HLA B27-positive ankylosing spondylitis professional soccer player with a successful return to sports

Abstract

Background: Ankylosing spondylitis (AS) is a disease of young adulthood and without adequate treatment it dramatically reduces physical activity. Aim of the work: To present a case with AS that successfully returned to professionally playing sports after treatment. Case presentation: A 19-year-old Eastern European Russian professional male soccer player with AS complained of right ankle joint pain not related to any trauma or injury and gradually intensified. The player's regular medical tests during the preceding years and were unremarkable. He would occasionally complain of discomfort in the ankle joints, sacroiliac area and symphysis but did not limit his regular training activity. He received the standard treatment of sports injuries that included: 14 days of limited physical activity, ibuprofen 200 mg twice/day, cryotherapy and joint taping. Examination revealed mobile joints, regular in shape without any swelling. Erythrocyte sedimentation rate (ESR) was 60 mm/1st hr and C-reactive protein (CRP) 50 mg/dl. AS was diagnosed as he fulfilled the classification criteria. HLA-B27 was positive, left sacroiliitis was present on magnetic resonance imaging (MRI). The Bath AS disease activity index (BASDAI) was 5.9. Treatment also included golimumab 50 mg/month. He improved, returned to regular training and continued playing sports professionally amid supportive therapy. The last BASDAI was 1.5. Conclusions: AS patients with initially high levels of physical activity may restore professionally playing of sports when a proper management plan is provided. Further monitoring is warranted and the creation of a database for athletes with rheumatic diseases is recommended in order to standardize treatment protocols.

Full text

Case reports
HLA B27-positive ankylosing spondylitis professional soccer player with
a successful return to sports
Eduard Bezuglov
a,b
, Maria Shoshorina
a,
⇑
, Mikhail Butovskiy
c
, Yuri Kuklin
d
, Kamila Kubacheva
e
,
Vladimir Khaitin
f,g
, Ryland Morgans
a
a
Department of Sports Medicine and Medical Rehabilitation, Sechenov First Moscow State Medical University (Sechenov University), Moscow, Russia
b
High Performance Sport Laboratory, Moscow Vitte University, Moscow, Russia
c
Football Club ‘‘Rubin”, Kazan, Russia
d
Football Club ‘‘Ural”, Ekaterinburg, Russia
e
Saint-Petersburg State Public Institution «City Hospital №40», Saint-Petersburg, Russia
f
Pavlov First Saint-Petersburg State Medical University, Saint-Petersburg, Russia
g
Football Club ‘‘Zenit”, Saint-Petersburg, Russia
article info
Article history:
Received 30 May 2023
Accepted 3 June 2023
Keywords:
Ankylosing spondylitis
Soccer
Spondyloarthritis
BASDAI
HLA-B27
abstract
Background: Ankylosing spondylitis (AS) is a disease of young adulthood and without adequate treatment
it dramatically reduces physical activity.
Aim of the work: To present a case with AS that successfully returned to professionally playing sports after
treatment.
Case presentation: A 19-year-old Eastern European Russian professional male soccer player with AS com-
plained of right ankle joint pain not related to any trauma or injury and gradually intensified. The player’s
regular medical tests during the preceding years and were unremarkable. He would occasionally com-
plain of discomfort in the ankle joints, sacroiliac area and symphysis but did not limit his regular training
activity. He received the standard treatment of sports injuries that included: 14 days of limited physical
activity, ibuprofen 200 mg twice/day, cryotherapy and joint taping. Examination revealed mobile joints,
regular in shape without any swelling. Erythrocyte sedimentation rate (ESR) was 60 mm/1st hr and C-
reactive protein (CRP) 50 mg/dl. AS was diagnosed as he fulfilled the classification criteria. HLA-B27
was positive, left sacroiliitis was present on magnetic resonance imaging (MRI). The Bath AS disease
activity index (BASDAI) was 5.9. Treatment also included golimumab 50 mg/month. He improved,
returned to regular training and continued playing sports professionally amid supportive therapy. The
last BASDAI was 1.5.
Conclusions: AS patients with initially high levels of physical activity may restore professionally playing
of sports when a proper management plan is provided. Further monitoring is warranted and the creation
of a database for athletes with rheumatic diseases is recommended in order to standardize treatment
protocols.
Ó2023 The Authors. Publishing services by ELSEVIER B.V. on behalf of The Egyptian Society of Rheumatic
Diseases This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/
licenses/by-nc-nd/4.0/).
1. Introduction
Ankylosing spondylitis (AS) is a synonym for radiographic axial
spondyloarthritis (axSpA), and along with non-radiographic axSpA
(less severe forms and early stages of AS) conform a broader dis-
ease of axSpA [1]. It is a relatively rare severe disease of the
musculo-skeletal system and other organs and is characterized
by prolonged inflammation of the spine and various joints. The
most commonly affected joints are the knees, sacroiliacs, ankles
and spine. Extra-articular conditions are reported and involve the
eyes and intestines [2,3]. Although, the exact cause of the disease
is unknown, the association between AS and HLA-B27 has been
confirmed [2,4–6,7]. AS is more prevalent in young adults, most
commonly men aged 25–35 years [6,8] with a significant restric-
tion of physical activity [9]. Diagnosis is based on the clinical pic-
ture and positive human leucocytic antigen B27 (HLA-B27) with
an increase in systemic markers of inflammation such as erythro-
cyte sedimentation rate (ESR) and C-reactive protein (CRP). In early
https://doi.org/10.1016/j.ejr.2023.06.001
1110-1164/Ó2023 The Authors. Publishing services by ELSEVIER B.V. on behalf of The Egyptian Society of Rheumatic Diseases
This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Peer review under responsibility of Egyptian Society of Rheumatic Diseases.
⇑
Corresponding author.
E-mail address: kaisough@yandex.ru (M. Shoshorina).
The Egyptian Rheumatologist 45 (2023) 267–272
Contents lists available at ScienceDirect
The Egyptian Rheumatologist
journal homepage: www.elsevier.com/locate/ejr

stages, magnetic resonance imaging (MRI) allows the identification
of inflammation in large joints [1,10].
The Bath AS disease activity index (BASDAI) allows a quick and
safe assessment of therapy and a score > 4 is considered an indica-
tor of an active process that requires a revision of the treatment
strategy [11,12]. The treatment options include non-steroidal
anti-inflammatory drugs (NSAIDs), analgesics, disease-modifying
antirheumatic drugs (DMARDs), tumor necrosis factor (TNF-
a
)
inhibitors and anti-interleukin-17 which may provide a prolonged
remission [13]. Local steroids can also be administered for symp-
tomatic relief.
Professional sports place specific demands on the musculo-
skeletal system and involve frequent post-exercise inflammation
of the muscles, tendons, bones and joints. In the vast majority of
cases, it is considered practically impossible to engage in profes-
sional sports with such a diagnosis.
This is a leading article that presents the clinical changes of a
young national team soccer player who returned to regular train-
ing after 16 months of treatment and medical rehabilitation.
2. Case presentation
In April 2018, a 19-year-old Russian (Eastern European) profes-
sional soccer player (height: 170 cm; weight: 65 kg; BMI: 22.5)
with a 13 year playing history at a leading Russian Premier League
soccer club and without any family history of inflammatory joint
diseases, subjectively reported to the club doctor. The main com-
plaints were pain in the right ankle joint, without prior connection
Fig. 1. Magnetic resonance imaging (MRI) of the right ankle. (A) Grade 1 on 2018 showing reactive bone marrow edema (BME) and (B) Grade 2 on 2020 showing reactive BME
and sclerosis with reduced signs of synovitis.
E. Bezuglov, M. Shoshorina, M. Butovskiy et al. The Egyptian Rheumatologist 45 (2023) 267–272
268

with injury that gradually intensified over the previous days. Upon
examination, the joint was considered normal with good mobility.
The study was approved by the local ethical committee (N 11–19)
and the written consent to participate was obtained from the
patient.
During the previous five years prior to treatment, the player
regularly completed medical tests twice a year with no remarkable
signs of inflammation or changes in blood tests. Previously during
the year, the player occasionally complained of discomfort in the
ankle joints, sacroiliac area and symphysis, although did not limit
his regular training activity and did not require therapy.
In April 2018, the reported negative symptoms were followed
by the standard treatment of sports injuries that included: 14 days
of limited physical activity, NSAIDs (ibuprofen 200 mg twice/day),
cryotherapy and joint taping. Examination revealed mobile joints,
regular in shape without any swelling. Regional lymph nodes were
of normal size and body temperature was normal.
Within a three-week period following the initial onset of pain in
the ankle joint, pain was reported in the knee joint, lumbar and
thoracic spine. Calf muscle hypotrophy also occurred and markers
of inflammation significantly increased (ESR 60 mm/1st hr, CRP
50 mg/dl). After a thorough examination, AS was diagnosed based
on the clinical picture, lesion location, blood tests (ESR, CRP) and
positive HLA-B27 as well as the MRI findings (Fig. 1). Proton den-
sity weighted-MRI (fat saturation) in adjacent sections of the calca-
neus and scaphoid bone with surrounding edema including the
region of the subtalar sinus and also showing synovitis in the ankle
joint. MRI showed changes of the left sacroiliac joint while there
were no signs on the right side (Fig. 2). X-ray of the thoracic and
lumbar spine, the hip and ankle joints were without any significant
features. He fulfilled the classification criteria for axSpA [1]. There
were no diagnostic challenges.
The BASDAI [11] was assessed and initial score was 5.9 denoting
activity. The rehabilitation program was implemented for
16 months during which he returned to the general group training
in August 2019 and was followed-up till May 2022.
Initially, for the first 6 months of therapy, the clinical picture
was stable, however during the 10th month, a regression in symp-
toms occurred. This prompted a change in therapeutic methods
that resulted in a positive progression. The patient has been stable
for more than two years (up to May 2022), continued to play pro-
fessional soccer and has completed 20 competitive matches of 70–
90 min each. The only treatment has been 50 mg golimumab once/
month. Outlines of therapeutic interventions are presented in
Table 1. Therapy included complete rest (no training), celecoxib
400 mg/day consumption, intra-articular injections of betametha-
sone into the inflamed joints of the foot (0.5 ml of
betamethasone + 0.5 ml of lidocaine once/week for two weeks),
ankle immobilization (orthotic splint-Bauerfeind
Ò
), cryotherapy
(20 min. twice/day), magnetotherapy (permanent magnetic field
up to 300 min./ day).
After six months of therapy, the severity of clinical symptoms
began to decrease and the patient’s condition stabilized. However,
by the 10th month of therapy, the condition worsened, which led
to a change in therapy and thus an improvement in the clinical pic-
ture. Secukinumab was initiated for its known effective and safe
use in axSpA [14,15].
Therapy in January 2019 included methotrexate (MTX) 20 mg/
week followed next day by folic acid 10 mg/week, and secuk-
inumab 150 mg subcutaneously (SC) once/month. Additionally,
the athlete received calcium (500 mg/day), vitamin D (5000 IU/day)
and alendronate (10 mg/week). Additionally, muscle strengthen-
ing, massage, and physiotherapy (magnetotherapy, electrical stim-
ulation, cryotherapy) were integrated. Consequently, the BASDAI
decreased. Blood parameters and acute phase reactant started to
Fig. 2. Magnetic resonance imaging (MRI) of the sacroiliac joints on 2019. Signs of sacroiliitis on the left side (taken in a circle). No sign of sacroiliitis on the right side.
E. Bezuglov, M. Shoshorina, M. Butovskiy et al. The Egyptian Rheumatologist 45 (2023) 267–272
269

be normalized. Since March 2019, secukinumab was replaced by
golimumab for its availability and in view of the suggested evi-
dence supporting its value in AS [16–18].
Over the following four months, therapy did not change, and
physical activity progressed while the condition remained stable.
No extra-articular lesions were observed during the entire dura-
Table 1
The characteristics and progress of the patient over the entire period of treatment.
Date Clinical picture Physical loading Blood Treatment Dose
Initially
(1–2 mo)
Apr-May 2018
Pronounced;
BASDAI
(5.9)
Rest ESR: 20
CRP: 20
Iron: 5
Hb: 11
Ferritin: 35
Celecoxib
Sulfasalazine
Betamethasone
400 mg/d
0.5–3 g/d/w/2 wks
IA (ankle/foot joints)
Immobilization
Cryotherapy
Magnetotherapy
Orthosis (Bauerfeind
Ò
)
20 min. twice/d
Up to 300 min/d
(3–6 mo) June-Sept
2018
Pronounced;
BASDAI
(5.2)
Light ex.
No football specific ex.
ESR: 60
CRP: 50
Iron: 3
Hb: 10.5
Ferritin: 30
Sulfasalazine
Celecoxib
Doxycycline Cryotherapy
Magnetotherapy
2 g/d
400 mg/d
200 mg/d
20 min twice /d
Up to 300 min/d
(7–9 mo) Oct-Dec 2018 Stable;
BASDAI
(4)
Training in the
gym and on the field
ESR: 15
CRP: 11
Iron: 15
Hb: 11.8
Ferritin: 38
Sulfasalazine Ibuprofen
Secukinumab
Cryotherapy
Magnetotherapy
500 mg/d
400 mg
150 mg SC 1–4/mo
20-min. twice/d
Up to 300 min/d
(10 mo)
Jan
2019
Pronounced;
BASDAI
(5.5)
BME (foot/ pubic bones)
Exercise
therapy
ESR: 60
CRP: 50
Iron: 3
Hb: 10
Ferritin: 30
Secukinumab
Cryotherapy
US therapy with
hydrocortisone
Magnetotherapy
150 mg SC 1/mo
20 min twice /d
20-min. once/d
Up to 300 min/d
(11–16 mo)
Feb-July 2019
Stable;
BASDAI
(3.6/2.9/
2.4/2.1)
From 16 mo
gp training
ms exercise
ESR: 6
CRP: 0.5
Iron: 17
Hb: 13.8
Ferritin: 15
Methotrexate
Golimumab
Folicacid
Gradual "till 20 mg/w
50 mg SC /mo
10 mg 12 h after MTX
(26 mo)
June
2020
Stable;
BASDAI
(1.5)
Regular training activities ESR: 6
CRP: 0.8
Iron: 17
Hb: 14.8
Ferritin: 40
Golimumab 50 mg SC /mo
(49 mo)
May
2022
Stable;
BASDAI
(1.5)
Regular training activities ESR: 5
CRP: 0.9
Iron:18
Hb: 14.6
Ferritin: 39
Golimumab 50 mg SC /mo
Side effects and allergic reactions were not recorded. ESR: erythrocyte sedimentation rate (mm/1st hr), CRP: C-reactive protein (mg/dl), Hb: hemoglobin (g/dl), Iron in umol/l,
Ferritin in ng/ml, BASDAI: Bath ankylosing spondylitis disease activity index, BME: bone marrow edema.
Fig. 3. The Bath ankylosing spondylitis disease activity index (BASDAI) during the treatment period. Activity is considered high when BASDAI is > 4.
E. Bezuglov, M. Shoshorina, M. Butovskiy et al. The Egyptian Rheumatologist 45 (2023) 267–272
270

tion. The patient returned to regular training activities in early
August 2019 performing in the first professional match on 19th
August 2019. Since June 2020, only golimumab 50 mg/month
was received under supervision of hospital medical staff after pre-
liminary blood tests.
During the entire treatment period, the patient did not experi-
ence any side effects and attended all training sessions. The patient
did not incur any injuries or illnesses that prevented match-play,
and no restrictions for participation in competitive matches were
recommended. During the following two year period, ESR and
CRP levels did not exceed 5 and 1.5, respectively, and the BASDAI
index was 1.5 (Fig. 3).
3. Discussion
The most common outcome for athletes diagnosed with AS is a
decrease in locomotor activity associated with inflammatory joint
damage. The course of the disease can be different, but commonly
joint mobility is irreversibly reduced and the diagnosis of AS makes
returning to sports participation impossible due to chronic inflam-
mation [19], a decrease in bone mineral density and an increased
risk of fractures [9], and a reduction of core stability and balance
[20]. Notably, only one publication describes the case of AS in an
athlete which described the successful treatment with a monthly
course of indomethacin. However, following the cessation of clini-
cal symptoms, the athlete was no longer involved in sports [21].
This is one of the few successful cases reported in the literature
of athletes returning to sport after rheumatic diseases [22–25]
(Table 2). As in the current case, diagnosis was not immediate. This
is important for professionals to detect the disease early and start
treatment to avoid career losses. However, in the studied patient,
inflammation did not occur even with a maximum training load,
arguably achieved by the administration of MTX and golimumab.
On-going therapy allows the continuation of a professional football
career. However, constant monitoring of inflammation markers is
necessary for timely correction of therapy. Provided the expensive
cost, it is essential that the medication is administered by an expe-
rienced medical practitioner.
This case study confirms the previously described efficacy of
golimumab in the treatment of AS. It was shown that monotherapy
with medium doses of this drug allows not only a return to the
usual daily activities but also to continue participation in compet-
itive games.
There are cases of athletes returning to sports with rheumatoid
arthritis (tennis player Caroline Wozniacki, a professional hockey
player from Finland) but they are not described in the existing
literature.
In conclusion, the case presented herein describes the first clin-
ical case of a young professional soccer player with AS returning to
his professional activity. Further monitoring is warranted and the
creation of an athlete database with similar diseases in order to
standardize patient treatment protocols with initially very high
levels of physical activity is essential.
Funding
This research did not receive any specific grant from funding
agencies in the public, commercial, or not-for-profit sectors.
Declaration of Competing Interest
The authors declare that they have no known competing finan-
cial interests or personal relationships that could have appeared
to influence the work reported in this paper.
Table 2
Athletes with rheumatic diseases successfully recovered and resumed sports.
Diagnosis Russia
this case
USA
[22]
Italy
[23]
USA
[24]
USA
[25]
RD AS PsA BD AS AS
Age (y) 19 39 29 32 young
Gender Male Male Male Male Male
Sport Soccer Football Athlete Military team Athlete
Family hx ve +ve – ve –
Features Arthralgia
Rt ankle joint
no inflammation.
Arthritis
Rt knee joint
(effusion).
ROU, skin rash,
EN, DVT, fever,
epididymitis, uveitis.
Radiating pain/LOM Rt
shoulder
Cx/hip pain
Sacroiliac joint
instability and
dysfunction
Prior injury ve +ve – ve –
Start therapy => Effect ;activity, NSAIDs, cryotherapy,
joint taping => stable
NSAIDs
=> improved
IA steroid => relief
AZA,
Prednisone
Exercise, heat, PT,
pain modalities
=> ;Cx/hip pain
NSAIDs,
gradual activity
=> no pain
After initial therapy Knee/lumbo-dorsal pain,
calf ms hypertrophy,
"ESR/ CRP, +ve HLA-B27
Large effusion
Synovitis, LOM
chondromalacia patellae
ROU, skin rash,
macular edema (ME),
;visual acuity (VA)
+ve HLA-B51
LOM shoulders /spine,
ms weakness,
"CRP, +ve HLA-B27
–
Continued treatment
=> Effect
Rest, celecoxib,
IA steroid foot,
cryo/magnetotherapy
=> Improvement
Synovitis resected
cartilage debrided
Steroid, CsA,
AZA => ;ME,
infliximab => ;skin lesions,
;ME, "VA
Cx stretches, ms ex,
ROM shoulder/spine,
NSAIDs, fluticasone =>
Improved
After continued therapy Regressing in
10th month
knee effusion, Ps,
"ESR, +ve ANA,
-ve RF/HLA-B27
Infliximab, AZA
=> remission
New therapy SKB, MTX, PT
=> improved, ;ESR/CRP.
then Golimumab.
Etanercept, MTX => remission
After arthroscopy => re-injury
Return to play PT 16 mo
Golimumab
without relapse after 2 years – Symptom free
gentle work
AS: ankylosing spondylitis, PsA: psoriatic arthritis, BD: Behҫets disease, ROU: recurrent oral ulcers, EN: erythema nodosum, DVT: deep venous thrombosis, LOM: limited range
of motion, NSAIDs: non steroidal anti-inflammatory drugs, IA: intra-articular, Ps: psoriasis, ESR: erythrocyte sedimentation rate, ANA: antinuclear antibody, RF: rheumatoid
factor, ME: macular edema, VA: visual acuity, Cx: cervical, CsA: cyclosporine A, AZA: azathioprine, SKB: secukinomab, MTX: methotrexate, ms ex: muscle exercise, PT:
physiotherapy.
E. Bezuglov, M. Shoshorina, M. Butovskiy et al. The Egyptian Rheumatologist 45 (2023) 267–272
271

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