British forirnal of’ Urology ( I 976), 48, 61 7-62 1

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The Endemic Bladder Stones of Indonesia-Epidemiology and Clinical Features KAMARDI THALUT, AHMAD RIZAL, J. G . BROCKIS, R. C. BOWYER, T. A. TAYLOR

and

2. S. WISNIEWSKI

Padang General Hospital, West Sumatra, Indonesia and Royal Pertli Hospital, Perth, Western Australia

Bladder stones are common amongst young male children of the lower social and economic classes of the Indonesian rural population. As a joint Indonesian-Australian project, we have studied 87 children suffering from the condition. Here we report our findings on the epidemiology and clinical features whilst a second paper provides details of biochemical investigations designed to disclose the basic cause of the condition. The area selected for study was West Sumatra, which has a population of approximately 24 million, stretching along the west coast of the island of Sumatra. Padang and Bukittingi hospitals serve the area. 200 new cases of children with bladder stones report to these hospitals each year, with an overall incidence of 8*3/100,000 population per year. Whilst the most populous area is the city of Padang, which is on the coast, very few patients come from there-the largest number are from Pariaman, a low-lying rice-growing rural area to the north The geographical distribution of our patients is shown in Figure 1. The area, within a degree of the equator, is a low-lying coastal plain growing rice, behind which are high mountains with jungle. Bukittingi is a hill town with a moderate climate. Most of the affected children come from poor homes built on land adjoining streams from which the rice paddies are flooded. The peak age of onset in our series is between 2 and 4 years (Fig. 2) and there is a ratio of 12:1 males to females (which compares with ratios of 20 males to 1 female in China and Syria (Thomson, 1921; Brown and Brown, 1941); 13:l in India (Andersen, 1962) and 1O:l in Thailand (Halstead and Valyasevi, 1967). An investigation of the children’s families shows that it is rare for more than 1 child to be affected, even though large families are the rule. The children are of average weight and height and do not show any external appearances of malnutrition. There is no association between the development of stones and kwashiorkor and no evidence of Vitamin A or I> deficiency so far as we are able to determine. The commonest clinical presentation is for a child to pull on his penis, which seems to help to initiate micturition and to relieve strangury. The sight of a child doing this is almost diagnostic! Other common symptoms are suprapubic pain and frequency of micturition. Retention of urine due to impaction of the stone is rare, and gross haematuria, as distinct from the presence of red cells in the urine is also infrequent. N o child has been known to suffer from renal colic and the only renal symptoms that have occurred can be attributed to ureteric refux and infection. 50 patients have had preoperative intravenous urograms: 8 showed bilateral urinary tract dilatation and 13 unilateral. Reflux was present in these children and postoperative X-rays taken much later have shown a trend to return to normality, suggesting an association with urinary tract infection. Diarrhoea is common amongst young children in this tropical area. Whilst mothers of the affected children frequently mentioned this, it has proved impossible to show that there was a higher incidence in children with stone; it is also very difficult to measure the severity of diarrhoea attacks. Read at the 32nd Annual Meeting of the British Association of Urological Surgeons in London, June 1976. 61 7

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Fig. I . Map of West Sumatra showing rural distribution of 87 patients.

Recurrence after removal of a stone is very rare, occurring only once in our series. The diet of the children who come from a rural low socio-economic group is of breast milk supplemented by polished rice. They do not receive any dairy products and the only other source of protein is a minute amount of fish. For the younger children, the rice may be predigested in the mother’s mouth. Thus almost the only source of protein is mother’s milk or rice. This diet is also low in fat and in inorganic phosphate. Human milk, unlike cow’s milk, does not have a high phosphate content. The water supply in country areas is from shallow wells-the water level being that of the paddy fields. It is sometimes cleared by filtration through river sand, but often has a high bacterial count. Padang water supply is piped and chlorinated but does not reach all the inhabitants of the town and there are many wells in the suburbs.

Discussion Endemic bladder stones were common in 18th- and 19th-century Europe. Civiale (1838) reported from France that 45 of 5,383 cases of stone occurred in children below the age of 20. William Cheselden (1723) of St Thomas’s Hospital, London, operated upon 213 cases of bladder stone, of whom 102 were under the age of 10 years and another 50 between 10 and 20.

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619

0Male Female

Age it1 )ear5 Fig. 2. Age and sex of 87 bladder stone patients.

Yelloly (1829) and Crosse (1835) reported from the Norfolk and Norwich Hospital, England, that endemic stones of childhood were very common in the eastern grain growing part of that county as compared with the west which had mixed farming. Vesical calculus practically ceased to exist as an endemic disease in northern Europe between the years 1910 and 1930 (Lett, 1936; Thomas, 1949). The condition still exists in a belt of countries beginning in southern Russia and the Balkans (Ratid, 1935), passing through Egypt (Loutfi, Hamid et at., 1972); Turkey (Eckstein, 1961); Israel (Levy and Falk, 1957); Lebanon Syria (Brown and Brown, 1941); Persia (Hedayat et a/., 1970); India (Anderson, 1962; Aurora, Ramalingaswami and Gaitonde, 1964); Thailand (Gershoff, Prien and Chandrapanond, I963 ; Halstead and Volyasevi, 1967); Laos (Westermeyer, 1971); Indonesia (Maruna, 1959; Tan, 1964); to China (Thomson, 1921) and Japan (Inada ct al., 1958). The absence of bladder stones amongst poor children in the maize-eating areas of Africa may be significant (Vermooten, 1941). Processing does not lower the phosphate content of this cereal. Lonsdale, Sutor and Wooley (1968) have shown that the major constituent of endemic bladder stone is ammonium acid urate, though in certain areas of Thailand where a diet high in oxalate content is consumed, bladder stones contain appreciable amounts of calcium oxalate (Suvachittanont, Meksongsee and Dhanamitta, 1973). In infected stones, the presence of calcium carbonate and magnesium ammonium phosphate has been detected, but not in those where the urine has been reported to be sterile. The remarkable feature of the stones we have analysed has been their very high content of ammonium acid urate with minimal quantities of inorganic material. Oxalate has been usually absent from the analyses. Urine analysis shows that the poor children have a high acid load to excrete through the 4817-H

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urinary tract. The urine of all children is usually moderately acid, but the rice diet consumed (Sherman and Gettler, 1912) and attacks of diarrhoea both add to urinary acidity. We have shown that the buffering of this acid load by phosphate buffer is inadequate (probablj, because of a poor phosphate content of the diet), and high levels of ammonium ion are produced by the kidneys. Urinary infection does not account for this excess ammonia as only about half the children with bladder stones have infected urinary tracts, and coliforni organisms, rather than 61. protcws, are often present. Ammonialcreatinine ratios are higher in the patients with non-infected urine than normal controls. It is possible that increased production of glutamine in the liver is a necessary precursor for the renal production of ammonia (Pitts, 1973). An excess of glutamine fed into the purine production cycle might be expected to result in increased formation and excretion of urate even though glutamine is not usually regarded as a ratcregulating factor in the production of uric acid. In Thailand, where oxalates are found to be components of children's vesical calculi due to ;I diet high in oxalate, a low urinary phosphate might be expected to encourage crystal growth. There is evidence that phosphate supplements to the diet of children i n this area reduce the incidence of bladder stones (Chulkaratana, Van Reen and Valyasevi, 1972).

Summary Bladder stone is a relatively common disease of childhood i n West Sumatra with an incidence of 8.31 100,000 population per year. We have studied 87 cases with a peak age of onset of 2-4 years and a male/female ratio of I2 : I . The majority of these are from poor families with a diet low in protein and phosphate. Diarrhoea is common. The composition of the stones is primarily ammonium acid urate. Patients with sterile urine have shown significantly higher levels of urinary ammonia than controls, and only approximately 50% of patients' urine were infected. This is consistent with excretion of a high acid load, due both to an acidogenic rice diet and diarrhoea, combined with a low level of phosphate. This condition was once endemic in Europe but is now confined to a belt of countries from the Balkans through Asia. The authors would like to acknowledge financial assistance received from the Royal Perth Hospital, Research Foundation and University of Western Australia, work done by the laboratories of the Royal Perth Hospital, Research Centre, and the co-operation of the Dean of the Faculty of Medicine, University of Andales. Dr Soemnrto and Dr Mispah of Bukittingi.

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The Authors Kamardi Thalut, MD, Surgeon, Padang General Hospital. Ahmad Rizal, MD, Surgical Registrar, Padang General Hospital. J. G. Brockis, ChM, FRCS, FRACS, Associate Professor, University of Western Australia. R. C. Bowyer, PhD, Biochemist, Royal Perth Hospital, Western Australia. T. A. Taylor, FRACS, Senior Surgical Registrar, Royal Perth Hospital. Z. S. Wisniewski, MBBS, Surgical Registrar, Royal Perth Hospital. Correspondence to: J. G. Brockis, University Department of Surgery, P.O. Box X2213, Perth, Western Australia.

The endemic bladder stones of Indonesia---epidemiology and clinical features.

Bladder stone is a relatively common disease of childhood in West Sumatra with an incidence of 8.3/100,000 population per year. We have studied 87 cas...
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