherapy and could be an indicator
for surgery.
Considering the diffuse nature of the malformation in the cheek, with a high risk
of iatrogenic facial nerve injury and bleeding with surgery, this patient was advised
to undergo sclerotherapy initially. The commonly used agents in sclerotherapy include
sodium tetradecyl sulfate (STS) and ethanol [2]. Since STS is a safer agent in the vicinity of the nerve, three sclerotherapy sessions
with 16, 14, and 9 mL of intralesional STS each were done in the interventional radiology
suite. One year after the last session of sclerotherapy, a repeat magnetic resonance
imaging scan demonstrated a marginal decrease in size of the lesion in addition to
decreased intensity when compared to imaging done 2 years previously, suggestive of
scar tissue following sclerotherapy. However, predictably, there was no change in
the size of phleboliths ([Fig. 3]). Surgery was planned for excision of phleboliths, not the VM, given its diffuse
nature and potential risk of injury to the facial nerve. We successfully removed seven
phleboliths of different sizes through a pre-auricular incision, the largest being
1.5 × 1.5 cm, without significant blood loss or any facial nerve injury ([Fig. 4]). Another solitary VM on the upper lip was excised, and primary closure was done.
Postoperatively, the patient was advised to wear a chin strap for compression. At
a 4-month follow-up visit, the patient was satisfied with a smooth surface on the
cheek ([Fig. 5]).
Fig. 3. Magnetic resonance imaging (from 2021).
Fig. 4. Intraoperative view of the excised phleboliths.
Fig. 5. Photograph at a 4-month follow-up visit.
Multiple palpable phleboliths in a VM of the head and neck are an uncommon indication
for combination treatment using sclerotherapy and surgery, which has not yet been
discussed in the literature. There have been reports of patients with individual palpable
phleboliths of the maxillofacial region, which have been excised, or multiple small
phleboliths for which only sclerotherapy was indicated [3]. This is an unusual case presentation, wherein a surgical intervention was indicated
for the deformity due to phleboliths. Using sclerotherapy before surgery was beneficial
by minimizing intraoperative bleeding, thereby enabling a reasonably clean dissection
that avoided any inadvertent nerve injury and led to a satisfactory outcome.
NOTES
Patient consent
The patient provided written informed consent for the publication and the use of his
images.
Author contribution
Conceptualization: S Agarwal. Visualization: S Agarwal. Writing - original draft:
F Cordelia M J, S Agarwal. Writing - review & editing: K Paul M.
