A case of histiocytic necrotizing lymphadenitis with bone marrow and skin involvement

Sumiyoshi, Yoshiaki; Kikuchi, Masahiko; Ohshima, Kohiti; Masuda, Yuiti; Takeshita, Morishige; Okamura, Takashi

doi:10.1007/bf01600281

Cited by 47 publications

(21 citation statements)

References 13 publications

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“…Several reports suggest that this virus infec tion may also be associated with histiocytic necrotizing lymphadenitis (Kikuchi's disease), which mainly affects adolescents and causes swelling of the cervical lymph nodes, leukopenia and an elevation of LDH [8], but does not cause hepatosplenomegaly or thrombocytopenia, which are often observed in VAHS. However, a case of Kikuchi's disease associated with findings of hemophagocytosis in the bone marrow was recently reported [9]. The roles of HHV-6 in these two diseases should be examined further in more detail.…”

Section: Human Herpesvirus 6 Infection Associated With Hemophagocyticmentioning

confidence: 99%

Human Herpesvirus 6 Infection Associated with Hemophagocytic Syndrome

Sugita¹,

Kurumada²,

Eguchi³

et al. 1995

Acta Haematol

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Section: Human Herpesvirus 6 Infection Associated With Hemophagocyticmentioning

confidence: 99%

Human Herpesvirus 6 Infection Associated with Hemophagocytic Syndrome

Sugita¹,

Kurumada²,

Eguchi³

et al. 1995

Acta Haematol

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“…Histopathologic findings show focal changes, characteristically pale at low magnification, corresponding to an infiltration by apoptotic plasmacytoid monocytes containing nuclear debris and large lymphocytes in the paracortical area. 4 On cutaneous histopathologic examination, under a normal epidermis, a perivascular and periadnexal dense identical infiltrate is observed in the dermis and hypodermis. 4 The principal differential diagnoses are malignant lymphoma and other etiologies of benign necrotizing lymphadenopathy, such as toxoplasmosis, infectious mononucleosis, and systemic lupus erythematosus.…”

Section: Discussionmentioning

confidence: 99%

“… 4 On cutaneous histopathologic examination, under a normal epidermis, a perivascular and periadnexal dense identical infiltrate is observed in the dermis and hypodermis. 4 The principal differential diagnoses are malignant lymphoma and other etiologies of benign necrotizing lymphadenopathy, such as toxoplasmosis, infectious mononucleosis, and systemic lupus erythematosus. 5 …”

Section: Discussionmentioning

confidence: 99%

Kikuchi disease associated with Still disease

et al. 1999

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A 32‐year‐old white man with no previous medical past was admitted to our department with an 8‐month intermittent history of acute spiking fever with axillary, inguinal, and neck lymphadenopathy, arthritis, and cutaneous eruption. Cutaneous examination showed nonpruriginous, indurated erythematous papules affecting the face, ears, and trunk, suggestive of subacute lupus erythematosus ( Fig. 1a,b). On general physical examination, no abnormalities were observed except for arthritis of the right ankle and, a few months later, arthritis of the left knee. Biological investigations revealed a raised erythrocyte sedimentation rate with a serum ferritin level at 800 ng/mL (normal < 200 ng/mL), a white blood cell count of 12,500/mm3 (with 80% neutrophils, 16% lymphocytes, and 4% monocytes), a hematocrit of 40%, and a hemoglobin level of 13.1 g/dL. Electrolytes and liver function tests were normal. Antinuclear antibodies and antibodies to deoxyribonucleic acid (DNA) were negative. Rheumatoid factor was absent. No evidence of the following infections/organisms was found on serologic screening: syphilis, Streptococcus, Toxoplasma, Mycoplasma, Brucella, Borrelia burgdorgferi, spotted fever, leptospirosis, hepatitis B and C virus, parvovirus, human immunodeficiency virus (HIV) 1 and 2, human T‐cell lymphotropic virus (HTLV) 1, Epstein–Barr virus (EBV), cytomegalovirus (CMV), herpes simplex virus (HSV) 6. Immunoglobulin M (IgM) HSV(1+2) by enzyme‐linked immunosorbent assay (ELISA) analysis with 1 year follow‐up was still detected. The articular synovial fluid of the involved ankle and knee was inflammatory but sterile. A bone marrow biopsy specimen revealed no lymphomatous involvement. Abdominal and chest scans were normal. Skin biopsy of a facial nodule showed, in the dermis and in the subcutaneous fat tissue, a dense perivascular and periadnexal infiltrate composed of numerous plasmacytoid histiocytes and large‐ or medium‐sized lymphocytes ( Fig. 2a,b). No epidermal changes, edema in the papillary dermis, or vasculitis were found. Immunofluorescence staining of frozen tissue sections of a facial nodule showed granular deposits of C3. Inguinal lymph node biopsy revealed, in the paracortical area, focal necrosis with a proliferation of transformed lymphocytes, histiocytes, and numerous nuclear debris typical of histiocytic necrotizing lymphadenitis ( Fig. 3a,b). On immunohistochemical staining examination, CD68 was positive in pathologic paracortical areas. Few mononuclear cells were positive for CD3, CD8, and L‐26 ( Fig. 4). Polymerase chain reaction (PCR) amplification did not show human herpes simplex viruses in the skin biopsy specimen, and culture was also negative. 1 (a) Clinical aspect: erythematous papulonodules on the face. (b) Clinical aspect: erythematous papulonodules of the upper part of the trunk 2 (a) Perivascular and periadnexal dense lympho‐ histiocytic infiltrate involving the dermis (hematoxylin and eosin, × 10). (b) Plasmacytoid histiocytic infiltrate in the subcutaneous fat tissue (hem...

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“…There are no characteristic skin lesions and the eruptions have been variously described as morbilliform, maculopapular or disseminated erythema [8] . In one instance [9] the bone marrow was affected. Recurrence of Kikuchi's disease has been reported [1,2] and multiple episodes have been recorded in the same Kikuchi's Disease ORL 1997;59:176-178 patient [10].…”

Section: Discussionmentioning

confidence: 99%