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Berkel, M. van

Boers, M.

Graaf, P. de

Meer, J.W.M. van der

Peeters, A.J.

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Shigellosis and AIDS : Report of a case and brief review of the literature

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Netherlands Journal of Medicine. 34 (1989) 93-97  
Elsevier
93
N JM  00193
Brief Report
Shigellosis and AIDS. 
Report of a case and brief review of the literature
M. Boers \  A.J. Peeters  \  M. van Berkel 2,
P. de G r a a f  4 a n d  J .W .M .  van  d e r  M eer  3
Department of Internal Medicine. Subdepartments of 1 Rheumatology, ~ Intensive Care, and ' Infectious 
Diseases, University Hospital. Leiden; 4 Department of Internal Medicine. St. Anthoniushove, Leidschendam
(Received 26 November 1987; revision received 11 March 1988; accepted 20 March 1988)
This report describes the fatal outcome of an infection with Shigella flexneri in a 
39-yr-old man with the acquired immunodeficiency syndrome (AIDS). The pertinent 
literature is reviewed. Neth J Med 1989;34:93-97.
Keywords:  AIDS; Shigella flexneri’, Cryptosporidium
Introduction
Shigellosis can be diagnosed in approximately 2% of male homosexuals evaluated 
for diarrhoea [1,2], and is regarded as a sexually transmitted disease in these 
individuals. Infections caused by Shigella flexneri predominate in these patients.
Although most pathogens responsible for chronic diarrhoea in homosexual men 
have been reported to cause infections in patients with the acquired im m uno­
deficiency syndrome (AIDS), only a few reports mention infections with Shigella. 
We report the case of a patient with AIDS, in whom the combination of atypically 
severe and protracted shigellosis together with cryptosporidiosis and unwarranted 
treatment with prednisone led to his early demise.
Case History
A 39-yr-old bank employee was admitted to a hospital because of persisting fever 
and diarrhoea after a holiday in the Himalaya region. Trophozoite amoebae had 
been reported in a stool specimen examined during the trip, and several unspecified 
drugs had failed to relieve the symptoms.
On admission the patient was found to be malnourished and obtunded.  Fever
Correspondence to: Dr. M. Boers. Dept, of Rheumatology. Building 1. C2-Q. University Hospital, P.O. 
Box 9600. 2300 RC Leiden.
0300-2977/89/S03.50  1 1989 Elsevier Science Publishers B.V. (Biomedical Division)
94
and hepatomegaly were noted. Sigmoidoscopy with biopsy revealed nonspecific 
haemorrhagic ulcerating colitis. Repeated examinations of stool, urine and blood 
failed to identify a causative organism. Serological testing revealed the presence of 
antibodies against hepatitis B “ c" and “ e" antigen. On a presumptive diagnosis of 
infectious colitis the patient was sequentially treated with ampicillin, chlor­
amphenicol and metronidazole over a period of 2 wk without clinical improvement. 
Inflammatory bowel disease was suspected and prednisone (initial dose 60 m g /d a y )  
and sulphasalazine were then administered. Unfortunately, this led to a toxic 
megacolon 20 days after the initiation of corticosteroid therapy, and massive rectal 
bleeding necessitated emergency right hemicolectomy and ileostomy.
A multiresistant strain of Shigella flexneri, type III was isolated from the resected 
colon specimen. Postoperative complications included Staphylococcus aureus and 
Candida albicans bacteraemia. Subsequent stool and ileal fluid specimens grew 
Shigella during a period of 4 wk despite adequate antibiotic therapy.
Ten weeks after admission all medications had been stopped. Profuse watery 
diarrhoea persisted, and bilateral pulmonary alveolo-interstitial infiltrates devel­
oped. Anaemia, hypovolaemia and azotaemia were followed by shock and cardiac 
arrest. After resuscitation the patient was transferred to the Leiden University 
Hospital.
On admission the patient was severely ill, cachectic and anuric. Ileal fluid 
production amounted to 300 m l /h .  Laboratory results included a low normal 
lymphocyte count with an almost complete absence of T-helper cells. Several species 
of Candida, but no other microorganisms were cultured from specimens of sputum, 
bronchial lavage fluid, urine and stool; cultures of blood and bone marrow 
remained sterile.
The patient improved temporarily after ventilatory support,  rehydration, 
parenteral nutrition and blood transfusions. However, diarrhoea and fever persisted 
and pulmonary infiltration worsened. The patient died of intractable respiratory 
failure, approximately 5 months after the start of symptoms. On the day of his 
death, his relatives finally disclosed his homosexual lifestyle.
Test results received after death revealed the presence of anti-HIV antibodies by 
enzyme linked immunosorbent assay, confirmed by Western blotting. Solid phase 
immunoassay [3] revealed the presence of HIV antigen. At autopsy, massive 
interstitial pneumonitis was found. Only Cytomegalovirus grew from lung tissue. In 
the colon and ileum, extensive infection with Cryptosporidium w'as noted. In 
retrospect, these organisms were also visible in the earlier sigmoid biopsy and 
colectomy specimens. No Shigella or other microorganisms were isolated from the 
intestinal tract. Other autopsy findings included mesenterial adenopathy with 
lymphocyte depletion, and granulomatous hepatitis and nephritis with microscopic 
and bacteriological evidence of Candida infection.
Discussion
This patient presented with intractable diarrhoea of unknown cause during a 
holiday in the Himalayas. Once corticosteroid therapy had led to the development
Reported cases of shigellosis in patients with AIDS *
TABLE 1
Source No. of 
patients
Organism Cultured from 
blood stool
R ecu rrence /  
persistence 
during therapy
Other intestinal 
infections
Remarks
Bell et al. [7] 5 4 flex 
1 sonn
1 /5 V 5 3 / 5  * * CMV colitis in all 5
Prummel et al. [8 ] 1 flex --------- + * * Adenovirus colitis Kaposi sarcoma in stomach 
and small bowel
Reiss el al. [9] 1 flex + HSV proctilis Contact  of patient described 
by Prummel, acute HIV infec- 
tion; AIDS-related complex; 
lost to follow-up
Turk et al. [10] 1 9• --------- + --------- Giardia. C. difficile, 
Cryplosporidi urn
Small bowel Kaposi sarcoma
Whimbey el al. [11] 1 sonn + + — 1 of 38 bacteraemias in 336 
patients with A lD S
Glaser et al. [5] 1 flex — + 9• 1 of 8 patients with 
S. typhimurium bacteraemia
Glupczynski et al. [4] 1 flex + female Zaire national; 
persistent diarrhoea with 
negative cultures after 
treatment
Mandell et al. [12] 1 flex + + ---------
Fever and diarrhoea reported in all cases. ** Maximum duration of persistence: 4 wk
96
of a toxic megacolon, S. flexneri was finally cultured from the resected specimen. It 
then proved difficult to eradicate. The Shigella was most probably acquired in 
Nepal; the initial therapy may have hampered a microbiological diagnosis. In 
retrospect the bowel was also infected with Cryptosporidium and heavily colonized 
with Candida. The diagnosis AIDS was made on the basis of his homosexuality, 
multiple opportunistic infections and virtual absence of T-helper cells, and con­
firmed by the presence of anti-HIV antibodies and circulating HIV antigen.
In most reviews Shigella species are not mentioned as pathogens for AIDS 
patients. A computerised literature search revealed 12 other Shigella infections 
associated with AIDS or AIDS-related complex (Table 1). S. flexneri was the 
predominant organism, with positive stool cultures in 11 and positive blood cultures 
in 4 cases. Watery diarrhoea, sometimes with an admixture of blood and mucus was 
the most frequent presenting symptom. In one patient, only the blood culture was 
positive [4], and the negative stool culture, as in our patient, suggests an enteritis 
with low numbers of bacteria or an extra-intestinal primary focus. Details concern­
ing therapy were available in 9 cases; in 4 of these patients recurrent or persistent 
episodes of infection (lasting for up to 4 wk) were noted in spite of adequate 
therapy, as in our patient.
The rarity of shigellosis in AIDS suggests that T-cell mediated immunity is less 
important in host defense against Shigellae than against other intestinal pathogens,
e.g. Salmonellae [5,6]. This may be the reason that shigellosis is an infrequent 
finding in patients with AIDS. In this setting, it is tempting to speculate that a 
second intestinal infection is necessary to trigger overt shigellosis in an AIDS 
patient who is a carrier of this organism. In our patient, extensive intestinal 
infestation with Cryptosporidium was noted, and systemic and enteric candidiasis 
w'as present. Bell et al. reported 5 patients with coexistent Shigella and CMV colitis 
[7]. In our patient CMV was cultured only from the lung, but CMV colitis cannot be 
completely ruled out.
In conclusion, shigellosis seems to be a relatively infrequent finding in patients 
with AIDS. Presentation may be atypical, and response to adequate therapy may be 
poor. The available literature and the present case report suggest that concomitant  
intestinal infections may play a role in the persistence of the Shigella. Finally, the 
case illustrates that persistent diarrhoea without a specific cause, or without im­
provement on adequate therapy should prompt a careful search for opportunistic 
enteric pathogens, e.g. Cryptosporidium.
Acknowledgement
We would like to thank Dr. J. Goudsmit  for performing the viral assays.
References
1 Sohn N, Robilotti JG. The gay bowel syndrome. A review of colonic and rectal condit ions in 200 
male homosexuals. Am J Gastroenterol 1977;67:478-484.


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