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Volume 38, Issue 1, Pages 62-64 (January 2003)


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HIV-positive African children with rectal fistulae

Presented at the 49th Annual Congress of the British Association of Paediatric Surgeons, Cambridge, England, July 23-26, 2002.

R Wiersma

Abstract 

Background/Purpose: Human immunodeficiency virus (HIV) disease is an increasingly common infection in children in sub-Sahara Africa. Rectal fistulation is one such condition with which these patients present to the paediatric surgeon. This appeared to be an exclusively female condition until 2 male patients were treated recently. Methods: A 6-year (1996 through 2001) retrospective study found 39 children presenting with HIV-related rectal fistulae. Thirty-seven girls were seen with rectovaginal fistulae (RVF), and there is supportive documentation showing an increase in this condition throughout Southern Africa. Until now, boys have not been described with this condition. The author presents 2 boys who complete this spectrum of HIV-related acquired rectal fistulae. Results: All patients were found to have rectal fistula at the dentate line. In girls it varied in size from pin-point to 5 mm diameter, tracking anteriorly into the vagina. When closure of the fistula was attempted, it broke down. The 2 boys had a large fistula, which tracked to the prostatic urethra on the right of the verumontanum. The first patient underwent a successful repair. The second patient had a “Y”-shaped fistula based at the dentate line, with the second limb passing into the bladder. The parents refused further treatment and took the child home. Conclusions: HIV disease affects increasing numbers of children. A spectrum of rectal fistulae now has been seen in both girls and boys. These acquired rectal fistulae arise at the dentate line in both genders. Girls with these fistulae are seen more commonly, presenting with RVF. The closure of a fistula has only been successful in one boy. J Pediatr Surg 38:62-64. Copyright 2003, Elsevier Science (USA). All rights reserved.

Article Outline

Abstract

Materials and methods

Results

Discussion

Discussion

References

Copyright

In a period of 20 years, human immunodeficiency virus (HIV) has changed from a newly recognized clinical entity to one of the most devastating diseases humankind has ever faced. HIV and AIDS now is the leading cause of death in sub-Sahara Africa. Worldwide, it is the fourth biggest killer.1 It is estimated that 2.4 million children infected with the HIV virus are living in sub-Sahara Africa.2 Seventy-five percent of these children die before their fifth birthday, and a significant proportion will require medical attention as they deteriorate with a loss of the CD4+ cell fraction, whereas some develop AIDS-defining conditions.2

Among the surgical presentations of HIV-positive children, the acquired rectal fistula has been a common pathology that has increased significantly over the past decade.3, 4 The aim of this report was to review the paediatric surgical spectrum of HIV-related rectal fistulae.

Materials and methods 

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The Department of Paediatric Surgery, University of Natal, in Durban, South Africa, saw 39 patients with HIV-related rectal fistula from 1996 to 2001 inclusive, on an in- and out-patient basis.

Of the 39 children with rectal fistulae, 37 were girls with ages ranging from 4 weeks to 11 years (mean, 15.5 months). All patients were born with a patent anus initially had normal passage of stool. The passage of stool per fistula was preceded by a severe diarrhoeal episode. In most, the fistula opened in the vagina, but a few were noted to track into the labia majora and perineum (Fig 1).


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Fig. 1. Rectal fistula opening on lower vagina/perineum.


No account of local anorectal trauma was elicited from any patient.

Two boys were seen in this period. The first was a fit 4 year old. He presented with the passage of stool per urethram after a severe diarhoeal illness at the age of 3 years. The second boy, a wasted 1 year old, presented with drainage of urine per urethram and per rectum. When he was born he passed urine through the urachus as a result of posterior urethral valves, but after a severe bout of diarrhoea, this had decreased, leaving him with a watery stool smelling of urine.

Results 

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Of the total 39 patients seen, only 13 patients were admitted, and the remainder were treated conservatively and reviewed on an out-patient basis.

Examination of the girls showed the fistula to be situated at the dentate line and tracking ventrally into the vagina or perineum. Those admitted for an examination under anaesthesia were noted to have fistulae ranging from pin size to 5 mm diameter. The rectal mucosa usually was irregular and inflamed with histology that showed nonspecific inflammatory features with ulceration and acute on chronic proctitis. No features of tuberculosis were seen, but 3 patients had a cytomegaloviral infection of the rectal mucosa.

Initially, girls with an acquired rectovaginal fistula were admitted for investigation and given a colostomy to divert the faecal stream before an attempted closure of the fistula. The various attempts at closing the fistulae were not successful in any girls because of poor wound healing and in many they were made worse. Once the fistulae were found to be HIV related and surgery unlikely to be successful, diverting colostomies were no longer done in girls, unless compelled by septicaemia or advancing perineal disease.

Two boys were seen and both were proven to be HIV positive on enzyme-linked immunosorbent assay (ELISA) tests. Examination under anaeasthesia showed the fistulae to be intersphincteric. The fistulae opening in the rectum were situated at the dentate line and found to track into the prostatic urethra, as shown on micturating cystourethrogram (Fig 2), in both patients on the right side of the verumontanum.


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Fig. 2. MCUG shows leakage through rectoprostatic urethral fistula.


The older child was given a diverting sigmoid colostomy for the rectourethral fistula. He subsequently underwent a “Peña approach,” separating the anterior rectum from the prostatic urethra, to close the fistula. His colostomy also has since been closed successfully. The younger boy was found to have one large “Y”-shaped intersphrinteric rectal fistula. The 2 ventral prongs of the fistula led into the prostatic urethra and bladder. The urethral valves were fulgurated, but the parents refused further treatment for the fistulae and he was sent home.

Histology of these fistulae showed an acute on chronic proctitis. In the older boy there was a nonspecific focal cryptitis, and colonic biopsy showed replacement of smooth muscle and nerve fibres by fibrous tissue. Viral and bacteriologic findings in both patients were negative. No other pathologic features were identified.

Discussion 

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Since HIV disease was first diagnosed in Southern Africa, there has been a steady increase in the number of patients affected. Although the official incidence in our area (Province of KwaZulu-Natal) has reached 36% in the year 2000,2 the true incidence of HIV disease among children in our population cannot be defined. However, an increase in number of patients with HIV-related rectal fistulae has been noted over the last years, from 5 patients in 1996 to 11 in 2001. An escalation in HIV-related rectal fistulae in girls has been well documented in Southern Africa.3, 4, 5, 6 No reports of a similar pathology in boys have to date been found in the literature.

The pathophysiology of HIV-related rectal fistulae is not known. Such fistulae have been noted to arise from the dentate line, and anal gland infections are proposed as the initiating factor.7 There is, however, a recognized clinical pattern in these patients, in which HIV-related rectal fistula follows a diarrhoeal infection, but the mechanism of this process has not been elucidated. Furthermore the significance of the female preponderance and the broad intimate relationship between the vagina with the rectum as opposed to the low male incidence and the comparatively thicker and smaller surface between anorectum and anterior male structures is a matter of speculation.

The management of girls with rectovaginal fistulae has changed over the years because of our inability to close these acquired fistulae surgically. These fistulae were found to have minor local effects; this, and the lack of systemic sequelae in the girls, has lead us not to intervene at all. These girls now are reviewed on an out-patient basis for systemic complications. None have been noted to date.

Our experience with the boys has been different, because the mixing of faeces and urine had given rise to urinary tract infections and urinary incontinence. A temporary diverting colostomy in one boy gave him relief. The subsequent closure of the fistula and later the colostomy has greatly improved the quality of life of this child, although his general condition is deteriorating in consequence of his HIV.

With the spread of HIV more children will be affected and the full spectrum of HIV-related acquired rectal fistulae will become more common. It is important to focus on the needs of the child and not be overwhelmed by the condition.

Discussion 

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A. Millar (Cape Town, South Africa): We have a slightly more aggressive approach and give them antiretroviral therapy for about 6 weeks, prophylactic therapy, raise the CD4 count, and then repair the fistula under colostomy cover, and they do very well.

R. Wiersma (response): The problem, as you know, is that the government does not recognise the condition of HIV/AIDS. The widespread use of HIV treatment therefore is not available to us. Hence, our conservative approach.

S. Suita (Fukuoka, Japan): We have many cases of rectal fistula in failure, and also in China and India. They are not associated with HIV problems. What is the racial background of your patient group?

R. Wiersma (response): All our patients were black African children.

References 

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1. 1 Joint United Nations Programme on HIV/AIDS update. December 2001. AIDS epidemic update. Washington, DC: WHO Press; December 2001;.

2. 2 Issued by the HIV & STD Directorate, Department of Health Department of Health: Managing HIV in children. March 2000;.

3. 3 Borgstein ES, Broadhead RL. Acquired rectovaginal fistula. Arch Dis Child. 1994;71:165–166. CrossRef

4. 4 Oliver MJ. Spontaneously occurring rectovaginal fistulae in children and adults with HIV infection. East & Central Afr J Surg. 1995;1:23–25.

5. 5 Schoeman CJ, Hallbauer U. Rectovaginal fistulas in HIV-infected children (letter). S Afr Med J. 2001;91:91–92. MEDLINE

6. 6 Manookian CM, Sokol TP, Headrick C, et al.  Does HIV status influence the anatomy of anal fistulas?. Dis Colon Rectum. 1998;41:1529–1533 (letter). MEDLINE | CrossRef

7. 7 Banieghbal B, Fonseca J. Acquired rectovaginal fistulae in South Africa. Arch Dis Child. 1997;77:94.

Department of Paediatric Surgery, Nelson R. Mandela School of Medicine, University of Natal, Durban, South Africa

 Address reprint requests to Mr R. Wiersma, Department of Paediatric Surgery, Nelson R. Mandela School of Medicine, University of Natal, Private Bag 7, Congella 4013, Durban, South Africa.

PII: S0022-3468(02)63019-2

doi:10.1053/jpsu.2003.50011


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