British Journal of Radiology (2007) 80, e24-e26
© 2007 British Institute of Radiology
doi: 10.1259/bjr/93847196
Unicentric and multicentric Castleman's disease
K Enomoto, MD
1,2
I Nakamichi, MD
3
K Hamada, MD
1
A Inoue, MD
4
I Higuchi, MD
1,5
M Sekimoto, MD, PhD
5
M Mizuki, MD, PhD
6
Y Hoshida, MD, PhD
3
T Kubo, MD, PhD
2
K Aozasa, MD, PhD
3 and
J Hatazawa, MD, PhD
1
Department of 1Nuclear Medicine and Tracer Kinetics 2Otolaryngology and Sensory Organ Surgery 3Pathology 4Radiology 5Clinical Oncology and Surgery and 6Hematology and Oncology, Osaka University Graduate School of Medicine, Suita, Osaka, Japan
Correspondence: Keisuke Enomoto, Department of Nuclear Medicine and Tracer Kinetics, Osaka University Graduate School of Medicine, 2-2, Yamadaoka, Suita City, Osaka, 565-0871, Japan. E-mail: keisuke.enomoto{at}tracer.med.osaka-u.ac.jp
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Abstract
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Castleman's disease (CD) appears at ubiquitous lymph nodes. To date, detection of the lesion focus for CD has mainly been carried out by physical examination and radiological findings, such as X-ray analysis, CT and MRI. 18F-FDG PET visualizes the active focus of glucose metabolism and the clinical value has been investigated for many different tumours. Previous studies of 18F-FDG PET for CD have only reported four cases of unicentric CD and no cases of multicentric CD. In this paper, we report two cases of CD, one with unicentric CD and one with multicentric CD. We demonstrate that the use of 18F-FDG PET for the detection and monitoring of patients with CD, especially multicentric CD, would be effective.
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Introduction
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Castleman's disease (CD) is an uncommon lymphoproliferative disease that was first described in 1956 [1]. The aetiology and pathogenesis are still under debate. Approximately 70% of CD cases occur in the thorax, 10–15% in the neck and 10–15% in the abdomen, retroperitoneum and pelvis [2]. CD is usually classified into two distinct subtypes: unicentric and multicentric [3]. The unicentric type is more common, and shows hyaline-vascular follicles and interfollicular capillary proliferation. Patients with this type of CD are treated surgically and have good prognoses. The multicentric type is histopathologically characterized by massive accumulation of polyclonal plasma cells in the interfollicular region. This type of CD is progressive and has a poor outcome. A more generalized lymphoadenopathy, accompanied by systemic symptoms, laboratory abnormalities, organomegaly and a more aggressive clinical course with the potential for malignant transformation, has been described. To date, detection of the lesion locus for CD has mainly been carried out by physical examination and radiological findings, such as X-ray analysis, CT, MRI and gallium scintigraphy. In the current report, we present two cases of CD, whose lesion locations were examined by positron emission tomography (PET) with 2-deoxy-[F-18]fluoro-D-glucose (18F-FDG).
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Case reports
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Case 1
A 44-year-old male presented with a large mesenteric mass. He had no abdominal symptoms, incontinence, or weight loss. Laboratory data were normal. A subsequent contrast-enhanced abdominal helical CT revealed a mesenteric mass (Figure 1a
). The patient underwent an 18F-FDG PET scan for pre-operative staging at 1 h after injection of 384 MBq of 18F-FDG. The 18F-FDG PET showed an increased uptake in the mesenteric region (maximum standard uptake value (SUV): 3.55) (Figure 1b). The mesenteric mass was surgically resected, and the histopathological diagnosis was unicentric CD (Figure 1c).
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Figure 1. Case 1.(a) A well-circumscribed 2.6 cmx2.4 cm mass with enhancement (arrow). (b) Accumulation of FDG in the mesenteric region (arrow). (c) Photomicrograph of the patient's lymph node demonstrating lymph follicles with abortive to relatively hyperplastic secondary follicles and hyalinized vessels in the interfollicular area.
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Case 2
A 64-year-old male was admitted with a 7-month history of fever. In his past history, he had suffered from chronic myelomonocytic leukaemia (CMMoL) without medication for 6 months. His blood biochemistry findings were notable for the high basal level of interleukin-6 (1210 pg ml–1). The white blood cell number was 4.77x109 l–1 (36% lymphocytes, 34% monocytes, 34% segmented neutrophils, 0% eosinophils and 0% blastic cells), C-reactive protein was 8.0 mg dl–1, immunoglobulin G (IgG) was 1810 mg dl–1, haemoglobin concentration was 7.4 g dl–1 and the platelet number was 493x109 l–1. Other laboratory tests, such as electrolytes and urine analysis, were normal. A contrast-enhanced chest CT scan showed multiple bilateral hilar and mediastinal lymphadenopathies, without any parenchymal lesions (Figure 2a). Their sizes ranged from 0.4 cm to 2.0 cm in diameter. The patient received an 18F-FDG PET scan for pre-operative staging at 1 h after injection of 392 MBq of 18F-FDG. The 18F-FDG PET images showed a focus of radiotracer accumulation in the axilla, bilateral hilar and mediastinal regions (SUV range: 3.03–5.38) (Figure 2b). An excisional biopsy with a mediastinoscope revealed multicentric CD (Figure 2c).
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Figure 2. Case 2.(a) Chest CT scan showing multiple bilateral hilar and mediastinal lymphadenopathies (arrows). (b) Increased uptake of FDG in the axilla, bilateral hilar and mediastinal regions (arrows). (c) Histopathological examination of the patient's lymph node showing lymphoid follicular hyperplasia and prominent plasma cell proliferation in the interfollicular region.
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Discussion
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A correct grasp of the lesion is very important for patient treatment. Physical examination and radiological findings have conventionally been used to identify lesions in CD cases. In particular, contrast-enhanced CT has been used routinely. CD generally manifests as a homogeneous or heterogeneous mass of soft tissue attenuation.
To our knowledge, previous studies of 18F-FDG PET for CD have only reported four cases of unicentric CD [4–7] and no cases of multicentric CD. We evaluated the lesions of unicentric and multicentric CD by 18F-FDG PET, and were able to detect the lesions clearly. In Case 1, the localized lesion seen on CT was shown to be metabolically active and to represent disease at a single site, and the patient proceeded to surgery. In Case 2, PET identified spread in the axilla, which was not detected on CT or guided biopsy. Our present findings demonstrate that PET examination is very useful for detecting the foci of CD, which can appear to be ubiquitous, and contribute to the management of the CD patients.
Although a CT scan has high spatial resolution, it can only evaluate the viability of a lesion by its dimensions. Arborizing, central and discrete calcifications have been reported on contrast-enhanced CT, but are unusual. The presence of oedema in the mucosal surface and distortion of the anatomic facial planes secondary to prior surgery or radiation therapy make differentiation of recurrent lesions from post-therapeutic changes difficult, if not impossible. Therefore, we consider that 18F-FDG PET imaging for CD, especially multicentric type, would be effective for assessment of disease extension and monitoring of treatment and detection of recurrent disease.
Received for publication June 27, 2005.
Revision received February 7, 2006.
Accepted for publication February 9, 2006.
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References
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- Castleman B, Towne VW. Case records of the Massachusetts General Hospital: case 40011. N Engl J Med 1954;250:26–30.[Medline]
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Abstract
Introduction
