Cutaneous multicentric Castleman's disease mimicking IgG4-related disease

Takeuchi, Mai; Sato, Yasuharu; Takata, Katsuyoshi; Kobayashi, Keita; Ohno, Kyotaro; Iwaki, Noriko; Orita, Yorihisa; Yoshino, Tadashi

doi:10.1016/j.prp.2012.09.006

Cited by 39 publications

(30 citation statements)

References 22 publications

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“…While cutaneous IgG4-RD lesions are mostly present with other extracutaneous lesions, skin involvement rarely precedes other lesions as the first manifestation (6,7,18). IgG4-related skin lesions usually present as erythematous and itchy plaques or subcutaneous nodules (19,20).…”

Section: Discussionmentioning

confidence: 99%

“…The present case was initially suspected to have IgG4-related skin lesions; however, as the histological findings did not fulfil these diagnostic features and the IgG4-/IgG+ plasma cell ratio was below 40%, we did not diagnose the patient with IgG4-RD at that time. On the other hand, because MCD patients can have elevated serum IgG4 levels and a high tissue IgG4+/IgG+ plasma cell ratio (18) and due to the presence of laboratory findings that are frequently observed in MCD patients, such as anemia, hypergammaglobulinemia and, importantly, elevated CRP and IL-6 levels, he was initially diagnosed with MCD. Patients with IgG4-RD do not usually have constitutional symptoms and have only modestly abnormal inflammatory marker levels with a normal IL-6 level.…”

Section: Discussionmentioning

confidence: 99%

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Overlap of IgG4-related Disease and Multicentric Castleman's Disease in a Patient with Skin Lesions

et al. 2017

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A 59-year-old man presented with multiple dark red erythemas with induration, anemia, and polyclonal hypergammaglobulinemia. A skin biopsy revealed the infiltration of lymphocytes and plasma cells and he was initially diagnosed with multicentric Castleman's disease (MCD). Glucocorticoid treatment was only partially effective. Four years later, the patient's bilateral lacrimal glands gradually became enlarged and a biopsy revealed dense lymphocyte and plasma cell infiltration with an IgG4+/IgG+ plasma cell ratio of 70%. The patient was diagnosed with IgG4-related disease (RD). Rituximab only had a slight effect. This case demonstrates that overlapping features of IgG4-RD and MCD may present in a single patient, which suggests a shared pathogenesis.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Overlap of IgG4-related Disease and Multicentric Castleman's Disease in a Patient with Skin Lesions

et al. 2017

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“…9,10 Recently, we encountered a case of cutaneous involvement of multicentric Castleman's disease that fulfilled the diagnostic criteria for IgG4-related disease. 11 Therefore, it is important to differentiate between IgG4-related skin disease and cutaneous involvement of multicentric Castleman's disease. In our cases, hyper-IL-6 syndromes were histologically and clinically excluded.…”

Section: Discussionmentioning

confidence: 99%

Clinicopathologic analysis of IgG4-related skin disease

et al. 2013

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IgG4-related disease is a recently recognized systemic syndrome characterized by mass-forming lesions with lymphoplasmacytic infiltration, increase in the number of IgG4 þ cells in affected tissues and elevation of serum IgG4 levels. In 2009, we were the first to report skin lesions in patients with IgG4-related disease, but no large case series has been reported and clinicopathological findings remain unclear. To clarify these features, we herein report 10 patients (9 men and 1 woman; median age, 64 years; age range, 46-81 years) with IgG4-related skin disease. All patients had erythematous and itchy plaques or subcutaneous nodules on the skin of the head and neck, particularly in the periauricular, cheek, and mandible regions, except for one patient, whose forearm and waist skin were affected. In addition, eight patients had extracutaneous lesions: these were found on the lymph nodes in six patients, the lacrimal glands in three patients, the parotid glands in three patients, and the kidney in one patient. Histologically examined extracutaneous lesions were consistent with IgG4-related disease; five of six lymph node lesions showed progressively transformed germinal centers-type IgG4-related lymphadenopathy. Cases of IgG4-related skin disease were classified into two histological patterns: those exhibiting a nodular dermatitis pattern and those with a subcutaneous nodule pattern. The infiltrate was rich in plasma cells, small lymphocytes, and eosinophils; the majority of the plasma cells were IgG4 þ . The IgG4 þ cell count was 49-396 per high-power field (mean±s.d., 172±129), with an IgG4 þ /IgG þ cell ratio ranging from 62 to 92%. Serum IgG4 levels were elevated in all examined patients. In conclusion, patients with IgG4-related skin disease had uniform clinicopathology. Lesions were frequently present on the skin of the periauricular, cheek, and mandible regions, and were frequently accompanied by IgG4-related lymphadenopathy.

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“…Since no eosinophilic abscess formation or proliferation of blood capillaries were observed among the lymphoid follicles, diagnoses of Kimura's disease (8) and angiolymphoid hyperplasia with eosinophilia (ALHE) were less likely. As the platelet count was not increased and the serum CRP and IL-6 levels were not remarkably elevated, Castleman's disease was also excluded (9). Considering the findings of chest CT and the laboratory tests, allergic bronchopulmonary aspergillosis and Churg-Strauss syndrome were also excluded.…”

Section: A B Cmentioning

confidence: 99%

IgG4-related Ocular Adnexal Disease Mimicking Thyroid-associated Orbitopathy

et al. 2013

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A 72-year-old man presented with bilateral eyelid swelling and redness. An orbital CT scan showed bilateral proptosis, extraocular muscle enlargement and swollen lacrimal glands, mimicking thyroid-associated orbitopathy (TAO) with Hashimoto's thyroiditis (HT). During the patient's clinical course, spontaneous remission of lung consolidation (35×26 mm) was seen. A diagnosis of IgG4-related disease (IgG4-RD) was made based on an elevated serum IgG4 level (1,020 mg/dL; normal, 4-108), predominance of IgG4-positive plasma cells (IgG4/IgG: 35/70 in HPF) in the lacrimal glands and typical features of Mikulicz's disease. This report provides a novel description of this unusual disease entity among HT, TAO and IgG4-RD.

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Cutaneous multicentric Castleman's disease mimicking IgG4-related disease

Cited by 39 publications

References 22 publications

Overlap of IgG4-related Disease and Multicentric Castleman's Disease in a Patient with Skin Lesions

Overlap of IgG4-related Disease and Multicentric Castleman's Disease in a Patient with Skin Lesions

Clinicopathologic analysis of IgG4-related skin disease

IgG4-related Ocular Adnexal Disease Mimicking Thyroid-associated Orbitopathy

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