Superficial lymph nodes were not palpable. Swelling

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Letters to the Editor
6 Smolen J, Landewe RB, Mease P et al. Efficacy and
safety of certolizumab pegol plus methotrexate in
active rheumatoid arthritis: the RAPID 2 study.
a randomised controlled trial. Ann Rheum Dis 2009;68:
797–804.
7 Fleischmann R, Vencovsky J, van Vollenhoven RF et al.
Efficacy and safety of certolizumab pegol monotherapy
every 4 weeks in patients with rheumatoid arthritis
failing previous disease-modifying antirheumatic
therapy: the FAST4WARD study. Ann Rheum Dis 2009;68:
805–11.
8 Keystone E, Fleischmann R, Smolen J et al. The efficacy of
certolizumab pegol added to methotrexate is sustained
over 2 years in the treatment of rheumatoid arthritis.
Arthritis Rheum 2009;60(Suppl.):S622.
Rheumatology 2012;51:580–582
doi:10.1093/rheumatology/ker350
Advance Access publication 24 November 2011
Arthropathy with infiltrate IgG4-positive plasma
cells in synovium
SIR, Recent reports of IgG4-related disease have
described various organ involvement [1, 2]. Chronic inflammation can affect lacrimal glands, salivary glands,
the thyroid, lung, pancreas, kidney and prostate. The concept includes Mikulicz’s disease, Riedel’s thyroiditis, pulmonary fibrosis, pulmonary pseudotumour, autoimmune
pancreatitis, a part of tubulointerstitial nephritis and
chronic prostatitis. It is important to note that these lesions can occur at different times and sites. The disease
is clinically characterized by elevated serum IgG4
concentration and infiltration of abundant IgG4-positive
plasma cells in the various tissues, mainly exocrine
tissue [3, 4]. Mikulicz’s disease presents with enlargement
of the lacrimal or salivary glands with infiltrate
IgG4-positive plasma cells. Numerous IgG4-positive
plasma cells were characteristically observed in pancreatic tissue with autoimmune pancreatitis. In IgG4-related
disease, mass-forming lesions (pseudotumours) that
consist of lymphoplasmacytic infiltrates and sclerosis,
such as retroperitoneal sclerosis, are observed [5].
Fluorodeoxyglucose PET (FDG-PET)/CT is a very useful
tool for evaluation of multiple organ involvement in
IgG4-related disease [6, 7]. Here, we would like to report
a case of arthropathy with infiltration of IgG4-positive
plasma cells in synovial tissue without any other organ
involvement. In the present case, we have not detected
any organ involvement or mass-forming lesion on
FDG-PET/CT except for the joints.
A 74-year-old female, who had been diagnosed with
OA, was admitted because of polyarthralgia and body
weight loss ( 15 kg/year). She was negative for fever
and swelling of symmetrical submandibular glands.
! The Author 2011. Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For Permissions, please email: [email protected]
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9 Van vollenhoven R, Smolen J, Schiff M et al. Safety
update on certolizumab pegol in patients with active
rheumatoid arthritis. Arthritis Rheum 2009;60(Suppl.):
S636.
Superficial lymph nodes were not palpable. Swelling
of the joints of the hands, knees and shoulders without
deformities were observed. CRP levels, serum IgG,
serum IgE and IgG4 were elevated to 80, 36, 150 mg/
l, 755 IU/ml and 2170 mg/l (normal range 700 mg/l), respectively. RF, anti-CCP antibody, anti-DNA and
anti-Sm antibodies were negative except for ANA in a
homogeneous pattern (1 : 80). Serum MMP-3 level was
high (>800 ng/ml). Multi-centric Castleman’s disease
(MCD) and Crow–Fukase syndrome were initially considered because serum IL-6 and VEGF levels were elevated at 41.3 and 1870 pg/ml, respectively. The patient
was negative for ascites, oedema in the extremities,
splenomegaly and polyneuropathy. Polyclonal hypergammaglobulinaemia existed, but M-band was not
found in immunoelectrophoresis. No mass lesions
were observed in thoracic or abdominal organs on
ultrasonographic examination and contrast CT imaging.
FDG-PET/CT showed abnormal accumulation in
multiple joints without organ involvement, including
spleen or lymph nodes (Fig. 1A). Therefore, MCD,
Crow–Fukase syndrome or SLE were unlikely in this
patient. Right knee MRI revealed osteophytes, joint
space narrowing and gadolinium enhancement of synovial tissue, and no bone destruction was observed.
She was diagnosed with OA, and right total knee
arthroplasty was performed. Histological examination
of the synovium of the right knee showed papillary
proliferation with marked infiltrate IgG4/CD138 doublepositive plasma cells without lymphoid follicles or
fibrosis (Fig. 1B–D). However, hyperplasia of the
synovial lining cells, the hallmark of RA [8], was not
observed in this case. Furthermore, a high concentration of serum IgG4 and abundant infiltration of IgG4positive plasma cells in the synovium were observed.
This contrasts greatly with RA, in which a high concentration of serum IgG1 and dominant IgG1-positive
plasma cells in the synovium are common [9]. Lip
biopsy was performed to rule out Mikulicz’s disease
and histopathological examination of minor salivary
glands revealed no infiltrate IgG4-positive plasma
cells. Based on these findings, she was diagnosed
with IgG4-related arthropathy and low-dose prednisolone (15 mg/day) was administered for 12 weeks.
Polyarthralgia disappeared, and CRP, serum IgG, IgE,
IgG4, IL-6, VEGF and MMP-3 were decreased to 0.1,
13 320 mg/l, 600 IU/ml, 1520 mg/l, 11.2, 147 pg/ml and
300 ng/ml, respectively.
Arthritis is reported to occur in 10% of patients with
IgG4-related systemic disease [4]. Recently, Shinoda
et al. [10] reported a case of Mikulicz’s disease with destructive arthritis in which histopathological examination of
the synovium showed infiltrate IgG4-positive plasma cells.
However, arthropathy without infiltration of IgG4-positive
plasma cells in the various tissues has not been reported.
Whole-body FDG-PET/CT scan can detect the active lesions in systemic IgG4-related diseases and active systemic lesions. In the present case, FDG-PET/CT scan showed
abnormal accumulation of FDG only in multiple joints
Letters to the Editor
FIG. 1 FDG-PET/ CT and histological findings of the synovium of the right knee. FDG accumulation was observed only in
multiple joints (A). Synovial tissue of haematoxylin and eosin staining (B), immunostaining for CD138 (C) and IgG4 (D)
indicated in filtration of abundant IgG4-positive plasma cells in the tissue (original magniEcation 100).
Accepted 15 September 2011
Correspondence to: Kunihiko Umekita, Department of Internal
Medicine, Division of Rheumatology, Infectious Diseases and
Laboratory Medicine, Faculty of Medicine, University of
Miyazaki, 5200 Kihara, Kiyotake, Miyazaki 889-1692, Japan.
E-mail: [email protected]
Rheumatology key message
.
We have described for the first time a case of IgG4related arthropathy without organ involvement.
Disclosure statement: The authors have declared no
conflicts of interest.
Kunihiko Umekita1, Yumi Kaneko1, Kenji Yorita2,
Yayoi Hashiba1, Motohiro Matsuda1,
Shunichi Miyauchi1, Shiro Ueno1, Ichiro Takajo1,
Norio Kusumoto1, Yasuhiro Nagatomo1,
Kousuke Marutsuka2 and Akihiko Okayama1
1
Department of Internal Medicine, Division of Rheumatology,
Infectious Diseases and Laboratory Medicine, Faculty of
Medicine, University of Miyazaki and 2Pathology Division,
University of Miyazaki Hospital, Miyazaki, Japan.
www.rheumatology.oxfordjournals.org
References
1 Takahashi H, Yamamoto M, Suzuki C et al. The birthday of
a new syndrome: IgG4-related diseases constitute a clinical entity. Autoimmune Rev 2010;9:591–4.
2 Sato Y, Notohara K, Kojima M et al. IgG4-related disease:
historical overview and pathology of hematological disorders. Pathol Int 2010;60:247–58.
3 Stone JH, Khosroshahi A, Deshpande V et al. IgG4-related
systemic disease accounts for a significant proportion of
thoracic lymphoplasmacytic aortitis cases. Arthritis Care
Res 2010;62:316–22.
4 Masaki Y, Dong L, Kurose N et al. Proposal for a
new clinical entity, IgG4-positive multiorgan
lymphoproliferative syndrome: analysis of 64 cases
of IgG4-related disorders. Ann Rheum Dis 2009;68:
1310–5.
581
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without any other organ involvement, such as lacrimal
glands, submandibular glands, lymph nodes and pancreas.
To the best of our knowledge, we have described for the
first time a case of IgG4-related arthropathy without organ
involvement.
Letters to the Editor
5 Cheuk W, Yuen HK, Chu SY et al. Lymphadenopathy of
IgG4-related sclerosing disease. Am J Surg Pathol 2008;
32:671–81.
6 Matsubayashi H, Furukawa H, Maeda A et al. Usefulness
of positron emission tomography in the evaluation of
distribution and activity of systemic lesions associated
with autoimmune pancreatitis. Pancreatology 2009;9:
694–9.
7 Suga K, Kawakami Y, Hiyama A et al. F-18 FDG PET-CT
findings in Mikulicz’s disease and systemic involvement of
IgG4-related lesions. Clin Nucl Med 2009;34:164–7.
8 Kraan MC, Haringman JJ, Post WJ et al.
Immunohistological analysis of synovial tissue for differential diagnosis in early arthritis. Rheumatology 1999;38:
1074–80.
9 Shakib F, Stanworth DR. IgG subclass composition of
rheumatoid arthritic sera and joint fluids. Ann Rheum Dis
1976;35:263–6.
10 Shinoda K, Matsui S, Taki H et al. Deforming arthropathy
in a patient with IgG4-related systemic disease: comment
on the article by Stone et al. Arthritis Care Res 2011;63:
172.
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