THE DIAGNOSIS OF CHRONIC INTUSSUSCEPTION CHILDREN · THE DIAGNOSIS OF CHRONIC INTUSSUSCEPTION IN CHILDREN FIG. 2. barium meal was requested. This was carried out on May 13. The stomach

THE DIAGNOSIS OF CHRONIC INTUSSUSCEPTION INCHILDREN

WITH AN ACCOUNT OF THREE CASES

BY

J. M. GARVIE AND F. H. KEMP

(From The Radcliffe Infirmary, Oxford)

Chronic intussusception in children is uncommon.As a clinical entity it is not well known. Still (1921)gives the clearest account:

'On a particular day the child seemed to havepain in the abdomen and vomited once or twice.During the next few days he occasionally vomited,generally in association with a colicky pain. Thevomiting has become less frequent, only once in twoor three days, and the pain is only occasional. Thebowels, which before were open daily, have sincethe onset been less regular, aperients have beennecessary, but when given have worked well and thestools have been normal; there has been no bloodin the stool or at most there has only been a streakonce or twice, such as might be seen on the stool ofany constipated child. The symptom which hastroubled the parents most is the wasting; the childafter three or four weeks of these vague troubles isobviously getting thin. The temperature throughouthas been normal. Such is the history, and with aduration of some weeks even the finding of a moreor less dJefinite lump in the abdomen may hardlyraise a sujspicion of intussusception, especially whenthe mother's chief complaint is the wasting. Never-theless, it is by the detection of the more or lesssausage-shaped tumour, and especially by its varyingconsistency from moment to moment during pal-pation, that the diagnosis is to be made; to thetactus eruditus also the unnatural emptiness of theright i!iac fossa (Dance's sign) may be usefulconfirmatory evidence, as in the ordinary acuteintussusception.'The purpose of this paper is to describe three

cases, which, though conforming to Still's descriptionin some respects, were not diagnosed for a long time,owing to the apparent absence of an abdominaltumour in two of the cases. Previous case reportsare given in an appendix.

Case recordsCASE 1324. (43). A boy aged seven-and-a-half

years was referred to the Radcliffe Infirmary byDr. W. Stobie on November 12, 1942.When two-and-a-half years old he was admitted

to a hospital in Chester with a sudden attack ofsevere abdominal pain. No operation was per-formed and he recovered within two days. InMarch, 1939, he was admitted to the private wardof the Radcliffe Infirmary with a purpuric rash and

pain and swelling of various joints. Some dayspreviously he had complained of abdominal pain.His bowels were constipated, but there was neverany blood in his stools and nothing abnormal wasfelt in his abdomen. The diagnosis was a purpuraof the Henoch-Schonlein type, but its exact etiologywas not determined.

After his discharge from hospital he was keptunder periodic observation. His general healthremained good but he continued to have attacks ofabdominal pain. Each attack seemed to followincreasing constipation. It was not accompaniedby fever, bruising or purpura, but he was said tolook very pale. The pain was of a colicky natureand it usually passed off in the night. These attacksrecurred about once a month.The last attack started five days ago and was

characterized by severe abdominal pain, whichcaused him to scream. The pain was centredaround the umbilicus and occurred in spasms aboutevery hour. It was severe for two days and thenseemed to subside, but it returned about twelvehours before admission, and his bowels were movedfor the first time since the onset of the pain. Themotions were normal.On examination, he was an alert, intelligent and

co-operative boy. He looked healthy, but at timeswas troubled with bouts of pain. His pulse andrespirations were normal. There was some resist-ance in the abdomen to the left and slightly belowthe umbilicus, which seemed to be due to hardeningof the left rectus. The signs were unaltered duringthe paroxysms of pain. The next day he was muchbetter and in the following ten days he had only oneor two spasms of pain. On account of his previoushistory, various haematological investigations werecarried out, but all revealed normal findings. Dr.V. Walker made certain skin tests but no foodallergy was detected. A barium meal was advisedand this was given on January 19, 1943. Thestomach and duodenum were normal, but the follow-through (fig. 1) revealed an intussusception with itsapex reaching just beyond the hepatic flexure of thetransverse colon. No attempt was made to reduceit. At operation (Mr. D. C. Corry) the intussus-ception was reduced by manipulation. There wereno adhesions, but the gut was swollen and slightlyreddened. Appendicectomy was performed andone large mesenteric gland was removed, thehistological examination of which did not show anyspecific lesion.

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ARCHIVES OF DISEASE IN CHILDHOOD

FIG. 1.

Convalescence was uneventful until February 10,when he was re-admitted with severe intestinal colicof seven hours duration. Laparotomy revealed aloop of small bowel bound down by adhesions.The adhesions were divided and he made a goodrecovery. Seven months later, his mother reportedthat he was quite well.

Comment. It is not known for certain how longthis boy had had an intussusception, but his historysuggests that it first happened when he was two-and-a-half years old, i.e. five years ago. On thisoccasion, spontaneous reduction must have occurred,for he remained free from symptoms for about twoyears. Three years ago he had an illness whichwas in the nature of a toxic reaction of the Henoch-Schonlein type, and there can be little doubt thatthe intussusception was caused at this time, forthereafter he had attacks of pain about once a

month. Despite the long standing illness, theboy's condition remained good and there was no

wasting. The true diagnosis was never suspectedbecause no abdominal tumour was palpated even

during the paroxysms of pain. The radiologicalexamination gave conclusive evidence of intussus-ception.

CASE 8340. (43.) R. S., a boy aged eight years,

four months, was admitted on April 25, 1943, withthe following history:On March 31, on returning home from school,

he collapsed on the sofa. During that night hecomplained of pain in the lower abdomen, vomitedundigested food and passed a loose motion in whichthere were streaks of blood. The following dayhis family doctor made a diagnosis of gastro-enteritis. He was kept in bed for three days on a

milk diet, and then as he seemed better, he wasallowed to get up. The same evening he hadanother attack, which was similar to the first, butmore severe. On this occasion he was sent to theVictoria Hospital at Swindon. Here, it was foundthat the right side of the abdomen was tender andthat there was some muscular guarding. Thepulse and temperature were normal. Among theconditions which were considered were intussus-ception, tuberculous enteritis and acute appendicitis.At first he was constipated, but on the third andfourth days his bowels opened naturally, abnormallylarge stools being passed. The tenderness dis-appeared and the Mantoux tuberculin test provednegative. A consulting physician suggested thatas he seemed quite well, he was discharged fromhospital (April 18).On the evening of his return home, he had another

attack. Three days later he had yet another, andon this occasion he complained of much pain andsix hours later passed about five ounces of blood.Two days later he was admitted to the RadcliffeInfirmary. He was now so pale that his doctorsent him for a blood transfusion.

His mother stated that before the illness beganthe boy had been a ravenous eater, but recently hisappetite had been very poor. She thought that hehad lost a lot of weight. He had never been arobust child, but had had no other severe illnesses.His mother's eldest brother died from tuberculousperitonitis, but there was no other case of tuber-culosis in the family.On examination he was a spare, rather sallow boy

with a slight malar flush. His rectal temperaturewas 1000 F. There was some slight resistance topalpation, particularly on the right side of theabdomen, but no apparent tenderness. The liverand spleen were not palpable. The rectum was fullof soft, normal coloured faeces and there was noblood on the examining finger.During the first ten days he lay listlessly in bed,

sometimes complaining of pain in the left hypo-chondrium. His appetite was poor and he vomitedfive times. On May 7 he was seen by Prof. L.Findlay, who noted that the abdomen was full,especially in the left epigastrium andhypochondrium.He detected an indefinite mass running from theleft hypochondrium to slightly above the midlinejust above the umbilicus; it could just be felt onrectal examination. Irritation of the abdominalwall induced peristaltic waves running from left toright along the line of the mass, apparently in thestomach.Three days later, careful palpation of the abdomen

revealed that the lower border of the mass extendedfrom below the costal margin to beyond the midline,just above the umbilicus, and it could be seenbulging the abdominal wall. The upper borderwas less well defined. The mass was not attachedto the abdominal wall and it moved with respiration.It was dull to percussion and continuous withsplenic dullness. Later the same day, the tumourchanged its position slightly and one of us thoughtthat it could be felt to grow harder and softer. Twodays later there was little change.The diagnosis was not clearly determined. The

question of intussusception was considered, butbecause of the position of the tumour and the routeof the peristalsis, it seemed that there might be atumour affecting the stomach and, for this reason, a

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THE DIAGNOSIS OF CHRONIC INTUSSUSCEPTION IN CHILDREN

FIG. 2.

barium meal was requested. This was carried outon May 13. The stomach and duodenum werenormal, but a follow-through (fig. 2) revealed anintussusception of the ileo-colic variety, with itsapex just beyond the splenic flexure. It wasdecided to attempt to reduce it with a barium enemaunder observation with the fluorescent screen. Theenema forced the apex of the intussusception back-wards into the ascending colon, but the intussus-ception could not be completely reduced. Theboy experienced only slight discomfort from thisprocedure and until his operation remained quitewell.On May 20 the child was operated upon and

found to have an ileo-caecal intussusception. Itcould not be reduced by manipulation, and thebase of the caecum and the appendix had to beexcised. No cause was seen. The child made anuneventful recovery, and six weeks later he wasquite well.

Comment. On looking back it will be seen thatthe history was typical. It began with a suddenattack, characterized by collapse, abdominal pain,vomiting and the passage of blood-stained faeces,from which he recovered within three days. Later,there were many other similar attacks and on one

occasion he passed a considerable quantity ofblood per rectum. The child's appetite failedand he lost a lot of weight. At first no abdominaltumour was recognized and for a long time thediagnosis of intussusception was never seriouslyentertained. Later, careful palpation disclosed a

tumour, but even then the diagnosis was notobvious, for in some respects its physical characterswere misleading. On the contrary, the radiological

findings gave conclusive evidence of the true natureof the complaint.

CASE 3. A.G., a boy aged seven years, wasadmitted to the Radcliffe Infirmary under the careof Dr. A. Cooke on August 31, 1944. He hadbeen perfectly well until two months ago, whenafter eating gooseberries he complained of a painin his stomach. The pain came on quite suddenly,but only lasted a minute or two and then passed off.Next day, he was off his food and vomited every-thing he took by mouth, but he had no further pain.His doctor saw him three days later. He wasemaciated and had a tumour running across theepigastrium, downwards and obliquely from left toright. The following day, the sickness had ceasedand the tumour had disappeared. He was allowedto resume solid food the next day and on the follow-ing day his sickness began again, his temperaturerose and the tumour reappeared. He was admittedto a hospital and a week later was discharged. Twodays later, the abdominal pain recurred. It cameon every two minutes and lasted a second or two.The child described the pain as ' not like a stomachache but like something pricking him.' He wasre-admitted to a hospital and a barium meal andenema were performed which were reported toreveal no evidence of obstruction.He was in hospital about six weeks and throughout

this time he seemed to be getting worse. Fluidswere taken and he appeared to improve on them,but if he was given solid food his temperature roseto 1020 F. and he began to vomit. Accordingly,his doctor decided to refer the child to the RadcliffeInfirmary for further investigation.On admission he was found to be undersized and

mentally retarded. He had marked generalizedmuscular wasting, an odd-shaped skull and a curiouscardiac murmur suggestive of congenital disease.The abdomen was protruberant but not tender, andno mass was felt. A barium enema was carried outon September 14, 1944, by Dr. B. Mannheimer andthis revealed a filling defect in the ascending colon-the appearances being typical of intussusception.There were large amounts of occult blood in thestools. His haemoglobin level was 60 per cent.For several days special efforts were made to improvehis general condition. On September 18, he wasreported to have vomited his food, but on examina-tion one hour later, no abdominal tumour could befelt. He said he had pain and pointed to theregion of his umbilicus. Operation was performedSeptember 23. Preliminary transfusion was unneces-sary as his haemoglobin had risen to 76 per cent.The operation (Mr. D. C. Corry) disclosed anintussusception of the ileo-caecal variety reachingas high as the ascending colon. Reduction bymanipulation was attempted but several areas ofnecrosis were revealed so the affected bowel wasresected. Examination of the specimen revealed nopredisposing causal factor. The child made asatisfactory recovery and was ultimately dischargedwithout complaint, but he was still very emaciated.

Comment. It is probable that this boy had hadan intussusception for about three months. At thebeginning the story was characteristic and a lumpwas palpable, but within twenty-four hours of thechild being brought under observation, the symptoms

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subsided and the lump disappeared. The sub-sequent history is more confusing. The boysuffered from repeated attacks of vomiting accom-panied by a rise in temperature. He had somepain but it was never severe. No lump was pal-pated. The doctor submitted the boy for a gastro-intestinal investigation, but gained little help forthe barium meal and enema were reported to showno signs of obstruction (it is not known whether theexaminations were performed by an experiencedradiologist). Since the child made no sustainedimprovement in health, he was referred to theRadcliffe Infirmary for a further opinion. A secondradiological examination (fig. 3) showed the intus-susception quite clearly, but though the child waskept under observation for three weeks, no lumpwas ever detected by ordinary clinical examination.The history was very difficult to analyse on accountof the child's mental state, but there is no doubtthat he did have some recurrent attacks of centralabdominal pain, though except on the first occasion,the pain was never very severe.No difficulty was experienced in making a correct

radiological diagnosis for the intussusception wasvisualized. (With regard to the findings of the firstexamination it should be noted that chronic intussus-ception does not cause intestinal otstruction.)

DiscussionJudging from the reports in the literature, the

diagnosis of chronic intussusception in children is

G. 3.

seldom easy. Generally, some weeks or monthselapse before the true nature of the condition isrealized, and sometimes it is never suspected untilrevealed by operation or at autopsy. It seems thatmistakes arise because the whole clinical picture isnot given careful consideration. The significanceof repeated attacks of abdominal cramp is notappreciated and too little attention is paid to thephysical examination.

Since Still wrote his paper many of the standardtextbooks have emphasized the presence of atumour; for instance: 'A child may have attacksof abdominal pain at hourly intervals for someweeks and on examination a mass is to be found'(Paterson). Again ' only the presence of a tumourleads to recognition of the condition' (Holt andMcIntosh). ' The correct diagnosis is arrived atby palpating a sausage-shaped mass, which con-tracts and expands beneath the fingers' (Pearsonand Wyllie).The question of whether a tumour can be palpated

or not is of great importance. Success obviouslydepends upon the skill of the examiner, the timeand conditions under which the examination iscarried out, and the position of the mass. Still(1921), in common with many successful paediatri-cians, believed that the child's abdomen should bepalpated during sleep. Clubbe (1906, 1921) whohad a very considerable experience of intussus-ception, said that, 'if there is any distension, orif the child resists or screams, it is quite hopeless

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THE DIAGNOSIS OF CHRONIC INTUSSUSCEPTION IN CHILDREN 77to expect to find a mass in the abdomen, even that the whole abdomen has been carefully palpated;though there be a large one.' He believed that an if necessary an anaesthetic should be given to secureanaesthetic ' should always be given for diagnostic proper relaxation. Likewise (sometimes in pre-purposes whenever you fail to feel a tumour, even ference to examination under anaesthesia), the childthough the child did not seem to resist your should be submitted for a careful radiographicmanipulation in any way.' study of the alimentary canal, preferably begun withNo one can seriously doubt the value of a properly a preliminary film of the abdomen and a barium

conducted clinical examination, yet it seems that enema, but supplemented, when negative, by ait is seldom performed efficiently, for in a series of barium meal. The clinician must remember thatsixty-two cases collected from the literature, a unless the radiologist has sufficient time to maketumour was detected at some time in only forty- himself familiar with the clinical picture, theseven. In many cases, the child has suffered for radiological examination is unlikely to provemonths, and in not a single instance was the child reliable on all occasions.examined under an anaesthetic.

Radiology offers another approach to the problem Summaryof how to detect the mass. Some experienced (1) Three cases of chronic intussusception inworkers, notably Nordentoft (1939) in Denmark, children are described. In two cases, the con-believe that radiography is reliable in a high pro- dition was not suspected for a long time owing toportion of cases. A barium enema is the usual the apparent absence of a palpable abdominalprocedure. Generally, the examination can be tumour, and in the third case because the tumourmade quickly and easily without undue stress to did not persist.the patient, but it is essential that it should be (2) The common features in the histories of thesecomplete and thorough (for Nordentoft has cases were repeated attacks characterized by theemphasized that intussusception can never be sudden onset of central abdominal pain.excluded unless the enema is seen to flow through (3) Any child who gives a suggestive historythe ileocaecal valve into the small bowel). Once should be carefully examined, for owing to thethe intussusception is visualized the diagnosis is not difficulty in securing proper relaxation the tumourdifficult, since the radiographic features are well can be very easily missed.known and quite distinctive. Occasionally the (4) Radiological studies should be made whetherexamination fails because the child will not take a a tumour is palpable or not, but it is essential thatfull enema, but in such cases a child may be given the examination should be thorough.an anaesthetic. In about five per cent. of cases the Thanks are due to Dr. A. Cooke, Professor L.intussusception is of the enteric variety and in such Findlay, and Professor L. J. Witts for their kindcases a barium enema is negative, but preliminary permission to record these cases, and Mr. D. C.films of the abdomen may show abnormal gaseous Corry for his notes on the operative findings.distension of a coil or coils of small bowel, and Pessor Findlay has kindly assisted by criticisingwhen necessary, the diagnosis can be confirmed by abarium meal (subject to there being no clinical REFERENCESindication which would render a barium meal (In addition to those given in Appendix)inadvisable). Clubbe, C. P. B. (1907, 1921). The diagnosis and treat-

In the light of the experiences reported here it is ment of intussusception, London. 1st and 2ndeditions.urged that the only reliable clinical feature is a Holt, L. E., and McIntosh, R. (1940). Diseases of

story of repeated attacks characterized by the children, New York. I ith edition.sudden onset of gripping abdominal pain. All Paterson, D. (1941). Sick children, London. 4th edition.other symptoms of intussusception may be absent Pearson, W. J., and Wyllie, W. G. (1935). Recentand are unreliable. A suspicious case should be NOrdadvances in diseases of children, London.andcarefunryeliablne.dA suspicious caseshouldbe Nordentoft, J. M. (1939). Sacrtvyk Fra-Nordishcarefully examined until the observer is satisfied Medicin, 1, 282.



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THE DIAGNOSIS OF CHRONIC INTUSSUSCEPTION CHILDREN · THE DIAGNOSIS OF CHRONIC INTUSSUSCEPTION IN CHILDREN FIG. 2. barium meal was requested. This was carried out on May 13. The stomach