Cutaneous Metaplastic Synovial Cyst in Ehlers-Danlos Syndrome: Report of a Second Case

Guala, Andrea; Viglio, Simona; Ottinetti, Antonio; Angeli, Giovanni; Canova, Gualtiero; Colombo, Enrico; Dardano, F.; Danesino, Cesare; Valli, Maurizia

doi:10.1097/dad.0b013e31815b9c82

Cited by 10 publications

(8 citation statements)

References 9 publications

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“…These cells are difficult to be distinguished by those, such as dermatologists, who are unfamiliar with synovial tissue. Positive immunoreactivity of lining cells against vimentin, but not against cytokeratin, CEA, or S-100, 3 together with the absence of expression of synovium marker CD68, supports the diagnosis.…”

Section: Discussionmentioning

confidence: 67%

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Cutaneous Metaplastic Synovial Cyst: Case Report and Literature Review from the Dermatological Point of View

et al. 2016

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Cutaneous metaplastic synovial cysts (CMSCs) are rare tumors typically comprising a solitary, wellcircumscribed cystic mass that is not connected to the joint. Synovial cysts have been reported predominantly by orthopedists or pathologists; however, the presence of CMSC is not generally well recognized by dermatologists. Herein, we report a CMSC in a 68-year-old woman receiving systemic corticosteroid therapy for the treatment of eosinophilic granulomatosis with polyangiitis (EGPA). We attempt to delineate the clinical characteristics of this unusual neoplasm by reviewing the literature, focusing especially on dermatological descriptions. Histologic examination of the surgical specimen in the current case revealed that the cystic wall was lined with layers of flattened synovial cell-like cells and connective tissues, mimicking the synovial membrane. Positive immunoreactivity of the lining cells against vimentin was detected, but no immunoreactivity against cytokeratin, carcinoembryonic antigen (CEA), CD68, or S-100 was detected. The pathogenesis of CMSC remains unclear, but it has been tightly linked to direct traumatic stimuli or relative tissue fragility, which potentially accounts for CMSC development in our case. Most CMSCs reported by dermatologists are located on the extremities, whereas those described by other specialists tend to be distributed more globally. Preoperative diagnoses are often either epidermal cyst or suture/foreign body granuloma. Incomplete surgical excision of usual synovial cysts may lead to local recurrence, which has been reported in oral and maxillofacial surgery, but not in dermatologic surgery. This fact could be explained by the technical difficulties of surgical excision related to anatomical location. Dermatologists need to be aware of CMSC, and CMSC should be included in the differential diagnosis of subcutaneous cysts.

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Section: Discussionmentioning

confidence: 67%

“…2 CMSC usually appears at the site of previous surgery or trauma and is not related to joint or synovial structures. 3 CMSC is not considered to be a true cyst because it lacks a definite epithelial lining. 1 Synovial cysts have been predominantly discussed in the field of orthopedic medicine or pathology.…”

Section: Introductionmentioning

confidence: 99%

Cutaneous Metaplastic Synovial Cyst: Case Report and Literature Review from the Dermatological Point of View

et al. 2016

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“…Since first described in 1987 by Gonzalez et al1, 23 cases of CMSC have been reported in 12 English language studies1-12. The current CMSC case was rare as it was on the sole without a history of previous trauma or surgical procedure.…”

Section: Discussionmentioning

confidence: 86%

“…Although the exact cause of CMSC has remained unclear, 13 of 23 reported cases have had histories of local trauma. Further, three of ten cases that have occurred spontaneously without local trauma were accompanied by Ehlers-Danlos syndrome3,10,12, which led us to believe that abnormal healing after microtrauma was a pathogenic mechanism of CMSC. Additionally, the fact that four cases of CMSC have accompanied rheumatoid arthritis6,9 and arthrosis10 suggested that chronic inflammation, constant pressure, and repeated manipulation have played critical roles in the pathogenesis of the lesions.…”

Section: Discussionmentioning

confidence: 99%

Cutaneous Metaplastic Synovial Cyst of the First Metatarsal Head Area

et al. 2011

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A cutaneous metaplastic synovial cyst (CMSC) is a cyst lined with metaplastic synovial tissue, which includes the formation of an intracystic villous structure resembling hyperplastic synovial villi. Clinically, the lesion is a tender, subcutaneous nodule that usually occurs at the site of previous surgical trauma and is frequently misdiagnosed as a suture granuloma. The actual cause remains unclear; however, trauma is presumed to be a precipitating factor, as most reported cases have demonstrated a history of antecedent cutaneous injury. Here, we present a case of CMSC in a 51-year-old woman who presented with a cystic mass localized in the left sole. She had no history of previous trauma or surgical procedures performed in the area. Although the case explained in this report is a spontaneous case of CMSC that occurred without a history of trauma, it is believed to have been caused by constant and chronic pressure since CMSC occurred in the first metatarsal head area, a part of the sole where heavy pressure is consistently applied.

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“…This lesion was reported in a wide age range (7 to 82 years), but most patients were older than 40 years old ( Table 1 ). In two reports, the reported lesions in a 7 and a 15 year-old patient were assumed to be a manifestation of Ehlers-Danlos Syndrome ( 12 , 13 ). Although our patient was young, no signs of Ehlers-Danlos syndrome and related manifestations were found.…”

Section: Discussionmentioning

confidence: 99%

Cutaneous Metaplastic Synovial Cyst: A Case Report and Literature Review

Kermani¹,

Dehghani²,

Dehghani³

et al. 2015

Iran Red Crescent Med J

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Introduction:Cutaneous metaplastic synovial cyst (CMSC) is a rare cutaneous lesion characterized by a tender subcutaneous nodule, which usually occurs at the site of previous surgical or local trauma. Histologically, the lesion includes a cystic structure with villous-like projections and a lining mimicking hyperplastic synovium.Case Presentation:We reported the first case of CMSC which developed at the surgical incision site of treatment of a maxillofacial fracture. In addition, we reviewed English literature to evaluate all previously reported CMSC cases and discussed its clinical and histopathological features and etiology. From 1987 to now, reviewing the English literature about CMSC includes 17 studies that described 28 cases, and our presented case was the 29th. There was no sex predilection and age of patients ranged from 7 to 82 years, but most of them were over 40 years. We can see this lesion in any site of the body and hand/arm is the most prevalent involved region (28% cases). Most of the patients had a history of previous local trauma or operation in the involved area.Conclusions:Although the actual etiology of CMSC remains unclear, trauma, as the most probable etiologic factor, plays a role in development of CMSC. Surgical excision of the lesion is the preferable treatment choice and rate of recurrence is low.

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Cutaneous Metaplastic Synovial Cyst in Ehlers-Danlos Syndrome: Report of a Second Case

Cited by 10 publications

References 9 publications

Cutaneous Metaplastic Synovial Cyst: Case Report and Literature Review from the Dermatological Point of View

Cutaneous Metaplastic Synovial Cyst: Case Report and Literature Review from the Dermatological Point of View

Cutaneous Metaplastic Synovial Cyst of the First Metatarsal Head Area

Cutaneous Metaplastic Synovial Cyst: A Case Report and Literature Review

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