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Intestinal schistosomiasis resembling regional ileitis
45 TRANSACTIONS OF THE ROYAL SOCIETY OF TROPICAL MEDICINE AND HYGIENE. Vol. 51. No. 1. January, 1957.
INTESTINAL
SCHISTOSOMIASIS
RESEMBLING
REGIONAL
ILEITIS
BY J O H N M. M Y N O R S , F.R.C.S.*
Senior Lecturer in Surgery, Kitchener School of Medicine, Khartoum.
I n t e s t i n a l s c h i s t o s o m i a s i s d u e to Schistosoma mansoni is p r e d o m i n a n t l y a n i n f e c t i o n o f t h e large i n t e s t i n e ; in t h e case r e p o r t e d h e r e t h e lesion h a d t h e a p p e a r a n c e of a r e g i o n a l ileitis b o t h at o p e r a t i o n a n d in t h e o p e n e d s p e c i m e n , even t h e m i c r o s c o p i c a p p e a r a n c e s r e s e m b l i n g t h a t c o n d i t i o n closely. CASE REPORT
A.E.T., a Sudanese carpenter, aged 21, living and working in Omdurman was admitted to hospital on 31st November, 1954 with a history of colicky umbilical pain and pain in the right iliac fossa for seven days, aggravated by meals and accompanied by vomiting and cough for seven days and constipation for five days. H e had already had a seven-day course of emetine without effect. No significant past history could be obtained at this time, but after the operation he admitted that he had had colicky abdominal pain and occasional diarrhoea for some months.
On Examination : H e was a wasted and dehydrated young man, with a dry furred tongue. T h e abdomen was fiat and there was no splash ; there was a tender, rounded, moderately mobile mass 3½ inches by 2 inches, lying directly under McBumey's point with its long axis parallel to the inguinal ligament. No other abnormality was discovered in abdomen, rectum, chest and lymphatic fields. Examination of the faeces did not reveal any parasites or abnormal constituents. His temperature was 100°F on admission and normal thereafter. A t Operation : On 4th December, 1954. U n d e r spinal anaesthesia. A right paraumbilical paramedian incision revealed that the terminal ileum and a small part of the caecum were reddened and much thickened, with numbers of small nodules projecting from the surface. T h e appendix was swollen and mildly hyperaemic but not involved in the process and definitely not inflamed. T h e small bowel proximal to the lesion was oedematous and a little distended. T h e mesentery was thickened and its lymph nodes enlarged and succulent. T h e liver and spleen appeared normal. A right hemicolectomy was performed removing about nine inches of the terminal ileum and completed by end-to-end anastomosis ; whilst the bowel was being manipulated that portion above the diseased segment went into intense spasm and during the subsequent resection was clearly seen to be obstructed. T h e patient had a certain amount of colic for the first 24 hours after operation but thereafter made a smooth recovery being discharged fit on 18th December, 1954. * I wish to thank Professor R. Kirk, University of Malaya, for his encouragement in the preparation of this paper and for his report upon t h e section ; Dr. Mansour Ali Haseeb, Director of the Stack Medical Research Laboratories, Khartoum ; Dr. Ahmed Ali Zaki, Director Medical Services, Sudan Government, for permission to publish ; and Mr. J. H. Airth, Mr. Vincent Nalpon and Dr. C. Weiss for producing the sections, photographs and photomicrographs.
46
SCHISTOSOMIASIS RESEMBLING REGIONAL ILEITIS
Scattered over the whole of the diseased bowel and projecting from the peritoneal surface were multiple small non-umbilicated nodules 1 to 2 mm. across ; there was a zone of closely applied nodules running around the bowel at the proximal edge of the diseased area in the ileum. Upon opening the bowel it was seen that the disease process affected the terminal four inches of ileum which was firm and much thickened ; the mucosa of the diseased portion had changed in character (Fig. 1) giving the effect of a closely velvety pile ; this stopped short quite suddenly (Fig. 3), the edge being overlapped by the swollen oedematous mucosa of the contiguous bowel ; there was thickening in all layers, marked in both submucous and subserous but maximal in the subserous ; the circular and longitudinal muscles fanned out more and more and eventually fragmented and almost disappeared as the diseased area was entered from the normal ileum, the longitudinal muscle fragmenting before the circular. The nodules appeared to lie principally within the subperitoneal tissue although projecting from the peritoneal surface. The disease process had destroyed the ileo-caecal valve and extended for one inch into the caecum where the mucosa had in the main ulcerated, though leaving a multitude of small islands of mucous membrane. The lumen of the affected bowel was a mere chink in the region of the ileo-caecal junction. The lymphatic nodes were succulent and did not show any caseation. The report on the section taken from the diseased area of the bowel was : " T h e section shows a chronic inflammatory or granulomatous condition, a fibrocellar tissue infiltrated with round cells, plasma cells and eosinophil polymorphs the last being specially prominent. Scattered throughout the section are numerous bodies which can be identified as ova of Schistosoma. I n one showing the complete structure of the ovum, the lateral spine is clearly visible (Fig. 2), thus enabling a specific determination of S. mansoni to be made. I n places, giant-cell systems and tuberclelike follicles are seen, sometimes resembling those seen in tuberculosis. At other times they are not very like those seen typically in tuberculosis, being smaller and very like the pseudo-tubercles which have sometimes been described in Schistosomiasis and containing eosinophils in addition to other cells. T h e cellular infiltration round some of the ova is composed almost entirely of eosinophils. Apart from the Schistosoma ova, no other parasites or other cause for the condition can be seen in the sections and no acid-fast bacilli could be made out in slides specially stained for this purpose."
Sections taken later of the lymph nodes showed them to be extremely active, cellular with many little bodies resembling tubercles, but no frank caseation anywhere. These sections did not show schistosome ova and only occasional small collections of eosinophils ; acid-fast bacilli were not seen in specially stained sections. The subperitoneal nodules were composed of the tubercle-like follicles which were surrounded with a peripheral zone of eosinophils. DISCUSSION
Regional ileitis, so-called because it most commonly affects the terminal ileum, may occur anywhere in the intestinal tract; its pathogenesis is not settled, a virus, specific organisms (including the tubercle bacillus), reticulosis, an agent in the faecal stream and allergy are perhaps the most commonly cited aetiological factors ; histologically it is a non-specific granuloma of the intestinal tract principally affecting young adults, and was first described in the Sudan by HORGAN and MORRIS (1943), the histological picture in this case being predominantly of lymphoid hyperplasia with some pseudo-tubercles. It is not impossible that the case described here could be a schistosomal infestation superimposed upon a regional ileitis ; on the other hand, the condition could be a primary schistosomal infestation in this area of the bowel which produced a picture very closely
FIG. 1. in the
The ileal end of the diseased bowel showing it in longitudinal lower part of the figure. Normal bowel to right, diseased bowel showing velvet-pile effect, muscle fragmentation and thickening.
L^.
-
from the distal part of the diseased FIG. 2. Section ileum showing the complete ovum with lateral spine. (Haematoxylin and eosin. x 510. For technical reasons this section is shown with a reversal of the black and white tones as the ovum is better revealed thereby). To face
$age 46)
“S
section to left
“-
FIG. 3. showing
Section transition
through mucosal surface from relatively normal area. (Haematoxylin
at the ileal end of the though infiltrated mucosa and eosin. x 47).
diseased to the
segment ulcerated
JOHN M. MYNORS
47
resembling that of regional i l e i t i s - in support of this are the scattering of schistosome ova throughout the section, the prominent eosinophil infiltration and the resemblance of the specimen in some respects to that from a case reported by KIRK (1949). This case was acute, the patient presenting with acute dysenteric symptoms and dying within five days. At postmortem the whole length of the peritoneal surface of the small intestine was studded with little nodules resembling miliary tubercles and the mesenteric lymph nodes were enlarged ; not unexpectedly, the mucosa showed congestion and minute submucous haemorrhages but not chronic changes ; ova of S. mansoni were found in the stool during life, and in the peritoneal nodules and mesenteric nodes postmortem. MANSONBAHR (1954) quoting Fairley and Lampe refers to these peritoneal nodules occurring in relation to the colon and closely resembling miliary tubercles. SUMMARY
A case is described of intestinal schistosomiasis occuring in a Sudanese man of 21, resembling regional ileitis, and treated by resection and anastomosis. REFERENCES MANSON-BAHR, P. H. (1954). Manson's Tropical Diseases, 14th Ed., p. 721. London : Cassell & Co., Ltd. SUDAN(1944). Report of the Sudan Medical Service for the year 1943, p. 35. (1950). Report of the Medical Services, Ministry of Health, Sudan Government for the year 1949, p. 37.
INTESTINAL
SCHISTOSOMIASIS
RESEMBLING
REGIONAL
ILEITIS
BY J O H N M. M Y N O R S , F.R.C.S.*
Senior Lecturer in Surgery, Kitchener School of Medicine, Khartoum.
I n t e s t i n a l s c h i s t o s o m i a s i s d u e to Schistosoma mansoni is p r e d o m i n a n t l y a n i n f e c t i o n o f t h e large i n t e s t i n e ; in t h e case r e p o r t e d h e r e t h e lesion h a d t h e a p p e a r a n c e of a r e g i o n a l ileitis b o t h at o p e r a t i o n a n d in t h e o p e n e d s p e c i m e n , even t h e m i c r o s c o p i c a p p e a r a n c e s r e s e m b l i n g t h a t c o n d i t i o n closely. CASE REPORT
A.E.T., a Sudanese carpenter, aged 21, living and working in Omdurman was admitted to hospital on 31st November, 1954 with a history of colicky umbilical pain and pain in the right iliac fossa for seven days, aggravated by meals and accompanied by vomiting and cough for seven days and constipation for five days. H e had already had a seven-day course of emetine without effect. No significant past history could be obtained at this time, but after the operation he admitted that he had had colicky abdominal pain and occasional diarrhoea for some months.
On Examination : H e was a wasted and dehydrated young man, with a dry furred tongue. T h e abdomen was fiat and there was no splash ; there was a tender, rounded, moderately mobile mass 3½ inches by 2 inches, lying directly under McBumey's point with its long axis parallel to the inguinal ligament. No other abnormality was discovered in abdomen, rectum, chest and lymphatic fields. Examination of the faeces did not reveal any parasites or abnormal constituents. His temperature was 100°F on admission and normal thereafter. A t Operation : On 4th December, 1954. U n d e r spinal anaesthesia. A right paraumbilical paramedian incision revealed that the terminal ileum and a small part of the caecum were reddened and much thickened, with numbers of small nodules projecting from the surface. T h e appendix was swollen and mildly hyperaemic but not involved in the process and definitely not inflamed. T h e small bowel proximal to the lesion was oedematous and a little distended. T h e mesentery was thickened and its lymph nodes enlarged and succulent. T h e liver and spleen appeared normal. A right hemicolectomy was performed removing about nine inches of the terminal ileum and completed by end-to-end anastomosis ; whilst the bowel was being manipulated that portion above the diseased segment went into intense spasm and during the subsequent resection was clearly seen to be obstructed. T h e patient had a certain amount of colic for the first 24 hours after operation but thereafter made a smooth recovery being discharged fit on 18th December, 1954. * I wish to thank Professor R. Kirk, University of Malaya, for his encouragement in the preparation of this paper and for his report upon t h e section ; Dr. Mansour Ali Haseeb, Director of the Stack Medical Research Laboratories, Khartoum ; Dr. Ahmed Ali Zaki, Director Medical Services, Sudan Government, for permission to publish ; and Mr. J. H. Airth, Mr. Vincent Nalpon and Dr. C. Weiss for producing the sections, photographs and photomicrographs.
46
SCHISTOSOMIASIS RESEMBLING REGIONAL ILEITIS
Scattered over the whole of the diseased bowel and projecting from the peritoneal surface were multiple small non-umbilicated nodules 1 to 2 mm. across ; there was a zone of closely applied nodules running around the bowel at the proximal edge of the diseased area in the ileum. Upon opening the bowel it was seen that the disease process affected the terminal four inches of ileum which was firm and much thickened ; the mucosa of the diseased portion had changed in character (Fig. 1) giving the effect of a closely velvety pile ; this stopped short quite suddenly (Fig. 3), the edge being overlapped by the swollen oedematous mucosa of the contiguous bowel ; there was thickening in all layers, marked in both submucous and subserous but maximal in the subserous ; the circular and longitudinal muscles fanned out more and more and eventually fragmented and almost disappeared as the diseased area was entered from the normal ileum, the longitudinal muscle fragmenting before the circular. The nodules appeared to lie principally within the subperitoneal tissue although projecting from the peritoneal surface. The disease process had destroyed the ileo-caecal valve and extended for one inch into the caecum where the mucosa had in the main ulcerated, though leaving a multitude of small islands of mucous membrane. The lumen of the affected bowel was a mere chink in the region of the ileo-caecal junction. The lymphatic nodes were succulent and did not show any caseation. The report on the section taken from the diseased area of the bowel was : " T h e section shows a chronic inflammatory or granulomatous condition, a fibrocellar tissue infiltrated with round cells, plasma cells and eosinophil polymorphs the last being specially prominent. Scattered throughout the section are numerous bodies which can be identified as ova of Schistosoma. I n one showing the complete structure of the ovum, the lateral spine is clearly visible (Fig. 2), thus enabling a specific determination of S. mansoni to be made. I n places, giant-cell systems and tuberclelike follicles are seen, sometimes resembling those seen in tuberculosis. At other times they are not very like those seen typically in tuberculosis, being smaller and very like the pseudo-tubercles which have sometimes been described in Schistosomiasis and containing eosinophils in addition to other cells. T h e cellular infiltration round some of the ova is composed almost entirely of eosinophils. Apart from the Schistosoma ova, no other parasites or other cause for the condition can be seen in the sections and no acid-fast bacilli could be made out in slides specially stained for this purpose."
Sections taken later of the lymph nodes showed them to be extremely active, cellular with many little bodies resembling tubercles, but no frank caseation anywhere. These sections did not show schistosome ova and only occasional small collections of eosinophils ; acid-fast bacilli were not seen in specially stained sections. The subperitoneal nodules were composed of the tubercle-like follicles which were surrounded with a peripheral zone of eosinophils. DISCUSSION
Regional ileitis, so-called because it most commonly affects the terminal ileum, may occur anywhere in the intestinal tract; its pathogenesis is not settled, a virus, specific organisms (including the tubercle bacillus), reticulosis, an agent in the faecal stream and allergy are perhaps the most commonly cited aetiological factors ; histologically it is a non-specific granuloma of the intestinal tract principally affecting young adults, and was first described in the Sudan by HORGAN and MORRIS (1943), the histological picture in this case being predominantly of lymphoid hyperplasia with some pseudo-tubercles. It is not impossible that the case described here could be a schistosomal infestation superimposed upon a regional ileitis ; on the other hand, the condition could be a primary schistosomal infestation in this area of the bowel which produced a picture very closely
FIG. 1. in the
The ileal end of the diseased bowel showing it in longitudinal lower part of the figure. Normal bowel to right, diseased bowel showing velvet-pile effect, muscle fragmentation and thickening.
L^.
-
from the distal part of the diseased FIG. 2. Section ileum showing the complete ovum with lateral spine. (Haematoxylin and eosin. x 510. For technical reasons this section is shown with a reversal of the black and white tones as the ovum is better revealed thereby). To face
$age 46)
“S
section to left
“-
FIG. 3. showing
Section transition
through mucosal surface from relatively normal area. (Haematoxylin
at the ileal end of the though infiltrated mucosa and eosin. x 47).
diseased to the
segment ulcerated
JOHN M. MYNORS
47
resembling that of regional i l e i t i s - in support of this are the scattering of schistosome ova throughout the section, the prominent eosinophil infiltration and the resemblance of the specimen in some respects to that from a case reported by KIRK (1949). This case was acute, the patient presenting with acute dysenteric symptoms and dying within five days. At postmortem the whole length of the peritoneal surface of the small intestine was studded with little nodules resembling miliary tubercles and the mesenteric lymph nodes were enlarged ; not unexpectedly, the mucosa showed congestion and minute submucous haemorrhages but not chronic changes ; ova of S. mansoni were found in the stool during life, and in the peritoneal nodules and mesenteric nodes postmortem. MANSONBAHR (1954) quoting Fairley and Lampe refers to these peritoneal nodules occurring in relation to the colon and closely resembling miliary tubercles. SUMMARY
A case is described of intestinal schistosomiasis occuring in a Sudanese man of 21, resembling regional ileitis, and treated by resection and anastomosis. REFERENCES MANSON-BAHR, P. H. (1954). Manson's Tropical Diseases, 14th Ed., p. 721. London : Cassell & Co., Ltd. SUDAN(1944). Report of the Sudan Medical Service for the year 1943, p. 35. (1950). Report of the Medical Services, Ministry of Health, Sudan Government for the year 1949, p. 37.