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Rheumatic Manifestations of Bartonella Infection in 2 Children MOHAMMAD J

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Rheumatic Manifestations of Bartonella Infection in 2 Children MOHAMMAD J Case Report Rheumatic Manifestations of Bartonella Infection in 2 Children MOHAMMAD J. AL-MATAR, ROSS E. PETTY, DAVID A. CABRAL, LORI B. TUCKER, BANAFSHI PEYVANDI, JULIE PRENDIVILLE, JACK FORBES, ROBYN CAIRNS, and RALPH ROTHSTEIN ABSTRACT. We describe 2 patients with very unusual rheumatological presentations presumably caused by Bartonella infection: one had myositis of proximal thigh muscles bilaterally, and the other had arthritis and skin nodules. Both patients had very high levels of antibody to Bartonella that decreased in asso- ciation with clinical improvement. Bartonella infection should be considered in the differential diag- nosis of unusual myositis or arthritis in children. (J Rheumatol 2002;29:184–6) Key Indexing Terms: MYOSITIS ARTHRITIS BARTONELLA Infection with Bartonella species has a wide range of mani- was slightly increased at 9.86 IU/l (normal 4.51–9.16), and IgA was 2.1 IU/l festations in children including cat scratch disease (regional (normal 0.2–1.0). C3 was 0.11 g/l (normal 0.77–1.43) and C4 was 0.28 (nor- mal 0.07–0.40). Antinuclear antibodies were present at a titer of 1:40, the anti- granulomatous lymphadenitis), bacillary angiomatosis, streptolysin O titer was 35 (normal < 200), and the anti-DNAase B titer was encephalitis, Parinaud’s oculoglandular syndrome, Trench 1:85 (normal). Urinalysis showed 50–100 erythrocytes and 5–10 leukocytes fever (Vincent’s angina), osteomyelitis, granulomatous per high power field. Routine cultures of urine, blood, and throat were nega- hepatitis, splenitis, pneumonitis, endocarditis, and fever of tive. Liver enzymes, electrolytes, HIV serology, cerebrospinal fluid analysis, unknown origin1-3. To our knowledge, there have been no radiographs of the legs and back, and a bone scan and bone marrow cytology were all normal. Computerized tomography (CT) of the abdomen and chest reports of myositis caused by Bartonella in immunocompe- revealed a small right pleural effusion. A skin biopsy showed intense histio- tent children, and very few reports of arthritis caused by this cytic inflammation. An excisional biopsy of one subcutaneous nodule showed organism in childhood. We describe 2 children in whom proliferative angiomatosis with features suggestive of intermediate behavior. Bartonella infection presented as rheumatological conditions: A lymph node biopsy showed a reactive appearance and the standard cultures one presented with skin nodules and arthritis and the second for bacteria, fungi, and tuberculosis were negative. Because hematuria and hypocomplementemia suggested acute poststrep- presented with bilateral proximal myositis of the lower limbs. tococcal glomerulonephritis, oral penicillin was given for 10 days. He subse- quently developed many more nodules over his trunk, face, fingers, scalp, and CASE REPORTS arms, but became afebrile. During the next month the nodules became small- Case 1. A 26-month-old healthy Caucasian boy was admitted to British er and he had no further skin desquamation. He developed cold induced Columbia’s Children’s Hospital with a 2 week history of low grade fever, Raynaud-like phenomenon in his fingers with pallor followed by erythema. fatigue, and a fine maculopapular truncal rash. On admission his temperature By the third month of his illness C3 and levels of immunoglobulins returned was 38°C and blood pressure 94/50. He had multiple firm, nontender, mobile to normal, and T and B lymphocyte quantification and their responses to mito- well defined cutaneous nodules over his knees, forearms, shins, elbows, and gens were normal. Serologic testing for Bartonella infection by immunofluo- scalp. They varied in size from 0.5 to 2.5 cm and some were erythematous. rescence assay showed an antibody titer of 1:2048 for both B. henselae and B. He had inguinal and antecubital lymphadenopathy, fine desquamation and quintana [serum for testing was forwarded by the Provincial Laboratory to erythema of his palms and soles, and mild hepatosplenomegaly. There was a the Centers for Disease Control (CDC) in Atlanta, Georgia]. Treatment with history of contact with kittens and rabbits. clarithromycin (20 mg/kg/day) was started. On the third day of treatment, he Initial blood tests showed leukocytosis (21 × 109/l) with a predominance became febrile (38.5°C), possibly representing a Jarish-Herxheimer reaction. of neutrophils. The erythrocyte sedimentation rate (ESR) was 47 mm/h, IgG One month after the initiation of antibiotic therapy, he developed acute arthri- tis of the right elbow and first, second, and third metatarsophalangeal joints. At this time he was afebrile, ESR was normal, and the urinalysis showed only From the Divisions of Rheumatology, Dermatology, Infectious and 2 WBC and 3 RBC per high power field. Naproxen (15 mg/kg/day) was Immunological Diseases, Departments of Pediatrics and Radiology, given. He continued to take clarithromycin for 3 months. At this time, 4 University of British Columbia at British Columbia’s Children’s Hospital, Vancouver, British Columbia, Canada. months after the initial testing, titers of antibodies to B. henselae and B. quin- tana fell to 1:512 and 1:128, respectively. The arthritis improved initially, and M. Al-Matar, MBBS, FAAP, Fellow; R. Petty, MD, PhD, FRCPC, Professor; D. Cabral, MBBS, FRCPC, Assistant Professor; L. Tucker naproxen was discontinued; however, it recurred in both elbows. At this time MD, FAAP, Associate Professor; B. Peyvandi, MD, Research Assistant; he was otherwise well except for the persistence of the Raynaud-like phe- J. Prendiville, MB, FRCPC, Associate Professor; J. Forbes, MBChB, nomenon. Radiographs of elbows revealed bilateral effusions and large shal- FRCPC, Associate Professor; R. Cairns, MD, FRCPC, Associate low erosions of the cortical surface of the olecranon (Figure 1). These find- Professor; R. Rothstein, MD, FRCPC, Professor. ings were confirmed on magnetic resonance images (MRI). Joint aspiration Address reprint requests to Dr. D. Cabral, British Columbia’s Children’s and synovial biopsy of the elbow were consistent with a nonspecific inflam- Hospital, 4480 Oak Street, Vancouver, BC V6H 3V4, Canada. matory synovitis, and cultures for Bartonella, tuberculosis, fungi, and bacte- Submitted April 23, 2001; revision accepted July 31, 2001. ria were negative. At this point, naproxen was restarted. There was no clini- Personal non-commercial use only. The Journal of Rheumatology Copyright © 2002. All rights reserved. 184 The Journal of Rheumatology 2002; 29:1 Downloaded on September 27, 2021 from www.jrheum.org Figure 1. Patient 1: lateral radiograph of the right elbow shows a large joint effusion and extensive irregular erosions of the articular surface of the ole- cranon (arrowheads). Figure 2. Patient 2: coronal fast spin echo fat saturation T2 weighted MR of the hips reveals bilateral changes of diffuse increased signal throughout the cal evidence of active arthritis 8 months later, and at last followup (18 months adductor muscles; changes are worse on the right. A small fluid collection after the symptom onset) radiographic findings had improved but not resolved (arrow) is present on the right. completely, and he had diminishing episodes of Raynaud’s phenomenon con- fined to his lower limbs. DISCUSSION Case 2. A previously well 12-year-old girl was hospitalized with a 2 week his- tory of fever, fatigue, and severe left thigh pain worsening with movement. Bartonella henselae infection is most commonly associated There was no history of trauma or viral infection. The medical history and with cat scratch disease, but B. quintana is the other causative family history were noncontributory. She had no history of close contact with organism of bacillary angiomatosis1,3. The former syndrome is cats, but she had an apparently healthy dog at home. A bone scan and hip radi- very commonly associated with a scratch from young cats ograph were normal. The ESR at that time was 90 mm/h. On admission she infected with fleas, but this association is less frequent for was febrile (39°C) and had pain on movement of the left leg, difficulty walk- 1-3 ing, and bilateral weakness of the hip girdle musculature. MRI indicated patients with bacillary angiomatosis . Bacillary angiomato- inflammatory changes involving the adductor muscles bilaterally, more pro- sis occurs primarily in patients with the acquired immune nounced on the right. A small fluid collection (2 cm) was observed in the infe- deficiency syndrome but also in immunocompetent patients1,4. rior right adductor magnus muscle. She was treated for presumed pyomyosi- This syndrome commonly involves the skin, but other organ tis, initially with intravenous cefotaxime and cloxacillin and then oral systems may be involved1,5. cephalexin 500 mg tid for a total of 6 weeks. Naproxen was also given. She had brisk reflexes in the upper and lower extremities; a Gowers’ sign In children, although Bartonella infection is common, was present and her gait was broad based. There was tenderness of the poste- rheumatic manifestations are very rare. Arthritis related to rior thighs bilaterally, worse on the left. Bartonella infection has previously been reported in only 2 Initial laboratory investigations showed a WBC count of 7 × 109/l with a children. Hayem, et al described a girl 2.5 years of age with × normal differential, hemoglobin 106 g/l, platelet count 574 10 g/l, ESR 123 fever, rash, and recurrent brief arthritis of hips, ankles, and mm/h. The following tests gave normal or negative results: HIV antibody, Monospot, bacterial cultures of blood, stool, urine and cerebrospinal fluid knees mimicking systemic onset juvenile rheumatoid arthri- 6 (CSF), CK, urinalysis, CSF analysis, autoantibodies (rheumatoid factor, anti- tis . Carithers described a 17-year-old girl with multiple ery- nuclear antibodies, antineutrophil cytoplasmic antibodies), coagulation stud- thema nodosum lesions and painful ankle swelling that lasted ies including Factor VIII related antigen, immunoglobulin levels, and levels for 3 to 4 days2. It was not clear whether the ankle swelling of hepatic enzymes and angiotensin converting enzyme. Serologic tests represented edema related to the skin lesions or true arthritis.
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