Lipomembranous fat necrosis in a patient with mixed connective tissue disease

Halvorson, Christian R; Kwon, Soonyou; Kao, Grace F.; Germanas, Juris P.

doi:10.1016/j.jaad.2010.01.054

Cited by 6 publications

(9 citation statements)

References 4 publications

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“…In the first case report, a skin biopsy from an affected patient's leg showed septal panniculitis with lymphocytic infiltrate and fat necrosis, multiple dermal lymphangiectasis, and dilation of venules between fat lobules [2]. In the second case report, a skin biopsy from a patient's abdomen revealed septal fibrosis and lipomembranous changes [3]. In common with these described findings, panniculitis with fat necrosis and fibrosis were seen in our patient, however lymphangiectasis was not seen.…”

Section: Discussionsupporting

confidence: 79%

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Breast involvement in mixed connective tissue disease

Varma

Szilágyi

Harshan

2019

Radiology Case Reports

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To date, mammographic involvement in mixed connective tissue disease has not been described in the literature to the authors’ knowledge. In addition, only 2 case reports have described panniculitis in mixed connective tissue disease. We present a patient with mixed connective tissue disease with coarse sheet-like calcifications on mammography bilaterally, which increased over time corresponding to rise in levels of anti-dsDNA. Biopsy showed fat necrosis with calcifications, chronic inflammation and fibrosis, vasculitis, as well as CD3, CD4, and CD8+ T cells and CD20+ B cells. These findings are similar histologically and mammographically to “lupus mastitis.”

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Section: Discussionsupporting

confidence: 79%

“…Panniculitis is a very rare entity in MCTD, with only 2 case reports described in the literature [2], [3]. Of these reports, MCTD panniculitis has never been described to affect the breasts.…”

Section: Discussionmentioning

confidence: 99%

Breast involvement in mixed connective tissue disease

Varma

Szilágyi

Harshan

2019

Radiology Case Reports

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“…Some authors have designated LFN as membranes with an 'arabesque' (7)(8)(9)11,16,33) or 'frost on a windowpane' (34) appearance. LFN is scattered singly or in some clusters within fatty and/or fibrous tissues and shows microcystic, macrocystic, and/or crushed features (7)(8)(9)(10)(11)(12)(14)(15)(16)19,(21)(22)(23)(24)(25)(26)28). Scattered LFN within fatty tissues represents early changes of LFN, and LFN within fibrous tissues may represent chronic phase lesions (9,12,(14)(15)(16)18,19,21,35).…”

Section: Morphological Characteristics Of Lfnmentioning

confidence: 99%

“…Such necrotic fat cells or lipogranulomatous lesions can be resolved during the relatively early stages of the disease. However, another unique form of white fat necrosis, designated lipomembranous fat necrosis (LFN) (7)(8)(9)(10)(11)(12)(13)(14)(15), also called lipomembranous changes (8,(15)(16)(17)(18)(19)(20)(21), membranous fat necrosis (1,15,(22)(23)(24)(25)(26)(27)(28)(29), membranocystic changes or fat necrosis (7,8,11,(14)(15)(16)(17)(28)(29)(30)(31)(32)(33)(34), membranous lipodystrophy-like changes (30,31), and pseudomembranous fat necrosis (7,35), is found in fibrotic tissues or later stages of various diseases (9,12,…”

Section: Introductionmentioning

confidence: 99%

Lipomembranous fat necrosis: A distinctive and unique morphology (Review)

et al. 2022

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Lipomembranous fat necrosis (LFN) is an uncommon but distinct form of fat necrosis, which is characterized by eosinophilic, crenulated and/or serpiginous membranes. LFN exhibits macrocystic, microcystic and/or crushed features. LFN is routinely detectable on hematoxylin and eosin (H&E)-stained sections, and is present both in the acute phase and in the later or fibrous stage of necrotic fatty lesions. Smaller crushed LFN embedded within fibrous tissues may be difficult to recognize on H&E-stained sections, but can be highlighted by some staining techniques, including Masson trichrome, periodic acid-Schiff, orcein, long Ziehl-Neelsen stain, silver impregnation, phosphotungstic acid-hematoxylin and luxol fast blue staining. LFN was initially considered a specific feature of Nasu-Hakola disease, but has since been identified in various subcutaneous or intraabdominal lesions related to ischemic conditions or venous insufficiency. In addition, LFN is detectable in intra-articular loose bodies and aortic valves with or without dysfunction, suggesting that LFN is also associated with ischemia-like hypoxic conditions or malnutrition. LFN is considered to be a histological hallmark of hidden ischemic or hypoxic/malnourished conditions in various diseases; however, the exact mechanisms of LFN remain poorly understood. The present review described the clinicopathological features of this interesting, but poorly characterized, condition.

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“…However, there continue to be case reports of connective tissue panniculitides that lack specific features of lupus, dermatomyositis or morphea, but are associated with other autoimmune diseases. For example there is a report of a hydroxychloroquine-responsive connective tissue panniculitis in a patient with Hashimoto's disease, insulin dependent diabetes mellitus and juvenile rheumatoid arthritis 2 , a report of lupus-like panniculitis in association with the autoimmune polyendocrinopathy, candidiasis, and ectodermal dystrophy (APECED) syndrome that improved with oral steroids 75 , and reports of lipomembranous panniculitis with mixed connective tissue disease 76,77 . Cases with overlapping features that blur the boundaries between definitive diagnoses are also present, such as the LEP and dermatomyositis overlap described by Yoo et al and an overlap between systemic sclerosis and LEP described by Oka et al 55,78 .…”

Section: Connective Tissue Panniculitismentioning

confidence: 99%

Update on management of connective tissue panniculitides

Braunstein

Werth

2012

Dermatol Ther

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In connective tissue diseases panniculitis can be the sole manifestation or occur along with the underlying disease process. The best described forms of connective tissue panniculitis are lupus erythematosus panniculitis (LEP) and lupus profundus, panniculitis associated with dermatomyositis, and morphea and scleroderma associated panniculitis. These processes cause significant morbidity, such as deep atrophic scars, cosmetic disfigurement and psychiatric sequelae. Due to the location of the inflammation in the subcutaneous adipose layer, topical therapies may not penetrate enough to be effective, and systemic agents are required. Despite the large number of reported cases and therapies, recommendations for treatment are based largely on case series and expert opinion due to a lack of controlled therapeutic trials. All treatments are off-label in the United States. The lack of validated clinical outcome measures makes systematic and controlled studies difficult. Nonetheless further investigation into the most effective therapies for these conditions are needed.

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Lipomembranous fat necrosis in a patient with mixed connective tissue disease

Cited by 6 publications

References 4 publications

Breast involvement in mixed connective tissue disease

Breast involvement in mixed connective tissue disease

Lipomembranous fat necrosis: A distinctive and unique morphology (Review)

Update on management of connective tissue panniculitides

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