97
CASE REPORT
Aseptic Abscess Syndrome in a Patient with
Rheumatoid Arthritis: A Case Report
Miguel González-Velásquez1 , Carlos Jaime Velásquez-Franco2 , Santiago Bernal-
Macías3 , Johann Gutiérrez-Bolaños4
1 Internal Medicine Resident, Universidad Pontificia Bolivariana, Medellin, Colombia.
2 Professor of Internal Medicine and Rheumatology, Universidad Pontificia Bolivariana, Medellin, Colombia.
3 Rheumatologist, San Ignacio University Hospital, Bogotá, Colombia.
4 Internal Medicine Physician, Clinica Universitaria Bolivariana, Medellin, Colombia.
ARTICLE INFORMATION
Keywords
Arthritis, Rheumatoid;
Abscess, Fever,
Hereditary Autoinflammatory Diseases
Received: April 21, 2023
Accepted: June 8, 2023
Correspondence:
Miguel González-Velásquez;
miguelgonv41@gmail.com
How to cite: González-Velásquez M, Velás-
quez-Franco CJ, Bernal-Macías S, Gutiérrez-Bo-
laños J. Aseptic Abscess Syndrome in a Patient
with Rheumatoid Arthritis: Case Report. Iatreia
[Internet]. 2024 Jan-Mar;37(1):97-105.
https://doi.org/10.17533/udea.iatreia.231
Copyright: © 2023
Universidad de Antioquia.
ABSTRACT
Aseptic abscess syndrome is a rare and possibly underdiagnosed condi-
tion characterized by the presence of sterile neutrophilic collections in
various tissues that do not respond to antimicrobial therapy but rather
require anti-inflammatory treatment. There is limited literature linking this
condition to rheumatoid arthritis. Here, we present the case of a patient
with rheumatoid arthritis, presenting with prolonged febrile syndrome
and multiple sterile abscesses. A diagnosis of aseptic abscess syndrome
was made, and successful resolution of the abscesses was achieved fo-
llowing treatment with glucocorticoid pulses and infliximab. Clinicians
should consider this condition in patients with inflammatory diseases
who develop abscesses in different tissues where no causative microbio-
logical agent can be identified.
IATREIA. Vol. 37 Núm. 1. (2024). DOI 10.17533/udea.iatreia.231
98
REPORTE DE CASO
Síndrome de abscesos asépticos en paciente con
artritis reumatoide: reporte de caso
Miguel González-Velásquez1 , Carlos Jaime Velásquez-Franco2 ,
Santiago Bernal-Macías3 , Johann Gutiérrez-Bolaños4
1 Residente de Medicina Interna, Universidad Pontificia Bolivariana, Medellín, Colombia.
2 Profesor titular de medicina interna y reumatología, Universidad Pontificia Bolivariana, Medellín, Colombia.
3 Reumatólogo, Hospital Universitario San Ignacio, Bogotá, Colombia.
4 Médico internista, Clínica Universitaria Bolivariana, Medellín, Colombia.
INFORMACIÓN ARTÍCULO
Palabras clave
Absceso;
Artritis Reumatoide;
Enfermedades Autoinflamatorias Hereditarias;
Fiebre
Recibido: abril 21 de 2023
Aceptado: junio 8 de 2023
Correspondencia: Miguel González-Velás-
quez; miguelgonv41@gmail.com
Cómo citar: González-Velásquez M, Velás-
quez-Franco CJ, Bernal-Macías S, Gutiérrez-Bo-
laños J. Síndrome de abscesos asépticos en pa-
ciente con artritis reumatoide: reporte de caso.
Iatreia [Internet]. 2024 Ene-Mar;37(1):97-105.
https://doi.org/10.17533/udea.iatreia.231
Copyright: © 2023
Universidad de Antioquia.
RESUMEN
El síndrome de abscesos asépticos es una condición poco frecuente, pro-
bablemente subdiagnosticada, que se caracteriza por la aparición de colec-
ciones neutrofílicas estériles en diferentes tejidos, las cuales no responden
a antimicrobianos y cuyo tratamiento es basado en terapia antiinflamato-
ria. La literatura que relaciona dicha condición con la artritis reumatoide
es escasa. Se presenta el caso de un paciente con artritis reumatoide, con
síndrome febril prolongado y múltiples abscesos estériles en quien se hizo
diagnóstico de síndrome de abscesos asépticos, con resolución de las co-
lecciones luego del tratamiento con pulsos de glucocorticoides e inflixi-
mab. Se debe sospechar esta condición en pacientes con enfermedades
inflamatorias que desarrollan abscesos en diferentes tejidos en los que no
se logra identificar ningún agente microbiológico causal.
IATREIA. Vol. 37 Núm. 1. (2024). DOI 10.17533/udea.iatreia.231
99González et al.
IATREIA. Vol. 37 Núm. 1. (2024). DOI 10.17533/udea.iatreia.231
INTRODUCTION
The Aseptic Abscess Syndrome (AAS) is a rare autoinflammatory condition, primarily associated
with Inflammatory Bowel Disease (IBD). It involves the formation of abscesses in various organs,
without the presence of infectious microorganisms (1). It is distinguished by a lack of response to
antibiotic therapy and the resolution of the abscesses following glucocorticoid treatment. Patients
often undergo prolonged antimicrobial treatments and unnecessary invasive procedures, which
can be avoided with proper diagnosis; hence, it is vital to recognize this condition (2). We report the
case of a patient with Rheumatoid Arthritis (RA) and prolonged febrile syndrome who developed
multiple abscesses in soft tissues and muscles without any microbiological isolation.
CLINICAL CASE
A 56-year-old male with a history of RA (positive rheumatoid factor and anti-citrullinated protein
antibodies, ACPA) and type 2 diabetes mellitus was under treatment with leflunomide 20 mg daily,
methotrexate 15 mg weekly, prednisolone 5 mg/day, insulin glargine 30 IU/day, and metformin 850
mg/day. The patient sought medical attention due to a four-month history of fever and a weight
loss of 20 kg. He had been hospitalized in another facility for one month, until five days prior to ad-
mission to our department. During that hospitalization, a left psoas abscess was documented and
drained twice (purulent material with negative cultures for aerobes, mycobacteria, and fungi). He
was treated with cefepime 1 g IV every 8 hours and metronidazole 500 mg IV every 8 hours for 28
days. He was discharged to continue outpatient antibiotic therapy, but persistent febrile episodes
were noted three weeks post-antibiotic therapy, prompting his return.
Upon admission, his vital signs were stable, but he appeared emaciated with mucocutaneous
pallor. Additionally, he had bilateral axillary lymphadenopathy and inflammatory changes in mul-
tiple joints (right elbow, left knee, both wrists, and metacarpophalangeal joints of the second and
third fingers of the left hand).
Laboratory tests on admission highlighted a leukocyte count of 13,130 cells/mm3 due to neu-
trophils, normocytic and normochromic anemia, C-reactive protein (CRP) of 25 mg/dL, and an
erythrocyte sedimentation rate of 68 mm/hour. Blood cultures, urine cultures, rapid plasma reagin
(RPR), fluorescent treponemal antibody absorption (FTA-ABS), and tests for HIV and hepatitis B and
C viruses were negative. A contrast-enhanced computed tomography (CT) scan of the abdomen
revealed a hypodense image measuring 10.5 x 6.4 mm in the left psoas consistent with a residual
collection. Given the suspicion of an active infectious process, prednisolone was prescribed at 10
mg orally daily, and both methotrexate and leflunomide were discontinued. From the time of ad-
mission, the patient was not on any antimicrobial medications. Figure 1 illustrates the chronological
sequence of significant events during the hospital stay. Figure 2 plots the evolution of CRP levels and
the glucocorticoid doses administered to the patient. Table 1 lists the relevant laboratory findings
and their progression during hospitalization, while Table 2 provides the microbiological studies.
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Figure 1. Significant events during hospitalization
Source: Own elaboration
Figure 2. C-reactive protein (CRP) and glucocorticoid dose (in prednisolone equivalence) during hospital stay.
X-axis: date, Y-axis: C-reactive Protein concentration (mg/dL). Equivalent prednisolone doses: * 10 mg, † 25 mg, ‡ 15 mg, §
20 mg, | 30 mg, ¶ 40 mg, ** 312,5 mg (methylprednisolone pulses: 250 mg IV/day for 3 days), †† 50 mg.
Source: Own elaboration
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Table 1. Laboratory tests during hospitalization
Test/ Date 30/04/21 04/05/21 18/05/21 21/04/21 31/05/21 05/06/21 19/06/21 25/06/21 Reference
value
Hemoglobin
(g/dL) 10,7 8,4 8,6 7,9 9,7 9,8 11,3 13 - 18
Leukocytes
(cells/mm3
) 13130 7720 13740 14290 11980 13310 10240 4500 -
11000
Neutrophils
(cells/mm3
) 10510 4910 10450 10320 8580 11540 8320 2000 -
7500
Platelets
(cells/mm 3
) 458000 410000 584000 591000 552000 482000 438000 140000 –
400000
C-reactive
protein (mg/dL) 25 15.56 13.18 9.3 7.5 3.63 2.31 1.02 0 - 5
Sedimentation
rate (mm/hour) 53 68 48 0 - 20
Ferritin (ng/mL) 9005 747.1 699.1 30 - 400
Source: Own elaboration
Table 2. Microbiological studies
Procedure (date) Sample Test Result
Blood culture (30/04/21) Blood Bacterial culture Negative
Urine culture (30/04/21) Urine Bacterial culture Negative
Arthrocentesis of left knee
(07/05/21)
Synovial fluid Gram Without bacteria
Aerobic culture Negative
Molecular test for M. tuberculosis Non detectable
M. tuberculosis culture Negative
Biopsy of right axillar lymphatic
node (11/05/21)
Lymph node Gram Without bacteria
KOH Without fungi
Bacilloscopy Negative
Molecular test for M. tuberculosis Non detectable
Aerobic culture Negative
Fungal culture Negative
M. tuberculosis culture Negative
Percutaneous drainage of right
axillary collection (15/05/21)
Secretion Gram Without bacteria
KOH Without fungi
Bacilloscopy Negative
Molecular test for M. tuberculosis Non detectable
Aerobic culture Negative
Fungal culture Negative
Percutaneous drainage of right
axillary collection (21/05/21)
Secretion Gram Without bacteria
Aerobic culture Negative
Percutaneous drainage of right
axillary collection (08/06/21)
Secretion Gram Without bacteria
KOH Without fungi
Bacilloscopy Negative
Molecular test for M. tuberculosis No detectable
Aerobic culture Negative
Fungal culture Negative
M. tuberculosis culture Negative
Source: Own elaboration
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The patient’s fever remained, furthermore, the inflammation of the left knee worsened, so an
arthrocentesis was performed, from which a cloudy, non-viscous fluid was obtained with 17,760
leukocytes (91% neutrophils). Microbiological studies were conducted on the fluid, yielding negati-
ve results. An excisional biopsy of the axillary lymph nodes was performed and sent for histopatho-
logical and microbiological examinations, both of which returned negative. Among the differential
diagnoses, there was a consideration that the patient might have a lymphoproliferative syndrome.
However, this was ruled out with histopathological and immunohistochemical studies of the axi-
llary lymph nodes and flow cytometry.
The patient developed inflammatory changes on the right dorsal side, leading to a CT scan of
the chest. This revealed a 75 x 75 mm collection in the right axilla and a 22 mm intramuscular collec-
tion behind the left scapula (Figure 3). The axillary collection was percutaneously drained, yielding
25 mL of seropurulent material. Various microbiological tests were conducted on this material, in-
cluding Gram stain, KOH, bacilloscopy, aerobic culture, fungal culture, and the GeneXpert molecular
test for Mycobacterium tuberculosis. All these tests were negative. Subsequently, the patient showed
worsening synovitis of the wrist, flexor tenosynovitis in both wrists, and persistent knee arthritis des-
pite adjustments made to the glucocorticoid doses. Because the patient presented with additive
polyarthritis, the following differential diagnoses were considered: infectious endocarditis, reactive
arthritis due to Chlamydia trachomatis, and disseminated gonococcal infection. A transesophageal
echocardiogram and a polymerase chain reaction for sexually transmitted pathogens were perfor-
med, ruling out the aforementioned diagnoses. The glucocorticoid dose was increased, and the
CRP decreased by approximately 50% (Figure 2). The patient developed progressive swelling of the
right upper limb, prompting an angio-CT scan. The scan showed the persistent collections in the
right axilla in close proximity to vascular structures but without compression and no associated
thrombosis. The patient underwent another percutaneous drainage of the collections, yielding 65
mL of citrine fluid with negative microbiological tests. During the drainage, a collection was obser-
ved anterior to the distal humerus with an apparent periosteal involvement, prompting a magnetic
resonance imaging (MRI) to rule out osteomyelitis. After the second drainage and adjustment of the
glucocorticoid dose, the fever subsided.
Figure 3. Contrast-enhanced chest CT scan.
Right axillary collection measuring 75 x 75 mm, with internal bubbles and air extending to the soft tissues of the anterior chest
wall (white arrow). Left intramuscular collection posterior to the scapula with internal air measuring 22 mm (red arrow).
Source: Own elaboration
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The magnetic resonance imaging (MRI) of the right upper limb revealed an osteitis zone in
the distal humerus without osteomyelitis along with multiple collections in the rotator cuff mus-
cles and the known axillary collection. The orthopedics department did not consider performing
surgical drainage; instead, they requested percutaneous drainage of the axillary collection. Having
reasonably ruled out both infectious and lymphoproliferative causes, combined with the presence
of recurrent abscesses, AAS was considered. As a result, pulses of methylprednisolone were ordered
(250 mg IV daily for 3 days due to hypoalbuminemia), and treatment with prednisolone continued
at 1 mg/kg/day. On 06/08/21, the last percutaneous drainage of the right axillary collection was per-
formed with negative microbiological studies. From 06/10/21, improvement in arthralgias and ar-
thritis began to be noticed, the swelling of the RUL decreased, and the acute phase reactants (APR)
normalized. A total body MRI was performed to rule out hidden abscesses, none were found, and a
colonoscopy, which did not show findings suggestive of IBD, was performed. Due to the presence
of active rheumatoid arthritis, despite receiving leflunomide, methotrexate, and glucocorticoids,
and the diagnosis of AAS, treatment with infliximab was initiated at a dose of 300 mg IV, with good
tolerance by the patient and without adverse effects.
After this, the swelling of the RUL completely improved, and the joint involvement significantly
decreased. The total body MRI showed synovitis in the shoulders, wrists, and hips; but these did not
present abscesses or collections, so it was decided to discharge the patient from the hospital with
management using corticosteroids and infliximab.
DISCUSSION
AAS is characterized by neutrophilic and sterile infiltration with a granulomatous reaction in diffe-
rent organs (1–3). Less than 100 cases have been reported in the literature (4), mainly in Europe,
although there are reports in Asia and America (1,2,5). This condition is primarily associated with
Inflammatory Bowel Disease (IBD) (in 66% of cases), but it is also associated with neutrophilic der-
matoses, relapsing polychondritis, monoclonal gammopathy of uncertain significance, and myelo-
dysplastic syndromes (1,5-6). There are two reported cases of AAS associated with rheumatoid ar-
thritis (7-8), one of them with concomitant disease activity (8), which is similar to the presented case.
Patients exhibit fever (which may precede the abscesses), weight loss, diaphoresis, myalgia, and
arthralgias. They may experience arthritis; skin involvement in the form of abscesses or lesions com-
patible with neutrophilic dermatoses (like pyoderma gangrenosum or Sweet’s syndrome), ulcers,
hepatosplenomegaly, or lymphadenopathy. In paraclinical examinations, leukocytosis, and elevated
acute phase reactants (APR) will be found. The spleen is the most affected site, but collections can
affect any tissue (liver, lungs, kidneys, pancreas, testicles, lymph nodes, brain, pharynx, and muscle)
(1,3,9-10). Regarding the symptoms related to this disease, the patient had abscesses in different
muscular planes and soft tissues, which did not resolve despite antibiotic therapy. Also supporting
the diagnosis was the fact that no microbiological isolation was achieved despite multiple studies
of the abscesses, and the patient had a constitutional syndrome with elevated APR. Additionally,
the patient improved after intensifying the immunosuppressive regimen. While the synovitis the
patient had could correspond to RA activity, AAS could have been the trigger for the activity and
contributed to the worsening of symptoms.
AAS should be suspected in patients with deep abscesses, in whom an infectious cause cannot
be demonstrated despite exhaustive studies (1). It is a diagnosis of exclusion, and while there are
no diagnostic criteria, André et al. (1) recommend the inclusion criteria used in the French registry.
To the criteria mentioned, the response to treatments other than glucocorticoids is added for diag-
nosis (Table 3). Due to the association between AAS and IBD, a colonoscopy is recommended as a
screening test (3).
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Table 3. Proposed Diagnostic Criteria for AAS *
Deep Abscesses on Imaging Studies: Predominantly neutrophilic when aspiration or biopsy is performed.
Negative Diagnostic Tests: Negative blood cultures, negative serologies, and, when punctures or biopsies
are carried out, negative microbiological studies.
Therapeutic Failure with Antibiotics: When prescribed for at least 2 weeks for conventional therapy and 3
months for antituberculous therapy.
Rapid Clinical Improvement After Glucocorticoid Prescription: Quick clinical improvement the day after
prescribing glucocorticoids (at least 0.5 mg/kg/day), followed by radiological improvement after one month.
A similar response with colchicine, anti-TNFα, and anti-IL-1 may also be considered.
*All criteria must be met
Source: Adapted form references (1, 11)
The majority of patients respond quickly to glucocorticoids. However, approximately 66% expe-
rience relapses when tapering off (1-2,11); therefore, adjuvant drugs are required for maintenance
(12). Among the drugs used as adjuvants are azathioprine, colchicine, cyclophosphamide, cyclos-
porine, methotrexate, mycophenolate mofetil, and dapsone. However, the optimal maintenance
therapy remains unknown (1,4). In refractory cases or those with frequent relapses, biological thera-
py with anti-TNFα (infliximab, adalimumab, and etanercept), anti-IL-1 (anakinra and canakinumab),
and anti-IL-6 (tocilizumab) has been used, achieving a complete response (4-5,9,11). In patients
receiving azathioprine, adalimumab, or infliximab as maintenance therapy, no relapses have been
reported (12). Splenectomy has been utilized due to the frequent involvement of the spleen, but its
use has become less common nowadays (1,13). Despite frequent relapses, the prognosis is good,
with no reported fatal cases to date (1-2).
CONCLUSIONS
AAS is a diagnosis of exclusion that should be considered in patients presenting abscesses and
where an infectious etiology cannot be demonstrated despite thorough investigation. Even though
it is mostly associated with IBD, it can occur in other inflammatory diseases. To the best of our
knowledge, this is the first reported case of AAS in Colombia in a disease other than IBD.
ETHICAL CONSIDERATIONS
Written informed consent was obtained from the patient for the publication of this case report, and
it was approved by the ethics committee of the institution where the care was provided.
CONFLICT OF INTEREST
The authors declare that they have no conflict of interest.
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