Case report

Pyomyositis and muscle due to Salmonella enteritidis in a patient with Behçet’s syndrome: a case report G. Kenar1, A. Köken Avşar1, A. Balcı2, G. Can1

1Department of Internal Medicine, ABSTRACT the patient was evaluated, he appeared Division of Rheumatology, 2Department of Salmonella usually present painful, with a blood pressure of Radiology, Dokuz Eylul University School with gastrointestinal manifestations 150/100 mm/Hg, a pulse rate of 100/ of Medicine, İzmir, Turkey. including enterocolitis especially in minute and a low-grade fever of 37.8 C Gökçe Kenar, MD immunocompromised patients. Haema- degree. He stated that he had difficulty Aydan Köken Avşar, MD togenous dissemination and in getting up from a sitting position, Ali Balcı, MD Gerçek Can, MD are very rare complications of Salmo- and the increased while walk- nella species. This case report docu- Please address correspondence to: ing. Diameter measurements of both Gökçe Kenar, MD ments a patient with Behçet’s syndrome lower extremities were similar. There Department of Internal Medicine, (BS) who has pyomyositis due to Sal- was a decrease in muscle strength in Division of Rheumatology, monella species. A 43-year-old male the proximal lower extremities. New- Dokuz Eylul University School of patient with BS presented to the out- onset erythema nodosum lesions were Medicine, İnciraltı, 35340, İzmir, Turkey. patient rheumatology clinic with bilat- observed on the anterior face of the pa- E-mail: [email protected] eral acute-onset lower extremity pain. tients’ tibia. Received on June 2, 2020; accepted in However, over a short time the pain In his previous medical history, he revised form on October 8, 2020. gradually increased and was accompa- had been diagnosed with BS in 2007 Clin Exp Rheumatol 2020; 38 (Suppl. 127): nied by fever. The magnetic resonance with posterior uveitis, recurrent oral S00-S00. scans demonstrated pyomyositis and aphthous ulcers, arthritis, erythema © Copyright Clinical and muscle abscess in the adductor and ob- nodosum, and positive pathergy test. Experimental Rheumatology 2020. turator muscles. The cultures showed Immunosuppressive treatment was ad- Salmonella enteritidis . The ministered for the first five years of the Key words: Behçet’s syndrome, patient was successfully treated with disease with azathioprine (AZA) and pyomyositis, abscess, Salmonella therapy. corticosteroids (CS), then the patient infections, immunosupression This case is important since it is one reported that he had been diagnosed of the first in the literature to report an with pulmonary tuberculosis (tbc). He adult patient with BS and Salmonella discontinued the immunosuppressive pyomyositis. treatment and was treated with anti- tuberculosis agents for a year. After remission of the tbc, he did not present Introduction at the hospital for four years. Then he Pyomyositis is a very rare infection had a new attack of posterior uveitis that arises after transient bacteremia, for which AZA, CSs and cyclosporine most often affecting the muscle groups treatments were started. It was learned of the pelvic girdle and lower extremi- that CS had been added at the dose of 1 ties (1). Pyomyositis due to Salmonella mg/kg/day two months ago due to a re- species has been reported in patients lapse of posterior uveitis. The dose was with diabetes mellitus (DM), cancer, gradually decreased, but he was still sickle cell anaemia, and human de- receiving 20 mg/day. He had no history ficiency virus (HIV) infections (2). of anatomic and functional asplenia Herein, we report on a patient with Be- and hyposplenism such as splenec- hçet’s syndrome (BS) who has pyomy- tomy, congenital asplenia, infarction, ositis due to Salmonella species. infiltration, sickle cell anaemia, thalas- semias, chronic liver diseases, HIV or Case report malignancies. A 43-year-old male presented at the The preliminary diagnoses were: psoas outpatient rheumatology clinic with abscess, muscle or soft tissue abscess Competing interests: none declared. acute-onset pain in both thighs. When due to tbc, infectious hip arthritis and

Clinical and Experimental Rheumatology 2020 1 Pyomyositis due to Salmonella in Behçet’s syndrome / G. Kenar et al.

Table I. The laboratory assessments of the patient. No microorganisms were isolated in the tissue cultures. Tbc polymerase Laboratory Parameter Results chain reaction (PCR) test was negative Leu/Neu/Lym 13.6/11.9/1.3 (103/µL) and no signs were observed for inflam- Hb/Plt 12.2 (gr/dL) / 221 (103/µL) matory in the pathological AST/ ALT 30/31 (U/L) sampling. However, S. enteritidis was CK 91 (U/L) Creatinine 0.55 (mg/dL) isolated in the blood cultures. ESR 104 (mm/h) The patient was treated with ceftriax- CRP 243 (mg/L) one 2000 mg/day for 8 weeks with cip- Procalcitonin 0.89 (ng/mL) rofloxacin 1000 mg/day for 6 weeks. Urine analysis Prt: neg, er: 1, dens: 1032, leu:1 Serum albumin 2.74 (mg/dL) Drainage of the abscess was not found Serum Na/K 132 / 4.5 (mmol/L) to be appropriate because of the dif- Brucella Rose Bengal and serum agglutination tests Negative ficult location of the abscess which Blood culture Growth of Salmonella enteritidis would mean very-deep invasion into Leu: leucocyte; Neu: neutrophil; Lym: lymphocyte; Hb: haemoglobin; Plt: platelet; AST: aspartate the muscle fibres. A clinic and labora- aminotransferase; ALT: alanine aminotransferase; CK: creatin kinase; ESR: erythrocyte sedimentation tory response were observed within the rate; CRP: serum C-reactive protein; Na: sodium; K: potassium; mg: milligram; prt: protein; dens: first week and remission was observed density; leu: leucocyte; er: erythrocyte. in MRI within the first month.

Discussion Pyomyositis might have serious com- plications such as diffuse muscle infec- tions, abscess formation, myonecrosis, , invasion to adjacent tissues and joints, and sepsis. S. aureus is the most common factor (more than 75%), while S. enteritidis in a very small proportion of cases (nearly 2%) (1). Infections caused by Salmonella are generally derived from the gastrointes- tinal system and generally self-limited. Bacteremia may be seen in 5-45% of the patients in the course of the disease (3). In a literature review including patients with Salmonella pyomyositis, S. enteritidis was found to be respon- sible for 50% of cases (2). Patients of- Fig. 1. The magnetic resonance imaging assessments of the patient. ten have diarrhoea as a primary infec- The contrast enhanced, SPIR and T1 weighted, transverse plane MR images showing fluid collection tion. However, in this case, the patient and edema in both adductor and obturator muscle groups, pyomyositis in thigh muscles, air view due presented with pain in the thighs and to abscess in muscles. fever, without any evidence of gastro- enteritis. inflammatory hip arthritis, inflamma- Magnetic resonance imaging (MRI) In previous cases it was observed that tory myositis or acute venous throm- was performed on the hips, lumbar re- Salmonella pyomyositis was reported bosis due to BS. Fever was observed gion, and proximal parts of the lower in elderly patients (4), children (5), pa- over 38.5°C during close monitoring. extremities, revealing fluid collection tients with DM (6), acquired immune AZA and cyclosporine were discon- and oedema in the adductor and ob- deficiency syndrome (AIDS) (7), can- tinued; only CS was continued at a turator muscle groups. Imaging of the cer (8), and sickle cell anaemia (9). lower dose. Blood culture tests were pyomyositis in the thigh muscles re- Pyomyositis due to Salmonella has made. The laboratory data revealed vealed a 2 cm-sized abscess and my- rarely been reported in immunocom- high serum C-reactive protein (CRP), onecrosis in these muscle groups (Fig. petent individuals (10). In previous normal creatinin kinase (CK) and high 1-2). There was also femoral avascular reports there was one case of systemic procalcitonin (Table I). In the Doppler necrosis bilaterally. lupus erythematosus (SLE) under CS ultrasonography of the lower extremi- Muscle tissue cultures and biopsy were treatment with Salmonella pyomyositis ties there was no finding for venous performed from the right thigh muscle (11). This case is first in the literature thrombosis. abscess with a local incisional . to report a BS patient with Salmonella

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strain producing CTX-M-15 extended-spec- trum beta-lactamase. J Clin Microbiol 2015; 53: 1439-41. 5. MINAMI K, SAKIYAMA M, SUZUKI H, YOSHI- KAWA N: Pyomyositis of the vastus medialis muscle associated with Salmonella enteritid- is in a child. Pediatr Radiol 2003; 33: 492-4. 6. JORRING S, KOLMOS HJ, KLARESKOV B: Myonecrosis in the leg caused by Salmonella enteritidis. Scand J Infect Dis 1994; 26: 619- 21. 7. MEDINA F, FUENTES M, JARA LJ, BARILE L, MIRANDA JM, FRAGA A: Salmonella pyomy- ositis in patients with the human immuno- deficiency virus. Br J Rheumatol 1995; 34: 568-71. 8. PUTCHAROEN O, SUANKRATAY C: Salmo- Fig. 2. The magnetic resonance imaging assessments of the patient. nella gas-forming femoral osteomyelitis and The contrast enhanced, SPIR and T1 weighted, coronal plane MR images showing fluid collection and pyomyositis: the first case and review of the edema in both adductor and obturator muscle groups, pyomyositis in thigh muscles, air view due to literature. J Med Assoc Thai 2007; 90: 1943-7. abscess in muscles. 9. WONG VK, LISSACK ME, TURMEZEI TD, MAITLAND JA: Salmonella pyomyositis complicating sickle cell anemia: a case re- pyomyositis. rates (30%) compared to other micro- port. J Med Case Rep 2010; 4: 198. MRI is recommended as the first choice organisms (17). Because the bactere- 10. CHAN O, NAWAZ SZ, HUGHES S, SKINNER in diagnosis. In this case, besides the mia is generally transient, it might not JA: Extensive pyogenic myositis of the hip in an immuno-competent patient. JRSM Short muscle oedema created by the infec- be possible to monitor the microorgan- Rep 2011; 2: 90. tion, abscess formation and air den- isms in blood cultures, and also some- 11. JIDPUGDEEBODIN S: Salmonella Crepitant sity areas due to gas formation within times from the tissue cultures. Pyomyositis in a Patient with Systemic Lu- muscle tissues were observed in the In summary, this case is important pus Erythematosus. J Infect Dis Antimicrob 2004; 21(1). MRI. Gas accumulation has also been since it is the first to report an adult BS 12. HATEMI G, SEYAHI E, FRESKO I, TALARICO previously reported in case reports of patient with Salmonella pyomyositis. R, HAMURYUDAN V: One year in review pyomyositis due to Salmonella strains These reports of cases have consider- 2018: Behçet’s syndrome. Clin Exp Rheuma- tol 2018; 36 (Suppl. 115): S13-27. (6, 8, 11). able importance for guiding clinicians 13. HATEMI G, SEYAHI E, FRESKO I, TALARICO The preliminary diagnosis of this case as rare microorganisms might cause R, HAMURYUDAN V: One year in review included inflammatory myositis. The rare clinic manifestations in patients 2019: Behçet’s syndrome. Clin Exp Rheuma- severe posterior uveitis, new-onset who have diseases and treatments tol 2019; 37 (Suppl. 121): S3-17. 14. SARUI H, MARUYAMA T, ITO I et al.: Necro- erythema nodosum lesions might also causing immunosuppression. tising myositis in Behçet’s disease: charac- be considered as clues that this patient teristic features on magnetic resonance imag- is in the active period of BS (12, 13). References ing and a review of the literature. Ann Rheum The rare findings of BS includes local- 1. BICKELS J, BEN-SIRA L, KESSLER A, WIEN- Dis 2002; 61: 751-2. TROUB S: Primary pyomyositis. J Bone Joint 15. CONWAY R, BERGIN D, CAREY JJ, ised or diffuse inflammatory myositis Surg Am 2002; 84: 2277-86. COUGHLAN RJ: A case of Behçet’s syndrome (14-16). However, having high fever, 2. COLLAZOS J, MAYO J, MARTINEZ E, BLAN- presenting as focal myositis. Rheumatology elevated procalcitonin, the detection of CO MS: Muscle infections caused by Salmo- (Oxford) 2012; 51: 975. abscess and gas formation in the MRI, nella species: case report and review. Clin 16. YAZICI H, TUZUNER N, TUZUN Y, YURDAKUL Infect Dis 1999; 29: 673-7. S: Localized myositis in Behçet’s disease. the absence of inflammatory myositis 3. AKKOYUNLU Y, CEYLAN B, IRAZ M, EL- Arthritis Rheum 1981; 24: 636. in the pathology samples and normal MADAG NM, ASLAN T: Muscle Abscess due 17. COLLAZOS J, MAYO J, MARTINEZ E, BLAN- CK excluded that patient had Behçet’s to . Iran Red Crescent CO MS: Comparison of the clinical and labo- myositis. Med J 2013; 15: 605-7. ratory features of muscle infections caused 4. HWANG JH, SHIN GW, LEE CS: Community- by Salmonella and those caused by other Pyomyositis due to Salmonella have onset pyomyositis caused by a Salmonella pathogens. J Infect Chemother 2001; 7: 169- been reported to have higher mortality enterica serovar enteritidis sequence type 11 74.

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