Full text of DOI:10.1007/s004170100374

Graefe’s Arch Clin Exp Ophthalmol
(2001) 239:882–885
L E T T E R T O T H E E D I T O R
DOI 10.1007/s004170100374
Harumi Fukushima
Jiro Numaga
Miwako Yoshimoto
Satoshi Kato
An ocular cat-scratch disease patient positive
for cytoplasmic anti-neutrophil cytoplasmic
antibody
Hidetoshi Kawashima
Abstract Background: We report a
case of ocular cat-scratch disease
Conclusion: C-ANCA is an indicator
of vasculitis and may be useful as an
Received: 26 April 2001
Revised: 6 August 2001
Accepted: 24 August 2001
Published online: 11 October 2001
© Springer-Verlag 2001
with permanent vision reduction in a indicator of severe cat-scratch dis-
patient who was cytoplasmic anti-
neutrophil cytoplasmic antibody
(C-ANCA) positive. Methods: Case
report and review of the literature.
Results: While taking steroids and
antibiotics, a 52-year-old man with
uveitis associated with cat-scratch
disease developed retinal vein occlu-
sion and a macular exudate. His final
visual acuity was poor because of re-
sidual macular degeneration and op-
tic atrophy. Serum C-ANCA in-
creased and decreased in parallel
with ocular inflammatory activity.
ease.
H. Fukushima ( ) · J. Numaga · S. Kato
✉
H. Kawashima
Department of Ophthalmology,
University of Tokyo School of Medicine,
7-3-1 Hongo, Bunkyo-ku, Tokyo,
113-8655, Japan
e-mail: fharumi@muc.biglobe.ne.jp
Tel.: +81-3-58413498
Fax: +81-3-38170798
M. Yoshimoto
Tokyo Teishin Hospital, Tokyo, Japan
eyes. Eye position, eye movement, and light reflexes were also
normal in both eyes. Slit-lamp examination showed a clear cornea
and lens bilaterally. However, 1+ cells in the anterior chamber and
3+ cells in the anterior vitreous body were recognized in the right
Introduction
Cat-scratch disease is caused by Bartonella henselae [5].
The prognosis of ocular cat-scratch disease is usually ex- eye. The right optic nerve head was swollen with dilated capillar-
cellent. We report a rare case with permanent vision re-
duction in a patient who was cytoplasmic anti-neutrophil
ies. No remarkable changes were found in the left eye.
A full systemic test was initiated. The erythrocyte sedimenta-
tion rate (ESR) was 47 mm/h (normal range 3–10 mm/h), and C-
cytoplasmic antibody (C-ANCA) positive.
reactive protein (CRP) was 5.43 mg/dl (normal range <0.3 mg/dl).
Results of other laboratory tests were normal (Table 1), including
complete blood cell counts and differential counts, chemistry pro-
file, serological tests, and immunologic tests. The purified protein
Case report
derivative tuberculin skin test was false positive. Brain computed
tomography and magnetic resonance imaging were negative.
Seven days after the first examination, although C reactive
protein had normalized and systemic symptoms had disappeared,
retinal vein occlusion in the superior nasal quadrant and a macular
exudate star had developed (Fig. 1a, b). Visual acuity was 0.01 in
the right eye. A fluorescein angiogram of the optic nerve head dis-
closed hyperfluorescence and an avascular zone of retinal vein oc-
clusion in the superior nasal quadrant (Fig. 2). Serum titers for
B. henselae were positive at 1 vs over 1024 for immunoglobulin G
(IgG) and 1 vs 20 for immunoglobulin M (IgM). Cat-scratch dis-
ease was diagnosed based on the history of cat scratches, the high
A 52-year-old man was referred because of uveitis of unknown or-
igin in his right eye with decreased vision. The patient had been
healthy and was not suffering from ocular disease until 10 days
before referral, when he began experiencing low-grade fevers and
fatigue. Two days before referral he had noted a fever of 38.4°C,
and the next day he awoke with decreased vision in his right eye.
He had sustained several cat scratches in a zoological garden just
before his initial symptoms manifested.
There were no enlarged cervical lymph nodes, and best-cor-
rected visual acuity was 0.15 in the right eye and 1.5 in the left
eye. Intraocular pressure was within the normal range in both

883
Table 1 Laboratory data at
first visit
Parameter
Value at first visit
Normal range
White blood cell count
Neutrophils
6000/µl
44%
(4000–9000)
(42–74)
(0–7)
Eosinophils
1%
Basophils
4%
(0–2)
Monocytes
13%
(1–8)
(18–50
Lymphocytes
36%
Red blood cell count
Hemoglobin
434*104/µl
13.1 g/dl
38.4%
(410–530)
(14–18)
(40–48)
(14–40)
(6.0–7.8)
(3.7–4.9)
(9–38)
Hematocrit
Platelets
25.9*104/µl
7.6 g/dl
4.0 g/dl
26 IU/l
Total protein
Serum albumin
Aspartate aminotransferase
Alanine aminotransferase
Gamma-glutamyl transpeptidase
Serum urea nitrogen
Serum creatinine
Blood sugar
28 IU/l
34 IU/l
(4–36)
(4–68)
11 mg/dl
1.3 mg/dl
141 mg/dl
139 mEq/l
3.8 mEq/l
101 mEq/l
47 mm
5.43 mg/dl
20 IU/ml
33.0 U/ml
Negative
73 U/ml
Negative
(9–21)
(0.6–1.3)
(<160)
Sodium
(132–148)
(3.8–5.0)
(100–110)
(<10)
(<0.3)
(<20)
(33.0–48.0)
Potassium
Chloride
ESR (1 h)
CRP
Rheumatoid factor
50% of hemolytic unit of complement
Coomb's test
Antistreptolysin O
Antinuclear antibodies
(<241)
AntiDNA antibodies
Double stranded
Single stranded
Negative
Negative
Antiextractable nuclear antigen antibodies
RNP
SM
SS-A
SS-B
Scl-70
Negative
Negative
Negative
Negative
Negative
Immunoglobulin
A
264 mg/dl (107–363)
1407 mg/dl (739–1649)
142 mg/dl (46–260)
11.8 IU/l
G
M
Angiotensin-converting enzyme
Cardiolipin antibody,
ß2-glycoprotein-1 complex
Serologic tests for syphilis
Antitoxoplasma antibody
Negative
Negative
1:128
(<1:160)
(<1:4)
Antibody titers for viruses
Cytomegalovirus
(cytomegalovirus antigen negative)
Herpes simplex
Herpes zoster
1:16
1:16
1:4
(<1:4)
(<1:4)
Human immunodeficiency virus
Hepatitis C
Hepatitis B antigen
Negative
Negative
Negative
Anti-B. henselae antibody
IgG
1:1024<
1:20
(<1:64)
(<1:20)
IgM
Anti-B. quintana antibody
IgG
1:64
1:20
(<1:64)
(<1:20)
IgM

884
Fig. 2 Fluorescein angiogram of the right eye obtained at the
same time as the ophthalmoscopic image in Fig. 1a. Hyperfluores-
cence of the optic nerve head and an avascular zone are seen in the
superior nasal quadrant
titer of B. henselae antibodies, and past medical history. Further-
more, serum C-ANCA was positive at 27 U/µl (normal range
0–10 U/µl).
Results
The patient was given oral ampicillin (2 g four times a
day) for 1 week, aspirin (81 mg/day) for 6 months and
prednisolone (tapering from 30 mg twice a day) for 4
months. He demonstrated gradual improvement of the
uveitis and neuroretinitis but no improvement in visual
acuity because of persistent macular degeneration and
optic atrophy (Fig. 1c). Serum titers for both B. hens-
elae and C-ANCA decreased in parallel with ocular dis-
ease activity (Table 2). His final right visual acuity was
0.3.
Discussion
This is the first report of cat-scratch disease associated
with C-ANCA positivity. Cat-scratch disease is usually
described as a benign, self-limited illness [2] Ocular
manifestations of cat-scratch disease include Parinaud’s
oculoglandular syndrome, papillitis, neuroretinitis [1, 3],
and peripapillary angiomatosis.[3] Appropriate treatment
(antibiotics and/or steroid administration) usually results
in markedly improved visual acuity, unless optic nerve
Fig. 1 a Ophthalmoscopy of the right eye 1 week after the initial
examination. Optic nerve head swelling, retinal vein occlusion in
the superior-nasal quadrant, and a macular exudate star are evi-
dent. b Three weeks after initial examination. c Four months after atrophy or cystoid macular edema persists [4].
initial examination. Optic nerve atrophy with proliferative tissue
and macular degeneration
Our patient had retinal vein occlusion with plasma C-
ANCA positivity. C-ANCA is thought to be an indicator
of vasculitis specific to Wegener’s granulomatosis. How-
ever, our patient had no convincing evidence of

885
Table 2 Changes in C-ANCA, P-ANCA, IgM and IgG for B. henselae, CRP, ESR, and visual acuity of the right eye during the clinical
course
Parameter
Date
Normal range
8 Sept 1999a
16 Sept 1999b 4 Oct 1999
11 Nov 1999
2 Feb 2000
31 May 2000
C-ANCA
P-ANCA
–
–
–
–
15
10
–
–
27
10
22
10
(0–10)
(0–10)
B. henselae
IgM
–
–
–
20
20
1024
0.3
20
512
0.3
9
20
64
0.3
9
(1:20)
(1:64)
(<0.3)
(<10)
–
IgG
–
1024
CRP
ESR
Visual acuity
5.43
47
0.3
0.16
10
0.01
0.3
–
–
0.03
0.06
0.1
0.3
a Initial examination
b The day our patient was referred to us
Wegener’s granulomatosis, such as upper respiratory Serum titers of C-ANCA decreased in parallel with ocu-
tract findings (by ear, nose, throat examination), pulmo- lar disease activity. This is the first report of a cat-scratch
nary involvement (by chest X-ray), and renal dysfunc- disease patient positive for C-ANCA. C-ANCA may
tion (by laboratory data). Other connective tissue diseas- thus be useful as an indicator of severe cat-scratch dis-
es were unlikely based on the absence of autoantibodies. ease with a poor visual prognosis.
References
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(1990) Neuroretinitis associated with cat
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patients. Am J Dis Child 139: 1124–1133
3. Golnik K, Marotto M, Fanous M,
et al (1994) Ophthalmic manifestations
of Rochalimaea species. Am J
5. Regnery R, Olson J, Perkins B,
et al (1992) Serological response to
“Rochalimaea henselae” antigen in sus-
pected cat-scratch disease. Lancet 339:
1443–1145.
Ophthalmol 118: 145–151
4. Kerkhoff F, Ossewaarde J, de Loos W,
et al (1999) Presumed ocular bartonello-
sis. Br J Ophthalmol 83: 270–275