the lancet, august 13, 1977 — page 343

Summary Some patients consistently produce false stories and fabricate evidence, so causing themselves needless hospital investigations and operations. Here are described parents who, by falsification, caused their children innumerable harmful hospital procedures — a sort of Munchausen syndrome by proxy.

Doctors dealing with young children rely on the parents' recollection of the history. The doctor accepts that history, albeit sometimes with a pinch of salt, and it forms the cornerstone of subsequent investigation and management of the child.

A case is reported in which over a period of six years, the parents systematically provided fictitious information about their child's symptoms, tampered with the urine specimens to produce false results and interfered with hospital observations. This caused the girl innumerable investigations and anaesthetic, surgical, and radiological procedures in three different centres.

The case is compared with another child who was intermittently given toxic doses of salt which again led to massive investigation in three different centres, and ended in death. The behaviour of the parents of these two cases was similar in many ways. Although in each case the end result for the child was "non-accidental injury", the long-running saga of hospital care was reminiscent of the Munchausen syndrome, in these cases by proxy.

Kay was referred to the pediatric nephrology clinic in Leeds at the age of 6 because of recurrent illnesses in which she passed foul-smelling, bloody urine. She had been investigated in two other centres without the cause being found.

In the child's infancy, her mother had noticed yellow pus on the nappies, and their doctor had first prescribed antibiotics for suspected urine infection when Kay was 8 months old. Since then, she had had periodic courses of antibiotics for presumed urine infection. Since the age of 3 she had been on continuous antibiotics which included co-trimoxazole, amoxycillin, nalidixic acid, nitrofurantoin, ampicillin, gentamicin, and uticillin. These treatments had themselves caused drug rashes, fever, and candidiasis, and she had continued to have intermittent bouts of lower abdominal pain associated with fever and foul-smelling, infected urine often containing frank blood. There was intermittent vulval soreness and discharge.

The parents were in their late 30s. Father who worked mainly in the evenings and at night, was healthy. The mother had had urinary-tract infections. The 3-year-old brother was healthy.

At the time of referral, she had already been investigated at a district general hospital and at a regional teaching hospital.

Investigations had included two urograms, micturating cys-tourethrograms, two gynaecological examinations under anaesthetic, and two cystoscopies. The symptoms were unexplained and continued unabated. She was being given steadily more toxic chemotherapy. Bouts were recurring more often and everyone was mystified by the intermittent nature of her complaint and the way in which purulent, bloody urine specimens were followed by completely clear ones a few hours later. Similarly, foul discharges were apparent on her vulva at one moment, but later on the same day her vulva was normal.

On examination she was a healthy girl who was growing normally. The urine was bloodstained and foul. It was strongly positive for blood and albumin and contained a great many leucocytes and epithelial cells. It was heavily infected with Escherichia coli.

The findings strongly suggested an ectopic ureter or an infected cyst draining into the urethra or vagina. Yet previous investigations had not disclosed this. Ectopic ureters are notoriously difficult to detect, and, after consultation with colleagues at the combined paediatric/urology clinic, it was decided to investigate her immediately she began to pass foul urine. No sooner was she admitted than the foul discharge stopped before cystoscopy could be done. More efficient arrangements were made for the urological surgeon concerned to be contacted immediately she should arrive in Leeds, passing foul urine. This was done three times (including a bank holiday and a Sunday). No source of the discharge was found. On every occasion it cleared up fast. Efforts to localise the source included further radiology, vaginogram, urethrogram, barium enema, suprapubic aspiration, bladder catheterisation, urine cultures, and exfoliative cytology. During these investigations, the parents were most cooperative and Kay's mother always stayed in hospital with her (mainly because they lived a long way away). She was concerned and loving in her relationship with Kay, and yet sometimes not quite as worried about the possible cause of the illness as were the doctors. Many of the crises involved immediate admission and urgent anaesthetics for examinations or cystoscopy, and these tended to occur most at weekend holiday periods. On one bank holiday, five consultants came into the hospital specifically to see her.

The problem seemed insoluble and many of the facts did not make sense. The urinary pathogens came and went at a few minutes' notice; there would be one variety of E. coli early in the morning and then after a few normal specimens, an entirely different organism such as Proteus or Streptococcus facalis in the evening. Moreover, there was something about the mother's temperament and behaviour that was reminiscent of the mother described in case 2, so we decided to work on the assumption that everything about the history and investigations were false. Close questioning revealed that most of the abnormal specimens were ones that at some stage or other had been left unsupervised in the mother's presence.

This theory was tested when Kay was admitted with her mother and all urine specimens were collected under strict supervision by a trained nurse who was told not to let the urine out of her sight from the moment it passed from Kay's urethra to it being tested on the ward by a doctor and then delivered to the laboratory. On the fourth day, supervision was deliberately relaxed slightly so that one or two specimens were either left for the mother to collect or collected by the nurse and then left in the mother's presence for a minute before being taken away. On the first 3 days, no urine specimen was abnormal. On the first occasion that the mother was left to collect the specimen (having been instructed exactly how to do so), she brought a heavily bloodstained specimen containing much debris and bacteria. A subsequent specimen collected by the nurse, was completely normal. This happened on many occasions during the next few days. During a 7-day period, Kay emptied her bladder 57 times. 45 specimens were normal, all of these being collected and supervised by a nurse; 12 were grossly abnormal, containing blood and different organisms, all these having been collected by the mother or left in her presence. All the specimens were meant to be collected in exactly the same way as complete specimens, and the mother was using the same sort of utensils as were the nurses. On one evening the pattern was as follows:

On that day the mother was persuaded to provide a specimen of urine from herself. She produced a very bloody specimen full of debris and bacteria which resembled the specimens she had been handing in as Kay's urine. The mother was menstruating. Kay was given xylose tablets so that we could identify which urine came from her. All the specimens handed in by the mother contained xylose which meant that each specimen contained some of Kay's urine. The help of the Yorkshire Police forensic laboratory was obtained. Kay and her mother had similar blood-groups, but erythrocyte acid phosphatase in the blood in the urine specimens was of group Ba which was similar to the mother's but not to Kay's.

At this stage, there was enough evidence to support the theory that the mother's story about her daughter was false, and that she had been adding either her own urine or menstrual discharge to specimens of her daughter's urine. Other abnormal findings could similarly be explained by the deliberate actions of the mother.

The consequences of these actions for the daughter had included 12 hospital admissions, 7 major X-ray procedures (including intravenous urograms, cystograms, barium enema, vaginogram, and urethrogram), 6 examinations under anaesthetic, 5 cystoscopies, unpleasant treatment with toxic drugs and eight antibiotics, catheterisations, vaginal pessaries, and bactericidal, fungicidal, and CEstrogen creams; the laboratories had cultured her urine more than 150 times and had done many other tests; sixteen consultants had been involved in her care.

The various fabrications occupied a major part in the mother's life and arrangements were made for her to see a psychiatrist at a hospital near her home. At first, she denied interfering with the management of her daughter. However, during the period of psychiatric outpatient consultation, Kay's health remained good. The urinary problems did not recur and her parents said that they felt that "since going to Leeds, Kay had been much better and their prayers had been answered".

Later it emerged that the mother had a more extensive personal medical history than she had admitted and that during investigation of her own urinary tract she had been suspected of altering urine specimens, altering temperature charts, and heating a thermometer in a cup of tea. She was a caring and loving mother for her two children. Kay was a long-awaited baby (in the hope of which the mother had taken a fertility drug), but after the birth she sometimes felt that her husband was more interested in the child than in her.

Charles had had recurrent illnesses associated with hyper-natraemia since the age of 6 weeks. He was the third child of healthy parents. The attacks of vomiting and drowsiness came on suddenly, and on arrival in hospital he had plasma-sodium concentrations in the range 160-175 mmol/1. At these times his urine also contained a great excess of sodium. The attacks occurred as often as every month; between attacks he was healthy and developing normally. Extensive investigations took place in three different centres. He was subjected to radiological, biochemical, and other pathological procedures during several hospital admissions. These showed no abnormality between attacks, and his endocrine and renal systems were normal. When given a salt load, he excreted it efficiently. The attacks became more frequent and severe, and by the age of 14 months it became clear that they only happened at home. During a prolonged hospital stay in which the mother was deliberately excluded, they did not happen until the weekend when she was allowed to visit. Investigation proved that the illness must be caused by sodium administration, and the time relationship clearly incriminated the mother. We did not know how she persuaded her toddler to ingest such large quantities of salt (20 g of sodium chloride given with difficulty by us raised the serum-sodium to 147 mmol/1 only). The mother had been a nurse and was presumably experienced in the use of gastric feeding tubes and suppositories.

"During the period in which the local paediatrician, psychiatrist, and social-services department were planning arrangements for the child, he arrived at hospital one night, collapsed with extreme hypernatraemia, and died.

Necropsy disclosed mild gastric erosions "as if a chemical had been ingested". The mother wrote thanking the doctors for their care and then attempted suicide.

She too was a caring home-minded mother. She had an undemonstrative husband, a shift worker who did not seem as intelligent as she. As a student she had been labelled hysterical, and during one hospital admission had been thought to be interfering with the healing of a wound.

These two cases share common features. The mothers' stories were false, deliberately and consistently false. The main pathological findings were the result of the mothers' actions, and in both cases caused unpleasant and serious consequences for the children. Both had unpleasant investigations and treatments, both developed illnesses as a result of the malpractice and the treatments, and the second child died.

Both mothers skilfully altered specimens and evaded close and experienced supervision. In case 1, a specimen of the child's urine collected under "close supervision" was abnormal, but it emerged that the mother had momentarily persuaded the nurse to leave the cubicle and leave the specimen unguarded for about a minute. Expressed breast milk collected from the mother of case 2 early in the course of the illness had a very high sodium content. It had been collected under supervision for chemical analysis, but when the supervisory nurse was instructed not to leave the specimen between its emergence from the mother's breast and its delivery to the laboratory, the next specimen was normal.

During the investigation of both these children, we came to know the mothers well. They were very pleasant people to deal with, cooperative, and appreciative of good medical care, which encouraged us to try all the harder. Some mothers who choose to stay in hospital with their child remain on the ward slightly uneasy, overtly bored, or aggressive. These two flourished there as if they belonged, and thrived on the attention that staff gave to them. It is ironic to conjecture that the cause of both these children's problems would have been discovered much sooner in the old days of restricted visiting hours and the absence of facilities for mothers to live in hospital with a sick child. It is also possible that, without the excellent facilities and the attentive and friendly staff, the repetitive admissions might not have happened. Both mothers had a history of falsifying their own medical records and treatment. Both had at times been labelled as hysterical personalities who also tended to be depressed. We recognise that parents sometimes exaggerate their child's symptoms, perhaps to obtain faster or more thorough medical care of their child. In these cases, it was as if the parents were using the children to get themselves into the sheltered environment of a children's ward surrounded by friendly staff. The mother of case 1 may have been projecting her worries about her own urinary-tract problems on to the child in order to escape from worries about herself. She seemed to project her own worries on to the child in many different ways, once informing another hospital that a specialist from Switzerland was coming to see her daughter in Leeds because she had an incurable kidney tumour which emptied into the vagina causing the discharge.

This sort of fabricated story is reminiscent of the Munchausen syndrome. The parents described, share some of the common features of that syndrome in which the persons have travelled widely for treatment, and the stories attributed to them are both dramatic and untruthful. But those with Munchausen syndrome have more fanciful stories, which are different at different hospitals. They tend to discharge themselves when the game is up. They cause physical suffering to themselves but not usually to their relatives. Munchausen syndrome has been described in children, the confabulations being made by the child.¹ Case 1 seems to be the first example of "Munchausen syndrome by proxy".

The repetitive poisoning of a child by a parent (case 2) has been described before. Rogers and colleagues² described six cases in 1976 and they suggested that such poisoning was "an extended form of child abuse". Larsky and Erikson³ suggested marital conflict as a possible cause for such poisoning, one spouse harming a child who was considered to be unfairly favoured by the other. The resulting illness of the child tended to restore marital relations at the child's expense.

None can doubt that these two children were abused, but the acts of abuse were so different in quality, periodicity, and planning from the more usual non-accidental injury of childhood that I am uneasy about classifying these sad cases as variants of non-accidental injury.

Whatever label one chooses to describe them, these cases are a reminder that at times doctors must accept the parents' history and indeed the laboratory findings with more than usual scepticism. We may teach, and I believe should teach, that mothers are always right; but at the same time we must recognise that when mothers are wrong they can be terribly wrong.

Asher began his paper on Munchausen's syndrome⁴ with the words "Here is described a common syndrome which most doctors have seen, but about which little has been written". The behaviour of Kay's mother has not been described in the medical literature. Is it because that degree of falsification is very rare or because it is unrecognised?

This paper is dedicated to the many caring and conscientious doctors who tried to help these families, and who, although deceived, will rightly continue to believe what most parents say about their children, most of the time.