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It is proposed to search if by immunological means it is possible to distinguish eosinophilia produced by the filaria Onchocerca volvulus from other eosinophiliae. The proposed methods also seem to allow the detection of the presence of the specific antibodies in other hematic cells.
Angiolymphoid hyperplasia with eosinophilia (AHE) is a distinct clinical and pathologic entity that demonstrates proliferation of endothelial cells associated with varying degrees of lymphocytic, histiocytic, and eosinophilic infiltration. Lymphoid hyperplasia with germinal centers often is present in older lesions and the patients may have an associated blood eosinophilia. Multiple deep nodules developed in a young man on the palmar aspect of the left hand, and subsequently the forehead, histologically diagnostic of AHE. Direct immunofluorescence of the tumor removed from the forehead demonstrated granular deposits of IgA, IgM, and C3 associated with small vessels. Cryoglobulins (IgA, IgM, and IgG) were found in the patient's serum. This new information lends support to the hypothesis that AHE is an inflammatory reactive lesion, possibly secondary to an immunologic injury, rather than a true vascular neoplasm with an associated inflammatory reaction.
Pulmonary artery hypertension is a rare feature in the eosinophilic pulmonary syndrome, it had been observed only in some cases of tropical pulmonary eosinophilia. A case of prolonged pulmonary eosinophilia with reversible pulmonary hypertension is presented.
A 23-year-old woman from the French Antilles was successfully treated for intestinal and hepatic schistosomiasis, as evidenced by the normalization of immunofluorescence titers and many negative histogogical controls. She presented, however, persistent colitis with marked eosinophilia (eosinophils in excess of 50,000/mm3). No precise diagnosis could be established in spite of all parasitological, allergological, hematological and gastroenterological investigations. The colon was the only organ involved and showed colitis of a type unclassifiable both endoscopically and histologically. Massive infiltration by eosinophils was present. Temporary relief was achieved with steroids but the clinical condition of the patient became dramatic. Three years after the onset of the illness total colectomy was performed, sparing the rectum. After surgery all manifestations of disease disappeared and for ten months the number of eosinophils was normal. Ileorectal continuity could be re-established. Ten months after reanastomosis there was recurrence of bleeding from the rectum and eosiophils once again reached levels of 2340/mm3. The differential diagnosis of this colitis associated with eosinophilia is discussed. To our knowledge it does not correspond to any clinical entity described so far.
A method of immunization has been described which consists of the i. v. injection into rats of large latex particles coated with human gamma-globulin (HGG). The coated particles embolize the pulmonary capillary bed and induce blood eosinophilia, which correlated strongly with the development of lymphocyte responsiveness to HGG in vitro. Sequential histologic studies of the lungs including ultrastructural analysis suggest a delayed hypersensitivity reaction to the coated particles. These studies support the concept that the induction of blood eosinophilia may be mediated by T lymphocytes.
The immunoglobulin class and subclass of cytophilic antibodies have been studied using peripheral leucocytes from twenty-two patients with allergic bronchopulmonary aspergillosis, aspergilloma and cryptogenic pulmonary eosinophilia. In patients with allergic bronchopulmonary aspergillosis, significantly increased histamine liberation occurred following challenge of their leucocytes with antisera to IgE, IgG2, IgG3 and IgG4 as well as with Aspergillus fumigatus antigen. The results were considerably modified if the patient was receiving corticosteroids at the time of the test. The presence of IgG2-specific antibody to A. fumigatus in the serum of one patient, capable of sensitizing donor leucocytes, was demonstrated in passive sensitization experiments. In two patients with uncomplicated aspergillomas no evidence of cytophilic antibody to any class was found although large amounts of precipitating IgG antibody was present in the serum. Two patients with aspergilloma and systemic symptoms of weight loss and fatigue (which have been interpreted by others as 'hypersensitivity' responses) had increased amounts of cytophilic antibody similar to those with allergic bronchopulmonary aspergillosis. Six patients with cryptogenic pulmonary eosinophilia were also studied. No evidence of specific antibody to A. fumigatus was found but, as a group, significantly increased histamine liberation using antisera to IgG2 was demonstrated. Individual patients also showed evidence of other classes of cytophilic antibody, one having IgE, three IgG3 and two IgG4. The relationship between heat-stable short-term sensitizing antibody (IgG STS) inducing immediate skin responses and the pattern of cytophilic antibodies found in our patients with bronchopulmonary aspergillosis having dual (immediate and late reactions) is discussed. Clinically these tests are of diagnostic value and they may be helpful in assessing symptomatic patients with aspergillomas for corticosteroid treatment.
Angiolymphoid hyperplasia with eosinophilia is manifested by benign vascular tumors that usually occur about the head in young adults. Clinical and histologic variation of this process has resulted in nosologic confusion, and the cases in the English literature were reviewed to characterize it within racial groups. Oriental patients tend to be younger at onset, usually male, and have marked blood eosinophilia; large, but relatively asymptomatic, tumors develop in these patients. White patients tend to be older at onset and have small, friable tumors. Large tumors that are painful or pruritic tend to develop in blacks and Middle Eastern patients.
Skin induration without internal organ involvement, blood and tissue eosinophilla, and fascilitis are features of diffuse fascilitis. However, cellular infiltrates (lymphocytes, plasma cells, and eosinophils) may also be present in the dermis, fat, and muscle. Blood eosinophilia (mild and transient) and skin eosinophilia were observed in about 20% of patients with systemic and localized scleroderma.
The recently described syndrome of diffuse fasciitis with eosinophilia consists of scleroderma-like cutaneous changes in the absence of Raynaud's phenomenon and without visceral involvement. Peripheral blood eosinophilia and hypergammaglobulinemia occur in the majority of patients. The biopsy specimen is characteristic and shows a diffusely thickened fascia with a noticeable inflammatory infiltrate. The etiology and pathogenesis of the entity are unknown. The majority of the cases have been reported to respond to corticosteroids. Pulmonary function abnormalities and rheumatoid factor elevation in one of our patients suggests that this syndrome may be a variant scleroderma or may have previously unrecognized systemic manifestations. The patients' objective responses to corticosteroids were minimal.
A female case of diffuse fasciitis with eosinophilia (or eosinophilic fasciitis) is reported. This disease is characterized by suddenly developed circumscribed subcutaneous indurations on the extremities, hyalinized fibrosis of the fascia and peripheral eosinophilia. Our patient further displayed Raynaud's phenomenon preceded by thrombophlebitis, and histological and electron microscopical features of her subcutaneous hyalinized area centering at the fascia closely resembled those of morphaea.
Two cases of granulomatous dermatitis with eosinophilla (Wells' syndrome) are reported. With Wells' original four cases, these two cases define a distinctive dermatosis with onset as cellulitis and formation of solid edema and either final spontaneous resolution or resolution with steroid therapy. Microscopic study showed diffuse tissue eosinophilia and fibrinoid flame figures, evolution of associated focal necrobiosis, and formation of focal microgranulomas associated with eosinophils. Biopsy of muscle and fascia showed comparable fasciitis and eosinophilic myositis. Immunofluorescence in one case disclosed fibrin in the dermis and lgM, lgA, and C3 in the blood vessels of the muscle. Recurrences of the lesions often appeared to be related to drug administration or surgery.
A patient with biopsy-proven papular mucinosis, plus the characteristic IgG lambda light chain paraproteinemia, also developed a severe proximal myopathy, seronegative inflammatory polyarthritis, and marked eosinophilia. Muscle enzymes were elevated, EMG was compatible with polymositis, and muscle biopsy revealed an atypical necrotizing vacuolar myopathy. Synovial biopsy revealed an inflammatory synovitis with Class II synovial fluid. No mucin deposition was detectable in muscle or synovium. During 7 years of observation, corticosteroids and various immunosuppressive agents were successively administered with little benefit. Recently, weekly intravenous methotrexate and low-dose oral corticosteroids have resulted in clinical and laboratory improvement. It is suggested that the pathology in papular mucinosis may include serious rheumatic manifestations in addition to the cutaneous involvement.
A patient with a giant cell carcinoma of the bronchus presented with headache and flushing attacks and a blood eosinophilia. He developed a right bundle branch block and later congestive cardiac failure. At post mortem he was found to have the appearance of Loeffler's endocarditis. A tentative mechanism for these events is proposed.
Reaginic antibodies to antigens from the human filarial parasites Wuchereria bancrofti and Brugia malayi and the animal parasite Dirofilaria immitis were studied by histamine release from basophils in 7 patients with tropical eosinophilia (T.E.) and 18 patients with other manifestations of filarial infection (lymphatic changes or symptomless microfilaraemia). All the patients had antibodies to all three filariae but T.E. patients were more highly sensitised. T.E. patients responded more to antigens from microfilariae than did patients with non-T.E. filariasis and responded more to microfilarial antigens from the human parasites than to those from the animal parasite. These findings support the view that T.E. is a form of occult filariasis which results from host hypersensitivity to the microfilarial stage of parasites which, in other individuals, cause the more common lymphatic manifestations of filarial disease.
The clinical and immunological features of fifteen cases of cryptogenic pulmonary eosinophilia are reported. There were ten women (mean age 35.4 years) and five men (mean age 42 years). Eight gave a previous history of asthma and seven had none. Thirteen of the fifteen patients had negative skin test to common allergens. Many features of a systemic illness were present in the asthmatic and non-asthmatic groups including anaemia, weight loss, fever and a grossly raised ESR. An absolute polymorphonuclear leucocytosis was frequent as well as the obligatory increase in blood eosinophils used as one of our criteria for inclusion. Hepatomegaly (three cases), splenomegaly (four cases) and hilar node enlargement (one case) were seen in the group without asthma. Evidence of renal involvement or necrotizing vasculitis was notably absent and the response to small doses of corticosteroids was dramatic. Immunologically the striking feature was a disproportionate increase in blood eosinophils compared with only minor elevations in the total serum IgE levels. This stands in contrast to patients with bronchopulmonary aspergillosis and helminth infestation. Studies of cytophilic antibodies using histamine liberation after challenge with antibodies to immunoglobulin sub-classes in six patients showed a marked increase in IgG2 and lesser increases of IgE and IgG3. No evidence of antibodies specific to A. fumigatus was found. The amount of cytophilic antibody was also in contrast to that found in bronchopulmonary aspergillosis.
A case of angiolymphoid hyperplasia with eosinophilia mimicking multiple cylindromas (turban tumor) is presented in detailed clinical and histologic features, and the literature on the condition is reviewed.
A 44-year-old woman with marked eosinophilia, leukocytosis, congestive heart failure, and the murmur of mitral stenosis had a restrictive type of pulse contour at cardiac catheterization. A right atrial angiogram revealed a huge right atrium, a small right ventricle, and a dilated contractile outflow tract consistent with the diagnosis of Loeffler's endocarditis. A marked conduction delay at the atrial level was demonstrated by His bundle electrogram studies.
A case of diffuse fasciitis with eosinophilia (DFE, or Shulman's disease) is presented. A review of the literature of the condition is presented, and the salient features are summarized. Diagnostic criteria and the management of patients with this unusual condition are discussed.