4L Eosinophilic Granuloma of Gastro-Intestinal Tract Caused by Herring Parasite Eustoma Rotundatum
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BsrrmsH 2 May 1964 MEDICAL JOURNAL 1141 4L Eosinophilic Granuloma of Gastro-intestinal Tract Caused by Herring Parasite Eustoma rotundatum B. STERRY ASHBY,* M.B., F.R.C.S.; P. J. APPLETONt M.B., B.S. IAN DAWSON,4 M.D., M.R.C.P. Brit. med.JY., 1964, 1, 1141-1145 For over 25 years sporadic reports have been appearing in the came from many countries in many different parts of the world. literature of cases of eosinophilic granuloma arising in various The essential details of these cases are recorded in Table I parts of the gastro-intestinal tract. Kaiiser (1937) described and Fig. 1. the first cases. In a search of the literature, which although The one feature common to all these case reports is the extensive is not claimed to be exhaustive, 47 papers were microscopical appearance of the lesion. No matter which part found, describing a total of 89 cases. They occurred through- of the alimentary tract is involved, the histological description out the alimentary tract from pharynx to rectum, though the is the same-an oedematous connective-tissue stroma with an majority were in the stomach and small intestine, and they increase of capillaries and lymphatics, and showing a massive diffuse eosinophil-cell infiltration, usually confined to the sub- * Surgical Registrar, Westminster Hospital and Medical School London. the muscularis mucosae and t House-Surgeon, Westminster Hospital and Medical School, Iondon. mucosa but sometimes splitting t Reader in Pathology, Westminster Hospital and Medical School, spreading into the muscle layer. The mucosa is almost always London. intact. TABLE I.-List of Published Cases, in Chronological Order Author Sex Age Site Author Sex Age Site M _5 Antrum Allergic to onions F 38 Pylorus, gall- Poultry farmer. Aller- F 38 Jejunum Allergic to neo- bladder, gic to gunpowder. Kaiiser (1937) *. { M arsphenamine jejunum Four operations for M 58 ,, separate lesions. Fragala (1942) Coon Orr et at. (1954) Eosinophilia 42%. F 64 Antrum Allergic to prunes A.C.T.H. used Possner and Vanek (1946){ M 55 F 42 Antrum Poultry farmer. Aller- Sison et al. (1947) F 31 Pentoneum Gross ascites. Pre- gic to chocolate operative eosino- de Santis (1954) .. Colon philia 38% Unnewehr and Ohrt (1954) F 55 Ileum Barrie and Anderson (1948) F 27 Antrum, duodenum Pre-op. eosinophilia Virshup and Mandelberg F 45 ,, Eosinophilia 20% 18% (1954) Herrera and de la Guardia M 52 Whole small bowel Diffuse involvement Judd et al. (1955) M 34 Antrum Pyloric stenosis. Asth- (1948) matic. Pre-op. Schneider and Dailey (1948) M 64 Antrum eosinophilla 68% Moloney (1949) 57 Antrum, duoden- Diffuse involvement. F 28 Stomach, pylorus Pyloric stenosis. F um, jejunum. Pre-op. eosinophilia McCune et al. (1955) Eosinophilia 59 % 12% M 54 Antrum Pyloric stenosis M 42 Antrum Pyloric stenosis M 30 Pylorus F 64 Pound (1956) .. Ileum M 55 M 59 Antrum M F 38 Vanek 47 ,, (1949) F 56 Died post-operatively 63 F 56 Stomach-lesser .1. 3.. .9. F 62 curve M 74 Coexistent pernicious anaemia F 54 Antrum F 51 Stomach-reater M 36 Stomach-greater Rigler et al. (1956) curve Z unes (1950) .. { curve F 57 Antrum M 50 Stomach Coexistent G.U. F 56 Coexistent lympgoma. M 44 Antrum ,, D.U. Diabetic M Polayes and Krieger (1950) 76 Jejunum M 83 Pre-existing perni- Spencer et al. (1950) M 40 Stomach Pre-op. eosinophilia cious anaemia. Co- 63% l existent carcinoma Booher and Grant (1951) F 57 Antrum stomach M 50 % Brunner (1951) 36 Pharynx Eosinophilia Smith (1956) F 54 Allergic to milk Doniach and McKeown M 39 Antrum Pre-op. eosinophilia ,, 10%/ F 54 (1951) Urban and Le~czyk (1956) M 62 IIleun F Intussusceptson Zak (1951) 31 Ileum Ferrier and Davis (1957) M 23 Jejunum Eosinophilia 30%. Barnett and Kazmann (1952) M 58 Antrum A.C.T.H. used Fennel (1952) M 30 Stomach, jejunum Diffuse involvement. Ileum Eosinophilic Koneman et al. (1959) M 45 Intussusception Toole and li~nitis plastica."~ Moochopoulos (1959) M 14 Eosinophilia 14% F 55 Antrumn Pardo and Rodriguez (1952) Colon Cox (1960) F 74 Ileum Pezzoli and Reggiani (1952) Caecum 51 ,, E. rotundatum larva Ruzic et al. (1952) Stomach Diffuse involvement. M present Eosinophilia 56% 49 ,.9 Taccani (1952) . Jejunum M 42 M ,, Frank (1953) 38 Antrum Severe allergic rhini- 40 ed post-op.-OP M Antrum,, tis, etc. Eosin 56% 51 ,, Anastomois leak M et al. M Ileums,, 58 Pre-op. eosinophilia Kulpers (1960a, 54 ,, Died post-op. 9% 1960b) M 25 M 35 Pre-existent perni- M 30 cious anaemia M 49 Died from other M 22 Pylorus F 52 causes M 43 Antrum M 63 Pre-existent perni- Ileum Heiwig and Ranier (1953)1 cious anaemia Rodenburg and Wielinga M 65 Antrum (1960). M 40 M 90 ,, Bauab (1961) F 50 Ileum, caecum, 4 separate lesions M 68 Died from other. colon causes and M 29 E. rorundarum larva sarcoid in Voorhuis Eijlers (1961) Stomach-Weater M 29 Coexistent curve present. Coexistent para-aortic nodes D.U. F 47 Szechy and Foldvari (1962) Duodenum Pyloric atenosis, Steger and Noto (1953) .. Rectum Hollmotz and Stepan (1963) _ Jejunum Marek (1954) .. F 57 Ileum Intussusception Bean 1142 2 May 1964 Eosinophilic Granuloma of Intestine-Ashby et al. MIDCAL JOURNAL The aetiology of these lesions has been discussed by many eosinophilic granulomata; in some there is already a possible authors, but until recently has remained obscure. Frequently aetiological factor, and in others such a source is not a practical it was suggested that the cause might well be an allergic reac- possibility; but in many of those occurring in western European territories it may well have been the causative agent. 301 25 in Case 1 20- The patient, a healthy man aged 37, had been well apart from 15 occasional episodes of abdominal colic during the previous year. LA Nine days before admission he had a severe attack of colic lasting 10. for 12 hours and relieved by pethidine. A barium-meal examina- => tion after the attack showed a Meckel's diverticulum but no other radiological abnormality or evidence of obstruction. On the day 5- of admission he had a further severe attack of colic without vomit- ing and with normal bowel action: by the time he reached hospital this had already subsided. 50 20 40 5o 6o 70 80 90 AGE On examination his temperature was 98.40 F. (36.9° C.) and FIG. 1.-Age and sex distribution, showing number of cases pulse 76. The abdomen was not tense or distended, there was no in each decade. (Hatched portion=female.) rigidity or guarding, qr indeed any abnormal physical signs, and he appeared perfectly well. A diagnosis of intermittent obstruction tion, possibly to some ingested foodstuffs. Some of the patients due to- a band associated with a Meckel's divertculum. was made, had an overt allergic history; of the three reported by Kaijser and laparotomy was performed the next day. (1937) one was allergic to onions and two were syphilitics Operation (Mr. K. James).-A small Meckel's diverticulum was allergic to neoarsphenamine. Orr et al. (1954), in an excellent found. No band or obstruction was present, but the small intestine review of the subject, described two cases, one allergic to gun- proximally was slightly dilated. The appendix appeared normal. powder, the other to chocolate; but perhaps the most signi- About 45 cm. proximal to the Meckel's diverticulum there was a ficant thing about these patients is that they-were both poultry sharply defined congested and oedematous segment of small intestine farmers. Others have been reported with an allergy to milk about 2j in. (6.4 cm.) in length, covered with fibrin on the surface. The lymph nodes draining the segment were not enlarged. The and prunes (Possner and Vanek, 1946). Some (Smith, 1956) affected segment was resected and an end-to-end anastomosis per- are cases have been asthmatics (Judd et al., 1955), and there formed. The appendix and the Meckel's diverticulum were also with a strong familial and constitutional tendency towards resected. The patient made an uneventful recovery. After the allergy, such as that described by Spencer et al. (1950), and histological sections had been examined (48 hours after operation), Frank's (1953) patient who suffered from severe rhinitis and the white-cell count was 9,800 cells/c.mm., of which 7% were conjunctivitis. eosinophils. No pre-operative white-cell count is available. W.R. were no could be in 1960 from Rotterdam came a series of papers and Kahn test negative and history of allergy Then obtained. describing 12 cases of eosinophilic granuloma of the small appendix was normal macroscopically and intestine and one of the stomach (Kuipers et al., 1960a, 1960b; Pathology.-The microscopically; no eosinophil infiltration was present. The and All the cases presented as Rodenburg Wielinga, 1960). Meckel's diverticulum was lined by small-intestinal epithelium. was resected. abdominal emergencies, and in each the lesion There was no ulceration, inflammation, or eosinophil infiltration The histological picture was again the eosinophific phlegmon microscopically. The third specimen consisted of 6 cm. of ileum. described so many times before; but in every specimen was The serosal surface was slightly congested and a patchy fibrinous found the remains of a larva which was identified as that of exudate was present on it, but this was nowhere conspicuous. The Eustoma rotundatum, a parasite commonly occurring in the mucosa did not appear to be ulcerated, but there was conspicuous peritoneum of the North Sea herring. thickening of the submucosa which had produced narrowing of the Kuipers and his colleagues investigated the parasite exten- lumen (Fig.