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PubMed1975

Systemic lupus erythematosus presenting as panniculitis (lupus profundus).

Six patients are described in whom panniculitis was a major manifestation of systemic lupus erythematosus. These patients were seen in a combined-clinics population of 270 patients with systemic lupus erythematosus for an incidence of approximately 2%. Panniculitis was the first symptom of systemic lupus erythematosus in three of these patients indicating that systemic lupus erythematosus should be considered as an underlying cause in patients with panniculitis or Weber-Christian's disease. Analysis of these six cases and those previously reported suggests that the addition of hydroxychloroquine to the treatment regimen may be beneficial in lupus panniculitis.

Adolescent↗
PubMedDate not supplied

Cytophagic histiocytic panniculitis: a diagnostic dilemma.

Cytophagic histiocytic panniculitis (CHP) is a recently recognized entity that frequently poses a perplexing diagnostic problem. Although the classical case presents with a relapsing fever, subcutaneous nodules, pancytopenia and liver dysfunction, most patients have in addition a multitude of other manifestations which confuse the clinical picture. Notwithstanding the variable clinical course, the disease frequently terminates in fatal hemorrhage. Diagnosis is based on histological features. A lobular panniculitis with an infiltrate of cytologically benign cytophagocytic histiocytes in skin nodules is the sine qua non of CHP. Hence, a deep skin biopsy which includes subcutaneous fat is mandatory to establish the diagnosis. Published information regarding this newly described entity remains scarce and we report two cases of CHP, one occurring in a 30-year-old Kadazan man and another in a 17-year-old Chinese woman seen at the University Hospital, Kuala Lumpur. The latter case presented with exudative ascites, an unusual feature, possibly due to intra-abdominal panniculitis. In addition, we record the development of cirrhosis in the same patient.

Adolescent↗
PubMedDate not supplied

Periduodenal panniculitis due to spontaneous rupture of a pancreatic pseudocyst into the duodenum.

We report a patient with transient periduodenal panniculitis due to spontaneous rupture of a pancreatic pseudocyst into the duodenum. He developed sudden onset of severe epigastric and back pain with jaundice, mimicking the symptoms of acute pancreatitis. However, the serum and urinary amylase levels did not increase. CT scans showed disappearance of his pseudocyst and periduodenal panniculitis without any evidence of acute pancreatitis. The CT findings of periduodenal panniculitis and his symptoms both improved within 3 weeks. A duodenal fistula leading to the remnant pseudocyst and narrowing of the periduodenal portion of the common bile duct were demonstrated by endoscopic retrograde cholangiopancreatography.

Aged↗
PubMed1998

Pancreatitis associated with pleural-mediastinal pseudocyst, panniculitis and polyarthritis.

We describe two patients with pancreatitis. One patient had acute pancreatitis of biliary origin and presented with small joint polyarthritis and panniculitis lesions. The other patient was originally hospitalised for dyspnoea with bilateral pleural effusion, and subsequently developed migratory polyarthritis. During his hospital stay he developed panniculitis lesions and a monoclonal IgG disorder of unknown significance. Very few patients with pancreatitis develop polyarthritis and panniculitis. The appearance of pseudocysts in the pleural and mediastinal cavity in the course of pancreatitis is an infrequent complication.

Adult↗
PubMed2000

Panniculitis, pancreatitis and very severe aplastic anemia in childhood: a challenge to treat.

In childhood, aplastic anemia is a rare disease of unknown etiology. Besides toxic effects, also an infectious or an autoimmune origin are discussed. We report on an 8-year-old boy with very severe aplastic anemia (VSAA) who developed pancreatitis together with panniculitis. Initially, active cytomegalovirus (CMV) infection was thought to be possibly contributive. Ganciclovir was tried resulting in clearance of CMV, but VSAA persisted. Two months after the onset of VSAA, oligosymptomatic pancreatitis was observed together with the onset of severe febrile panniculitis, occurring with multiple painful enlarged subcutaneous infiltrates of up to 7 cm in diameter. Treatment according to the Severe Aplastic Anemia-94 (SAA-94) protocol consisting of glucocorticoids, cyclosporin A (CsA), anti-thymocyte globulin and granulocyte colony-stimulating factor was instituted. Since this treatment did not lead to remission after day 110, escalation of the CsA dose up to 8 mg/kg body weight was tried. This regimen resulted in complete recovery of panniculitis and symptoms of pancreatitis. Incomplete hematological remission was reached and, to date, the patient has not required transfusions for 6 months. Because this boy suffered simultaneously from three rare disorders, which all responded to intense immunosuppression, this observation may underline common autoimmune mechanisms of these distinct diseases.

Anemia, Aplastic↗
PubMed2004

Mesenteric panniculitis: US and CT features.

Mesenteric panniculitis is an aseptic inflammation of mesenteric fat. Before the age of ultrasonography (US) and computed tomography (CT), mesenteric panniculitis was rarely diagnosed, but today the disorder is more commonly encountered, often as an incidental imaging finding. Its exact cause remains unknown. This review illustrates the characteristic US and CT features of mesenteric panniculitis and discusses its differential diagnosis.

Acute Disease↗
PubMed2006

Subcutaneous panniculitis-like T-cell lymphoma presenting with hemophagocytic lymphohistiocytosis and skin lesions with characteristic high-resolution ultrasonographic findings.

Subcutaneous panniculitis-like T-cell lymphoma (SPTCL) is an unusual type of skin lymphoma, characterized by subcutaneous soft tissue infiltration with pleomorphic T-cells and benign macrophages that mimic panniculitis. Hemophagocytic lymphohistiocytosis is a rare but potentially fatal disorder which is thought to result from uncontrolled activation and proliferation of T-cells and excessive activation of macrophages. Hemophagocytic lymphohistiocytosis have been reported as the presenting feature in 37% of SPTCL patients. High-resolution ultrasonographic appearance of SPTCL with a characteristic finding has been described in only one patient. We report a 28-year-old woman with hemophagocytic lymphohistiocytosis presented with nonremitting fever, thrombocytopenia, hypofibrinogenemia, hyperferritinemia, increased serum levels of soluble interleukin (IL)-2 receptors and hemophagocytosis in bone marrow examination. Markedly elevated level of serum IL-18 was also demonstrated in our patient. The fever subsided and the ferritin level declined to normal after treatment with oral steroid and etoposide. No evidence of hemophagocytosis was revealed in a follow-up bone marrow examination. However, nonremitting high fever and panniculitis-like skin lesions over her back, arm, lower abdominal wall, and bilateral pretibial regions developed 1 month later. A high-resolution ultrasonography revealed thickening of the subcutaneous fat layer with homogeneous hyperechogenicity pattern and poor-defined margin over the skin lesions. An ultrasound-guided excision biopsy of the nodular lesion over left upper arm was performed and the histopathology showed neoplastic cells with hyperchromatic nucleoli, which extended from deep dermis to subcutaneous fat. The immunochemical stain showed a T-cell lineage of tumor cells. SPTCL was diagnosed and the skin lesions subsided gradually after treatment with anthracycline-based combination chemotherapy.

Adult↗
PubMed1995

Edematous, scarring vasculitic panniculitis: a new multisystemic disease with malignant potential.

BACKGROUND: Hydroa vacciniforme (HV) is a disease of unknown origin characterized by erythema, vesicles, necrosis, and varicelliform scars in light-exposed skin. Systemic involvement is absent. A few patients have been reported with "severe HV" with systemic involvement, development of non-Hodgkin's lymphoma, and a poor prognosis. OBJECTIVE: Our purpose was to characterize and differentiate our patients' disease from HV. METHODS: We performed a retrospective clinicopathologic study of 14 children previously diagnosed as having "severe HV." RESULTS: The extension and severity of the cutaneous lesions, fever, wasting, failure to thrive, hepatosplenomegaly, vasculitis, panniculitis, and potential development of lymphoma are features that clearly differentiate edematous scarring vasculitic panniculitis from HV. CONCLUSION: Edematous scarring vasculitic panniculitis is a novel multisystemic disease with malignant potential that is not related to classic HV.

Adolescent↗
PubMed1995

Pancreatic panniculitis.

BACKGROUND: Pancreatic panniculitis is a rare disease in which necrosis of fat in the panniculus and other distant foci occurs in the setting of pancreatic disease. OBJECTIVE: Our purpose was to study the clinical and histopathologic features of this disease. METHODS: We conducted a retrospective review of 11 patients. RESULTS: In five patients subcutaneous fat necrosis preceded the diagnosis of pancreatic disease by an average of 13 weeks. Five of our patients had underlying pancreatitis, and the remainder had carcinoma of the pancreas. Joint manifestations were prominent in six patients. Intestinal submucosal fat necrosis developed in two cases and caused massive gastrointestinal bleeding in one. Acute lesions demonstrated focal fat necrosis with "ghost cells," pathognomonic of this condition. Chronic lesions showed granulomatous and lipophagic panniculitis, with rare areas of basophilic fat necrosis. Cholecystectomy for gallstone pancreatitis was curative in two patients. CONCLUSION: Pancreatic panniculitis has distinctive clinical and pathognomonic histopathologic findings that can be the presenting features of pancreatic disease.

Acute Disease↗
PubMed2005

[Mesenteric panniculitis].

Mesenteric panniculitis also named retractile mesenterite is a rare tumor-like lesion that thickens and shortens the mesentery. It is characterized by the association of inflammation, necrosis or fibrosis involving the adipose tissue of the bowel mesentery. The pathophysiology of this disease remains unclear despite associations with inflammatory diseases or malignancies, especially lymphomas that have been described. When symptomatic, patients may present with abdominal pain, palpable abdominal mass or intestinal obstruction. The disease remains asymptomatic in 30 to 50% of cases. Abdominal CT plays an important role in suggesting the diagnosis and can be useful in distinguishing the several conditions that can mimic mesenteric panniculitis. Nevertheless, pathologic examination of surgical excisional biopsies or sometimes percutaneous biopsies remains necessary to confirm the diagnosis and exclude an underlying infection or malignancy. Medical treatment may consist of therapy with anti inflammatory or immunosuppressive agents and can be proposed in highly symptomatic diseases. Surgical treatment should be exclusively attempted when intestinal obstruction or ischemia occur. Most of the time, it consists in intestinal derivation or segmental resection because complete excision of the lesions is often not possible. Mesenteric panniculitis usually has an uneventful clinical course and resolves spontaneously in a variable delay.

Diagnosis, Differential↗
PubMed1993

Perinephric panniculitis.

Panniculitis is an inflammatory condition of adipose tissue that is mainly seen subcutaneously or intra-abdominally. There have been rare case reports of retroperitoneal involvement but no reported cases of perinephric panniculitis. To our knowledge we report the first documented case of perinephric panniculitis.

Humans↗
PubMed2000

Recurrent eosinophilic panniculitis associated with Fasciola hepatica infection.

Eosinophilic panniculitis is characterized by a prominent infiltration of subcutaneous fat with eosinophils. We report a case of Fasciola hepatica infection presenting with eosinophilic panniculitis successfully treated with bithionol. To our knowledge, this is the first report of recurrent eosinophilic panniculitis associated with Fasciola hepatica infection.

Animals↗
PubMed1989

Clinical and pathologic correlations in 96 patients with panniculitis, including 15 patients with deficient levels of alpha 1-antitrypsin.

alpha 1-Antitrypsin levels were determined for 96 patients with various forms of biopsy-proved panniculitis. Fifteen of the 96 patients had alpha 1-antitrypsin deficiency, and 12 of these also had an abnormal alpha 1-antitrypsin phenotype. This group of patients showed substantial and characteristic clinical and histopathologic differences from the group of patients with normal levels and phenotypes of alpha 1-antitrypsin. Spontaneous ulceration and drainage of panniculitis lesions were much more common in patients with alpha 1-antitrypsin deficiency, and biopsy specimens from lesions of panniculitis in these patients were much more likely to show large areas of normal fat adjacent to necrotic lobular and septal areas, which contained many polymorphonuclear leukocytes and histiocytes. Destruction of elastic tissue was more frequent and extensive in patients with alpha 1-antitrypsin deficiency.

Female↗
PubMed1997

Blind loop syndrome: an unusual cause of panniculitis.

We describe a 52-year-old woman with panniculitis and blind loop syndrome. She had undergone a gastrectomy for peptic ulcer 4 years before. Tender erythematous nodules on her palms and soles were associated with diarrhea and weight loss. A biopsy specimen revealed septal and lobular panniculitis. A glucose hydrogen breath test was consistent with bacterial overgrowth. These results were consistent with panniculitis associated with a blind loop syndrome. Only four cases of this association have been reported previously.

Animals↗
PubMed1993

[Relapsing polychondritis and mesenteric panniculitis: apropos of 2 cases].

We report two cases of polychondritis associated with mesenteric panniculitis. Case 1. In February 1989, a woman born in 1949 presented with 40 degrees C fever accompanied by pain in the abdomen and pelvis. Eight days later, nodular skin lesions appeared on her lower limbs. The abdomen was swollen with gas and undepressible. An abdominal CT scan revealed partitioned peritoneal collections, and a guided needle aspiration produced a chylous fluid. Direct and indirect bacteriological examinations gave negative results. Histology showed intense inflammatory reaction with giant cells and lipophages, thereby confirming the presence of mesenteric panniculitis. Six months later, the development of chondritic lesions on the nose and the helix of the ear clinched the diagnosis of polychondritis. The patient was put on corticosteroid therapy for a few months, and in January this year (1993) she is durably asymptomatic. Case 2. In October 1977, a woman born in 1937 presented with polychondritis with prolonged fever, inflammatory syndrome and chondritic lesions of the nose, larynx and helix of the ear. In December 1978, she developed signs of abdominal obstruction. Laparotomy revealed infiltration by multiple nodular formations of the entire posterior line of attachment of the mesentery. Biopsies withdrew a puriform fluid. Histology showed a partly necrotic adipose tissue with giant cells and lipophages. High-dose corticosteroid therapy partially controlled the chondritic and abdominal manifestations. The occurrence of abdominal pain in patients with polychondritis may result from several disorders, such as iatrogenic complications, digestive tract vasculitis or ulcerative colitis, but also associated mesenteric panniculitis.

Abdomen, Acute↗
PubMed2000

A fatal case of pancreatic panniculitis presenting in a young patient with systemic lupus.

BACKGROUND: Subcutaneous fat necrosis associated with pancreatic disease is a rare event. The clinical cutaneous findings are non-specific erythematous nodules with central softening located predominantly on the lower extremities. The histopathologic features of these lesions are very characteristic and diagnostic. METHODS: We present an unusual case of pancreatic panniculitis associated with lupus pancreatitis in a 21-year-old African American female. The patient presented with lower extremity skin nodules, arthralgia, and serositis prior to the diagnosis of systemic lupus and pancreatitis. The skin lesions progressed despite normalization of serum pancreatic enzymes. Following femoral vein catheterization for renal dialysis, she developed a large indurated area over the left lower quadrant, flank, groin, and upper thigh measuring 25 cm. She was treated with repeated debridement, tissue grafts, and hyperbaric oxygen because of a clinical suspicion of necrotizing fasciitis. RESULTS: Examination of skin biopsies and debrided tissue revealed the pathognomonic features of pancreatic panniculitis without any evidence of necrotizing fasciitis. Organisms were not detected by tissue examination or microbiologic cultures. CONCLUSIONS: This case illustrates the potential role of vascular trauma in the pathogenesis of pancreatic panniculitis.

Adipose Tissue↗
PubMed2001

Lupus profundus, indeterminate lymphocytic lobular panniculitis and subcutaneous T-cell lymphoma: a spectrum of subcuticular T-cell lymphoid dyscrasia.

INTRODUCTION: The diagnosis and classification of lymphocytic lobular panniculitis (LLP) has historically proven to be a difficult challenge. We encountered 32 cases of primary LLP which could be categorized as: 1) lupus erythematosus profundus (LEP) (19 patients); 2) an indeterminate group termed indeterminate lymphocytic lobular panniculitis (ILLP) (6 patients); and 3) subcutaneous T-cell lymphoma (SCTCL) (7 patients). OBJECTIVE: We attempted to better define the subtypes of LLP by morphologic, phenotypic and genotypic features and to correlate those features to clinical presentation and outcome. METHOD: Skin biopsy material was studied by conventional light microscopy, through immunophenotyping performed on sections from paraffin-embedded, formalin-fixed tissue and in some cases on sections of tissue frozen after receipt in physiological (Michel's) medium, and by polymerase chain reaction single-stranded conformational polymorphism analysis to assess for clonality of T-lymphocytes. Clinical features were correlated to histologic, phenotypic, and genotypic analyses. RESULTS: Patients with LEP had a prior diagnosis of LE or overlying skin changes which light microscopically were characteristic of LE. Patients with ILLP had no concurrent or prior history of LE, no systemic symptoms or cytopenias, and a clinical course not suggestive of lymphoma. Cases of SCTCL showed hemophagocytic syndrome and/or lesional progression with demise attributable to the disease. Lesions in all groups showed proximal extremity predilection. Females predominated in the LEP group. The average age of onset was 38, 40 and 55 years in the LEP, ILLP and SCTCL groups, respectively. Cytopenia was seen in 4 LEP patients; 1 also developed fever. In LEP and ILLP, lesions resolved with hydroxychloroquine and/or steroid therapy, with recurrences following cessation of therapy. In the SCTCL group 4 developed hemophagocytic syndrome, 4 died within 2 years of diagnosis, and 3 went into remission following chemotherapy. The LEP and SCTCL groups manifested histological similarities: dense perieccrine and lobular lymphocytic infiltration, lymphoid atypia, histiocytes with ingested debris, eosinophilic necrosis of the fat lobule and thrombosis. The atypical lymphocytes although pleomorphic did not have a cerebriform morphology. The infiltrate in ILLP had a similar cytomorphology and distribution with variable angioinvasion which in all save one case was of lesser intensity and was not associated with significant fat necrosis or vasculitis. Germinal centers, dermal/subcuticular mucin deposition and an atrophying interface dermatitis with hyperkeratosis and follicular plugging were largely confined to the LEP group. Erythrophagocytosis, characteristic of SCTCL, usually indicated a supervening subcuticular lymphoid dyscrasia when encountered in ILLP and LEP. SCTCL showed a selective loss of CD5 expression with or without diminution in CD7 and monoclonal CD3 expression. Of 4 cases studied, 3 showed a CD8 dominant infiltrate while 2 others exhibited CD56 and CD30 positivity, respectively. All cases of SCTCL with amplifiable DNA showed T-cell clonality. Similar molecular and phenotypic features indicative of subcuticular lymphoid dyscrasia were encountered in cases of LEP and ILLP including a reduction in CD5, CD7, and/or monoclonal CD3 expression, a preponderance of CD8 lymphocytes within the subcutaneous fat and T-cell clonality. These cases showed lymphoid atypia with variable erythrophagocytosis. Cases of phenotypically abnormal and/or clonal LEP showed one or more of local destruction, lesional size progression, fever, and cytopenias, but lesions responded to hydroxychloroquine and/or prednisone therapy and death attributable to panniculitis could not be documented. Cases that were phenotypically normal and without clonality had none of the aforesaid atypical clinical features. CONCLUSION: Lymphoid atypia, erythrophagocytosis, loss of certain pan T-cell markers, a reduced CD4/8 ratio and TCR rearrangement define subcuticular T-cell lymphoid dyscrasia, including a subset of LEP and ILLP. The subcuticular lymphoid infiltrates represent a spectrum of histologic, immunophenotypic, and molecular abnormalities which range from those which are clearly benign to those which are clearly neoplastic, and also encompasses those cases which defy precise classification into the two aforesaid poles.

Adult↗