Mahne et al
The Pediatric Infectious Disease Journal • Volume 34, Number 1, January 2015
2. Ferrieri P, Gewitz MH, Gerber MA, et al; Committee on Rheumatic Fever, Endocarditis, and Kawasaki Disease of the American Heart Association Council on Cardiovascular Disease in the Young. Unique features of infective endocarditis in childhood. Circulation. 2002;105:2115–2126. 3. Das M, Badley AD, Cockerill FR, et al. Infective endocarditis caused by HACEK microorganisms. Annu Rev Med. 1997;48:25–33. 4. Kaplan AH, Weber DJ, Oddone EZ, et al. Infection due to Actinobacillus actinomycetemcomitans: 15 cases and review. Rev Infect Dis. 1989;11:46–63. 5. Carlile JR, Beckman EN, Arensman RM. Actinobacillus actinomycetemcomitans pneumonia. Clin Pediatr (Phila). 1984;23:578–580. 6. Chen AC, Liu CC, Yao WJ, et al. Actinobacillus actinomycetemcomitans pneumonia with chest wall and subphrenic abscess. Scand J Infect Dis. 1995;27:289–290. 7. Hagiwara S, Fujimaru T, Ogino A, et al. Lung abscess caused by infection of Actinobacillus actinomycetemcomitans. Pediatr Int. 2009;51:748–751. 8. Nash CB, Hendrickson BA, Alexander KA. Pneumonia with chest wall invasion in a school aged child. J Ped Infect Dis. [published online ahead of print February 16, 2014]. 9. Morris JF, Sewell DL. Necrotizing pneumonia caused by mixed infection with Actinobacillus actinomycetemcomitans and Actinomyces israelii: case report and review. Clin Infect Dis. 1994;18:450–452. 10. Meyer BR, Buttone E, Hirschman SZ, Schneierson SS, Gershengorn K. Infection due to Actinobacillus actinomycetemcomitans. Am J Clin Pathol. 1971;56:204–211. 11. Leys EJ, Griffen AL, Strong SJ, et al. Detection and strain identification of Actinobacillus actinomycetemcomitans by nested PCR. J Clin Microbiol. 1994;32:1288–1294.
PANUVEITIS CAUSED BY BORRELIA BURGDORFERI SENSU LATO INFECTION Jasna Mahne, MD*, Branka Stirn Kranjc, MD, PhD†, Franc Strle, MD, PhD*, Eva Ružić-Sabljić, MD, PhD‡, and Maja Arnež, MD, PhD* Abstract: A 13-year-old boy who presented with a red left eye, painful eye movement, blurred vision, photophobia and increased lacrimation, was diagnosed with 1-sided panuveitis with optic disk edema. Diagnostic workup revealed borrelial antibodies in serum. Diagnosis of Lyme borreliosis was substantiated by demonstration of lymphocytic pleocytosis, intrathecal borrelial antibody synthesis, improvement after treatment with ceftriaxone and exclusion of other causes. Key Words: Panuveitis, Lyme neuroborreliosis, Borrelia burgdorferi sensu lato, pediatric, tick bite Accepted for publication June 25, 2014. From the *Department of Infectious Diseases, University Medical Centre Ljubljana, Japljeva 2, 1525 Ljubljana; †Department of Ophthalmology, University Medical Centre Ljubljana, Grablovičeva 46; and ‡Institute of Microbiology and Immunology, School of Medicine, Zaloška 4, 1000 Ljubljana, Slovenia. Franc Strle’s institution is receiving a grant from the Slovenian Research Agency (No P3-0296). For the remaining authors none were declared. Address for correspondence: Jasna Mahne, MD, Department of Infectious Diseases, University Medical Centre Ljubljana, Japljeva 2, 1525 Ljubljana, Slovenia. E-mail: [email protected]. Copyright © 2014 by Lippincott Williams & Wilkins DOI: 10.1097/INF.0000000000000473
L
yme borreliosis (LB) is a multisystem infectious disease caused by Borrelia burgdorferi sensu lato and transmitted to humans by tick bite. It is endemic in several regions of North America, Asia and Europe, including Slovenia. In European patients, the most common clinical manifestation of the disease is erythema migrans (EM), followed by Lyme neuroborreliosis (LNB).1,2 Data on ocular manifestations of LB are scarce and predominantly restricted to adults. Eyes can be affected primarily as a result of inflammation of eye tissue or secondarily due
102 | www.pidj.com
to extraocular manifestations of LB such as paresis of cranial nerves and orbital myositis.3 We report a case of acute, 1-sided panuveitis with optic disc edema in a child in whom diagnostic work-up revealed LNB.
CASE REPORT On April 13, 2011, a 13-year-old boy was referred to the University Medical Centre Ljubljana, Slovenia, with a 7-day history of a red left eye with pain provoked by eye movement, blurred vision, photophobia and increased lacrimation. His previous medical history was unremarkable with the exception of numerous tick bites (the last 1 being several months before the onset of the present illness) and of solitary EM in 2005 that had been treated with azithromycin. The only abnormal finding on physical examination was a red and painful left eye. No skin lesions were uncovered, and neurologic examination disclosed no abnormalities. Ophthalmologic examination revealed impaired vision in the left eye (0.4 Snellen equivalent) while visual acuity in the right eye was normal (1.0 Snellen equivalent). A few tiny keratic precipitates and cells in the aqueous humor and vitreous body, with some floaters, predominantly in the posterior vitreous, were present in the left eye. The pupil was normal, and the lens was clear. The left optic disk was swollen (also seen on optic coherence tomography and ultrasound), pink, without evident hemorrhages, the vessels were slightly congested, the posterior pole, fovea and peripheral chorioretina appeared normal. No clinical abnormalities were seen in the right eye. The initial ocular pressure was ≤16 mm Hg in both eyes and gonioscopy showed normal angle structures with a slightly more pigmented ciliary body bilaterally. Color vision and visual fields were not affected. A diagnosis of panuveitis with left optic disk edema was made and its cause was searched for. Initial laboratory findings revealed a normal C-reactive protein value (3 mg/L) and a slightly elevated leukocyte count (14 × 109/L) with normal differential white cell counts. Basic biochemical blood tests as well as concentration of serum protein, fibrinogen, antinuclear antibodies and C3 were normal or negative, C4 and serum immunoglobulin G were slightly elevated, and human leukocyte antigen-B27 was not expressed. Negative angiotensin-converting enzyme result pointed against sarcoidosis. Testing for serum antibodies to Toxoplasma gondii was unremarkable, however, the diagnostic work-up revealed the presence of borrelial antibodies in serum. The latter result, the presence of unilateral disc edema and the fact that the patient was from a highly endemic region for LB, prompt further testing including lumbar punction and cerebrospinal fluid (CSF) examination in spite of normal findings on neurologic examination. The evaluation revealed lymphocytic pleocytosis and intrathecal borrelial immunoglobulin M as well as immunoglobulin G synthesis (Table 1). B. burgdorferi sensu lato blood and CSF cultures were negative. Therefore a diagnosis of LNB was made and intravenous ceftriaxone 2 g once daily for 14 days was added to therapy with systemic acetazolamide, topical steroids and mydriatics, and triamcinolone sub Tenon. The symptoms started improving within a few days, the signs of the uveitis and optic disk edema regressed in a few weeks, while elevated intraocular pressure (26–36 mm Hg) in the boy’s left eye had to be treated over the next few months. The further course was smooth; there was no relapse of the ophthalmic problems and no clinical signs of LB during 12-month follow-up.
DISCUSSION Panuveitis is an inflammation of the uveal tract involving both the anterior segment (iris and ciliary body) and the posterior segment (chorioretina and optic disc). It can be caused by © 2014 Lippincott Williams & Wilkins
The Pediatric Infectious Disease Journal • Volume 34, Number 1, January 2015
Panuveitis
TABLE 1. Cerebrospinal Fluid and Microbiologic Test Results in the Patient with Panuveitis Variable
CSF Findings
White blood cell count (normal
The Pediatric Infectious Disease Journal • Volume 34, Number 1, January 2015
2. Ferrieri P, Gewitz MH, Gerber MA, et al; Committee on Rheumatic Fever, Endocarditis, and Kawasaki Disease of the American Heart Association Council on Cardiovascular Disease in the Young. Unique features of infective endocarditis in childhood. Circulation. 2002;105:2115–2126. 3. Das M, Badley AD, Cockerill FR, et al. Infective endocarditis caused by HACEK microorganisms. Annu Rev Med. 1997;48:25–33. 4. Kaplan AH, Weber DJ, Oddone EZ, et al. Infection due to Actinobacillus actinomycetemcomitans: 15 cases and review. Rev Infect Dis. 1989;11:46–63. 5. Carlile JR, Beckman EN, Arensman RM. Actinobacillus actinomycetemcomitans pneumonia. Clin Pediatr (Phila). 1984;23:578–580. 6. Chen AC, Liu CC, Yao WJ, et al. Actinobacillus actinomycetemcomitans pneumonia with chest wall and subphrenic abscess. Scand J Infect Dis. 1995;27:289–290. 7. Hagiwara S, Fujimaru T, Ogino A, et al. Lung abscess caused by infection of Actinobacillus actinomycetemcomitans. Pediatr Int. 2009;51:748–751. 8. Nash CB, Hendrickson BA, Alexander KA. Pneumonia with chest wall invasion in a school aged child. J Ped Infect Dis. [published online ahead of print February 16, 2014]. 9. Morris JF, Sewell DL. Necrotizing pneumonia caused by mixed infection with Actinobacillus actinomycetemcomitans and Actinomyces israelii: case report and review. Clin Infect Dis. 1994;18:450–452. 10. Meyer BR, Buttone E, Hirschman SZ, Schneierson SS, Gershengorn K. Infection due to Actinobacillus actinomycetemcomitans. Am J Clin Pathol. 1971;56:204–211. 11. Leys EJ, Griffen AL, Strong SJ, et al. Detection and strain identification of Actinobacillus actinomycetemcomitans by nested PCR. J Clin Microbiol. 1994;32:1288–1294.
PANUVEITIS CAUSED BY BORRELIA BURGDORFERI SENSU LATO INFECTION Jasna Mahne, MD*, Branka Stirn Kranjc, MD, PhD†, Franc Strle, MD, PhD*, Eva Ružić-Sabljić, MD, PhD‡, and Maja Arnež, MD, PhD* Abstract: A 13-year-old boy who presented with a red left eye, painful eye movement, blurred vision, photophobia and increased lacrimation, was diagnosed with 1-sided panuveitis with optic disk edema. Diagnostic workup revealed borrelial antibodies in serum. Diagnosis of Lyme borreliosis was substantiated by demonstration of lymphocytic pleocytosis, intrathecal borrelial antibody synthesis, improvement after treatment with ceftriaxone and exclusion of other causes. Key Words: Panuveitis, Lyme neuroborreliosis, Borrelia burgdorferi sensu lato, pediatric, tick bite Accepted for publication June 25, 2014. From the *Department of Infectious Diseases, University Medical Centre Ljubljana, Japljeva 2, 1525 Ljubljana; †Department of Ophthalmology, University Medical Centre Ljubljana, Grablovičeva 46; and ‡Institute of Microbiology and Immunology, School of Medicine, Zaloška 4, 1000 Ljubljana, Slovenia. Franc Strle’s institution is receiving a grant from the Slovenian Research Agency (No P3-0296). For the remaining authors none were declared. Address for correspondence: Jasna Mahne, MD, Department of Infectious Diseases, University Medical Centre Ljubljana, Japljeva 2, 1525 Ljubljana, Slovenia. E-mail: [email protected]. Copyright © 2014 by Lippincott Williams & Wilkins DOI: 10.1097/INF.0000000000000473
L
yme borreliosis (LB) is a multisystem infectious disease caused by Borrelia burgdorferi sensu lato and transmitted to humans by tick bite. It is endemic in several regions of North America, Asia and Europe, including Slovenia. In European patients, the most common clinical manifestation of the disease is erythema migrans (EM), followed by Lyme neuroborreliosis (LNB).1,2 Data on ocular manifestations of LB are scarce and predominantly restricted to adults. Eyes can be affected primarily as a result of inflammation of eye tissue or secondarily due
102 | www.pidj.com
to extraocular manifestations of LB such as paresis of cranial nerves and orbital myositis.3 We report a case of acute, 1-sided panuveitis with optic disc edema in a child in whom diagnostic work-up revealed LNB.
CASE REPORT On April 13, 2011, a 13-year-old boy was referred to the University Medical Centre Ljubljana, Slovenia, with a 7-day history of a red left eye with pain provoked by eye movement, blurred vision, photophobia and increased lacrimation. His previous medical history was unremarkable with the exception of numerous tick bites (the last 1 being several months before the onset of the present illness) and of solitary EM in 2005 that had been treated with azithromycin. The only abnormal finding on physical examination was a red and painful left eye. No skin lesions were uncovered, and neurologic examination disclosed no abnormalities. Ophthalmologic examination revealed impaired vision in the left eye (0.4 Snellen equivalent) while visual acuity in the right eye was normal (1.0 Snellen equivalent). A few tiny keratic precipitates and cells in the aqueous humor and vitreous body, with some floaters, predominantly in the posterior vitreous, were present in the left eye. The pupil was normal, and the lens was clear. The left optic disk was swollen (also seen on optic coherence tomography and ultrasound), pink, without evident hemorrhages, the vessels were slightly congested, the posterior pole, fovea and peripheral chorioretina appeared normal. No clinical abnormalities were seen in the right eye. The initial ocular pressure was ≤16 mm Hg in both eyes and gonioscopy showed normal angle structures with a slightly more pigmented ciliary body bilaterally. Color vision and visual fields were not affected. A diagnosis of panuveitis with left optic disk edema was made and its cause was searched for. Initial laboratory findings revealed a normal C-reactive protein value (3 mg/L) and a slightly elevated leukocyte count (14 × 109/L) with normal differential white cell counts. Basic biochemical blood tests as well as concentration of serum protein, fibrinogen, antinuclear antibodies and C3 were normal or negative, C4 and serum immunoglobulin G were slightly elevated, and human leukocyte antigen-B27 was not expressed. Negative angiotensin-converting enzyme result pointed against sarcoidosis. Testing for serum antibodies to Toxoplasma gondii was unremarkable, however, the diagnostic work-up revealed the presence of borrelial antibodies in serum. The latter result, the presence of unilateral disc edema and the fact that the patient was from a highly endemic region for LB, prompt further testing including lumbar punction and cerebrospinal fluid (CSF) examination in spite of normal findings on neurologic examination. The evaluation revealed lymphocytic pleocytosis and intrathecal borrelial immunoglobulin M as well as immunoglobulin G synthesis (Table 1). B. burgdorferi sensu lato blood and CSF cultures were negative. Therefore a diagnosis of LNB was made and intravenous ceftriaxone 2 g once daily for 14 days was added to therapy with systemic acetazolamide, topical steroids and mydriatics, and triamcinolone sub Tenon. The symptoms started improving within a few days, the signs of the uveitis and optic disk edema regressed in a few weeks, while elevated intraocular pressure (26–36 mm Hg) in the boy’s left eye had to be treated over the next few months. The further course was smooth; there was no relapse of the ophthalmic problems and no clinical signs of LB during 12-month follow-up.
DISCUSSION Panuveitis is an inflammation of the uveal tract involving both the anterior segment (iris and ciliary body) and the posterior segment (chorioretina and optic disc). It can be caused by © 2014 Lippincott Williams & Wilkins
The Pediatric Infectious Disease Journal • Volume 34, Number 1, January 2015
Panuveitis
TABLE 1. Cerebrospinal Fluid and Microbiologic Test Results in the Patient with Panuveitis Variable
CSF Findings
White blood cell count (normal
