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PubMed1987

Sea-blue histiocyte syndrome with cutaneous involvement. Case report with ultrastructural findings.

A patient with infiltration of the skin resulting in eyelid swelling and facial nodules was recognized as a case of sea-blue histiocyte syndrome with cutaneous involvement. Typical sea-blue histiocytes were found in the skin and confirmation was provided by electron microscopy. Hepatosplenomegaly, lung infiltrates and bone marrow involvement were the other symptoms. The relationship between sea-blue histiocyte syndrome and adult Niemann-Pick disease is also discussed.

Adult↗

PubMed1985

T-zone histiocytes in granulomatous skin diseases.

We investigated the distribution of T-zone histiocytes by immunohistochemical demonstration of S-100 protein in granulomatous skin diseases (granuloma annulare, 8 cases; necrobiosis lipoidica, rheumatoid nodule, sarcoidosis, lupus vulgaris, and foreign-body granuloma; 5 cases each). T-zone histiocytes were regularly found in the lymphohistiocytic mantle, but also occasionally between epitheloid cells. Our results show that, besides the monocyte-macrophage system, T-zone histiocytes consistently contribute to the formation of cutaneous granulomas. These findings may indicate a role of delayed-type hypersensitivity.

Foreign-Body Reaction↗

PubMed1991

Parvovirus B19 infection can induce histiocytic necrotizing lymphadenitis (Kikuchi's disease) associated with systemic lupus erythematosus.

We report three cases of systemic lupus erythematosus (SLE) associated with necrotizing histiocytic lymphadenitis (Kikuchi's disease) and immunologically proven human parvovirus B19 infection. Simultaneous occurrence of SLE and Kikuchi's disease was a characteristic of the three cases. Kikuchi's disease is an uncommon disease that usually affects young women and is characterized by painless unilateral cervical lymph-node enlargement. T-cell regions of affected lymph nodes are exclusively involved with patchy paracortical necrosis surrounded by a polymorphous cell population of histiocytes and macrophages. However, lymphadenopathy in patients with SLE may be histologically indistinguishable from Kikuchi's necrotizing lymphadenitis. The cause of Kikuchi's disease remains uncertain, although infectious agents have been proposed. A positive IgM-specific anti-human parvovirus B19 antibody test in our three cases suggests that B19 can induce a necrotizing histiocytic lymphadenitis and possibly a clinical SLE flare. High-dose (1 mg/kg/day) and medium-dose (0.5 mg/kg/day) oral prednisone was an effective treatment for constitutional and visceral symptoms of Kikuchi's and SLE diseases.

Adolescent↗

PubMed2004

Histiocytic proliferations in childhood.

Histiocytic disorders occur with increased frequency in children and young adults. The wide spectrum of neoplastic and nonneoplastic diseases characterized by histiocytic proliferation was reflected in the cases submitted to the 2003 Society for Hematopathology and the European Association for Haematopathology Workshop. This review focuses on the entities represented at the workshop. It includes an overview of the classification and immunophenotype of the histiocytic disorders. The clinical and pathologic features of each entity are illustrated with representative cases submitted to the workshop, with an emphasis on recent biologic and genetic insights. In addition, a brief review of the pathologic features of the lysosomal storage disorders is included, with an emphasis on findings relevant to the practicing hematopathologist, exemplified with cases presented at the workshop.

Child↗

PubMed1980

Liberation of a fibrogenic factor from human blood monocytes, ascites cells, cultured histiocytes and transformed mouse macrophages by treatment with SiO2.

Human monocytes and ascites macrophages from cirrhotic patients were isolated in Percoll-gradient and cultured with and without silica. Similar experiments were carried out also with cultured malignant human histiocytes and transformed mouse macrophages. The fibrogenic activity of the culture media was tested by measuring the incorporation of [3H]proline and [3H]thymidine into cultured rat granuloma and human synovial cells. Media from silica-treated monocytes, ascites macrophages and certain histiocyte and mouse macrophage lines caused an increase in the incorporation of both [3H]proline and [3H]thymidine into collagen and DNA, respectively, in both cell systems. Alkaline RNase activities were decreased markedly in the media from silica-treated ascites macrophages but not in the media of the monocytes or histiocytes.

Adult↗

PubMed2005

Fulminant multisystem non-langerhans cell histiocytic proliferation with hemophagocytosis: a variant form of Erdheim-Chester disease.

Hemophagocytosis (HP), a feature seen in malignant histiocytosis and infection- and lymphoma-associated disorders, has not been previously emphasized in Erdheim-Chester disease (ECD). Generally, ECD is recognized as a rare, systemic, non-Langerhans cell histiocytosis with a variable clinical course. Herein, we describe a unique case of multisystem non-Langerhans cell histiocytic proliferation with a fulminant clinical course (death occurred within 3 months of presentation) that showed prominent HP and extensive involvement of multiple organs, including the lungs, resulting in respiratory failure. Hemophagocytosis led to severe anemia that required transfusion and thrombocytopenia. Antemortem lung and bone marrow biopsy specimens revealed involvement by a histiocytic infiltrate with features highly suggestive of ECD and HP. Furthermore, the autopsy documented the presence of HP and the histiocytic infiltrate in multiple other organs. This case is best categorized as a variant form of ECD. Recognizing this variant has the following important implications: (1) HP may be a marker for fulminant clinical course in ECD, (2) the presence of HP does not exclude a diagnosis of ECD, and (3) ECD should be considered in the differential diagnosis of HP.

Aged↗

PubMedDate not supplied

[Histiocytic necrotizing lymphadenitis (Kikuchi-Fujimoto disease)].

INTRODUCTION: Histiocytic necrotizing lymphadenitis is a disease of unknown etiology, favourable course and outcome, mostly encountered in young women. It exhibits increased temperature and lymphadenopathy. The illness resolved spontaneously after a few months. A CASE REVIEW: In a 32-year-old patient the disease developed gradually, with pain in the right axilla, within enlarged lymph. Two weeks later, her temperature increased to 39 degrees C, accompanied by fever, shivering, malaise and loss of appetite. The ultrasonographic finding showed a conglomerate of hypoechogenic lymph nodes, with a few enlarged lymph glands. By immunohistochemical examination of a gland tissue sample the diagnosis was established Kikuchi-Fujimoto disease ("Histiocytica necrotizing lymphadenitis"--"Apoptotic lymphadenitis"). Corticosteroid therapy had been administered for one year, which resulted in both subjective improvement and regression of enlarged lymph nodes. DISCUSSION: A connection between this disease and viruses has not been proved. Presence of intracellular tubuloreticular formations suggests autoimmune etiology of histiocytic necrotizing lymphadenitis. It is considered as hyperimmune reaction of the organism induced by a viral infection. Differential diagnosis should consider taxoplasmosis, tuberculosis, lymphadenitis and malignant lymphoma. CONCLUSION: Although histiocytic necrotizing lymphadenitis is a rare disease, this case shows that it should be considered when lymphadenopathy, particularly cervical, occurs in young women, accompanied by increased temperature.

Adult↗

PubMed1975

Sea-blue histiocytes in mycosis fungoides.

A patient with tumor-stage mycosis fungoides and lymph node involvement had sea-blue (ceroid-containing) histiocytes in her bone marrow. Leukopenia, eosinophilia, and a mild type II beta-hyperlipoproteinemia were also features. Sea-blue histiocytes were demonstrated in the skin after the mycosis fungoides infiltrate had cleared with treatment. There is a possible interrelationship between the sea-blue histiocytes, the abnormal mycosis cells in the skin, and lipid metabolism.

Adult↗

PubMed2002

[Gastric interdigitating reticulum cell sarcoma: unusual presentation of a histiocytic tumor].

Histiocytic sarcoma, proliferation arising from immunoregulatory effector system cells, is a very rare and recently recognized tumor. Diagnosis is based on immunohistochemical and molecular genetic study, which allows to distinguish histiocytic sarcoma from lymphocytic proliferation, such as non-Hodgkin's lymphoma. We report the case of a gastric interdigitating reticulum cell sarcoma, treated by resection and chemotherapy. We review here clinical, histological, immunohistochemical and genotypic features of histiocytic tumors of the digestive tract.

Antigens, CD↗

PubMed2006

[The clinical and pathological characteristics of histiocytic necrotizing lymphadenitis: analysis of 52 cases].

OBJECTIVE: To study the clinical manifestation and pathological features of histiocytic necrotizing lymphadenitis (Kikuchi's disease, KD). METHODS: 52 patients with KD were collected to analyze the clinical manifestation, pathological features of biopsy lymph nodes, diagnosis and treatment. RESULTS: 41 cases (79%) were female of the 52 patients. The main clinical features included persistent fever (100%), single (23%)/multi (77%)-lymphadenopathy (always in cervical region), pleomorphism erythra (35%), neutropenia (76%), elevated erythrocyte sedimentation rate (100%), insensitivity to antibiotics (100%) and sensitivity to small dosage glucocorticoid (81%). 26 cases (50%) had elevated aspartate aminotransferase and/or alanine aminotransferase, but only 7 cases (13%) had upper respiratory tract symptom like influenza. Pathological features included distinctive necrosis, loss of lymph node structure, infiltration with histiocytes and lymphocytes, absence of neutrophils. Immunohistochemical stainings showed CD(68) positive for histiocytes and CD(3), CD(45) RO positive for T lymphocytes. CONCLUSION: Diagnosis of KD relies on the pathological examination and immunohistochemical staining.

Adolescent↗

PubMed1991

Uterine müllerian adenosarcoma with histiocytic (xanthomatous) mesenchymal component.

We present an endometrial Müllerian adenosarcoma in which the sarcomatous component showed prominent nests of foamy cells that accounted for 50% of the neoplastic mesenchyma. Such foamy cells showed occasional cytological atypias and immunohistochemical features of histiocytic (macrophagic differentiation in the absence of changes that could substantiate the presence of an inflammatory infiltration of foamy histiocytes. These facts suggest histiocytic differentiation from neoplastic mesenchymal cells. Such differentiation has been reported in association with malignant mixed mesodermal tumor, but not in Müllerian adenosarcoma.

Biomarkers, Tumor↗

PubMed1991

Histiocytic medullary reticulosis occurring with small cell lung carcinoma.

Haematophagocytic activity is a salient feature of malignant histiocytic medullary reticulosis. This potency has also been observed as an aberrant property of non-histiocytic malignant cells or as a benign histiocytic process in response to infections or malignancies. We report the case of a patient who presented simultaneously with a limited stage small cell lung carcinoma and a fatal reactive form of histiocytosis. We conclude that such a haematophagocytic syndrome may occur in the setting of small cell lung carcinoma s previously described in other malignancies.

Aged↗

PubMed1990

[A case of neurovisceral storage disease with sea-blue histiocyte and severe horizontal supranuclear ophthalmoplegia].

Neville and coauthors (1973) reported several cases of neurovisceral storage disease with vertical supranuclear gaze paresis, ataxia and other central nervous disorders. This disease is classified into Niemann-Pick disease type C because of the presence of foamy cells or sea-blue histiocytes in bone marrow, and the accumulation of sphingomyelin, cholesterol and other glycosphingolipids. In this paper, we reported a rare case of neurovisceral storage disease with severe horizontal supranuclear ophthalmoplegia and sea-blue histiocyte in bone marrow. The patient was a 9-year-old boy. He was hospitalized for unstable gait. The neurological examination revealed severe horizontal supranuclear ophthalmoplegia, moderate ataxia of four extremities and trunk, and mild dystonia of neck and four limbs on walking and standing. The ocular movement in the vertical direction was less impaired and his mentality was almost normal. The bone marrow aspiration showed a few sea-blue histiocytes. The activities of fibroblast lysosomal enzymes including sphingomyelinase were normal. The rectal biopsy revealed many foamy cells in mucous membrane and submucosa. The cell had PAS-positive and acid phosphatase-positive substances, which showed rose-red metachromasia with Feyrter's thionin method. But these abnormal cells were never stained by Sudan black B. These histochemical reactions were compatible with those of Neville's neurovisceral storage disease (Lake, 1983). Therefore we supposed the pathogenesis of this case was the same as that of Neville's cases. In this case, the horizontal supranuclear ophthalmoplegia was a unique symptom.

Bone Marrow↗

PubMed1985

[Immunohistochemical characterization of lymphocytes and histiocytes in bladder cancers in terms of local immune responses].

Ten resected bladder cancers were studied immunohistochemically using monoclonal antibodies to clarify the subsets of infiltrating lymphocytes and histiocytes. Leu 4+ T-lymphocytes infiltrated diffusely both in the stroma and the submucosal tissue, but Bl+ B-lymphocytes usually formed follicle-like clusters in the submucosal tissue. The number of Leu 4+, Leu 3 a+ helper/inducer T-lymphocytes was considerably more than that of Leu 4+, Leu 2 a+ suppressor/cytotoxic T-lymphocytes. Leu 7+ natural killer cells were sparsely present in the submucosal tissue, and there was no suggestion of any significant contact with cancer cells. OKT9+ OKM1+ macrophages were prominent in the inflammatory lesions, but OKT6+ T-zone histiocytes were characteristically present among the cancer cells. Patients with dense infiltration of OKT6+ T-zone histiocytes were expected to have a good prognosis.

Aged↗

PubMed1988

Virus-associated histiocytic proliferations in children. Frequent association with Epstein-Barr virus and congenital or acquired immunodeficiencies.

Nineteen children who presented with fever, hepato-splenomegaly, bone marrow and/or hepatic failure, and biopsy evidence of histiocytic proliferations were evaluated for lymphocyte dysfunction and evidence of prior viral infection. Seventeen of the children had erythrophagocytosis consistent with the previously described virus-associated hemophagocytosis syndrome (VAHS) or Familial erythrophagocytic lymphohistiocytosis (FEL). The other two had benign histiocytic proliferations in the central nervous system (CNS) with liver and bone marrow dysfunction. There were two sibling pairs and six patients with known disorders of immune deficiency. The remaining nine cases appeared to be sporadic and idiopathic. Epstein-Barr Virus (EBV) was identified in patients by serologic or DNA hybridization studies (15), EBV and cytomegalovirus (CMV) (1), adenovirus plus EBV and CMV (1), or adenovirus and EBV (1). Herpes zoster was associated with reactivation of symptoms in one patient. Immunologic impairment was evidenced by lymphopenia in 10 of 19 patients. More extensive evaluations could be done at diagnosis on only some of the children because the histiocytic proliferative syndrome was not recognized or because there were insufficient numbers of lymphocytes in samples obtained. For those who could be evaluated, the following immune deficiencies were found: decreased lymphocyte proliferation to mitogens (4 of 9), absent or markedly decreased natural killer function (5 of 5), and decreased cytotoxic lymphocyte reactivity to allogenic EBV-infected target cells (3 of 3). A new finding reported here is a higher than expected prevalence of HLA types A30, B8, and A1/B8 among the patients tested.

Child↗

PubMed1988

Aschoff bodies of rheumatic carditis are granulomatous lesions of histiocytic origin.

The histogenesis of the Aschoff body of rheumatic carditis is controversial. Proliferative Aschoff bodies in heart sections from 6 patients with acute rheumatic heart disease were tested by avidin-biotin peroxidase labeling methods for the presence of desmin, muscle-specific actin, S-100, neurofilament, leukocyte common antigen, receptor for Ulex europeus I lectin, Factor VIII-related antigen, vimentin, alpha 1-antichymotrypsin, and myeloid/histiocyte antigen. Lack of Aschoff body labeling for desmin and muscle-specific actin, S-100 and neurofilament, and Ulex europeus I and Factor VIII-related antigen is not consistent with histogenesis from smooth or striated cardiac muscle, nerve or nerve sheath, and lymphatic or vascular endothelium, respectively. Strong labeling of Aschoff body cells for vimentin is evidence for a mesenchymal origin, and labeling for myeloid/histiocyte antigen is consistent with a histiocytic origin. Furthermore, weak, variable labeling of Aschoff body cells for leukocyte common antigen suggests that at least some Aschoff body cells were originally derived from blood-borne monocytes. Both multinucleated Aschoff cells and "owl's eye," Anitschkow cells label identically, suggesting a common origin. Alpha 1-Antichymotrypsin, a widely utilized marker of histiocytes, was unexpectedly negative. Perhaps histiocytes that form Aschoff bodies do not express this lysosomal enzyme. Aschoff bodies appear to be a unique and distinctive form of granuloma.

Cell Line↗

PubMed1986

Expression of interleukin-1 in Reed-Sternberg cells and neoplastic cells from true histiocytic malignancies.

Interleukin-1, a peptide produced by monocytes, histiocytes, and interdigitating reticulum cells, plays an important role in the regulation of immune function. In this styde, we examined the production of interleukin-1 in 115 patients with a variety of human lymphomas by using a rabbit anti-interleukin-1 antibody and the immunoperoxidase technique. Interleukin-1 was detected in Reed-Sternberg cells from 20 patients with Hodgkin's disease as well as in neoplastic cells from 9 patients with true histiocytic lymphoma or malignant histiocytosis. In the other 86 cases, which included T- and B-cell lymphomas, no interleukin-1 could be detected. This result indicates a close relationship between Hodgkin's disease and true histiocytic malignancies and provides additional evidence to support our hypothesis that Reed-Sternberg cells are related to interdigitating reticulum cells.

Animals↗

PubMed1979

[Sea-blue histiocyte syndrome (author's transl)].

The literature published before October, 1977 on the so-called sea-blue histiocyte syndrome is reviewed. This is a new lipid thesaurismosis, and from a morphological point of view it is characterized by the appearance of large histiocytes in the organs of the reticuloendothelial system with numerous intracytoplasmic granules which take on a typical sea-blue or greenish color with Wright's or Giemsa stain. The exact nature of the accumulated substance has not yet been specifically determined, though it appears to be gluco- and/or phosphosphingolipid, essentially sphingomyelin. The specific biochemical alteration responsible for this chronic deposit has not been established, though a partial sphingomyelinase deficiency has been detected. Sea-blue histiocytes have been observed in two different situations, either as an acquired phenomenon or as a primary condition. Of the latter there have been sporadic cases and cases with a definite familial incidence. The clinical manifestations include enlargement of the liver and spleen, neurological symptoms, cirrhosis of the liver, hemorrhagic diathesis and purpura, chronic pneumopathies, eye or cutaneous disturbances, or no symptoms at all. The disease has a benign clinical course, and the prognosis is less favourable when clinical manifestations appear early in life. These cases have a greater tendency to develop neurological alterations. The final definition of the syndrome must await the clear identification of the accumulated material and the altered enzyme or metabolic pathway.

Diagnosis, Differential↗