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AIDS in Pre-AIDS Era
AIDS in Pre-AIDS Era
AIDS in Pre-AIDS Era
NOVEMBER-DECEMBER 1987
© 1987 by The University of Chicago. All rights reserved. 0886-0162/87/0906-0003$02.00
A search of the medical literature published since 1950 disclosed 19 cases of probable
AIDS reported before the start of the current epidemic. These cases retrospectively met
the Centers for Disease Control's surveillance definition of the syndrome and had a clini-
cal course suggestive of AIDS. The reports originated from North America, Western Eu-
rope, Africa, and the Middle East. The mean age of patients was 37 years, and the ratio
of male to female patients was 1.7:1.Sixteen patients had opportunistic infection(s) with-
out Kaposi's sarcoma. The remainder had disseminated Kaposi's sarcoma. The commonest
The acquired immunodeficiency syndrome (AIDS) the presence of reliably diagnosed disease at least
was first described in 1981 [1,2]. The first recognized moderately indicative of underlying cellular immu-
cases of this syndrome were seen in the United States nodeficiency, such as Kaposi's sarcoma in a patient
in 1978 [3,4]. Since then more than 20,000 cases have under 60 years of age, pneumocystis pneumonia, or
been identified all over the world [5]. other opportunistic infections (table 1);and second,
One of the paramount questions regarding the cur- the absence of known causes of underlying immu-
rent AIDS epidemic is whether the disease occurred nodeficiency and of any other reduced resistance
prior to its general recognition. In search of an an- reported to be associated with the disease (im-
swer to this question, we have reviewed the medical munosuppressive therapy, lymphoreticular malig-
literature of the three decades antedating the begin- nancy).
ning of this epidemic. We have identified cases that Such cases were included in the present review if
met the surveillance definition of AIDS and had a they could be classified in one of the following two
clinical course suggestive of this syndrome. Herein, categories: cases whose descriptions were published
we describe the spectrum of disease in patients who before the first publication on AIDS in May 1981,
may have had sporadic, unrecognized cases of AIDS or cases whose descriptions were published after 1981
in the pre-AIDS era, and we discuss additional clin- but that were either diagnosed before 1978 or diag-
ical and serologic evidence supporting the occurrence nosed between 1978 and 1981 but unrecognized as
of AIDS in the past. AIDS at the time of publication. The year of ap-
pearance of Kaposi's sarcoma or the first opportunis-
tic infection was considered the time of the first
Methods
manifestation fitting AIDS.
We searched the medical literature from 1950 until Excluded from the present review were cases lack-
January 1986 for case reports fulfilling the Centers ing sufficient data, cases occurring in children un-
for Disease Control's (CDC's) surveillance definition der 5 years of age and in patients more than 60 years
of AIDS [3, 4, 6-9] on the basis of two criteria: first, of age, cases in which disease was cured, and cases
of Kaposi's sarcoma in Africa.
1102
AIDS in the Pre-AIDS Era 1103
1
1953 [lOJ United States 1952 28/M Cytomegaloviral pneumonia No
1982 [30]*
2, 1960 [11] Canada 1958 36/M Pneumocystis carinii pneumonia No
3
1960 [12] United Kingdom 1959 251M Cytomegaloviral pneumonia, P. carinii No
pneumonia
1983 [31J*
4
AIDS [45-55]. We also excluded cases occurring in cause it was difficult to differentiate the aggressive,
the extremes of age [56-59] because of the possible human immunodeficiency virus (HIV)-related type
existence of either congenital or age-related immu- of malignancy from the endemic type frequently
nodeficiency. In addition, we did not include cases found on this continent. These exclusion criteria
of Kaposi's sarcoma occurring in Africa [60, 61] be- prevented the inclusion of doubtful cases and left
AIDS in the Pre-AIDS Era 1105
Aggressive Kaposi's sarcoma in young Africans, 1969-1970 [61] Detection of antibodies to human immunodeficiency
virus in stored serum samples from
Cases reported to the CDC· occurring in the United States Africa: Zaire 1959 [64]
before 1978 [3] Upper Volta 1963 [65]
Zaire 1970 [66]
Uganda 1972-1973 [67]
Zaire 1976 [68]
Probable AIDS in France, 1976-1978 [63] America: Venezuela 1968 [69J
United States 1971-1972 [70]
Cases included in the present report [10- 29]
only those cases with a high probability of being true and the United States [70]. Although some of the
AIDS. detected antibodies have been proved to be or are
suspected of being false-positive findings [66, 70],
these observations are further evidence for the
Discussion
sporadic occurrence of AIDS in the past (table 3).
The present review suggests that sporadic cases of On the other hand, several studies of various
AIDS occurred before the beginning of the current aspects of the syndrome have failed to detect evi-
epidemic of this syndrome. Even since the discovery dence of cases occurring before 1978. These studies
of HIV, which is the etiologic agent of AIDS [6], include revisions of cancer registries and autopsy files
the diagnosis of the syndrome has continued to be in searches for Kaposi's sarcoma in the United States
based on clinical criteria [7], e.g., the occurrence of [71], Denmark [72], and Haiti [73]; data on pentami-
either life-threatening, opportunistic infection with- dine use in the United States as an indicator for
out known underlying immune defects or Kaposi's P. carinii pneumonia [3, 4]; and data on HIV anti-
sarcoma before age 60 [8]. According to the revised body status in sequential samples of plasma from
CDC case definition of AIDS [9], retrospectively American [74] and Scottish [75] hemophiliacs.
studied cases lacking serologic and immunologic test A critical analysis of the clinical and laboratory
results continue to be regarded as AIDS if they satisfy data may call into question our assumption that the
the clinical criteria. Since all 19 cases included in our patients included in the present review really suffered
review meet these criteria, they may be regarded as from AIDS. For example, some patients had received
probable cases of AIDS in the pre-AIDS era. corticosteroid therapy, but only for short periods and
The high specificity of the CDC case definition late in the course of their disease (cases no. 1-6, 9,
of AIDS was confirmed after the introduction of lab- 15, and 19). In one case (no. 13), malnutrition could
oratory tests for the detection of antibody to HIV not be absolutely excluded as a factor predisposing
[9,62]. It was found that "most persons whose clin- to development of the opportunistic infection; on
ical illness fulfills the CDC surveillance definition the other hand, the malnutrition could have resulted
for AIDS will have been infected with the virus" [62]. from AIDS. Only one of the patients had his T cell
One of the cases reviewed (no. 4) was retrospec- subsets defined (case no. 18). The immunologic
tively reported to the CDC [3]. Another case of prob- workup in the majority of cases was incomplete, and
able AIDS occurring in the United States before 1978 some cases had features that are atypical for but do
was also reported to the CDC [3], and five similar not exclude AIDS, such as lymphocytosis (cases no.
cases were retrospectively detected in France [63]. We 4 and 17) and reactive skin tests (cases no. 2, 5, and
have not included these cases in our review because 9). Despite these difficulties, we believe that at least
of insufficient clinical data. some of the 19 cases reviewed were true AIDS.
After the discovery of HIV, antibodies to this vi- In one case of a patient with P. carinii pneumo-
rus were detected in stored specimens of serum ob- nia [16], the same fatal opportunistic infection was
tained before 1978 in Africa [64-68], Venezuela [69], documented almost simultaneously in the patient's
1106 Huminet; Rosenfeld, and Pitlik
husband, who had acute lymphocytic leukemia as mocystis carinii pneumonia in an adult: report of a case.
well. In another instance [26] immune dysfunction Am 1 Clin Pathol 1961;35:353-64
15. Schonell ME, Crofton JW, Stuart AE, Wallace A. Dissemi-
and oral candidiasis were observed in the 3-month- nated infection with Mycobacterium avium. I. Clinical-
old daughter of the index case. In view of recent evi- features, treatment and pathology. Tubercle 1968;49:12-30
dence of heterosexual and maternal-neonatal trans- 16. Watanabe 1M, Chinchinian H, Weitz C, Mcilvanie SK. Pneu-
mission of HIV, these familial cases of opportunis- mocystis carinii pneumonia in a family. JAMA 1965;193:
tic infection may represent further evidence for the 685-6
17. Fermaglich J, Hardman JM, Earle KM. Spontaneous progres-
retrospective diagnosis of AIDS. sive multifocal leukoencephalopathy. Neurology 1970;20:
The historical data presented in our review lead 479-84
us to believe that AIDS is an old disease that has 18. Bolton CF, Rozdilsky B. Primary progressive multifocal leu-
been unrecognized in the past because of its sporadic koencephalopathy: a case report. Neurology 1971;21:72-7
occurrence. The magnitude of the current epidemic 19. Hagmar B, Kutti J, Lundin P, Norlin M, Weinfeld A, Wahlen
P. Disseminated infection caused by Mycobacterium kan-
tivity in 1971-1972: parenteral drug abusers-a case of false nodeficiency syndrome. Ann Intern Med 1983;98:1020-1
positives or evidence of viral exposure? [letter] N Engl J 74. Evatt BL, Gomperts ED, McDougal JS, Ramsey RB. Coin-
Med 1986;314:1387-8 cidental appearance of LAVlHTLV-III antibodies in
71. Gilkey FW. Opportunistic infections and Kaposi's sarcoma hemophiliacs and the onset of the AIDS epidemic. N Engl
in homosexual men [letter]. N Engl J Med 1982;306:934 J Med 1985;312:483-6
72. Jensen OM, Mouridsen HT, Petersen NS, Jensen KH, Thom- 75. Madhok R, Melbye M, Lowe GOO, Forbes CD, Froebil KS,
sen K, Ulrich K. Kaposi's sarcoma in homosexual men: Bodner AJ, Biggar RJ. HTLV-III antibody in sequential
is it a new disease? [letter] Lancet 1982;1:1027 plasma samples from haernophiliacs 1974-84 [letter]. lan-
73. Leonidas J-R, Hyppolite N. Haiti and the acquired immu- cet 1985;1:524-5