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At least 145 records · Page 8Linked to original sources
PubMed2002

Panniculitis-like T-cell lymphoma clinically manifested as alopecia.

A 45-year-old women presented with multifocal scalp lesions with the clinical impression of alopecia areata. Histological findings first suggested cytophagic histiocytic panniculitis, although a 'burned-out' panniculitis-like T-cell lymphoma could not be excluded. After a 20-month follow-up period, assessment of the T-cell receptor gamma-chain gene rearrangement verified the diagnosis of subcutaneous panniculitis-like T-cell lymphoma. This case is interesting because of its isolated scalp manifestation as well as its indolent course.

Alopecia Areata↗
PubMed1999

A case of natural killer/T cell lymphoma of the subcutis resembling subcutaneous panniculitis-like T cell lymphoma.

A case of nasal type natural killer (NK)/T cell lymphoma of the subcutis showing clinical and morphological features that resemble subcutaneous panniculitis-like T cell lymphoma (SPTCL) is presented. A 73-year-old man presented with swelling of the left arm and was diagnosed with panniculitis by a dermatologist. It was concluded from a skin biopsy specimen that the patient had non-Hodgkin's lymphoma of the large cell, NK/T cell type because the neoplastic cells showed polyclonal CD3 immunoreactivity. Treatment with interferon-gamma was initiated, but the patient died of disseminated intravascular coagulation and multiple organ failure 2 months after the initial symptoms appeared. However, involvement of additional organs by the lymphoma was not apparent clinically. An autopsy was not performed. A routinely stained section of the biopsy skin specimen revealed massive necrosis of the subcutaneous fat, karyorrhexis admixed with reactive histiocytes, and large atypical lymphoid cells. Immunoreactivity for polyclonal CD3 was present in the perinuclear region, but absent in the neoplastic cell membranes. CD56, CD45RO (UCHL-1), CD43 (MT1), CD45 (leukocyte common antigen), and the cytotoxic molecules perforin, granzyme B and TIA-1 were positive, but CD20 (L26), CD4, CD8, and betaF1 were negative. Epstein-Barr virus (EBV) mRNA was detected in the nuclei of neoplastic cells by in situ hybridization. Subcutaneous panniculitis-like T cell lymphoma is reported to be an EBV-negative, clonal T cell neoplasm. Although this case showed clinical and morphological features that resembled SPTCL, perinuclear polyclonal CD3 staining and membranous CD56 reactivity seen in neoplastic cells were suggestive of NK cells. Furthermore, the neoplastic cells were positive for EBV. This case is considered to be a NK/T cell lymphoma of the subcutis resembling SPTCL. It is believed that it is important to recognize such a tumor because patients may undergo a fulminant clinical course, despite the tumor being localized in the subcutaneous adipose tissue.

Aged↗
PubMed2000

A case of cytophagic histiocytic panniculitis associated with exertional rhabdomyolysis.

An 18-year-old man who suffered from panniculitis involving the entire left lower limb after exertional rhabdomyolysis is reported. A high fever (>39 degrees C) and leukocytosis (>20,000/microL) persisted for 1 week, and his general status deteriorated rapidly into pre-disseminated intravascular coagulation, complicated by pleural effusion and prolonged clotting time. His condition was dramatically improved by steroid pulse therapy and he has remained in good health for the 20 months since discharge. Histologic examination of subcutaneous tissue from the swollen left lower limb revealed pleomorphic small, medium or large lymphocytes, macrophages and neutrophils infiltrating the edematous subcutaneous adipose tissue in a lobular panniculitis-like pattern. The majority of inflammatory cells were T lymphocytes, with equal proportions of CD4+ and CD8+ cells. As polymerase chain reaction did not show bands suggesting T cell receptor gamma gene rearrangement, the proliferation of T lymphocytes was considered to be polyclonal. The T lymphocytes also expressed Fas ligand, suggesting the involvement of Fas-mediated cytotoxicity. This case may represent a new category of cytophagic histiocytic panniculitis induced by exertional rhabdomyolysis.

Adolescent↗
PubMed2000

Acute Crohn's colitis with lobular panniculitis--metastatic Crohn's?

One of the common lesions in Crohn's colitis is erythema nodosum, a septal panniculitis that may appear before diagnosis or in conjunction with flare up. We report a case of Crohn's colitis where the presenting sign was neutrophilic lobular panniculitis with few granulomas. The possibility that this case represents a forme fruste of metastatic Crohn's is suggested. As skin biopsies are not usually performed by gastroenterologists in erythema nodosum-like skin lesions in Crohn's patients it may well be that the incidence of lobular panniculitis is higher than reported in the literature.

Aged↗
PubMed2001

Monitoring of phagocytic activity in histiocytic cytophagic panniculitis.

Histiocytic cytophagic panniculitis presents with subcutaneous panniculitis. Histologically, it is characterized by phagocytosis of blood cells in the subcutaneous tissue and bone marrow. One patient with histiocytic cytophagic panniculitis is described in whom hemophagocytosis macrophages and histiocytes was observed histologically and was confirmed in vitro measuring phagocytosis by peripheral blood monocytes by means of chemiluminescence. In vitro measurements of phagocytosis corresponded well with the clinical course. Chemiluminescence for measuring phagocytosis in vitro may be suitable for analyzing disease activity and for testing therapeutic compounds in vitro.

Adult↗
PubMed1987

Gout presenting as lobular panniculitis.

A 37-year-old man presented with panniculitis of the legs that, on biopsy, showed aggregates of crystals whose appearance suggested sodium urate. Based on this finding, a uric acid level was obtained which proved to be markedly elevated. The patient subsequently developed gouty arthritis. Gout should be added to the differential diagnosis of causes of panniculitis, and biopsy specimens of panniculitis should be carefully examined for crystals.

Adult↗
PubMed2000

Panniculitis: definition of terms and diagnostic strategy.

Inflammation of the subcutaneous tissue represents a dynamic process that shows different histopathologic findings at different stages of development; therefore, the stage of evolution of a lesion at the time of biopsy influences the microscopic appearance significantly. Furthermore, location and type of inflammation may vary among different examples of the same panniculitis independent of the stage of evolution. For these reasons, the histopathologic diagnosis of panniculitides is often difficult. Currently, the most common approach to diagnosis is differentiation between predominantly septal and predominantly lobular panniculitis, followed by the distinction between lesions with and without vasculitis. Although these criteria are important for diagnosis, they are often insufficiently specific. To determine an alternative method of diagnosis, 329 cases of panniculitis were histopathologically analyzed using the following parameters: location and type of inflammatory infiltrate within and around the subcutaneous tissue, presence or absence of fat necrosis, type of necrosis, presence or absence of vascular changes, and presence or absence of associated findings (e.g., hemorrhage, sclerosis). On the basis of the results of this study and of an extensive review of the literature, tables of histopathologic findings for the diagnosis of panniculitides are presented.

Humans↗
PubMed1994

Successful treatment of cytophagic histiocytic panniculitis with modified CHOP-E. Cyclophosphamide, adriamycin, vincristine, predonisone, and etoposide.

A 27-year-old woman developed generalized subcutaneous painful nodules, fever, abnormal liver function, a bleeding tendency, and pancytopenia. Skin biopsies revealed the lobular panniculitis with a morphologically benign histiocytic infiltration with prominent phagocytosis. Leukophagocytosis and erythrophagocytosis were also present in the bone marrow. The diagnosis of cytophagic histiocytic panniculitis was made. The patient received polychemotherapy with cyclophosphamide, Adriamycin, and vincristine on day 1, prednisone on day 1-5 (modified CHOP), with the addition of etoposide (E). This regimen was repeated 8 times every 3 weeks. The patient obtained a complete clinical remission that has lasted almost 2 years after the completion of chemotherapy. Thus we suggest modified CHOP-E chemotherapy for an effective treatment for the aggressive form of cytophagic histiocytic panniculitis.

Adult↗
PubMed1998

Subcutaneous panniculitis-like T-cell lymphoma: clinicopathologic, immunophenotypic, and genotypic analysis of alpha/beta and gamma/delta subtypes.

Subcutaneous panniculitis-like T-cell lymphoma (SPTCL) is an uncommon cutaneous lymphoma that has been proposed as a distinct clinicopathologic entity, but studies of SPTCL are limited. We studied the clinicopathologic, immunophenotypic, and genetic features of 11 SPTCLs. All cases had a variable admixture of pleomorphic small, medium, or large lymphocytes and histiocytes infiltrating the subcutis in a lobular panniculitis-like pattern. A granulomatous reaction was seen in three cases and erythrophagocytosis in four. Karyorrhexis and fat necrosis were present in all cases. Angioinvasion was seen in seven SPTCLs; four had areas of coagulation necrosis. All cases expressed T-cell-associated antigens (CD3epsilon, CD45RO, or CD43) and T-cell receptors (TCR); nine expressed alphabeta TCRs and two expressed gammadelta TCRs. T-cell receptor-gamma, TCRbeta, or TCRdelta genes were clonally rearranged in 8 of 10 cases studied. Both gammadelta SPTCLs expressed Vdelta2+ TCRs and were CD4-, CD8- and CD56+. CD56 was negative in seven of nine alphabeta SPTCLs and inconclusive in the other two. Six of nine alphabeta SPTCLs were CD8+; the CD4/CD8 phenotypes were indeterminate in the other three. Cytolytic granule-associated proteins were expressed by all SPTCLs (11 of 11 were TIA-1+, 4 of 4 were perforin+). In situ hybridization for Epstein-Barr virus-encoded RNA (EBER-1) was negative in all cases. Most patients responded to systemic chemotherapy or local radiation therapy. Seven patients are alive: four without disease (19-73 months) and three with disease (32-72 months); four died: three of disease (3-25 months) and one without disease (42 months). We conclude that SPTCLs are clonal, EBV-, cytotoxic T-cell lymphomas derived from alphabeta T-cells or gammadelta T-cells. The gammadelta SPTCLs appear to be preferentially derived from the Vdelta2+ subset. Subcutaneous panniculitis-like T-cell lymphoma may be rapidly fatal or indolent; local therapy may be appropriate for some patients.

Adolescent↗
PubMed1997

alpha 1-antitrypsin deficiency-associated panniculitis: resolution with intravenous alpha 1-antitrypsin administration and liver transplantation.

Panniculitis is a rare complication of alpha 1-antitrypsin (A1AT) deficiency that is characterized by acute inflammatory infiltrate and fat necrosis. Different treatment strategies are used to provide symptomatic relief. Here we describe two patients with homozygous A1AT deficiency who developed panniculitis and were successfully treated with A1AT replacement. The patient who received a liver transplant experienced complete resolution of the skin lesions. The patient who received A1AT intravenously showed complete response, but the skin lesions recurred when the levels of A1AT fell below 50 mg/100 ml. Panniculitis secondary to A1AT deficiency can be successfully treated with liver transplantation or intravenous infusion of A1AT.

Female↗
PubMed2004

Atypical lymphocytic lobular panniculitis.

BACKGROUND: Although subcutaneous T-cell lymphoma (SCTCL) is considered an aggressive form of lymphoma, some patients manifest a long waxing and waning phase unaccompanied by constitutional symptoms. METHODS: Twelve patients were prospectively encountered, presenting with a lymphocytic panniculitis accompanied by lymphoid atypia, although not fulfilling criteria for SCTCL. Clinical, histologic, phenotypic, and genotypic analyses were conducted. RESULTS: There were five men, one boy, and six women; none had symptoms compatible with lupus erythematosus or aggressive SCTCL. All but two had a waxing and waning course of years. Four patients had periodic cytopenias accompanied by fevers. While responding somewhat to prednisone, the lesions relapsed. In one patient, treatment with alemtuzumab (CAMPATH-1) led to complete lesional resolution with no recurrence. Light microscopy showed expansion of the interstices of the fat lobule by mildly atypical lymphocytes of the CD4 subset in 10 biopsies from eight patients; in the other four patients, there was an increase in CD8 lymphocytes. There was diminished expression of CD5 and/or CD7 in the majority of biopsies. Ten of 13 biopsies showed clonal T-cell receptor-gamma rearrangements. CONCLUSIONS: We apply the term atypical lymphocytic lobular panniculitis to this distinctive form of lymphocytic panniculitis manifesting this light microscopic, phenotypic, and genotypic profile.

Adipose Tissue↗
PubMed2005

Lupus erythematosus panniculitis (lupus profundus): clinical, histopathological, and molecular analysis of nine cases.

BACKGROUND: The diagnosis of lupus erythematosus panniculitis (LEP) may be very difficult in cases in which involvement of the subcutaneous fat is the only manifestation of the disease. The main differential diagnosis is subcutaneous panniculitis-like T-cell lymphoma (SPTCL). METHODS: We performed a retrospective study reviewing the histopathologic features of 11 biopsy specimens from nine patients with LEP (M : F = 2 : 7; median age: 48 years; range: 20-71 years). RESULTS: Histopathologically, all biopsies revealed a lobular panniculitis, with concomitant septal involvement in 82% of them. Dermal changes included the presence of superficial and deep infiltrates (82%) and mucin deposition (73%). The majority of cases (73%) presented also some form of epidermal involvement. The subcutaneous infiltrate was composed of lymphocytes in all cases, admixed with plasma cells in 91% of cases. Lymphoid follicles with reactive germinal centers were detected in 45% of cases. Immunohistochemistry showed a predominance of alpha/beta-T-helper and cytotoxic lymphocytes in 80% of cases admixed with B lymphocytes. The polymerase chain reaction analysis of the T-cell receptor (TCR)-gamma gene showed a polyclonal smear in all cases. CONCLUSIONS: Our study shows that the most useful histopathologic criteria for distinguishing LEP from SPTCL are the presence of involvement of the epidermis, lymphoid follicles with reactive germinal centers, mixed cell infiltrate with prominent plasma cells, clusters of B lymphocytes, and polyclonal TCR-gamma gene rearrangement.

Adult↗
PubMed1985

Panniculitis associated with cutaneous T-cell lymphoma and cytophagocytic histiocytosis.

A 36-year-old woman had a 6-year history of recurrent panniculitis with development of an angiocentric and angiodestructive cutaneous T-cell lymphoma (CTCL) of the helper cell phenotype. She subsequently developed a rapidly progressive fatal syndrome characterized by cytophagocytic histiocytosis and hyperlipidaemia. Cytophagocytic histiocytosis has previously been reported in association with panniculitis, malignancy and infection, but not with CTCL and the precise relationship between panniculitis, CTCL, cytophagocytic histiocytosis and hyperlipidemia is unclear.

Adult↗
PubMed2004

Whipple's disease presenting with symmetrical panniculitis.

Specific cutaneous involvement in Whipple's disease is extremely rare. The condition usually runs a chronic course, with symptoms preceding diagnosis by years or even decades. We report a 44-year-old man who presented with a rapid onset of progressive, extensive, symmetrical plaques of panniculitis affecting the inner thighs and forearms. He had accompanying large joint arthritis and was profoundly anaemic. Biopsy of the subcutis revealed a florid septal panniculitis with infiltration of the septa by foamy macrophages containing intracellular granules that stained strongly with periodic acid-Schiff reagent. A similar but more intense infiltrate was seen in the small bowel lamina propria, and a diagnosis of Whipple's disease was made. Symmetrical panniculitis has not previously been reported as a manifestation of Whipple's disease.

Adult↗
PubMed2005

Calcifying panniculitis following subcutaneous injections of nadroparin-calcium in a patient with osteomalacia.

Calcifying panniculitis is a rare form of calcinosis cutis belonging to the spectrum of calciphylaxis that has almost invariably been described in patients with severe renal disturbances. We report a patient with osteomalacia without chronic renal failure, who developed calcifying panniculitis following subcutaneous administration of nadroparin-calcium. Light microscopy studies of biopsy specimens revealed multiple foci of microcalcification within the adipose lobules, in the interadipocyte spaces, in connective tissue septa and in the media of small arteries in the subcutis. The patient had an elevated level of intact parathyroid hormone, whereas the calcium-phosphorus product was normal. The lesions slowly resolved upon discontinuation of nadroparin. We conclude that calcifying panniculitis is a rare complication associated with the subcutaneous administration of nadroparin-calcium that may rarely also occur in the absence of renal disturbances. Low molecular weight calcium-containing heparins should probably be used with caution in the presence of hyperparathyroidism.

Anticoagulants↗
PubMed1993

Systemic nodular panniculitis in a patient with alpha-1 antitrypsin deficiency (PiSS phenotype).

The association of alpha-1 antitrypsin deficiency (PiZ phenotype) with systemic nodular panniculitis has been well documented. Despite reports of cases of systemic nodular panniculitis associated with other alpha-1 antitrypsin deficiency phenotypes, it is still not known if this association is fortuitous rather than causal. We report a case of systemic nodular panniculitis associated with alpha-1 antitrypsin deficiency (PiSS phenotype), with clinico-pathological features similar to those reported in cases associated with the PiZ phenotype.

Adult↗
PubMed2005

Factitious panniculitis masquerading as pyoderma gangrenosum.

We report a case of factitious panniculitis masquerading as florid pyoderma gangrenosum in a 35-year-old woman. At presentation, she had tender, ecchymotic plaques over the lower trunk and limbs, and several biopsies showed active lobular panniculitis. However, the extensive ulceration that ensued was clinically persuasive for pyoderma gangrenosum. We elected to treat the inflammatory element symptomatically with a range of topical and systemic medications including clobetasol propionate, tacrolimus 0.1% ointment, prednisolone, dapsone, cyclosporin A and mycophenolate mofetil, none of which effected an improvement. The possibility of a factitious aetiology had been suspected from the outset, and when signs of clinical depression emerged, antidepressant therapy was initiated and the ulcers were encased in fibreglass casts. Within a short period, healing commenced and slowly progressed with scar formation. In retrospect, we consider the diagnosis to have been factitious panniculitis on the basis of strong circumstantial evidence and the disparity between the histological and clinical features.

Adult↗
PubMed2004

Unusual case of subcutaneous panniculitis-like T-cell lymphoma.

An unusual case of subcutaneous panniculitis-like T-cell lymphoma is presented involving multiple organ systems, which eventually culminated in rapid demise from the haemophagocytic syndrome, after an initial protracted course. A 44-year-old man presented in April 2001 with bronchiolitis obliterans organising pneumonia that initially responded well to corticosteroids. However, the condition relapsed on attempted prednisone withdrawal in January 2002 and the patient was noted to have developed truncal subcutaneous nodules. Initial skin biopsy revealed lobular panniculitis, with negative microbiological culture. In July 2002, mononeuritis multiplex was diagnosed after the patient presented with paresthesiae and was treated with pulse cyclophosphamide therapy. By November 2002 there was ulceration of the subcutaneous nodules. Repeat skin biopsy revealed subcutaneous panniculitis-like T-cell lymphoma. The clinical manifestations were supportive of an unifying diagnosis of malignancy involving pulmonary, cutaneous and nervous systems. Combination chemotherapy with fludarabine, mitoxantrone and dexamethasone was commenced. However, the patient deteriorated, with fevers, weight loss, pancytopenia and laboratory features consistent with the haemophagocytic syndrome. Despite maximal supportive therapy the patient succumbed to his disease.

Adult↗