A rare occurrence of tumoural calcinosis of long head biceps tendon post-COVID-19 infection

Mohamad Azwan Aziz; Harmizie Osman; Mohanakumar Kalimuthu; Rozaiman Ebrahim

doi:10.4103/mohe.mohe_28_22

CASE REPORT

Year : 2022 | Volume : 11 | Issue : 2 | Page : 115-119

A rare occurrence of tumoural calcinosis of long head biceps tendon post-COVID-19 infection

Mohamad Azwan Aziz¹, Harmizie Osman², Mohanakumar Kalimuthu², Rozaiman Ebrahim²
¹ Department of Sports Medicine, University Malaya Medical Centre; Sports Medicine Unit, Faculty of Medicine, University of Malaya, Kuala Lumpur, Malaysia
² Department of Orthopedic, Sports Medicine Unit, Hospital Tuanku Jaafar, Seremban, Malaysia

Date of Submission	12-Aug-2022
Date of Decision	22-Oct-2022
Date of Acceptance	08-Nov-2022
Date of Web Publication	22-Feb-2023

Correspondence Address:
Mohamad Azwan Aziz
Department of Sports Medicine, University Malaya Medical Centre; Sports Medicine Unit, Faculty of Medicine, University of Malaya, Kuala Lumpur
Malaysia

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/mohe.mohe_28_22

Abstract

Tumoural calcinosis is a rare entity commonly caused by hyperphosphatemia due to bone mineral disease, hyperparathyroidism of chronic renal failure. However, our case demonstrated a normo-phosphatemic tumoural calcinosis post-COVID-19. This is a 36-year-old with a multiple history of soft-tissue calcification presented with acute onset severe right shoulder pain associated with anterior shoulder swelling at day 20 post-COVID-19. The clinical examination reveals anterior shoulder swelling at bicipital groove with severe restriction of range of motion due to pain. Ultrasound revealed an initial solid mass arising from the sheath of long head of biceps tendon which turns into cystic mass at week 4 of the disease. Computed tomography scan demonstrate sedimentation sign. His blood parameters revealed normo-calcemic, normo-phosphatemic bone profile, normal renal function and no sign suggestive of rheumatological disease. He was started on short course on non-steroidal anti-inflammatory drugs (NSAIDs) for 3 week and does not require surgical intervention. His symptoms completely resolved after 4 weeks with persistent shoulder swelling. He was started with prophylaxis low phosphate diet to prevent future recurrence. Our case demonstrates that conservative management using the short course of NSAIDs can be beneficial in treating primary normophosphatemic tumoural calcinosis.

Keywords: Calcification, long COVID, post-COVID, shoulder pain, tumoural calcinosis

How to cite this article:
Aziz MA, Osman H, Kalimuthu M, Ebrahim R. A rare occurrence of tumoural calcinosis of long head biceps tendon post-COVID-19 infection. Malays J Mov Health Exerc 2022;11:115-9

How to cite this URL:
Aziz MA, Osman H, Kalimuthu M, Ebrahim R. A rare occurrence of tumoural calcinosis of long head biceps tendon post-COVID-19 infection. Malays J Mov Health Exerc [serial online] 2022 [cited 2023 Mar 28];11:115-9. Available from: http://www.mohejournal.org/text.asp?2022/11/2/115/370239

Introduction

Tumoural calcinosis is characterised by the presence of tumour-like calcific mass adjacent to the large joint, which was first described by Inclan et al., 1943. The most common region involved is the shoulder, elbow and hip region, leading to painful joint limitation, especially when it is enlarged (Fathi and Sakr, 2014). Current aetiology remains uncertain as normo-calcemic and normo-phosphotemic are the hallmark of this primary tumoural calcinosis (Fathi and Sakr, 2014). Secondary tumoural calcinosis is commonly due to chronic renal failure with secondary hyperparathyroidism (Fathi and Sakr, 2014). To author's knowledge, this would be the first report on tumoural calcinosis post-COVID-19 infection. Thus, our main aim is to describe report a rare occurrence of tumoural calcinosis post-COVID-19 and outline our approach on managing tumoural calcinosis post-COVID-19.

Case Report

This is a 36-year-old gentleman with acute onset, severe right shoulder pain associated with swelling over the anterior shoulder following COVID-19 infection. He was diagnosed with COVID-19 category 2A on end of April 2022, presented with fever for 3 days associated cough. He was tested positive at day 3 of illness using RTK antigen and was on home quarantine for 7 days. He recovered well from COVID-19 infection after few days of rest. However, on day 20 post-COVID-19, he developed sudden onset, atraumatic, excruciating right shoulder pain with pain score of 8 at rest, radiating to lateral shoulder. The pain was aggravated by slight movement which prevent him from moving his right shoulder to perform activity of daily living. He also complained of night pain which prevent him to have a good sleep. The pain was associated with anterior right shoulder swelling, progressively increasing in size and it is firm in nature. He denied any history of trauma or fall, instability, skin changes or discharge over his right shoulder. He visited staff health clinic at day 3 and he took a 3-week course of non-steroidal anti-inflammatory drugs (NSAIDs). Fortunately, the condition improving. The severe resting pain that is associated with very limited range of motion lasted for 2 weeks, followed by improvement of pain and range of motion for the next 2 weeks. He was pain free after the 4^th week of the disease with full range of motion, swelling disappear on the 8^th week.

He denied any systemic symptoms such as fever, prolong cough, loss of weight, loss appetite, night sweat, rashes or other joint pain or swelling. He denies any hypercalcemia symptoms such as haematuria with abdominal pain, constipation, low mood or bone pain. He does have multiple episodes of bilateral shoulder calcific tendinitis, which resolved after short course of NSAIDs. His diet involved normal balance diet, and he did not consume diary and milk frequently. He did not have any family history of recurrent calcification, recurrent kidney stone or chronic renal failure. He works as pharmacist and does not get exposed to any hazardous chemical or carcinogen. He is nonsmoker and does not consume any alcohol. The clinical examination of his right shoulder at the beginning of presentation reveals swelling at anterior shoulder, firm, tender and associated with limited range of motion in all direction. Summary of his right shoulder physical examinations are listed at [Table 1]. Systemic examination does not reveal any other joint swelling, deformities, scleroderma, rashes or tophi suggestive of rheumatological disease.

Table 1: Right shoulder examinations

Click here to view

In terms of investigations, we performed numerous blood investigations to rule out rheumatological disease and metabolic bone disease. The summary of important investigations is listed in [Table 2]. He had normal renal function test, normal bone profile including parathyroid hormone and negative test for rheumatological screening. His X-ray of his right shoulder was unremarkable and no joint erosion noted suggestive of gout. However, his musculoskeletal (MSK) ultrasound findings were interesting. At 2 weeks of the disease, we found that there is a swelling arising from the sheath of long head of biceps tendon (LHBT) anterior to the bicipital groove and attached to it, with well encapsulated, moves as the LHBT moves, and it is a solid mass with hyper echogenicity inside the mass [Figure 1]. These findings were confirmed with computed tomography (CT) scan of the right shoulder showing anterior bicipital groove mass with sedimentation sign and calcification at the centre [Figure 2]. There was no joint erosion noted. Repeated MSK ultrasound at the 4^th week of the disease showed changes in consistency of the swelling, with cystic in nature and hypoechoic characteristic inside the mass [Figure 3]. The lesion also demonstrated acoustic shadow sign suggestive of presence of hard calcification around the mass. Resolution of the mass was seen at the 8^th week.

Figure 1: Comparison between week 2 and week 4 in the short axis view at bicipital groove of right shoulder. The arrows demonstrate the mass anterior to bicipital groove changing from solid to cystic lesion from weak 2 to weak 4

Click here to view

Figure 2: Axial view of CT scan of shoulder reveals calcification, anterior to bicipital groove

Click here to view

Figure 3: This is a longitudinal axis of bicipital groove demonstrating a cystic mass arising from LHBT by arrowhead. Noted that the arrow demonstrates an acoustic shadow sign suggestive of hard calcification. LHBT: Long head of biceps tendon

Click here to view

Table 2: Blood investigations at 3^rd week of the disease

Click here to view

These findings were suggestive of normo-calcemic, normo-phosphatemic primary tumoural calcinosis of the LHBT of right shoulder post COVID-19. The exact aetiology of this disease is unknown, but we believed that COVID-19 has triggered the cascade of inflammation leading to formation of tumoural calcinosis. Ideally, we should perform excision biopsy for histopathological examination. However, there is a risk of poor wound healing and recurrent. Thus, the patient decided not to take the risk. With short course of NSAIDs, the symptoms improved.

In terms of pain management, he was on short course of NDAIDs for 3 weeks and regular ice therapy. In terms of rehabilitation, after initial inflammatory phase subsides, he was started with isometric shoulder exercise including rotator cuff and scapular. As the pain improving, we progressed him isotonic eccentric rotator cuff exercise with scapular stabilisation exercise using tera band. We also start the patient on low dietary phosphate meals to prevent future recurrence.

Discussion

Post-COVID MSK pain is a rising issue occurred in this pandemic. Meta-analysis by Fernández-de-Las-Peñas et al., 2021 showed that the prevalence of 10% of post COVID patients suffered from MSK pain within 30–60 days. The common MSK pain described were post-COVID reactive arthritis, myalgia, chest pain (Fernández-de-Las-Peñas et al., 2021). However, this is the first case to describe tumoural calcinosis post-COVID-19 infection.

The postulated theory post-COVID tumoural calcinosis in normo-calcemic, normo-phosphotemic conditions lies on the hyper-inflammatory state of COVID-19 infection. Taha et al., 2021 found a strong association of inflammatory markers including ESR, CRP and IL-6 with COIVD-19. This can lead to inflammation around peri-articular structure, and repetitive microtrauma causing haemorrhage. Subsequently, there will be neobursae formation from the reparative process together with friction force (Slavin et al., 1993). Then, transient hyperphosphatemia occurs resulting in the formation of multifactorial calcification due to local released from the injured tissue along with increase in collagenolysis activity (Slavin et al., 1993). Finally, there is declining in collagenolysis activity along with calcific debris formation leading to the formation of tumoural calcinosis (Slavin et al., 1993).

To approach this case, we recommend a detailed history and examination ruling out other possible cause of systemic calcification such as gout, hyperparathyroidism; primary or secondary due to chronic kidney disease, myositis ossificans due to trauma and more sinister cause such as osteosarcoma. A detailed bone profile examination including serum calcium, urine calcium, serum alkaline phosphatase, serum phosphate, Vitamin D level and parathyroid level should be done to differentiate primary and secondary tumoural calcinosis. Connective tissue screening with anti-nuclear, anti-smith and anti-centromere should be done to exclude rheumatological causes. Plain radiograph will demonstrate cloud like calcification at periarticular distribution and CT scan will help confirms the diagnosis with sedimentation sign demonstrating multiple fluid calcium level (Hug and Gunçaga, 1974). Absent of joint erosion is a hallmark of tumoural calcinosis (Olsen and Chew, 2006). Bone scan can be done to investigate multiple lesions, new lesion formation or monitoring disease activity after treatment, but it is not done in our case (Fathi and Sakr, 2014). Biopsy can be done to confirm the diagnosis. However, it is not routinely recommended as it has increased risk of infection, poor wound healing and heterotopic ossificans.

Treatment should be tailored based on patient's best interest, size of lesion, location as well as symptoms. Although few literatures recommend surgical excision for primary tumoural calcinosis, there are high risk of recurrent, high risk of infection, prolong wound healing and high risk of developing heterotopic ossification, which could worsen the symptoms (Steinbach et al., 1995; Lykoudis et al., 2012; Farzan and Farhoud, 2011; King et al., 2011). Thus, we opted for conservative management short course of NSAIDs. Non-calcium-based phosphate binder can be used to prevent recurrence of calcification. Non calcium based should be opted as calcium-based phosphate binder can increase serum calcium level and increase risk of calcification (Mozaffarian et al., 1972; Kirk and Simon, 1981). Low phosphate diet such as reduction in milk and diary products, nuts and fish could be tried to reduce recurrence (Fathi and Sakr, 2014; Mozaffarian et al, 1972; Kirk and Simon, 1981). The use of steroids, diphosphonates, or calcitonin and radiation therapy in managing tumoral calcinosis is not recommended as there is lack of evidence (Fathi and Sakr, 2014). [Figure 4] is the algorithm recommended in management of primary tumoral calcinosis.

Figure 4: Algorithm for management of primary tumoural calcinosis

Click here to view

Conclusion

Primary tumoural calcinosis post-COVID-19 is a rarely reported. Although literature mentioned that first line management is surgical excision, our case has proven that short course of NSAIDs is beneficial in improving pain and reduced the inflammation. For preventive measure, non-calcium-based phosphate binder with low phosphate diet can be used to prevent future recurrence. It is important that we ruled out more common causes such as gouty tophi, hyperparathyroidism or chronic renal failure before concluding the diagnosis of primary tumoural calcinosis.

Declaration of patient

The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given his consent for images and other clinical information to be reported in the journal. The guardian understands that names and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Fathi, I., & Sakr, M. (2014). Review of tumoral calcinosis: A rare clinico-pathological entity. World Journal of Clinical Cases, 2(9), 409-414. doi: 10.12998/wjcc.v2.i9.409.
2.	Farzan, M., & Farhoud, A. R. (2011). Tumoral calcinosis: What is the treatment? Report of two cases of different types and review of the literature. American Journal of Orthopedics (Belle Mead, N.J.), 40(9), E170-E176.
3.	Fernández-de-Las-Peñas, C., Palacios-Ceña, D., Gómez-Mayordomo, V., Florencio, L. L., Cuadrado, M. L., Plaza-Manzano, G., & Navarro-Santana, M. (2021). Prevalence of post-COVID-19 symptoms in hospitalized and non-hospitalized COVID-19 survivors: A systematic review and meta-analysis. European Journal of Internal Medicine, 92, 55-70. doi: 10.1016/j.ejim.2021.06.009.
4.	Hug, I., & Gunçaga, J. (1974). Tumoral calcinosis with sedimentation sign. The British Journal of Radiology, 47(562), 734-736. doi: 10.1259/0007-1285-47-562-734.
5.	Inclan, A., Leon, P. P., & Camejo, M. (1943). Tumoral calcinosis. The Journal of the American Medical Association, 121, 490-495.
6.	King, J. J., Brennan, K. B., Crawford, E. A., Fox, E. J., & Ogilvie, C. M. (2011). Surgical complications associated with extensive tumoral calcinosis. American Journal of Orthopedics (Belle Mead, N.J.), 40(5), 247-252.
7.	Kirk, T. S., & Simon, M. A. (1981). Tumoral calcinosis. Report of a case with successful medical management. The Journal of Bone and Joint Surgery. American Volume, 63(7), 1167-1169.
8.	Lykoudis, E. G., Seretis, K., & Ristanis, S. (2012). Huge recurrent tumoral calcinosis needing extensive excision and reconstruction: Report of a rare case and brief literature review. Aesthetic Plastic Surgery, 36(5), 1194-1197. doi: 10.1007/s00266-012-9923-0.
9.	Mozaffarian, G., Lafferty, F. W., & Pearson, O. H. (1972). Treatment of tumoral calcinosis with phosphorus deprivation. Annals of Internal Medicine, 77(5), 741-745. doi: 10.7326/0003-4819-77-5-741.
10.	Olsen, K. M., & Chew, F. S. (2006). Tumoral calcinosis: Pearls, polemics, and alternative possibilities. Radiographics: A Review Publication of the Radiological Society of North America, Inc., 26(3), 871-885. doi: 10.1148/rg.263055099.
11.	Slavin, R. E., Wen, J., Kumar, D., & Evans, E. B. (1993). Familial tumoral calcinosis. A clinical, histopathologic, and ultrastructural study with an analysis of its calcifying process and pathogenesis. The American Journal of Surgical Pathology, 17(8), 788-802.
12.	Steinbach, L. S., Johnston, J. O., Tepper, E. F., Honda, G. D., & Martel, W. (1995). Tumoral calcinosis: Radiologic-pathologic correlation. Skeletal Radiology, 24(8), 573-578. doi: 10.1007/BF00204854.
13.	Taha, S. I., Samaan, S. F., Ibrahim, R. A., El-Sehsah, E. M., & Youssef, M. K. (2021). Post-COVID-19 arthritis: Is it hyperinflammation or autoimmunity? European Cytokine Network, 32(4), 83-88. doi: 10.1684/ecn.2021.0471.