INTERPOSITION OF THE COLON BETWEEN LIVERAND DIAPHRAGM (CHILAIDITI'S SYNDROME) IN

CHILDRENBY

A. D. M. JACKSON and C. J. HODSONFrom the Institute of Child Health, University ofLondon, and the Queen Elizabeth Hospital for Children,

London

(RECEIVED FOR PUBLICATION SEPTEMBER 24, 1956)

In 1910 Chilaiditi published a paper in which hedescribed three adult patients with gaseous ab-dominal distension and intermittent displacementof the liver by a distended loop of colon. Althoughthere had been some earlier brief reports, for examplethose of Gontermann (1890), Cohn (1907) andWeinberger (1908), this was the first really detailedaccount of the condition which has come to beknown as Chilaiditi's syndrome.During the last 45 years many more cases have

been reported but very few of these have beenchildren. It will be of some interest, therefore,to record the clinical and radiological findings infour further cases of Chilaiditi's syndrome in child-hood and to discuss the aetiology of the condition assuggested by the study of these children.

Case ReportsCase 1. Andrew F. was born in August, 1949. He

was first seen in December, 1950, at the age of 16 months.Abdominal distension had been noticed for about a yearand more recently there had been some abdominal painand vomiting. The past history was otherwise unevent-ful and the parents and a sibling were healthy.On examination the abdomen was considerably dis-

tended and tympanitic. With the child lying supine theliver edge could be felt below the right costal margin.In the erect and left lateral positions, however, there wasresonance to percussion over the right hypochondriumcontinuous with the resonance over the lung, and theedge of the liver could be palpated with some difficultyon the left side of the abdomen. The urine was normaland there were no other important findings.RADIOLOGICAL EXAMINATION. Radiological investi-

gations were carried out on several occasions over aperiod of several months. There was invariably gaseousdistension of the stomach and intestines. Films takenin the erect position showed the liver to be displaceddownwards and to the left, its place being taken by adistended loop of colon (Fig. 1). In the supine positionthe liver and colon usually occupied their normal

positions. On one occasion during the fluoroscopy theascent of the colon and the displacement of the liver wereactually observed as the patient was rotated from thesupine to the left lateral position. Fluoroscopy after anopaque meal excluded any form of intestinal obstructionand the interposed loop of bowel was identified as thehepatic flexure.

PROGRESS. During the six months following his firstvisit to hospital the child had recurrent attacks of severeabdominal pain with vomiting and his mother becameaware of his swallowing air. He was admitted to hos-pital in June, 1951, and put to bed. The attacks promptlyceased, although the abdominal distension persisted andthe position of the liver could still be altered with posture.The patient's progress over the next few years was

satisfactory and when last seen in April, 1956, he washaving only occasional, mild abdominal pain in theevening and this was rapidly relieved by sitting or lying

FIG. 1.-Radiograph of the abdomen (erect) in Case 1.

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152 ARCHIVES OF DISE,down. His mental and physical development wasnormal. However, apart from considerable diminutionof the abdominal distension, the clinical and radiologicalfindings were unchanged.

Case 2. Ann S. was born in November, 1951. Shewas admitted to hospital in November, 1954, at the ageof 3 years with a history of abdominal distension formost of her life and recent abdominal pain. The motherhad also noticed that the child gulped her food andfrequently passed flatus, especially if any pressure wasapplied to the abdomen. The bowels were constipatedbut there was no history of vomiting. Details of thefamily and past history were not relevant.On examination the abdomen was extremely dis-

tended. A few dilated veins were seen on the upperabdominal wall and there was visible intestinal peristalsis.The liver was displaced downwards and to the left in theerect position, its lower edge being palpable in the lefthypochondrium. Occasionally with the patient lyingdown the liver could be felt in the normal position. Onpercussion the whole abdomen was tympanitic and therewas no liver dullness. During her stay in hospital thechild occasionally complained of slight abdominal painin the evening. Relief of the constipation did not affectthe abdominal distension which was found to be muchless marked in the early morning and to increase gradu-ally during the day irrespective of whether the child waskept in bed or allowed up. The displacement of the liver,however, was only demonstrated in the erect position.RADIOLOGICAL EXAMINATION. In view of the experi-

ence with Case 1 radiological investigations were moreextensive in this case. Comparison of films taken soonafter the child awoke in the early morning with thosetaken in the late afternoon showed a striking differencein the degree of the abdominal distension. In themorning there was very little gas in the stomach and onlyslight distension of the colon confined to the descendingpart, whereas later in the day there was gross distensionof the stomach and most of the intestinal tract. Thesefindings confirmed the clinical observation of increasingdistension during the day. There were also differences inthe position of the liver depending on the time of day atwhich the films were taken. In the early morning theliver shadow was seen in its normal position below theright diaphragm in both supine and erect films. In theafternoon with the patient erect the liver was displaceddownwards, forwards and medially, its exact positionbeing difficult to make out, and a loop of the distendedcolon occupied the sub-diaphragmatic space (Figs.2 and 3); in the supine position the liver returned to itsnormal place on some occasions but not on others.Air swallowing was observed during screening of theabdomen.

PROGRESS. This patient has now been-followed for18 months. For the first year or so shevcontinued tohave marked abdominal distension, and occasionalabdominal pain during the later part of the day whichwas relieved by lying down. Recently ihe symptomshave almost completely subsided but at the latest exami-nation in April, 1956, although the abdominal distension

'ASE IN CHILDHOODwas considerably less, the clinical and radiological signsof interposition with the liver displacement were un-altered.

Case 3. Naomi P. was born in December, 1950, andwas 5 years old when first admitted to hospital. Hercomplaint was of attacks of abdominal pain which hadbegun some five weeks previously. The pain was quitesevere, lasted about half an hour and was relieved bybelching and passing flatus; it occurred most commonlyin the late afternoon. She never vomited but was some-times constipated and her appetite, previously good, hadbecome poor. Her abdomen had been unduly distendedfor as long as the parents could remember. There wereno siblings and the family and past history were notrelevant to the present condition.The clinical findings were similar to those in the

previous cases. The increasing daytime distension ofthe abdomen was particularly well marked, the circum-ference of the abdomen increasing from 18 in. at 5 a.m.to 24 in. at 2 p.m. In the erect position and occasionallyin the afternoon in the supine position the liver dullnesswas absent and the liver itself was not palpable. The-child's physical and mental development was normal.There were no abnormal physical signs and laboratoryexamination of the urine and faeces showed no abnor-mality.RADIOLOGICAL EXAMINATION. The typical features of

the syndrome were again demonstrated. There wasprogressive daytime distension of the whole alimentarytract. Supine films (Fig. 4) showed the normal anatomi-cal relations of liver and colon but in the erect positionthe colon was interposed between liver and diaphragmwith displacement of the liver (Fig. 5). On one occasionthe interposition was only partial, the liver being dis-placed forwards but not downwards. An opaque mealpassed down the alimentary tract without obstructionand air swallowing was observed during the examination.

PROGRESS. This child's progress has so far beenfollowed for only three months. During this time someattempt has been made to lessen the abdominal distensionwithout much success. Oral administration of charcoalbiscuits and a carminative mixture produced no obviousimprovement, and the wearing of an elastic abdominalbelt has, if anything, increased the child's discomfort.She still vomits occasionally and is troubled by theprogressive daytime distension but is otherwise quite welland has gained weight.

Case 4. Elizabeth F. was born in September, 1951,and was 44 years old when admitted to hospital inFebruary, 1956. Following an attack of gastro-enteritistwo months before admission she had complained ofanorexia, recurrent abdominal pain and vomiting,frequent passage of flatus and abdominal distension,which was most marked at the end of the day. For sometime past she had been noticed to swallow air. Theparents and a sibling were healthy and there were noincidents of note in the past history.On examination the physical signs of liver displace-

ment were present in the erect position and the typicalprogressive abdominal distension was observed. The

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CHILAIDITI'S SYNDROME IN CHILDREN 153

Fi;(. 2. Fi(;. 3.FIGS. 2 and 3.-Antero-posterior and lateral radiographs of the abdomen (erect) in Case 2. These films were taken in the late after-

noon and illustrate the extreme degree of gaseous distension of the stomach and intestines.

. s.ea }] e s¢-_ ~~~AE x,..

FI;. 4. Fi(;. 5.FiGs. 4 and 5.-Radiographs of the abdomen (supine and erect) in Case 3. In the supine position (Fig. 4) the liver and colonoccupy their normal positions. In the erect position (Fig. 5) there is interposition of the colon and displacement of the liver. The

huge gas bubble in the stomach is typical of aerophagy.

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ARCHIVES OF DISEASE IN CHILDHOOD

urine was normal and there were no other abnormalitieson clinical examination.RADIOLOGICAL EXAMINATION. The familiar radio-

logical features were present (Fig. 6). Gaseous disten-sion of the alimentary tract was far greater in the laterpart of the day. Air swallowing was observed on

screening and there was no evidence of intestinal obstruc-tion. Interposition of the colon and varying degrees ofdisplacement of the liver was seen in films taken with thepatient erect; the normal anatomy was restored in thesupine position.

PROGRESS. This patient has been under observationfor about four months. During this time she has madea complete recovery. She wore an abdominal belt forthree months after her discharge from hospital but hadleft it off for one month before the last examination whenshe was found to have no clinical or radiologicalabnormality.

DiscussionThese cases illustrate a well-defined clinical

syndrome, the main features of which are markeddiurnal abdominal distension and interposition ofthe colon between the liver and the diaphragm in theerect position. The commonest symptom is abdo-minal pain which is sometimes quite severe andassociated with vomiting. This pain occurs typicallytowards the end of the day when the abdominaldistension is maximal and is less frequent and muchless severe when the patient is in bed. Other, lesscommon symptoms are anorexia, constipation andthe frequent passage of flatus. The distinctivephysical signs are those of abdominal distensionwith absent liver dullness, and the liver edge may bepalpable somewhere on the left side of the abdomen.There are no other abnormalities or signs ofretardation of physical or mental development.The diagnosis may be confirmed by radiology if,

in the erect position, the striking appearance of adistended loop of colon under the right dome of thediaphragm is present and the liver is to a greater orlesser degree displaced. In addition, haustralmarkings establish the fact that the subphrenic gasis within the colon and not free in the peritonealcavity. The generalized nature of the distension,which extends-right down to the lower rectum, andthe absence of hIrge faecal masses help to distinguishthe radiological picture from that of Hirschsprung'sdisease and chronic constipation of the 'terminalreservoir' type. It is important to ensure thatradiographs are taken in the erect position anrthat a

lateral view is included, since interposition does notusually occur when the patient is supine and theposition of the liver may be in doubt on an antero-posterior film.The literature on interposition of the colon in

adults is extensive and several authors have eachcollected over 20 cases (Uspensky, 1928; Tremolieres

and Tardieu, 1931; Kolju, 1938; Hubacher, 1946;Torgersen, 1951). The incidence of the condition inadults appears to be about 1 per 1,000 radiologicalexaminations made in the erect position, i.e., chestfilms or screening (Uspensky, 1928; Kolju, 1938).This figure might, however, be considerably less iflateral radiographs had more often been taken toconfirm the displacement of the liver. A distendedcolon lying anterior to the liver, and therefore nottruly interposed, may in an antero-posterior filmobscure the liver shadow, giving the false impressionthat the liver is displaced. The scarcity of publishedreports concerning children suggests that the con-dition is rare in this age group but cases may havebeen missed because radiographs of children withabdominal distension are not normally taken in theerect position. The fact that the present series offour cases* was collected over the space of only a fewyears suggests that such cases are not exceptionallyuncommon in children. In adults both sexes areaffected, males more frequently than females, andthere is a suggestion of a geographical distributionin the fact that cases have been reported mostfrequently by authors on the continent of Europe.A recent extensive review of the subject (Baum andKarpati, 1954) lists 57 references of which only fourare in the English language.Baum and Karpati (1954) discuss the aetiology of

Chilaiditi's syndrome in some detail. They pointout that the liver is held in position mainly by asort of sub-diaphrasgmatic suction and suggest thatcertain abnormalities can disturb this relationshipbetween liver and diaphragm, allowing the liver todescend under the influence of gravity and the colonto take its place. Such abnormalities, which mayact singly or in combination,-fiiclude intestinaldistension, diaphragmatic paralysis, abnormalitiesof the size or suspension of the liver, traction onthe liver by adhesions in the region of the duodenum,and ascites. The evidence that some of these factors,e.g., abnormal suspension of the liver and thetraction effect of the peri-duodenal adhesions, playany part is, however, unconvincing.There may be different degrees of interposition

and liver displacement. In the complete form,illustrated by the present cases, the colon lies abovethe liver, contact between liver and diaphragm isbroken and the liver is displaced downwards,forwards and medially. Interposition is incompleteor partial when the colon does not get above theliver but lies laterally (Fig. 7) or posteriorly (Figs.8 and 9). The liver is then displaced medially or

* In addition to the four cases studied in detail three further casesof complete interposition have come to our notice during the prepara-tion of this paper and one case of partial interposition is brieflydescribed in the legend to Fig. 7.

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CHILAIDITI'S SYNDROME IN CHILDREN 155

F i 7

FIG. 6.-Radiograph of the abdomen (erect) in Case 4.

FIG. 7.-Part of a radiograph of the abdomen (erect) of a girl aged8 years showing a partial (lateral) interposition of the colon withmedial displacement of the liver. This was a chance finding; thepatient had pneumonia and there were no abdominal symptoms.The interposition has not been seen again despite repeated radio-

logical examinations.

FIGS. 8 and 9.-Antero-posterior and lateral radiographs of theabdomen (erect) in Case 3. On this occasion the interposition wasincomplete (posterior). The colon has risen behind but not above

the liver which is displaced forwards.FIG. 6.

FIG. 8. FIG. 9.

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forwards but not downwards. Chilaiditi's (1910)original cases were of this type. Presumably suchfactors as the intra-abdominal pressure, the degreeof distension of the stomach and intestines and themobility of the colon determine whether the inter-position shall be partial or complete. Occasionallythe interposition is transient, being detected for onlybrief periods without further recurrence. In suchcases there is usually a very temporary cause for thedistension of the gut such as an acute respiratoryinfection (Gruiber, 1937; see also Fig. 7) or preg-nancy (Hunter and Rapp, 1953). In most cases,however, including the four children under dis-cussion, the interposition persists, although onlyin the erect position, over a much longer period andoccasionally the colon becomes permanently fixedin its abnormal position by adhesions between it andthe liver or diaphragm (Rogers, 1935; Choussat andChoussat-Clausse, 1937). Very rarely other partsof the intestinal tract, such as the ileum (Baum andGrasser, 1952), may become interposed.Abdominal symptoms such as pain, vomiting,

flatulence and constipation, were present in abouthalf the reported adult cases and in addition to thedevelopment of adhesions a further important com-plication may arise, namely torsion of the inter-posed loop of bowel which gives rise to the signs andsymptoms of acute intestinal obstruction. Thismay lead to an unnecessary laparotomy eitherbecause in the radiograph the presence of a gas-filledloop of colon under the diaphragm is misinterpretedas free gas in the peritoneal cavity (Pendergrass andKirk, 1929) or because the importance of posturein the relief of interposition is not realized. Hunterand Rapp (1953) reported a case of temporaryinterposition with torsion of the interposed hepaticflexure demonstrated by barium enema. Thesymptoms of obstruction were rapidly relieved whenthe colon was released and the normal anatomyrestored by alteration of the patient's postureduring radiological examination.As far as children are concerned very little has

been added in recent years to the clinical descriptionby Gontermann (1890) of an infant with abdominaldistension and the physical signs of liver displace-ment (Wanderleber). Gruber (1937) and Arce(1946) also described cases and in addition gavebrief accounts of the radiological findings.

Such is the historical background of Chilaiditi'ssyndrome in adults and children against which theobservations on the present cases were analysed.The primary abnormality in these children isabdominal distension almost certainly due to theaerophagy which was observed in all four caseseither by the parents or by the radiologist. The

progressive distension of the alimentary tract duringthe day with tremendous dilatation of the stomach,the diminution of the distension by night and theabsence of intestinal obstruction are further facts insupport of a diagnosis of aerophagy. The reasonwhy these children should acquire the air swallowinghabit to such a marked degree is not obvious. Noneof them appeared to have any significant emotionaldisturbance to suggest a psychological cause. Sincethe abdominal distension persists even when thepatient is kept in bed it is apparently independent ofcolonic interposition and liver displacement whichonly take place in the erect position. Kolju (1938)studied the mechanism of interposition in adults byfluoroscopy. With the patient upright the colonwas distended with air through a rectal tube and wasseen to rise up and insert itself between the liver andthe diaphragm. At this point displacement of theliver took place. It is probable that this is exactlythe sequence of events in the present cases with airswallowing as the cause of the distension. Thereis no reason to suggest any abnormality of the liveror its suspensory ligaments since the normal liver issufficiently mobile to allow considerable displace-ment to take place as, for example, in cases ofpneumoperitoneum either artificially induced (Fig.10) or spontaneous (O'Donoghue, 1956).

G--::.-

FIG. 10.-Radiograph of the chest and abdomen of a boy aged 12in whom an artificial pneumoperitoneum has been induced forpulmonary tuberculosis. There is considerable infero-medial

displacement of the liver.

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CHILAIDITI'S SYNDROME IN CHILDRENOne further fact requires explanation. In a

search for further examples of this syndrome radio-graphs, taken in the erect position, of other childrenwith abdominal distension were examined. Theseincluded 10 cases of Hirschsprung's disease and fourcases of aerophagy, in all of which the distension ofthe colon was comparable with that of the cases ofChilaiditi's syndrome. Nevertheless, in no instancewas there any evidence of interposition or liverdisplacement. In addition, therefore, to colonicdistension and the effect of gravity some furtherfactor is necessary if interposition is to take place.It seems probable that this factor is an unusualmobility of the ascending colon and hepatic flexuredue to the presence of an ascending mesocolon.There is evidence from operation reports (Rogers,1935; Herget, 1951) and necropsy reports (Pender-grass and Kirk, 1929; Soupault, 1930) that thismobility of the colon exists in adult cases. Inchildren a highly-placed caecum and an apparentredundancy of the hepatic flexure are normalanatomical variants (Bryant, 1924; Caffey, 1956) sothat abdominal distension might by chance occurin a child with a mobile ascending colon.

It is concluded, therefore, that in the presentgroup of children the aetiology of interposition ofthe colon is primarily abdominal distension of asevere degree resulting from aerophagy. Unusualmobility of the ascending colon combined with theeffect of gravity result in ascent of the distendedbowel into the sub-diaphragmatic space with con-sequent infero-medial displacement of the liver.These abnormalities associated with the symptomsand distinctive radiological picture already describedconstitute a particular form in children of the syn-drome which is probably best termed 'interpositionof the colon between liver and diaphragm' or moresimply 'interposition of the colon'. This title ispreferable on the grounds of euphony to 'hepato-diaphragmatic interposition of the colon' whichhas been used by some authors. The eponymoustitle 'Chilaiditi's syndrome' is not strictly correctsince the cases described by Chilaiditi (1910) wereadults with a partial form of interposition and notentirely comparable with those under discussion.The pain and vomiting in these children could be

entirely explained by the degree of intestinal dis-tension and it might be argued that the abnormalposition of the liver and colon play no part in theirproduction. However, children with quite markedabdominal distension due to aerophagy withoutinterposition seldom complain of severe pain,whereas in three of the present cases (Cases 1, 3 and4) the symptoms were at times bad enough to suggestthe possibility of intestinal obstruction. In Case 1,

before the nature of the diagnosis has been appre-ciated, laparotomy was seriously considered and wasonly avoided by the timely disappearance of thesymptoms after the patient had been put to bed.The frequent observation of this relief of acuteabdominal symptoms by a change of posture fromupright to supine indicates that, at least on someoccasions, the symptoms are due to colonic or liverdisplacement rather than to distension. Sinceexperience in patients with artificial pneumo-peritoneum suggests that liver displacement per sedoes not cause abdominal pain it is probably theinterposition of the colon which is to blame. Apossible explanation is that the symptoms are due,as in certain adult cases previously mentioned,to obstruction brought about by torsion or incar-ceration of the interposed loop of colon as itbecomes more and more distended in the relativelyconfined space above the liver. Releasing the inter-position by placing the patient supine would relievethis obstruction and hence the symptoms. Theamount of distension varies from case to case andfrom time to time in any one case and it is possibleto imagine a degree of distension of the colonsufficient to give rise to interposition but not severeenough to cause obstruction. Hence the fact thatin Cases 1 and 2 there has been a reduction in dis-tension and in the severity and frequency of symp-toms, although in both cases interposition of thecolon persists. This theory would also account forthe absence of symptoms in many adult cases andfor such cases as the child whose radiograph isillustrated in Fig. 7, in which the distension isminimal and the resulting interposition incompleteand symptomless.

In view of the relatively short period of obser-vation and the small number of children studied itis not possible to be certain of the natural history ofthe syndrome in these children. The anatomicalabnormality has so far persisted for five and a halfyears in Case 1 and for 18 months in Case 2, althoughin both cases the air swallowing and consequentlythe distension and pain are steadily diminishing. Ifthis improvement is maintained and the colonbecomes less mobile a spontaneous cure, such asappears to have occurred in Case 4, might beexpected. If, however, the mobility of the colonis permanent the persistence or recurrence of inter-position would always be possible depending on thepresence of abdominal distension, whether it weredue to aerophagy or to any other cause. Since thecondition is comparatively common in adults it ispossible that some childhood cases do, in fact,persist into adult life.

Treatment for the condition has been attempted

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158 ARCHIVES OF DISEASE IN CHILDHOODin only two of the children (Cases 3 and 4). In thelight of the spontaneous improvement in Cases 1and 2 the necessity for any treatment is doubtfuland the assessment of its value difficult. Preventionof the aerophagy is obviously desirable but inchildren of this age difficult to achieve. Carmina-tives had no effect on the intestinal distension inCase 3. The effect of wearing an abdominal beltwas tried in Cases 3 and 4, and, although in Case 4this measure was accompanied by an apparentrecovery, it is difficult to believe that this was any-thing but coincidental. On balance there does notso far appear to be any significant difference inprogress between the treated and untreated cases.Operative fixation of the colon (Rogers, 1935) orliver (Schenck, 1938) has been suggested to preventinterposition taking place and to avoid complica-tions. Until the possibility of permanent spon-taneous cure is excluded such drastic therapy doesnot seem to be justified. Surgical measures todivide established adhesions between the colon andthe diaphragm would perhaps be more reasonablebut it is difficult to see how this complication wouldbe diagnosed on clinical grounds.

In conclusion it is suggested that the syndromeof interposition of the colon in children is a definiteentity, easily recognized by a typical clinical andradiological picture, and is of some importance byreason of the symptoms it may produce and thepossible complication of intestinal obstruction whichmay lead to an unnecessary laparotomy if the natureof the condition is unrecognized. It is very probablethat more cases would be discovered if the existenceof the syndrome were more widely appreciated.

SummaryThe syndrome of interposition of the colon

between liver and diaphragm is described.

A clinical and radiological study of four casesof this syndrome in childhood is reported. It isconcluded that in these cases the primary abnor-mality is abdominal distension due to aerophagyand that unusual mobility of the distended colonpermits its ascent into the sub-diaphragmatic spacein the erect position, with consequent displacementof the liver.The syndrome is probably not uncommon in

children despite the rarity of published reports.

We are indebted to the following paediatricians whohave allowed us to study and record their cases and havegiven much helpful advice in the preparation of thispaper: Dr. Helen Mackay (Case 1), Dr. Richard Dobbs(Case 2), Dr. Bernard Schlesinger (Case 3), Dr. GeorgeOrmiston and Dr. Wilfrid Sheldon (Case 4). We shouldalso like to thank Drs. W. F. Richards and B. C. H. Wardof High Wood Hospital for Children for their adviceconcerning artificial pneumoperitoneum and for theradiograph reproduced in Fig. 10. Dr. L. G. Blairkindly provided several of the other radiographs.

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Fr., 25, 147.Cohn, M. (1907). Verh. dtsch. RontgGes., 3, 66.Gontermann (1890). Dtsch. med. Wschr., 16, 1043.Gruber, E. (1937). Ibid., 63, 1113.Herget, R. (1951). Beitr. klin. Chir., 183, 83.Hubacher, 0. (1946). Schweiz. med. Wschr., 76, 554.Hunter, H. L. and Rapp, R. (1953). Radiology, 61, 67.Kolju, K. J. (1938). Amer. J. Roentgenol., 39, 928.O'Donoghue, P. D. (1956). Lancet, 1, 189.Pendergrass, E. P. and Kirk, E. (1929). Amer. J. Roentgenol.,

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