5 Kinney JS, Gross TP, Porter CC, Rogers MF, Schonberger LB,

Hurwitz ES. Hemolytic-uremic syndrome: a population-based

study in Washington, DC and Baltimore, Maryland. Am J Public

Health 1988;78:6456 ORyan M, Prado V. Risk of the hemolytic-uremic syndrome

after antibiotic treatment of Escherichia coli O157:H7 infections.

N Engl J Med 2000;343:127137 Sheth KJ, Swick HM, Haworth N. Neurological involvement in

hemolytic-uremic syndrome. Ann Neurol 1986;19:9038 Lynn RM, OBrien SJ, Taylor CM, et al. Childhood hemolytic

uremic syndrome, United Kingdom and Ireland. Emerg Infect Dis

2005;11:59069 Bhimma R, Rollins NC, Coovadia HM, Adhikari M. Post-dys-

enteric hemolytic uremic syndrome in children during an epidemic

of Shigella dysentery in Kwazulu/Natal. Pediatr Nephrol1997;11:5604

Frontoethmoidalmeningoencephalocoelerepair in Cambodia:outcomes and costcomparisonsJ Gollogly* N Oucheng* G Lauer,

T Pinzer, F Lauwers F E Roux

W Singleton** S Douglas

*Childrens Surgical Center, Kien Khleang, Chroy Changvar,Phnom Penh, Cambodia; Department ofCranio-maxillofacial Surgery; Department of Neurosurgery,Carl Gustav Carus University Hospital, Dresden, Germany;Department of Cranio-facial Surgery and Federation ofNeurosurgery, University Hospitals, Toulouse, France;**University of Oxford Medical School, Oxford, UK

Correspondence to: Dr J Gollogly, Childrens Surgical Centre,Kien Khleang, Chroy Changvar, PO Box 1060, Phnom Penh,CambodiaEmail: jim@csc.org

TROPICAL DOCTOR 2008; 38: 167170DOI: 10.1258/td.2007.070124

SUMMARY In Cambodia, spina bifida is rare, butfrontoethmoidalmeningoencephalocoeles (MECs) arecommon. Mean life expectancy for patients with congeni-tal MECs may be ,20 years, but the complex treatmentrequired has not been available in the country untilrecently. During visits by combined neurosurgical/cranio-facial teams from both Germanyand France, a method ofrepair has been developed that is suitable for the localconditions, affordable and has allowed Cambodian sur-geons to learn how to successfully treat MECs.The surgicaltechnique and initial results with 30 patients have beendescribed in a previous publication.This paper presents theoutcomes of128 cases and illustrates that it is cost-effectivefor these patients to be treated in Cambodia.

Introduction

Most Western health-care professionals are familiar withspina bifida. It is caused by failures of the closure of the

caudal neural tube, most commonly affecting the lumbo-sacral spine and presenting with an obvious meningocoeleor meningomyelocoele, with severe neurological deficits.More rarely, occipital encephalocoeles are also seen in theWest. Frontoethmoidal meningocoeles or meningoencepha-locoeles (MECs), conversely, are congenital failures of theclosure of the bony sheath of the neural tube at the rostralend (sinciput), which are seen in certain areas of southeastAsia. A defect between the frontal and ethmoidal bones inthe skull allows herniation of the meninges, cerebrospinalfluid (CSF) and usually parts of the anterior frontal lobesof the brain into the forehead, nose or orbits. These three des-tinations of the hernial sac give rise to three subtypes ofMEC: nasofrontal, nasoethmoidal and naso-orbital.1 Often,this deformity does not seem to be associated with any neuro-logical deficit, but it is strongly associated with gross changesin facial features: the presence of a mass, pathology in theorbit and eye and an elongated nose.2 These cosmeticabnormalities result in extremely low patient self-esteemand social exclusion.

The aetiology of MEC in Cambodia is poorly understoodand multiple hypotheses have been advanced. Thu and Kyudemonstrated variation in MEC incidence by season ofbirth in Burma. It has been suggested that, in the Burmesecontext, aflatoxin growing on moldy rice in the rainyseason and consumed by the mother at the critical periodof pregnancy may interfere with embryonic folic acid metab-olism in such a way that mesodermal ingrowth fails to separ-ate neuroectoderm from the skin of the frontonasal process,resulting in MEC through failure of formation of interposingbone.3

Cambodias health infrastructure was destroyed by decadesof war in the late 20th century, including the infamous KhmerRouge regimen of 19751979. Since the Vietnamese occu-pation in 1979, Cambodia has received billions of dollars inforeign aid, which has accelerated since the United Nationsoversaw the first democratic elections in 1993. Despite this,the medical system remains grossly deficient compared withthe neighbouring nations of Thailand and Vietnam.4

Secondary and tertiary care is scanty and often availableonly in facilities funded and run by non-governmental organ-izations (NGOs). The training of Cambodian doctors isimproving and around 36 doctors per year are now receivinga year of training abroad, mainly in France. However, a func-tioning neurosurgical or craniofacial service manned byCambodian personnel has yet to emerge.

Many foreign surgical teams visiting Cambodia since1979, usually doing plastic surgery, have encounteredpatients with MECs. Their attempts at operative MECrepair were carried out through the face, with no neurosurgi-cal input and generally had poor results. In recent years,however, neurosurgeons have begun visiting the countryand some Cambodian surgeons have received neurosurgicaltraining. In addition, some patients have been sent abroadto have their deformities corrected. However, the costsassociated with this were substantial, and obviously fewpatients would be lucky enough to be given that option.

In nine years of working as a surgeon in Cambodia at theChildrens Surgical Center (CSC), the senior author (JG) hasseen well over 150 patients with frontoethmoidal MECs, butonly three with a lumbar meningocoele and two with an occi-pital meningocoele. The present population of Cambodia,following 30 years of war, is now preponderantly made upof younger people, with 8 million (63%) of the total popu-lation of 13 million being under the age of 25 years. If theprevalence of MEC in Cambodia is similar to that inBurma (15 cases in 100,000 people3) there are likely to beapproximately 1400 young patients suffering from this

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condition in the country (assuming that there is no excessmortality in Cambodia).

From over 150 MEC patients JG has only encounteredthree older than 25. This suggests a surprisingly low preva-lence in this older age group, even after taking into consider-ation increased mortality during the Khmer Rouge period.Such a skewed age distribution implies that untreatedMECs lead to premature mortality. The parlous state ofmany of the MECs that have been seen at CSC suggeststhat the mechanism of this may well be CSF leakage result-ing in fatal meningitis.

Personal and anecdotal experiences of sending patientswith MECs to the developed world for treatment revealedthat hospital and surgical charges alone had beenUS$35,000 for one patient in Canada; US$70,000 foranother in USA; US$50,000 each for two patients in Japanand about US$20,000 each for four patients treated inSingapore. Given the likely numbers of patients, these costsare unacceptable. It was therefore necessary to devise anoperation that could be carried out in Cambodia, byCambodian staff, under Cambodian conditions.

Methods

Starting in 2004, Medecins du Monde (MDM), (anInternational NGO with branches in Germany and France),supplemented their plastic/craniofacial surgical teams withneurosurgeons. Attention was focused upon finding a sol-ution to the problem of MECs, after a frontal approachalone had resulted in many recurrences. There was no lackof available patients .70 were awaiting treatment atCSC, and many others were referred once word spread thatthe condition could be treated. The German team consistedof a neurosurgeon and a craniofacial surgeon alone, whilethe French team had two similar surgeons plus nurses andanaesthetists. From the Cambodian point of view, the anaes-thetists and nurses were not essential, but the surgeons wereindispensable. Over the course of four years, in eight differ-ent time periods, 128 patients were operated upon.6

Conditions for surgery in a small Cambodian hospital suchas CSC are not ideal. Laboratory reports are not immediatelyavailable. Radiology and other investigations are not on site,blood transfusions are difficult to arrange and postoperativecare is of variable quality. Patients are usually nursed bytheir families, and medications are handed to the patientscaregiver, with instructions for use that may, or may not,be followed. Nevertheless, if a solution was to be found, itwould have to be found using the facilities and personnelat hand, and at affordable costs.

In Cambodia, computerized tomography scanning has beenavailable since about 2003 (currently at a cost of aboutUS$100) and magnetic resonance imaging since 2004(US$200). It was decided, in view of our understanding ofthe pathology, that we should not employ either of theseinvestigations, nor even a simple radiograph: instead, weopted to explore the face at surgery, and take the necessarycorrective measures. Our preoperative assessment was solelyclinical in the vast majority of cases, and no laboratory orradiological investigations were routinely performed unlessthe patient was clinically unwell in which case referral toan appropriate level of care was arranged. Meticulous intrao-perative haemostasis allowed us to use only one postoperativeblood transfusion in this series of cases.

A Cambodian surgeon who had worked as a paediatricsurgeon, who had returned from two years of further trainingin France, was designated as the one to learn the operativetechnique. He subsequently participated in the last 41 ofthe operations, while more junior surgeons assisted on a

rotating basis to allow them to become familiar with the pro-cedures and skills.

Results

It was decided at the outset that teenagers and preteens wouldbe the population most easy to treat, as they were the largestgroup of patients and more likely to have shorter recoverytimes. The age range was expanded to include younger chil-dren and infants as experience was gained. By January 2007,the youngest patient to receive an operation was only fourmonths old and weighed 7 kg. At the end of February2007, 128 patients had been treated and had their MECsrepaired during the programme (16 of had complicationsrequiring a second operation, and one had four operations).Unfortunately, follow-up is always difficult in Cambodia,but the known postoperative complications of MEC repairin our series can be seen in Table 1.

In 2004, CSC calculated the approximate cost of eachoperations by dividing the total amount of money spentduring the year by the number of operations done, resultingin a figure of US$85 for each operation. This was donewithout differentiating between the resources consumed bylarger or smaller operations. Obviously, this figure could berevised up or down using different methods of accounting,but it does provide an average cost per surgical operation.Because these patients stay two or three weeks longer inthe hospital than many other patients, we have estimatedthat about US$15 extra is spent on them rounding upCSCs costs at about US$100 per MEC operation. By theend of 2006, this figure had increased to about US$150using the same accounting procedure, since the totalnumbers of operations had dropped slightly over the yearalthough the complexity had increased.

For the German team of just two members only (one cra-niofacial surgeon and one neurosurgeon), the total cost ofspending two weeks in Cambodia was approximatelyUS$4000, as each surgeon spent approximately US$1350on airfares and US$650 on hotel and living expenses.Given that, on average two MEC operations were completedeach working day (giving a total of 20 over the two weeks),the total average cost per operation was US$300: the Germanteams expenses were US$200 per operation and the ROSEcharity expenses were roughly US$100.

The French team usually consisted of at least fourmembers, as they routinely brought an anaesthetist and anurse in addition to the two surgeons. Their costs were

Table 1 Postoperative complications ofmeningoencephalocoeles repair at the Childrens SurgicalCentre, Cambodia

Complication No. of children %

Death 4 3.1Blindness 1 0.7Meningitis (successfully treated) 3 2.3Recurrence 4 3.1Proptosis 1 0.7Epiphora 21 16.4Strabismus 1 0.7Transient CSF leakage 10 7.8Fever 43 33.6Headache 44 34.3Seizures 5 3.9Vomiting 39 30.4Transient facial/forehead swelling 46 35.9Abdominal pain 2 1.4Local infection 13 10.1Lacrimal duct obstruction 1 0.7

CSF, cerebrospinal fluid

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therefore essentially double those of the Germans. Hence, thecosts for operations by the French team were at least US$400,plus US$100 ROSE costs i.e. a total of US$500.

Discussion

These results have been accepted in the Cambodian andexpatriate surgical community as very encouraging.However, this series did include four deaths and one veryserious complication. The first occurred in a 10-year-oldboy, who asphyxiated following surgery due to the delayedrecognition of his respiratory difficulties by the postoperativestaff in the recovery room. The second occurred in an11-month-old baby boy, who did not recover normal con-sciousness after his operation. He died three days later ofan unknown cause. A 15-year-old boy developed diarrhoeathree days after the operation, therefore requiring a bloodtransfusion, and fluctuated in and out of consciousnessbefore dying suddenly and unexpectedly. Anotherseven-year-old girl was noted to have bossing of the insideof the calvarium upon removal of the frontal bone flap, andher brain seemed very tense. The operation had beenuneventful, but when she had not recovered consciousness8 hours later, she was taken back to theatre for re-explorationto exclude cerebral compression by an intracranial haema-toma. No haematoma was found, and attempts to tap thelateral ventricles were unsuccessful, suggesting compressedor obliterated lateral ventricles. Her brain was decompressedas much as feasible. She did not recover consciousness afterthe second operation and died the following day. Since thisdeath, a policy of performing a fundoscopy in order to ident-ify papilloedema and raised intracranial pressure (ICP) afterthe induction of general anaesthesia has been instituted toavoid operating on patients with a raised ICP. Anine-year-old girl went blind after being taken home by herparents, against medical advice, after developing postopera-tive proptosis. She was driven across country in very windyand dusty conditions resulting in exposure keratitis to bothher corneas, with subsequent scarring and loss of sight.

These tragedies gave us pause for thought, but it wasdecided that if the natural outcome of not operating onpatients was death before the age of 25, then the risks of oper-ation were not great enough to cause us to terminate theprogramme. We believed that the improvement of social func-tioning and the increase in the lifespan of at least 90% of thepatients some perhaps to normal longevity justified therisks involved.

Using the figures published in reports from Burma andThailand, we have estimated that there are possibly about1500 cases of frontal MEC in Cambodia. It is quite obviousthat these patients cannot all travel to foreign neurosurgicalcentres for treatment. The financial sustainability of theMEC repair technique described is of paramount importance.

Cambodian government hospitals operate, in practice if notofficially, on a fee-for-service basis. Even if the MEC repairoperation was available via the government system, it wouldcost at least US$350 for the surgery alone prohibitivelyexpensive for the vast majority of patients who come fromvery poor rice-farming families. We believe that CSCscosts of US$300US$500 per operation compare favourablywith the likely costs of treatment given via the governmentsystem and very favourably with the cost of treating MECsabroad. While the long-term follow-up is incomplete, wehope that the procedure will result in the improved socialfunction and reduced mortality of patients. Given the poorprognosis for unoperated MECs, it seems quite possiblethat each operation will result in a net gain of 10 or more dis-ability adjusted life years (DALYs). Therefore, the cost per

DALY gained would compare favourably with publichealth interventions, including many of those aimed at pre-venting HIV transmission in Africa or southeast Asia.5

This seems to be a highly efficient use of charitablefunding. Furthermore, we believe that CSCs approachoffers the best of foreign expertise and enables local surgeonsto be trained in a procedure adapted to local conditions.

Local surgeons participated in every operation: no oper-ation was done without at least one Cambodian surgeon onthe team. One surgeon learned the whole technique so wellthat he was able to do the operation by himself by the endof the series. Another local plastic surgeon had his skillsimproved so much that he was able to do the facial recon-struction after the neurosurgery was completed when necess-ary (Figure 1). All the local doctors were fascinated by theprocedures and were grateful to be given the opportunity toparticipate in the operations and to learn the techniques.

The expatriate experts also benefited from this experience.Not only did they learn to cope in difficult conditions, butthey also learned to question their own practices in theirhome countries. Did they really need such a detailedwork-up for their own preoperative patients? Could theyoperate in a more efficient and conservative way, withoutall the frills and fancies of modern operating suites, usingfewer consumable materials such as sutures and gelfoam?Did they need so many assistants? Could they rememberhow to take care of patients themselves, without alwaysrelying on their juniors? It was a learning experience for all.

Conclusion

The cooperation between CSC and the German and Frenchteams of MDM led to the development and application of asimplified method of repairing frontal MECs, which was suit-able for a less developed country. Although there were fourdeaths and one severe complication, these constituted ,5%of the series. The skills necessary to treat these childrenwere passed on to local surgeons. This report seeks merelyto outline the costs and obvious benefits of doing this oper-ation where the disease is found, rather resorting to using ter-tiary care facilities in a more developed country. We wouldalso argue that our results demonstrate that surgery may, incertain circumstances, be as cost-effective as most otherhealth-care interventions.

Acknowledgements

The Childrens Surgical Centre is a registered Charity inAlaska, USA (710470) the Kadoorie Charitable Foundation,western embassies in Cambodia and private donors.

Figure 1 Pre- and postoperative images of a successfulmeningoencephalocoeles repair

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References

1 Suwanwela C, Suwanwela N. A morphological classification of

sincipital encephalomeningoceles. J Neurosurg 1972;36:201112 Rojvachiranonda N, David DJ, Moore MH, Cole J.

Frontoethmoidal encephalomeningocele: new morphological find-

ings and a new classification. J Craniofac Surg 2003;14:847583 Thu A, Kyu H. Epidemiology of frontoethmoidal encephalome-

ningocoele in Burma. J Epidemiol Community Health

1984;38:89984 Gollogly L. The dilemmas of aid: Cambodia 19922002. Lancet

2002;360:93355 Hogan DR, Baltussen R, Hayashi C, Lauer JA, Salomon JA.

Cost effectiveness analysis of strategies to combat HIV/AIDS indeveloping countries. Br Med J 2005;331:14317

6 Pinzer T, Lauer G, Gollogly J, Schackert G. A complex therapy

for treatment of frontoethmoidal meningoencephalocele in a

developing third world country: neurological aspects. J

Neurosurg 2006;104(Suppl.):32631

The unreported morbidityof suicidal poisoningsduring an insurgency: a16-year Kashmir experienceZaid Ahmed Wani MDShabir Ahmed Dhar MSArshad Hussain MDWaseem Qureshi MDThe Government SMHS Hospital, Srinagar, Kashmir, India

Correspondence to: Dr Shabir Ahmed DharEmail: shabirdhar@yahoo.co.in

TROPICAL DOCTOR 2008; 38: 170171DOI: 10.1258/td.2007.070158

SUMMARY Around amillion people commit suicide, andat least10 times this number attempt suicide, worldwideevery year. No nationwide epidemiological studies havebeen undertaken in India but a significant rise in suicideshas been observed in Kashmir in recent years.This studywas carried out on patients reporting to the GovernmentSMHS Hospital in Srinagar with a history of suicidalpoisoning.

Introduction

Worldwide, around a million people die from suicide, and atleast 10 times this number attempt suicide.1 A review of theliterature shows that attempted suicide rates vary from100 to300 per 100,000 people per year, the greatest number ofwhom are women.2 In the USA, around five million poisonexposures occur yearly and up to 30% of psychiatric admis-sions are prompted by suicidal poisoning.3 In India, nonationwide epidemiological studies have been undertaken,so it is not possible to know the extent of the problem andthe change in pattern over the years.4 However, a significantrise in suicides has been observed in Kashmir in recent timesand poisoning is the most common method employed.1,5,6 In

a recent study of 364 poisoning cases by Khan et al. 83.5%were found to be suicidal in nature.5,6 Similar results weredescribed by Malik et al.6 There has been an alarmingincrease of suicidal poisoning in Kashmir mostly as a resultof the presiding insurgency. This study was conducted inorder to examine the rise in suicidal poisoning in the areaand to compare it with the pre-turmoil data.

Material and methods

This study was carried out on patients reporting to theGovernment SMHS Hospital in Srinagar with a history ofsuicidal poisoning. The cases were referred to our hospitalfor emergency treatment from peripheral primary and sec-ondary health-care institutions from January 1989 toDecember 2004. The study population mainly included thecivilian population of the Kashmir state and a fewmembers of the armed forces. The data of poisoning casesduring in the pre-turmoil period from January 1985 toDecember 1998 was collected from the medical recordsdepartment and studied retrospectively for comparison.

The sociomedical history was obtained from the patients,their attendants and the accompanying legal administrators.These included the nature of the poison, the amount con-sumed, the time since intake and the circumstances thatprompted the suicide attempt. Containers of poisons (e.g.bottles, strips of tablets and sachets) were searched, examinedand sent for chemical analysis whenever possible. After abrief history and clinical examination, treatment was insti-tuted. Gastric lavage, antidotes, supportive therapy andother required measures were taken. All gastric contents,blood and urine samples were preserved and sent for chemi-cal analysis.

Exclusion criteria

Patients with a doubtful history of ingestion, accidentalexposure, poor cooperation and patients leaving the hospitalagainst medical advice were excluded from the study.

After stabilization the patients were subjected to a detailedpsychiatric evaluation to try and pin point the basic precipi-tating causes and counselling was given to the patient.

All the data obtained from the history, examination, inves-tigations, psychiatric evaluation, as well as the death, wererecorded for each patient.

Results

The study included a total of 13,157 cases of suicidal poison-ing, of which 11,829 were studied over a period of 16 years(19892004). The data of the pre-turmoil period was studiedretrospectively. The present insurgency in the Kashmir valleycame into being in 1989. The post insurgency cases com-prised 5543 men (46.85%) and 6286 women (53.15%). Ofthese cases, the majority (10,823 [91.49%]) were Muslimsand 82.43% came from a rural background.

The trend showing the increase of suicidal poisoning isshown in Table 1. The substances used for suicidal purposeswere organophosphorous compounds, the most commonlyused (57.59%), rodenticides (20.99%), drugs, such as bezo-diazepines, acids, antihistamines (16%) and dhatura andalcohol (5.42%).

The various precipitating factors for suicidal poisoningwere loss of a loved one, loss of property, torture, witnessinga death and violence. Analysis of selected groups of patientsby psychiatrists revealed that depression, with a post-traumatic

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