We believe this case is the first reported Purtscher-like retinopathy and PAMM following HA fillers involving breast tissue. We postulate that there was an inadvertent inoculation of HA into the artery supplying the breast which then passed on to the subclavian artery, the brachiocephalic trunk and onward to the arch of aorta, ascending to both common carotid arteries and to the internal carotid arteries. Finally, the molecules reached the ophthalmic arteries and then the distal branches of the central retinal artery. (Figure 4). The tendency for embolic occlusion is higher in distal arteries than in proximal arteries as the luminal diameter decreases with distality. [5]
Although the patient had presented to the ophthalmologist only two weeks later, the clinical features of Purtscher’s retinopathy were apparent in both fundi, mainly the haemorrhages and cotton wool spots in the posterior pole. The intact choriocapillaris supplying the outer one-third of the retina through the choroidal circulation gave rise to a pseudo-cherry-red spot appearance in contrast to the perifoveal rim of retinal whitening. Small patches of retinal whitening also known as Purtscher’s fleckens (Figure 1A and 1B) were visible. The SD-OCT confirmed the typical findings of PAMM with increased band-like hyperreflectivity seen in the inner nuclear layer.
PAMM is believed to occur when there is ischemia of the intermediate and deep capillary plexus. [6] In this case, the smaller size of HA soft tissue fillers is postulated to pass downstream to arterioles and precapillary vessels causing hypoperfusion and retinal ischaemia. The inner nuclear layer and outer plexiform layer may act as a watershed zone being more susceptible to ischaemia. The secondary inflammation provoked by this mechanical occlusion may cause perivascular oedema and further exacerbate hypoperfusion by vaso-compression.
There were four previous case reports on PAMM following non-facial filler injection. Khatibi reported a case after PMMA injection into patient’s buttock muscles, with good visual improvement after a course of systemic steroid therapy [7]; Bruno et.al. reported a similar case following silicone injection in both thighs and buttocks [8]. Table 1 compares our case with other cases of visual loss following non-facial filler injections. [9][10] To our knowledge, there are no reported cases using HA for breast tissue enhancement presenting with visual loss.
There is no standard evidence-based management for visual loss secondary to filler. However, early and prompt intervention may be potentially sight-saving. [11] Hyaluronidase enzyme is a proposed treatment modality to reverse blindness from HA filler injection. [12] Sharudin et.al. reported a case of full visual recovery following subcutaneous hyaluronidase in a patient with HA-induced visual loss. [13] Carruthers et.al. suggested that early hyaluronidase administration within 60-90 minutes can possibly disintegrate HA emboli [14]. Hyaluronidase was not used in our patient as she presented two weeks after the onset of symptoms, which is delayed beyond the aforementioned golden period. Furthermore, hyaluronidase was not readily available when the patient presented to us.
Systemic corticosteroids in the form of intravenous or oral steroids have been used in the management to reduce inflammation which may further compromise blood flow. Although our patient presented only after two weeks, she had already received systemic hydrocortisone and oral steroids for the pulmonary involvement. While the visual acuity was still poor at counting fingers, there was gradual visual recovery noted with the continuation of an anti-inflammatory dose of oral corticosteroids. The possibility of this being part of the natural course of PAMM which may be self-limiting cannot be ruled out.
This patient first developed shortness of breath soon after the breast filler procedure and was initially managed by the physicians for life-threatening respiratory distress syndrome. This delayed her referral to the ophthalmologist. Pulmonary complications following HA injections are less common and usually related to non-thrombotic pulmonary embolisms (NTPEs). [15][16] Very few cases have reported diffuse alveolar haemorrhage (DAH) the cause of which remains unclear. Some have postulated an immune-mediated response to excessive HA, while others have attributed it to abnormal haemostasis and disruption of the alveolar-capillary integrity. [17] Besides this, she also developed an altered sensorium and was found to have a left corona radiata infarct, raising the possibility of filler-induced cerebral embolism (FICE) which has been reported in the literature [18].