close

Вход

Забыли?

вход по аккаунту

?

000207260

код для вставкиСкачать
Acta haemat. 64: 227-229 (1980)
Multiple Myeloma in Husband and Wife
M. Brugiatelli, M. Comis, P. Iacopino, F. Morabito, F. Nobile, V. Trapani Lombardo
and A . Neri
Divisione di Ematologia, Ospedali Riuniti, Reggio Calabria
Key Words. Mutiple myeloma • Myeloma cluster • Spouses
Abstract. An instance of multiple myeloma (MM) in a married couple is discussed,
since cases of MM in husband and wife have rarely been observed. The patients were aged
60 when the disease was discovered, and the interval between the diagnosis in the mates
was 44 months. Genetic or environmental factors that could have explained the occurrence
of myelomatosis were not found in these spouses. Therefore, on absence of a defined cause,
our observation, like others previously reported in the literature, should be considered,
although exceptional, a chance event.
The occurrence of multiple myeloma
(MM) in husband and wife represents a
very unusual observation. It was first re­
ported by Kyle et al. [5] who published four
sets of spouses with MM: the time lag be­
tween the diagnosis in the mates varied
broadly, ranging from 1 month to 15 years.
More recently, Pietruszka et al. [7] de­
scribed a patient who developed MM 5
years after his wife had died of myelomato­
sis. All these patients were over 50 when
the disease was discovered. No other cases
of MM in spouses, to our knowledge, have
been so far reported in the literature. We
feel, therefore, that it is of interest to pre­
sent in this communication a new instance of
MM in a married couple, who came to our
observation.
Case Reports
Husband. A 60-year-old joiner was admitted to
our hospital in April 1976 because of a long histo­
ry of fatigue and lassitude. On admission, renal
failure (creatinine 4.6 mg/dl) and severe anemia
(Hb 8.3 gAll) were found. A bone marrow aspirate
contained 72% of immature, atypical plasma cells;
a skeletal survey displayed diffuse osteoporosis.
Serum electrophoresis revealed no peaks, but a
Bence Jones protein, lambda type, was detected by
immunoelectrophoresis, consistent with the diag­
nosis of light chain disease. Chemotherapy with
alkylating agents and prednisone was soon after
started, and his clinical and hematological condi­
tions rapidly improved.
Downloaded by:
Vanderbilt University Library
129.59.95.115 - 10/27/2017 3:10:56 AM
Introduction
Brugiatelli/Comis/Iacopino/Morabito/Nobile/Trapani Lombardo/Neri
Wife. A 60-year-old previously healthy house­
wife was complaining of weakness and low back
pain for several months. When admitted to our
department in December 1979 she was anemic
(Hb 8.0 g/dl) and a bone marrow aspirate yielded
60% plasma cells, mostly abnormal. Serum protein
Table I. Main laboratory findings of the patients on
their first hospitalization
Husband
Wife
Hemoglobin, g/dl
Leukocytes
per pi (x 103)
Platelets
per pi (X 103)
ESR in 1 h
Calcium, mg/dl
Creatinine, mg/dl
Bence Jones
proteinuria
160
68
9.9
4.6
150
88
9.8
0.82
yes
yes
Serum immunoglo­
bulins, mg/dl
IgG
IgA
IgM
750
38
29
410
5,460
10
HLA typing
A1-B5-B17
8.3
8.0
5.7
3.6
A9-AW3031BW21
Table II. The incidence of MM in Reggio Calabria
and its surrounding area (420,000 inhabitants) in the
last 5 years
Year
Cases of MM
1975
1976
1977
1978
1979
6
7
6
8
7
Total
34
Data have been obtained from all the hospitals of
this area.
electrophoresis showed a beta globulin spike, and
immunoelectrophoresis disclosed a homogeneous
IgA, k, paraprotein. Roentgenograms revealed dif­
fuse osteoporosis and some osteolytic lesions in
the skull. The diagnosis of MM, IgA, k was made
and a treatment with melphalan and prednisone
was administrated.
The main laboratory data of both patients arc
summarized in table I.
Family Investigations. The patients do not
have children. Sera from two living brothers of
the wife and one sister of the husband were exam­
ined; one brother of the husband could not be in­
vestigated. Serum proteins of all these relatives re­
sulted normal when studied by electrophoresis and
immunoelectrophoresis.
Discussion
In commenting upon our case reports, the
question if MM in this married couple is to
be considered as mere chance or if a com­
mon etiological factor, albeit unknown,
might be involved, was taken into consider­
ation. The interval of 44 months between
the clinical onset of the disease in the hus­
band and the diagnosis in the wife could not
be an argument against the latter hypothe­
sis, since it is recognized that the develop­
ment of an overt myeloma may take a long
time [2, 8].
As far as the chance hypothesis is con­
cerned, we estimated that the calculated risk
of having MM among the population living
in our province is 1.7/100,000/year (table
II); then the probability for these spouses to
have been afflicted with the same disease is
statistically very small.
On the other hand, we were unable to
explain the occurrence of MM in this mar­
ried couple on the ground of a defined
cause. The etiology of myeloma is largely
unknown. Nevertheless, some factors have
been claimed: thus genetic factors have
been considered, since various instances of
Downloaded by:
Vanderbilt University Library
129.59.95.115 - 10/27/2017 3:10:56 AM
228
Myeloma in Spouses
References
1 Goldstone, A. R.: Wood, J. K„ and Cook, M.
K.: Myeloma in mother and daughter. Acta
haemat. 49: 176-181 (1973).
2 Hokanson, J. A.; Brown, B. W.; Thompson, J.
R.; Drewinko, B., and Alexanian, R.: Tumor
growth patterns in multiple myeloma. Cancer
39: 1077-1084 (1977).
3 Ishimaru, T. and Finch, S. C.: More on radia­
tion exposure and multiple myeloma. New
Engl. J. Med. 301: 439-440 (1979).
4 Kyle, R. A.; Herber, L.; Evatt, B. L., and
Health, C. W., Jr.: Multiple myeloma. A com­
munity cluster. J. Am. med. Ass. 213:
1339-1342 (1970).
5 Kyle, R. A.; Heath, C. W., Jr., and Carbone,
P.: Multiple myeloma in spouses. Archs intern.
Med. 127: 944-946 (1971).
6 Maldonado, G. E. and Kyle, R. A.: Familial
myeloma. Report of eight families and a study
of scrum proteins in their relatives. Am. J.
Med. 57: 875-884 (1974).
7 Pietruszka, M.; Rabin, B. S., and Srodes, C.:
Multiple myeloma in husband and wife. Lan­
cet /: 314 (1976).
8 Salmon, S. E.: Immunoglobulin synthesis and
tumor kinetics of multiple myeloma. Semin.
Hematol. 10: 135-144 (1973).
Received: March 6, 1980
Accepted: June 6, 1980
Alberto Neri, Divisione di Ematologia,
Ospedali Riuniti, 1-89100 Reggio Calabria
(Italy)
Downloaded by:
Vanderbilt University Library
129.59.95.115 - 10/27/2017 3:10:56 AM
familial myeloma have been reported [1, 6],
But this is not the case with our patients:
there is no consanguinity, their family histo­
ries were unremarkable for malignancies,
and finally the results of serum investiga­
tions in the relatives were negative for immunoproliferative disorders. The role of ra­
diations in determining MM has also been
emphasized [3]. However, we could exclude
exposure in both patients. Moreover, a
careful investigation ruled out the possibility
to take into account other environmental
factors, such as chemicals or drugs. Finally,
evidence was achieved that we were not
dealing with a community cluster of MM
cases [4]: they had been married for 20
years, and always lived in the same house;
this is a single house, located on an area
where we are not aware of cases of myelo­
matosis recorded in the past years.
In conclusion, all that is lacking in our
case reports is the identification of a possi­
ble, if any, etiological factor(s). In this way,
the occurrence of MM in these spouses, like
in others previously reported in the litera­
ture, remains unexplained, and it should be
considered, however exceptional, a chance
event.
229
Документ
Категория
Без категории
Просмотров
2
Размер файла
334 Кб
Теги
000207260
1/--страниц
Пожаловаться на содержимое документа