A Variant of AESOP Syndrome (Adenopathy and Extensive Skin Patch Overlying a Plasmacytoma) in a Malignant Blue-Cell Tumor

Foo, Chong Wee; Florell, Scott R.; Petersen, Marta J.

doi:10.1001/archdermatol.2012.2858

Cited by 6 publications

(6 citation statements)

References 5 publications

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“…Histological examination of this plaque revealed a similar vascular proliferation without dermal mucin deposition and with pleomorphic, multinucleated histiocytes. While there has been some discussion regarding whether this case was an example of AESOP or MCAH, the plaque interestingly resolved with radiation and chemotherapy as is reported in AESOP …”

Section: Discussionmentioning

confidence: 83%

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Adenopathy and extensive skin patch overlying a plasmacytoma with unusual histologic findings in a patient with polyneuropathy, organomegaly, endocrinopathy, monoclonal protein and skin changes syndrome and Castleman disease

et al. 2019

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A 56-year-old previously healthy man presented to the dermatology clinic with a 2-year history of an expanding, violaceous, infiltrated plaque on the right flank. Biopsy revealed a diffuse dermal vascular proliferation of bland, capillary-sized vessels admixed with conspicuous fibrohistiocytic cells including scattered multinucleated floret cells. Further workup revealed a monoclonal gammopathy, an osteolytic chest wall plasmacytoma underlying the plaque, and regional lymphadenopathy leading to a diagnosis of adenopathy and extensive skin patch overlying a plasmacytoma (AESOP).Biopsy of an enlarged lymph node revealed Castleman disease. The patient subsequently developed polyneuropathy and peripheral edema, which supported an additional diagnosis of polyneuropathy, organomegaly, endocrinopathy, monoclonal protein, and skin changes (POEMS) syndrome. Herein, we discuss the unique findings of our patient, the potential pathogenesis of AESOP, and the link between these three rare paraneoplastic entities along with review of the literature. K E Y W O R D S adenopathy and extensive skin patch overlying a plasmacytoma, AESOP, Castleman disease, POEMS, polyneuropathy, organomegaly, endocrinopathy, monoclonal protein and skin changes

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Section: Discussionmentioning

confidence: 83%

“…Treatment of the plasmacytoma with radiation, surgery plus radiation, or radiation plus chemotherapy has a good prognosis and can be curative . It often leads to regression of the skin lesion over several months and resolution of POEMS manifestations .…”

Section: Discussionmentioning

confidence: 99%

Adenopathy and extensive skin patch overlying a plasmacytoma with unusual histologic findings in a patient with polyneuropathy, organomegaly, endocrinopathy, monoclonal protein and skin changes syndrome and Castleman disease

et al. 2019

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“…To date, 20 cases of AESOP syndrome have been published, including ours (Table 1). 3–6,8–17 In all cases, patients presented with an extensive ill-defined violaceous patch on the trunk, in 19 an underlying plasmacytoma was identified, and in 1 case it was a blue-cell sarcoma. 10,15 The most common location of the plasmacytoma was ribs (10/19), followed by sternum (5/19), scapula (1/19), clavicle (1/19), skull (1/19), and cervical spine/ribs (1/19).…”

Section: Discussionmentioning

confidence: 99%

“…3–6,8–17 In all cases, patients presented with an extensive ill-defined violaceous patch on the trunk, in 19 an underlying plasmacytoma was identified, and in 1 case it was a blue-cell sarcoma. 10,15 The most common location of the plasmacytoma was ribs (10/19), followed by sternum (5/19), scapula (1/19), clavicle (1/19), skull (1/19), and cervical spine/ribs (1/19). Among 20 reported cases (including ours), 15 patients presented with polyneuropathy and 7 with Castleman’s disease.…”

Section: Discussionmentioning

confidence: 99%

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Adenopathy and extensive skin patch overlying plasmacytoma syndrome—the clue to early diagnosis of polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin changes: A case series and literature review

Sanchez

Ramos

Ratnarajah

et al. 2022

SAGE Open Medical Case Reports

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Importance: Adenopathy and extensive skin patch overlying plasmacytoma syndrome is a paraneoplastic syndrome characterized by a cutaneous vascular patch overlying a plasmacytoma and systemic manifestations. It is thought to be an early stage of polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin changes syndrome, which is a rare, but potentially fatal multisystemic disease that is associated with plasma cell dyscrasia. Thus, a high index of suspicion is required to identify patients with adenopathy and extensive skin patch overlying plasmacytoma as they may present with early polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin changes, which is curable if detected early. Objective: To report additional cases of adenopathy and extensive skin patch overlying plasmacytoma syndrome, describe dermatoscopic and histologic findings of the cutaneous patch and review all up to date literature on adenopathy and extensive skin patch overlying plasmacytoma syndrome. Design: Case series from a single tertiary care center. Participants: Here, we present the second case series of three patients with adenopathy and extensive skin patch overlying plasmacytoma syndrome who all meet the diagnostic criteria for polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin changes. The diagnosis was suspected based on the presence of the violaceous cutaneous patch along with symptoms of systemic involvement (fatigue, weight loss, weakness). Dermoscopy revealing regular dilated parallel capillaries was suggestive of a benign/reactive vascular process. Histopathology in all three cases showed reactive vascular proliferation with a characteristic 90° branching. To date only 20 cases of adenopathy and extensive skin patch overlying plasmacytoma have been published, including ours. All patients presented with cutaneous lesions (violaceous patch and others) and most, at least 15/20, met the diagnostic criteria for polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin changes. When clinical follow-up was reported, most patients had a favorable prognosis with partial or complete symptom resolution following treatment of the underlying plasmocytoma.

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