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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 2  |  Issue : 1  |  Page : 10-13

Coexistence of ankylosing spondylitis and seropositive rheumatoid arthritis


1 Department of Rheumatology, Prince Mohammed Bin Abdulaziz Hospital, Riyadh, Saudi Arabia
2 Department of Radiology, Prince Mohammed Bin Abdulaziz Hospital, Riyadh, Saudi Arabia

Date of Submission01-Nov-2021
Date of Decision09-Jan-2022
Date of Acceptance20-Jan-2022
Date of Web Publication25-May-2022

Correspondence Address:
Dr. Hanan Ali Alhamzi
Prince Mohammed Bin Abdulaziz Hospital, Riyadh
Saudi Arabia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ara.ara_8_21

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  Abstract 


Although rheumatoid arthritis (RA) and ankylosing spondylitis (AS) are among the most common rheumatic diseases, reports of patients who had a combination of both diseases are rare. There are no one classification criteria to diagnose the coexistence of both. This coexistence is a rare presentation that has an implication on the patient management. We are presenting the case of a 69-year-old male who had an unusual combination of AS and RA manifestations and we reviewed similar cases that previously reported to see what the features of those cases are and how can this combination affect the patient management.

Keywords: Ankylosing spondylitis, rheumatoid arthritis, sacroiliitis


How to cite this article:
Aldahneen FA, Almotairy AS, Alhamzi HA. Coexistence of ankylosing spondylitis and seropositive rheumatoid arthritis. Ann Rheumatol Autoimmun 2022;2:10-3

How to cite this URL:
Aldahneen FA, Almotairy AS, Alhamzi HA. Coexistence of ankylosing spondylitis and seropositive rheumatoid arthritis. Ann Rheumatol Autoimmun [serial online] 2022 [cited 2022 Jun 29];2:10-3. Available from: http://www.ara.ssr.com/text.asp?2022/2/1/10/345955




  Introduction Top


Rheumatoid arthritis (RA) and ankylosing spondylitis (AS) are different in their symptomatology and etiology. Both diseases can lead to a reduction in physical activity with increased joint destruction.[1],[2] Advanced diagnostic and laboratory techniques can differentiate the two conditions.[3]

It is rare to diagnose RA and AS in the same patient because RA and AS have widely different pathophysiology and underlying genetic parameters.[2]

Here, we report one patient with these two diseases, with a literature review of similar cases, to see the common features of the coexistence of both diseases. This is the first case reported in the Middle East and North Africa region.


  Case Report Top


A 69-year-old male patient, a smoker with coronary artery disease and chronic kidney disease, presented to the rheumatology clinic with a history of 25 years of inflammatory low back pain and treated himself as needed with nonsteroidal anti-inflammatory drugs (NSAID) without regular follow-up with a rheumatologist.

Five years later, he started to complain of hand, knee, shoulder, and elbow joint pain associated with 30 min early morning stiffness with episodic swelling of the joints.

He denied any history of eye symptoms, oral, genital ulcers, chronic diarrhea, or skin rash.

There was a limitation in neck rotation (turn 70° right, 70° left). Modified Schober test was 4 cm, occiput to wall test was normal. There was symmetrical polyarthritis that is typical for RA, involving both wrists and metacarpophalangeal (MCP) joints. There was left elbow and ankle joints swelling with limitation of the movement. He had Z deformity of both thumbs. His hemoglobin was 10.8 g/dL (normal 12-15 g/dL), white blood cell count 8.5 × 109/L (normal 4 × 109/L-11 × 109/L), and platelet 359 × 109/L (normal 150 × 109/L-500 × 109/L). He has high inflammatory markers (erythrocyte sedimentation rate 76 mm/h [normal 0–15] and C-reactive protein (CRP): 3.4 mg/dl [normal 0.01–0.50]) and normal serum uric acid 389 micromole/l (normal: 210–420). Other tests showed positive human leukocyte antigen (HLA)-B27, positive anti-cyclic citrullinated peptide (anti-CCP) (169 units) (normal range <20), and positive rheumatoid factor RF (93 IU/ML) (normal range <20).

Radiologically, there were the features of both diseases. Sacroiliac X-ray showed fusion of both sacroiliac joints [Figure 1] that also was clear on sacroiliac magnetic resonant image (MRI) [Figure 2]. There were bilateral erosions of the MCP joints, intercarpal and radiocarpal joint space narrowing in hand X-ray [Figure 3]. Syndesmphytes had been found in lateral spine radiograph and lumber spine MRI [Figure 4]. In addition, there was bone marrow increase signal intensity of anterior superior endplate of L5 indicative of bone marrow edema [Figure 5].
Figure 1: Anterior posterior axial sacroiliac joint radiograph (top) demonstrates ankylosis of both sacroiliac joints with right posterior oblique view (lower left) and left posterior oblique view (lower right)

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Figure 2: A coronal oblique magnetic resonant imaging T1-weighted images of the sacroiliac joints showing bilateral ankylosis more pronounced on the right side

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Figure 3: Bilateral hand radiograph AP view showing generalized osteopenia, bilateral erosions of the metacarpophalangeal joints (white arrows), intercarpal and radiocarpal joint space narrowing

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Figure 4: Left side; lateral spine radiograph and right side; sagittal magnetic resonant imaging T2-weighted images of the lumbar spine showing multilevel lumbar spine syndesmophytes (arrows)

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Figure 5: Axial and coronal T2 with fat suppression showing bone marrow increase signal intensity of anterior superior endplate of L5 indicative of bone marrow edema

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Based on clinical features, laboratory and radiological features, the patient was diagnosed with both seropositive RA with AS.

There were features of latent tuberculosis (TB) (positive tuberculin skin test and normal chest x-ray). Anti-TB prophylaxis was started (the plan is to continue for 9 months).

After 1 month of anti-TB, etanercept was started. Within 3 months, the patient reported excellent outcome. He did not use any NSAID and had no more back or joint pain. Bath ankylosing spondylitis disease activity index was 0.9 (remission) and disease activity score 28-CRP: 2.5 (remission).


  Discussion Top


The prevalence of AS in various countries ranges from 0.7 to 49 per 10,000,[1] while the prevalence of RA varies from 0.7% to 3%.[2],[3]

AS is more common in males, and RA is more common in females. AS onset usually occurs before 30, but RA happens later between 40 and 50 years of age.[4]

In RA cases, HLA-DR4 and HLA-DR1 are known to be significant genetic backgrounds. HLA-B27 is strongly associated with AS, but HLA-DRB1 has also been linked to this disease too. HLA-B27 is not associated with RA.[5]

The research by Toussirot and Acquaviva[6] revealed that 6.6% of patients with RA have positive HLA-B27. RA patient with positive HLA-B27 does not have increased incidence of enthesitis or sacroiliitis. From those with positive HLA-B27, about 5% can have AS.[7] Tumor necrosis factor (TNF) is a pro-inflammatory cytokine that plays an essential role in the pathogenesis of both disorders.

There is a variation in the clinical presentation of both diseases. RA patients have symmetrical synovitis of multiple small hand and feet joints, mainly in the proximal interphalangeal, MCP joints, wrist, and metatarsophalangeal joints. While in AS one-third of cases usually have asymmetric peripheral arthritis that involves a single large joint. The most characteristic symptoms of AS are bilateral sacroiliitis and enthesitis. In RA, sacroiliitis is rare.[4]

RF and anti-CCP are the characteristic serological markers of RA. The positivity of one or both is one of the classification criteria of RA. In another way, RF should not be positive in AS, and it is very rarely seen positive (low titer) in patients with AS.[4]

Radiological features of RA are usually present in the wrist and hand small joints. The radiological features include bone erosions, joint space narrowing with periarticular osteopenia, and lastly joint distortion and bone fusions. The sacroiliac joints and spine are the sites for characteristic radiological features of AS and revealed as syndesmophytes and bone fusions.[4],[6] In the case described here, radiological changes were typical for both RA and AS, as shown in [Figure 1], [Figure 2], [Figure 3], [Figure 4].

Synthetic disease-modifying antirheumatic drugs (DMARDs) should be started as soon as possible in newly diagnose RA. No benefit from synthetic (DMARDs) in the AS without peripheral joint inflammation. TNF inhibitors are biological drugs with high efficacy in both diseases.

In 1995, Toussirot and Acquaviva[6] reported three patients and analyzed 44 similar cases. Positive RF and sacroiliitis were present in all patients, 29 patients had syndesmophytes, and 40 out of 43 patients had HLA-B27 positive. Typical erosions for RA and rheumatoid nodules were found in 38 and 18 patients, respectively.

In 2011, Guo et al.[7] reported a 30-year-old female patient diagnosed with coexisting RA and AS and analyzed 20 similar cases described in China.

In 2014, Dundar et al.[8] reported a 63-year-old female patient diagnosed with both RA and AS. Rituximab treatment, in that case, showed good result after anti-TNF failure.

In 2015, Barczyńska et al.[4] described three cases. All of them have inflammatory back pain, symmetrical hand joints arthritis, positive HLA B27 and RF. One of them had positive anti-CCP and rheumatoid nodules.

In 2019, Bryan-Josué et al.[9] reviewed 73 patients with concurrent diagnoses of RA and AS are described. It includes 71 patients detected in the literature and two of his cases. Rheumatoid nodules, low back pain, syndesmophytes, erosions in the hand and feet X-rays and radiographic sacoiltits were present in 56%, 85%, 78%, 88%, and 99%, respectively. RF and HLA B-27 were positive in 90% of these cases.

Interestingly, it has been found that mucosal microbial dysbiosis occurs more commonly in RA patients than controls. While change in microbial diversity within subject has been more reported as a risk factor more than specific pathogen.[10]

Porphyromonas gingivalis is one of these pathogens that may drive citrullination of peptides and anti-CCP.[11] Moreover, anti–P. gingivalis and anti–Prevotella intermedia antibody titers have been found to be higher in patients with spondylarthritis than in healthy people.[12] The role of mucosal microbial dysbiosis in coexistence of both diseases, especially with smoking needs to be examined further.

From our case and the cases mentioned in the literature review, we deduce the following:

  • The presence of inflammatory back pain and symmetrical hand arthritis should alert the rheumatologist to the possible coexistence of RA and AS
  • RF plays a role in the coexistence of AS and RA in these patients because most of the patients that have been reported have RF positive
  • In most of the reported cases HLA B27 was positive. This may indicate that patients with positive rheumatoid factor and positive HLA B27 should be monitored for development RA and AS
  • The use of anti-TNF may mask the feature of one of these diseases and make the diagnosis of coexistence AS and RA rare
  • The presence of triple-positive factors (RF, ACCP, and HLA B27) may make the response to a specific treatment, for example, anti-TNF more than the other.


Coexistence AS and RA needs to be managed based on the more active and aggressive symptoms.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Dean LE, Jones GT, MacDonald AG, Downham C, Sturrock RD, Macfarlane GJ. Global prevalence of ankylosing spondylitis. Rheumatology (Oxford) 2014;53:650-7.  Back to cited text no. 1
    
2.
Laine VA. Rheumatic complaints in an urban population in Finland. Acta Rheumatol Scand 1962;8:81-8.  Back to cited text no. 2
    
3.
Al-Dalaan A, Al Ballaa S, Bahabri S, Biyari T, Al Sukait M, Mousa M. The prevalence of rheumatoid arthritis in the Qassim region of Saudi Arabia. Ann Saudi Med 1998;18:396-7.  Back to cited text no. 3
    
4.
Barczyńska TA, Węgierska M, Żuchowski P, Dura M, Zalewska J, Waszczak M, et al. Coexistence of rheumatoid arthritis and ankylosing spondylitis. Reumatologia 2015;53:279-85.  Back to cited text no. 4
    
5.
Hossein A, Matthew V. Rheumatoid Arthritis and Ankylosing Spondylitis. Treasure Island (FL): StatPearls Publishing; 2022.  Back to cited text no. 5
    
6.
Toussirot E, Acquaviva PC. Coexisting rheumatoid arthritis and ankylosing spondylitis discussion of 3 cases with review of the literature. Clin Rheumatol 1995;14:554-60.  Back to cited text no. 6
    
7.
Guo YY, Yang LL, Cui HD, Zhao S, Zhang N. Coexisting ankylosing spondylitis and rheumatoid arthritis: A case report with literature review. Chin Med J (Engl) 2011;124:3430-2.  Back to cited text no. 7
    
8.
Dundar U, Çevik H, Demirdal US, Toktas H. Use of rituximab to treat a patient with coexistence of rheumatoid arthritis and ankylosing spondylitis: 18 months follow-up. Int J Rheum Dis 2018;21:1869-72.  Back to cited text no. 8
    
9.
Bryan-Josué FR, Valvanera P, Angel EI, Eztizen LS, Leticia MM, Juan A LM, et al. Coexistence of ankylosing spondylitis and rheumatoid arthritis (analysis of 73 cases). Sci Abstr 2019:473. doi: 10.1136/annrheumdis-2019-eular.4233.  Back to cited text no. 9
    
10.
Tsuji Y, Tamai M, Morimoto S, Sasaki D, Kawashiri SY, Yanagihara K, et al. Oral dysbiosis reflects the immunological alteration of RA regarding to HLA DRB1*SE, ACPA and cigarette smoking: NAGASAKI Island study. Ann Rheum Dis 2021;80:998.  Back to cited text no. 10
    
11.
Mikuls TR, Thiele GM, Deane KD, Payne JB, O'Dell JR, Yu F, et al. Porphyromonas gingivalis and disease-related autoantibodies in individuals at increased risk of rheumatoid arthritis. Arthritis Rheum 2012;64:3522-30.  Back to cited text no. 11
    
12.
Öğrendik M. Oral anaerobic bacteria in the etiology of ankylosing spondylitis. Clin Med Insights Arthritis Musculoskelet Disord 2017;10:1-2.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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