Familial Hodgkin's Disease Research Articles

Genetic Anticipation in Familial Hodgkin's disease.

Genetic Anticipation in Familial Hodgkin's disease.

Genetic Anticipation in Familial Hodgkin's disease.

Genetic Anticipation in Familial Hodgkin's disease.

Familial Hodgkin's disease: a disease of young adulthood?

Familial Hodgkin's disease (FHD) is estimated to represent approximately 4.5% of all cases of Hodgkin's disease (HD). Shared environmental factors, such as Epstein-Barr virus and other viral agents, and genetic determinants have all been proposed to explain familial aggregation of HD. In order to compare the characteristic features of FHD with those of the much more common sporadic form, we reviewed 28 articles on FHD, published between 1972 and 1995, and analyzed in further detail data from 18 papers, reporting on a total of 328 patients. The male-to-female ratio of the FHD population examined was 1.5, similar to that reported for sporadic HD, and lower than the one suggested for FHD by some authors. On the other hand, a significant difference was found between sporadic and familial HD according to age at diagnosis; that is, only one major peak between 15 and 34 years was present in the group of patients with FHD. Further investigation of FHD in young adulthood may provide insight into the hypothesis of a genetic or infectious etiology of the disease.

Epstein-Barr Virus and Familial Hodgkin's Disease

Several studies suggest that the Epstein-Barr virus (EBV) is etiologically linked to Hodgkin's disease (HD). This study was undertaken to examine the role of EBV in familial HD (FHD). Among 60 FHD patients from 27 families with two or more cases per family, we tested available paraffinized tumor tissues from 46 cases by in situ hybridization for EBV-encoded RNA (EBER1) expression. Thirteen of 46 FHD patients (28%) had EBER1 expressed in the Reed-Sternberg cells. Concordance rate of EBV positivity was evaluated among 34 first-degree related pairs from 17 families for which both cases had available paraffinized tumor tissues. Only two of 17 pairs were concordant for EBER1 positivity. There was no excess of positive concordance (P = .18). Serologically, FHD patients had higher geometric mean antibody titers (GMTs) to the viral capsid antigen (VCA) and early antigen D (EA-D). There was no difference in seroprevalence between patients and control groups, nor was there concordance in elevated serology among 15 pairs of first-degree related FHD cases. Young adult unaffected family members (UFM) may not react to EBV in the same way as the general population as evidenced by the lower titer of VCA, although not statistically significant, and significantly lower titers of EA-D, compared with age-matched controls. While EBV might have some role in a subset of HD, lack of concordance of EBER1 expression and EBV serology among the FHD cases in the same family suggest that EBV does not play an important role in FHD.

Epstein-Barr virus in familial Hodgkin's disease.

Hodgkin's disease (HD) has been found to be linked to Epstein-Barr virus (EBV). Familial HD (FHD) may be related to a possible unknown agent. We have determined whether EBV small RNAs (EBERs) were found in Reed-Sternberg cells from FHD. Five families were studied for histological subtype and EBER presence. There was a striking similarity in FHD subtypes of each family and 3/11 (27%) of the cases were EBER positive. In conclusion, EBV EBERs are only infrequently found in FHD and other factors including viruses different from EBV should be further investigated in FHD.

Myasthenia and familial Hodgkin's disease associated in one patient

Myasthenia and familial Hodgkin's disease associated in one patient

Familial Hodgkin's disease a clinical and laboratory investigation

A three to sevenfold increased risk of Hodgkin's disease has been noted in families of patients. We report the first family in which all four of a four member sibship had Hodgkin's disease. In these four sibling cases, we were able to explore markers of infectious etiologic factors by measuring antibodies to Epstein-Barr virus (EBV) and human T-cell lymphotropic virus (HTLV), and possible genetic risk factors by human leukocyte antigen (HLA) typing and chromosome analysis. All three men were diagnosed within six months of their 30th birthday. HLA typing revealed that all four siblings share the DR5 antigen, and two siblings share HLA-B27. Chromosome analysis on peripheral lymphocytes revealed no abnormalities. A detailed, structured interview failed to elicit evidence of unusual exposures. The EBV antibody titers are probably within the normal range and none of the family had antibodies to HTLV-III. Observations on this family suggest that genetic factors play a greater role than environmental factors in the etiology of familial Hodgkin's disease.

Incidence of familial Hodgkin's disease.

The family histories of 131 patients with histologically defined Hodgkin's disease (HD) were studied and 2,517 first and second degree relatives and spouses were identified and followed-up. The causes of death in deceased relatives were ascertained from death certificates. The numbers of deaths from selected causes were compared with the numbers that would be expected if the relatives had suffered the same mortality rates as the Scottish national population. A 4-fold increase in deaths due to HD was found among first and second degree relatives of patients with the disease (6 cases observed compared with 1.4 expected). Five of the 6 familial cases were related to index patients with the mixed cellularity form of the disease, the remaining case was the brother of a patient with the lymphocyte-depleted form of the disease. The increased risk was seen among relatives of both young and older patients and there was no consistent intrafamilial similarity in age of onset or time of onset of disease.

HLA genotypes in familial Hodgkin's disease. Excess of HLA identical affected sibs

The observations of HLA-genotyped 13 family cases of Hodgkin's disease including 7 pairs of affected sibs, 4 parent/child cases and 2 pairs of first cousins are reported. Among the pairs of affected sibs, an excess of HLA identical patients ( 6 observed out of 7) ( 1.75 expected) do not reach the level of statistical significance. However when the results are pooled with international data, the increased frequency of HLA identity among the affected sibs reaches a significant level (P < 0.05). This observation strongly supports the hypothesis of the role of the HLA complex as one of the factors of susceptibility to Hodgkin's disease.

HLA antigens in familial Hodgkin's disease

In a study of 13 families prone to Hodgkin's disease, the probands showed significant excesses of the HLA antigens Bw35 (7 cases) and Bw37 (3 cases). Although based on a small number of patients, the results suggest that immunogenetic mechanisms account at least partly for the familial predisposition to Hodgkin's disease.

Famili&amp;auml;rer Morbus Hodgkin Fallbericht und Literatur&amp;uuml;bersicht

In a family of five sisters four developed malignant lymphomas. A case report is given together with a literature survey of familial Hodgkin's disease. The observations fit the hypothesis that for the development of Hodgkin's disease two requirements must be met: a genetically determined susceptibility as well as some causative agent.

Genetical Investigations in Familial Hodgkin's Disease

Genetical Investigations in Familial Hodgkin's Disease

Immunological depletion contributing to familial Hodgkin's disease

The occurrence of Hodgkin's disease in three sisters of a sibship of five girls, over a period of six years, provided an ideal situation for the investigation of possible aetiological factors. Of note was the occurrence of recurrent herpes labialis in the fourth girl, infectious mononucleosus in the fifth sister and recent Herpes Zoster in the mother. The main findings were (a) a familial depletion of T lymphocyte function as evidenced initially in all members of the family by depressed lymphocyte transformation and (b) the presence of HL-A 1,8 in two affected sisters, the two unaffected sisters and the mother, who had, however, transmitted these antigens in the normal fashion. Of significance was consistent skin anergy to DNCB and tuberculin in the mother (HL-A 1,8) and in the fourth daughter (HL-A 1,8) even two and a half years after she had glandular fever. In addition, the mother, who has no outward signs of disease, had a paraprotein in her serum forming almost 60% of the gammaglobulin fraction.

HLA in familial Hodgkin's disease. Results and a new hypothesis;.

A familial aggregate of seven cases of Hodgkin's disease (HD) has been investigated by HLA typing. Over 600 people in the immediate population (i.e. about half) have been HLA typed and haplotypes have been obtained for 95% of them. It was expected that the cases would share a particular HLA haplotype or at least that they would have one or two HLA antigens of the same series in common. However, this was not the case so no simple idea of association of HLA with HD cases was upheld. When antigen frequencies were examined in the whole population, it was found that HLA B18 increased progressively in incidence from 0.08 to 0.4 in successive groups of individuals each one more closely related to the HD cases. Similarly the community with the highest incidence of HD also had the highest incidence of B18. Thus B18, which in the world figures carries the highest relative risk, emerged as important in this study. Of four proposed interpretations of the data, we are most interested in the idea that the important HLA association is at a population level rather than at the level of the individual patient. A hypothesis, based on the concept of a "healthy carrier" for the HD agent, explains how such an association might operate. It is possible that B18-linked complement deficiency could be the basis for such a carrier state.

Familial Hodgkin's disease and associated cancer.A Clinical-Pathologic Study

The etiology of Hodgkin's disease is complex, as is evident in studies suggesting the importance of horizontal transmission, occupational factors, racial and ethnic background, and familial, genetic factors, or both, including HL-A associations. The present study is of a remarkable kindred in which Hodgkin's disease was histologically verified in two sibships involving second-cousins related through maternal great-grandparents. Cancer of the lung, breast, endometrium, ovary, pancreas, and brain, as well as leukemia and Wilms' tumor, occurred in first and second-degree relatives of the Hodgkin's patients. HL-A haplotypes in patients with Hodgkin's disease in this family showed HL-AB5 or HL-ABW35, an association confirmed in other reports. The findings of associated malignant neoplasms in familial Hodgkin's disease, here and elsewhere in the literature, suggest that in the quest for etiology of Hodgkin's disease one must view the disorder eclectically, with a painstaking search for multiple etiologies, genetic and environmental.

Letter: Familial Hodgkin's disease.

Letter: Familial Hodgkin's disease.

FAMILIAL HODGKIN'S DISEASE: AN ENVIRONMENTAL AND GENETIC DISORDER

Evaluation of cases of familial Hodgkin's disease among first-degree blood-relations suggests that the time-intervals between diagnoses are shorter than the age differences between family members. This and the observation that the time-intervals between diagnoses for relatives living in the same household were shorter than for those living apart are consistent with an environmental aetiology. However, since many familial pairs had the same Rye subtypes at diagnosis, regardless of their relationship or proximity, host reactivity may possibly be related to genetic factors.

Proceedings: Immunological depletion contributing to familial Hodgkin's disease.

Proceedings: Immunological depletion contributing to familial Hodgkin's disease.

Malignant lymphomas.

CA: A Cancer Journal for CliniciansVolume 15, Issue 5 p. 198-213 ArticleFree Access Malignant lymphomas Lloyd F. Craver M.D., Lloyd F. Craver M.D. Attending Physician Emeritus, Memorial Hospital for Cancer and Allied Diseases; and Professor of Clinical Medicine Emeritus, Cornell University Medical College, New York CitySearch for more papers by this authorDaniel G. Miller M.D., Daniel G. Miller M.D. Head, Lymphoma Section, Division of Clinical Chemotherapy, Sloan-Kettering Institute for Cancer Research; Associate Attending Physician, Memorial Hospital for Cancer and Allied Diseases: and Assistant Professor of Medicine, Cornell University Medical CollegeSearch for more papers by this author Lloyd F. Craver M.D., Lloyd F. Craver M.D. Attending Physician Emeritus, Memorial Hospital for Cancer and Allied Diseases; and Professor of Clinical Medicine Emeritus, Cornell University Medical College, New York CitySearch for more papers by this authorDaniel G. Miller M.D., Daniel G. Miller M.D. Head, Lymphoma Section, Division of Clinical Chemotherapy, Sloan-Kettering Institute for Cancer Research; Associate Attending Physician, Memorial Hospital for Cancer and Allied Diseases: and Assistant Professor of Medicine, Cornell University Medical CollegeSearch for more papers by this author First published: September/October 1965 https://doi.org/10.3322/canjclin.15.5.198AboutPDF ToolsRequest permissionExport citationAdd to favoritesTrack citation ShareShare Give accessShare full text accessShare full-text accessPlease review our Terms and Conditions of Use and check box below to share full-text version of article.I have read and accept the Wiley Online Library Terms and Conditions of UseShareable LinkUse the link below to share a full-text version of this article with your friends and colleagues. Learn more.Copy URL Share a linkShare onFacebookTwitterLinkedInRedditWechat References 1 Scheerer, P. P.; Pierre, R. V.; Schwartz, D. L. and Linman, J. W.: Reed-Sternberg-cell leukemia and laetic acidosis. Unusual manifestations of Hodgkin's disease. New England J. Med. 270: 274– 278, 1964. 2 Agress, C. M., and Fishman, H. C.: Lipomelanie retieulasis, J.A.M.A. 143: 957– 960, 1950. 3 Squire, R. A.: Hematopoietic tumors of domestic animals, Cornell Vet. 54: 97– 150, 1964. 4 Murphy, E. D.: Personal communication. 5 Craver, L. F.: Hodgkin's disease. Tice-Harrey Practice of Med. 6: 1017– 1063, 1964. 6 Aisenberg, A. C.: Hodgkin's disease—prognosis, treatment and etiologic and immunologic considerations. New England J. Med. 270: 508– 514, 565–570, 617–622, 1964. 7 Fox, H.: Presentation of microscopic preparations made from some of the original tissue described by T. Hodgkin, 1832. Ann. Med. History. 8: 370– 374, 1926 8 Razis, D. V.; Diamond, H. D., and Craver, L. F.: Familial Hodgkin's disease: Its significance and implications. Ann. Int. Med. 51: 933– 971, 1959. 9 Gilliam, A. G.: Age, sex, and race selection at death from leukemia and the lymphomas. Blood 8: 693– 702, 1953. 10 Allen, A. C.: Persistent “insect bites” (dermal eosinophilic granulomas) simulating lymphoblastomas, histiocytoses, and squamous cell carcinomas. Am. J. Path, 24: 367– 387, 1948. 11 Fisher, E. R., and Hazard, J. B.: Differentiation of megakaryocyte and Reed-Steruberg cell, with reference to the periodic acid-Schiff reaction. Lab. Invest. 3: 261– 269, 1954. 12 Bodansky, O.: New functional principles in diagnostic aspects of liver disease. Bull. New York Acad. Med. 38: 711– 726, 1962. 13 Williams, H. M.; Diamond, H. D.; Craver, L. F., and Parsons, H.: Neurological complications of lymphomas and leukemias; p. 134. Springfield, III.: C. C. Thomas, 1959. 14 Levitan, R.; Diamond, H. D., and Craver, L. F.: The liver in Hodgkin's disease. Gut 2: 60– 71, 1961. 15 Levitan, R.; Diamond, H. D., and Craver, L. F.: Jaundice in Hodgkin's disease. Am. J. Med. 30: 99– 111, 1961. 16 Richmond, J.; Sherman, R. S.; Diamond, H. D., and Craver, L. F.: Renal lesions associated with malignant lymphomas. Am. J. Med. 32: 184– 207, 1962. 17 Aisenberg, A. C.: Immunologic aspects of Hodgkin's disease. Medicine (Balt., Md.) 43: 189– 193, 1964. 18 Barry, R. M.; Diamond, H. D., and Craver, L. F.: The influence of pregnancy on the course of Hodgkin's disease. Am. J. Obst. & Gynec. 84: 445– 454, 1962. Volume15, Issue5September/October 1965Pages 198-213 ReferencesRelatedInformation

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