An innovative hybrid training method for ultrasound in giant cell arteritis, polymyalgia rheumatica and large vessel vasculitis

doi:10.21203/rs.3.rs-2754910/v1

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An innovative hybrid training method for ultrasound in giant cell arteritis, polymyalgia rheumatica and large vessel vasculitis

https://doi.org/10.21203/rs.3.rs-2754910/v1

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To circumvent the lack of hands-on ultrasound training resulting from inability to conduct face-to-face workshops during the COVID-19 pandemic, especially as local expertise was not available, we developed a novel hybrid method combining virtual and physical training. The objective was to achieve appropriate expertise in ultrasound for the safe and accurate diagnosis of giant cell arteritis, polymyalgia rheumatica, and large vessel vasculitis. To evaluate the effectiveness of training, competency assessment was conducted using the hybrid platform.

COVID-19

giant cell arteritis

hybrid

medical education

ultrasound training

virtual education

The pandemic in Singapore was part of a global pandemic caused by the severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2). National travelling restrictions were mandated in Singapore and many other countries worldwide, aimed at curbing the COVID-19 virus’ spread. For the ensuing 2 years, all overseas leave for public institution healthcare workers were frozen due to manpower needs. As local and overseas physical training for ultrasound (US) was halted, there was an acute unmet need for greater innovative approaches for the delivery of US training in the context of safe-distancing and accessibility. Hands-on training is essential for the rheumatologists’ professional development of US skills. Therefore, to circumvent the lack of hands-on training resulting from inability to conduct face-to-face workshops, we developed a novel hybrid method of US training combining virtual and physical training to enable continuity of training. This article reports on how we set out to achieve our objectives and explores our journey on how postgraduate training in US can be transitioned to a hybrid method combining virtual and physical platforms as an effective method to acquire US skills.

Since the advent of COVID-19 pandemic, there has been several articles on medical student education using electronic or online platforms,¹ barriers and impact faced by hybrid virtual teaching in medical student education,^2,3,4 and hybrid model for organising scientific events.⁵ Notably, many difficulties were faced by medical education during the pandemic such as lack of social interactions and impact on educators who are required to learn new skills to engage students and manipulate the electronic platform.² Other concerns reported were the lack of clinical exposure, for example in surgery. ⁵ Similar concerns would apply to postgraduate US training whereby the training cannot be effectively delivered entirely by digital means. To the best of our knowledge, there has not been any publication on this method of training for US.

Hybrid Training Method

We focused on ultrasound training for giant cell arteritis (GCA), polymyalgia rheumatica (PMR), and large vessel vasculitis (LVV), as there was a clinical need to develop this subspecialty service. The objective was to achieve appropriate expertise in US for the safe and accurate diagnosis of GCA, PMR and LVV. The training programme consisted of a series of physical and virtual hybrid sessions. Initially, we had attended a 2-day introductory on-site workshop prior to the pandemic (conducted in Southend University Hospital, United Kingdom), with demonstration of scanning techniques by an international teaching faculty, followed by hands-on practice on actual patients.

In March 2020, when the World Health Organization (WHO) declared the COVID-19 outbreak a global pandemic, the on-site physical course was replaced by a virtual webinar. The latter involved didactic lectures via Zoom videoconferencing and online interactive cases led by the speaker (BD). The key objectives for these first 2 sessions were to develop the theoretical knowledge and practical skills of the practitioner, be familiar with the clinical applications for diagnosis and assessment and recognise imaging findings of specific lesions in GCA. The latter was reinforced through hands-on practical scanning techniques.

We (CLC and CH, Rheumatologists at Singapore General Hospital, Singapore), intended to complete the formal training and accreditation despite the COVID-19 disruptions. As local trainers with the necessary expertise were not available, we decided to introduce the hybrid-physical and -virtual training into our skills acquisition and training programme in 2021, as many countries globally were still under ‘lockdown’ measures.

The training was conducted by the trainer (BD) who was physically based in England over 2 consecutive sessions. Total hands-on scanning and didactic lectures were 4.5 and 1.5 hours, respectively. The anatomical areas covered were cranial arteries (common superficial temporal, parietal and frontal branches, facial, occipital, vertebral, and common carotid arteries), as well as extra-cranial arteries, such as axillary and subclavian arteries. The US scans were performed using a linear probe with grey-scale frequency ranging from 18MHz to 24MHz and a colour Doppler frequency of 9MHz. The pulse repetition frequency (PRF) was set at 2-3kHz.

We invited 4 subjects with cranial signs and symptoms for the hands-on scanning. The trainer was able to view the entire protocolised scanning in real-time through Zoom videoconferencing using a portable laptop device. A laptop camera was set-up to focus on the trainee performing the scans. The real-time B-mode and color Doppler US images from the US machine monitor were also visible to the trainer via Zoom video. This enabled the trainer to effectively assess the trainees’ scanning techniques. The trainer then provided verbal feedback and critique on the scanning techniques and images obtained, akin to a face-to-face training session.

Training Assessment and Outcomes

The trainer assessed the individual trainees through the hybrid-physical and -virtual methods, as described above. A crucial part of the training was maintaining a logbook of all the cases scanned. The minimum requirement from each candidate was 40-50 cases, including normal and abnormal scans. Logbook images with sonographic pathology (for example, halo sign, non-compressive halo, stenosis, and intimal media thickening) were sent electronically to the trainer to be appraised, whereby the quality of the images was assessed based on accuracy of diagnosis and interpretation, precision in measurements, as well as appropriate labelling. Intimal media thickness (IMT) was measured at the point of maximum thickness of the relevant vessels in the longitudinal plan. Halo sign was morphologically defined as US finding of a dark, hypoechoic, non-compressible area around the vessel lumen. Selected clinical cases, including mimics of GCA, were discussed. This provided the trainees with an authentic learning experience which simulates daily clinical practice.

Upon completion of the full training, the trainees were granted certification of competency in US for GCA, PMR and LVV. Ongoing continuing professional development (CPD) is paramount for maintaining standards and updating skills. To fulfill CPD, the trainees were required to demonstrate achieving a specified minimum number of scans per year and attending relevant US-vasculitis workshops/conferences at recommended intervals.

Mimics of GCA

Here, we describe 3 cases of GCA mimics. Case 1: ANCA-associated vasculitis (AAV) overlap with GCA. A 74-year -old Chinese lady who does not smoke nor drink, presented to the Ear, Nose and Throat (ENT) clinic with a 2-week history of left sided hearing loss and tinnitus. Magnetic resonance imaging (MRI) of the internal auditory meatus showed otomastoiditis and hemorrhagic labyrinthitis. She was treated with a course of oral prednisolone 50mg (1mg/kg body weight) daily for 2 weeks. However, 3 months later, there was progressive hearing loss involving bilateral ears. Repeated MRI showed worsening bilateral otomastoiditis with granulation tissue and effusion, for which she was treated with intratympanic dexamethasone injections. Subsequently, 3 months later, she developed acute left eye visual loss associated with anterior ischaemic optic neuropathy (AION) and cranial nerves III and VII palsies. Further investigations revealed acute kidney injury (baseline 60umol/L rising to 214micromol/L), significantly raised ESR 135mm/hr. and CRP 142 mg/dL, and positive anti-MPO 24.1 RU/ml (NR <20). A comprehensive malignancy screen was unyielding, and brain MRI/MRA as well as lumbar puncture were unremarkable. Renal biopsy showed necrotising pauci-immune crescentic glomerulonephritis with small vessel vasculitis. Her GCA probability score (GCAPS) was 14 (high-risk category). She was hence treated as GCA, but additional tests were recommended given the alternative diagnosis of AAV which fulfilled the 2022 American College of Rheumatology/European League Against Rheumatism (ACR/EULAR) classification criteria for microscopic polyangiitis. She underwent a left temporal artery biopsy which was consistent with temporal arteritis, as evidenced by marked intimal thickening causing severe luminal narrowing and, in some places, complete obliteration of the lumen and presence of multinucleated giant cells.⁶ She was subsequently diagnosed with AAV with ENT, renal and neurological involvement, overlap with GCA, and was treated with pulsed intravenous methylprednisolone and cyclophosphamide as induction therapy, with oral methotrexate as maintenance therapy. An 8-vessel ultrasound including bilateral cranial (superficial temporal, parietal and frontal) and axillary arteries were performed for monitoring of treatment response 2 months after she received high dose glucocorticoids. The sono-features were normal (no thickening of intima media thickness (IMT), no halo sign, and negative compression sign; considering that she had already received a significant cumulative dose of glucocorticoids. Overlap of biopsy proven GCA and AAV has been reported, albeit rare. The duration between presentations of GCA and AAV can range from 1 week to 8 years, whereas some cases were diagnosed simultaneously.⁷ A ten-year retrospective Swiss AAV cohort found a significantly higher total prevalence of AAV with associated LVV, than the estimated prevalence of LVV in the normal Swiss population (OR 234.9, 95% CI 91.18–605.2, p < 0.001).⁸A differential diagnosis of AAV with temporal arteritis⁹ as a manifestation was considered, given that the patient presented with CN VII palsy which is unusual for GCA. Notably, in GCA, apart from the optic nerve, the most commonly affected cranial nerves are CNs III, IV, and VI. Lower cranial nerve involvement, such as CN VII is unusual. Other features which may indicate non-GCA include, refractoriness to treatment or early relapse despite high dose glucocorticoid therapy, presence of fibrinoid necrosis and small branch vasculitis, and a negative US (halo sign is a sensitive test which has high negative predictive value). This case illustrates that manifestations of GCA are protean and heightened suspicion of an alternative diagnosis in the presence of overlapping and atypical manifestations of GCA is critical. A wider workup for mimics and overlap conditions is crucial in establishing the correct diagnosis and appropriate treatment. Importantly, cranial US scan should be performed in a timely manner, ideally within 1-2 weeks from the onset of symptoms, to minimise false negative findings masked by the glucocorticoids. Fast-track pathways play a key role in expeditious GCA diagnosis and hence limiting the risk of sight loss.¹⁰

Case 2: Idiopathic hypereosinophilic syndrome. A 59-year-old Indian man with a history of childhood asthma and gout presented with recurrent episodes of amaurosis fugax (3 episodes within 24 hours), low grade fevers, and lethargy. He was found to have raised Immunoglobulin (Ig) E with significant eosinophilia with absolute eosinophil count ranging at 15-17x10(9)/L for 1 month prior to the onset of amaurosis fugax. Eye examination showed no signs of AION and fundal examination was unremarkable. He was empirically treated with ivermectin for presumptive parasitic infection. He had extensive workup for hypereosinophilia which revealed negative stool parasites, autoimmune serology, and normal ECHO and pan-CT. ESR and CRP 106 mg/dL were both raised at 70mm/hr and 106 mg/dL respectively. Peripheral blood lymphoma flow cytometry showed no clonal B cells and myeloma screen was negative. Bone marrow trephine showed normocellular trilineage hematopoiesis with eosinophilic hyperplasia without histological features of myeloproliferative or lymphoproliferative neoplasm. His GCAPS was 8 (low probability category). The US of cranial and axillary arteries showed borderline IMT and halo sign, at the left common superficial temporal and frontal arteries, and right common superficial temporal artery. However, compression sign was negative for all 6 cranial vessels. Amaurosis fugax was likely a manifestation of a thrombotic phenomenon associated with hyperviscosity from hypereosinophilia. He was diagnosed with presumptive idiopathic hypereosinophilic syndrome by the hematologist. The patient declined TAB and his peripheral eosinophilia did improve with prednisolone 60mg daily which was rapidly tapered over 2 months before cessation. A rare case of hypereosinophilia with corticosteroid-responsive temporal region swelling and digital ischemia due to non-granulomatous eosinophilic vasculitis has been reported, for which US revealed occlusion and corkscrew-like changes of the temporal arteries and TAB histology showed eosinophilic vasculitis without giant cell formation.¹¹

Case 3: Atherosclerosis. A 48-year-old Chinese lady with a history of rheumatoid arthritis was seen by a private rheumatologist who noticed that she had palpable thickened temporal arteries and was subsequently referred to a tertiary referral centre to exclude GCA. She did not have any ischaemic symptoms or signs, such as visual symptoms or jaw claudication, however she reported bilateral shoulder, posterior scalp tenderness and dizziness. Eye examination showed no ophthalmic features of GCA. CRP was slightly raised at 30 mg/dL but ESR was normal. Her GCAPS was 8 (low probability category). 8-vessel US scan showed halo signs in bilateral parietal and left superficial temporal arteries, with normal axillary arteries. She had a 18FDG PET-CT which showed no evidence of GCA or large vessel vasculitis. MRA of the brain showed no evidence of intracranial vasculitis, however there were patchy areas of minimal mural thickening and mild mural enhancement along bilateral superficial temporal artery branches without luminal narrowing. In view of the suspicious MRI findings, she underwent a TAB which showed marked luminal narrowing of artery due to sub-intimal atherosclerotic plaque with no evidence of granulomatous inflammation or giant cells. Atherosclerosis may raise suspicion of large vessel GCA (LV-GCA), but certain non-GCA characteristics can differentiate between these two conditions.¹² For example, in atherosclerosis, CRP is usually not significantly elevated (<10 mg/L), and PET-CT FDG-tracer uptake is generally lower (grade 1) than in GCA (grade 2 or 3). Atheromatous lesions are often located in vessels of the lower limbs and form focal lesions at the origin of collaterals and appear heterogeneous and irregular, whereas GCA lesions are diffuse and commonly involve the ascending aorta, supra-aortic trunks, and the aortic arch, and are more homogeneous and linear.¹³ One study found that atherosclerotic disease with a carotid IMT >0.9 mm increases the temporal artery IMT and might mimic the halo sign, thus producing false positives in GCA diagnosis.¹⁴

Although this form of US training has limitations, such as, the lack of face-to-face contact, we were still able to acquire effective training by harnessing technology and combining physical and virtual training methods. Given the hands-on nature of US training and the ongoing circumstances surrounding the pandemic, training would otherwise be unattainable. In 2022, with the easing of the travel restrictions, and reopening for large group physical conferences, the trainees attended a refresher course in Southend Hospital, U.K. as part of continuing professional development.

GCA is associated with irreversible vision loss in 15–25% of cases.⁶ Therefore early recognition is essential for timely institution of treatment. GCA diagnosis is made by a combination of clinical, US, and laboratory findings. A study by Patil¹⁰, showed that a staggering 93% patients with sight loss in whom temporal artery US was performed had a positive result, and more than half of the patients with uncomplicated GCA without ischemic injury were also US positive. One of the key components is the fast-track clinic pathways, first developed in Southend Hospital, where patients with suspected GCA undergo the 8-vessel GCA scan (bilateral common superficial temporal arteries, the parietal and frontal branches, and axillary arteries in longitudinal and transverse axes), after algorithmic stratification based on probability score triage.¹⁵ The algorithm categorises low-risk category as < 9, intermediate risk category as 9–12, and high risk category as > 12, respectively. The probability score, used in conjunction with US, enhances the test performance of US and aids interpretation of US in context. This value adds by excluding mimics and identifying diagnostic uncertainty, and hence the need for re-evaluation and using alternative tests to confirm diagnosis. This evidence-based approach of probability-based diagnostic algorithm using US may be applied to other arthritides (such as gout, rheumatoid arthritis, seronegative spondyloarthritis, and polymyalgia rheumatic) to facilitate diagnosis and management in daily clinical practice. In fact, a rheumatology point of care ultrasound (POCRUS) approach using scenario-based teaching is currently being developed and has been recently adopted by the Asia-Pacific League of Associations for Rheumatology (APLAR) to deliver online courses.

In summary, we proposed a novel hybrid approach of virtual and physical hands-on training complemented by online learning, as an effective method to acquire US skills. Furthermore, it provides an invaluable access to distant specialist cognitive and procedural expertise during pandemic-times, when conventional means may not be feasible. Even as the world gradually moves out of ‘lockdown’, the pandemic has serendipitously led to novel ways of doing things; the lessons learnt here can be harnessed for future hybrid training models, especially where hands-on experience is crucial. Ultimately, by combining virtual technology and physical teaching, we strive to reduce worldwide training inequalities, particularly in specialised areas where the expertise may not be available locally.

Challenges faced during the pandemic, including the need for physical distancing, limitation in group size, and overseas travel restrictions, may be mitigated by developing a novel hybrid training method, notwithstanding the trainer/preceptor and trainee situated in different countries or physical locations.
Hybrid physical and virtual hands-on ultrasound training strategy in GCA may be an effective method for acquiring ultrasound skills.
The pre-requisite of successful virtual hands-on training is to have an appropriate setting. These includes adequate video conferencing platforms, as well as documentation in the form of stored images and logbook records. Additionally, it is important for the trainees to possess prior basic ultrasound skills.

Ethics approval and patient consent to participate: All methods were carried out in accordance with the relevant guidelines and regulations. Informed consent was obtained from all subjects. Ethical approval waiver has been obtained from SingHealth Centralised Institutional Review Board (CIRB).

Consent for publication: Informed consent was obtained from all subjects.

Availability of data and materials: The datasets used/or analysed during the current study are available from the corresponding author on reasonable request.

Competing interests: The authors have declared no conflicts of interests.

Funding: No funding was received to carry out the work described in this article.

Authors’ contributions: CLC conceived the idea and design of the work. BD contributed to the design of the work and supervised the ultrasound scanning. CLC and CH contributed to acquisition of data including case studies of the scans. CLC was the major contributor in writing the manuscript. All authors were involved in the drafting and editing of the manuscript content and approved the final version published.

Acknowledgements: Not applicable.

GCA Giant cell arteritis

GCAPS Giant cell arteritis probability score

IMT Intima media thickness

LVV Large vessel vasculitis

MRI Magnetic resonance imaging

PMR Polymyalgia rheumatica

PRF Pulse repetition frequency

US Ultrasound

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No competing interests reported.

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An innovative hybrid training method for ultrasound in giant cell arteritis, polymyalgia rheumatica and large vessel vasculitis

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