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Disseminated Eosinophilic Infiltration of a Newborn Infant, with Perforation of the Terminal Ileum and Bile Duct Obstruction

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Disseminated Eosinophilic Infiltration of a Newborn Infant, with Perforation of the Terminal Ileum and Bile Duct Obstruction Arch Dis Child: first published as 10.1136/adc.56.1.66 on 1 January 1981. Downloaded from 66 Shinozaki, Saito, and Shiraki infant who acquired hepatitis from her mother. Br Med J 6 Yoshida A, Tozawa M, Furukawa N, Oya N, Kusunoki T, 1970; iv: 719-21. Kiyosawa N. HBsAg-positive chronic active hepatitis in 3 Bancroft W H, Warkel R L, Talbert A A, Russell P K. a 1 and 1/2 year-old-child (in Japanese). Shonika Shinryo Family with hepatitis-associated antigen. JAMA 1971; 1977;40: 1246-50. 217:1817-20. McCarthy J W. Hepatitis B antigen (HBAg)-positive chronic aggressive hepatitis and cirrhosis in an 8-month- Correspondence to Dr T Shinozaki, Department of old infant. A case report. JPediatr 1973; 83: 638-9. Paediatrics, Teikyo University School of Medicine, 11-1 5 Fujiwara T, Abe M, Tachi N, Jo M, Shiroda M. Kaga, 2 Chome, Itabashi-ku, Tokyo 173, Japan. HBsAg-positive infantile hepatitis associated with chronic aggressive hepatitis (in Japanese). Shonika Rinsho 1975; 28:1303-6. Received 26 November 1979 Disseminated eosinophilic infiltration of a newborn infant, with perforation of the terminal ileum and bile duct obstruction S M MURRAY AND C J WOODS Department ofPathology and Department ofPaediatrics, Victoria Hospital, Blackpool Case report SUMMARY A preterm boy died 4 days after delivery from septicaemia which at necropsy was found to be A white boy, weighing 1490 g, was born by spon- due to perforation of an eosinophilic lesion of the taneous vertex delivery at 35 weeks' gestation to a copyright. terminal ileum. Eosinophilic infiltration was also healthy 19-year-old rubella-immune group A Rh- found in kidney, lymph node, bone marrow, portal positive mother, who had a severe antepartum tracts of liver, gall bladder, and bile duct with associ- haemorrhage due to placenta praevia. The pregnancy ated obstruction of the cystic duct and mucocele of had been uneventful, apart from vaginal bleeding at the gall bladder. No allergic cause for the infiltrate 13 and 18 weeks which had settled without treatment was found in either the infant or his mother. Eosino- on each occasion. No drugs had been taken during the philic infiltration of neonatal spleen, lymph node, pregnancy. http://adc.bmj.com/ intestinal mucosa, epicardium, thymus, pancreas, There was no birth asphyxia, regular respirations portal tracts of the liver, and skin has been reported were established within 1 minute and, apart from but the aggressive behaviour of the infiltrate in this being preterm, there were no abnormalities on patient bears more resemblance to the eosinophilic clinical examination. 1-mg of vitamin K1 was gastroenteritis that has been described in older given intramuscularly. Intermittent intragastric tube children and adults. feeding with low solute milk was started and, apart from occasional regurgitation of small amounts of Eosinophilic infiltration of the alimentary tract of the feed, the infant made satisfactory progress. on September 30, 2021 by guest. Protected adult occurs in two forms-namely eosinophilic Meconium was first passed at age 8 hours. gastroenteritis and inflammatory fibroid polyp of the At age 48 hours the infant's condition deteriorated gastrointestinal tract-each has been compre- suddenly with the clinical picture of peripheral hensively reviewed by Johnstone and Morson.1-2 circulatory failure. There was no significant The condition is described much less often in children abdominal distention. Cerebrospinal fluid showed and in two reviews3-4 only 13 cases were recorded in only 1 lymphocyte/mm3 and no organisms were children under 12 years, the youngest being a 2-year- found on microscopical examination or culture. old boy. Eosinophilic infiltration of the gastro- The haemoglobin was 10 9 g/dl, packed cell volume intestinal tract has also been described in neonatal 33.9%/, and white blood count 1000 x 109/l. The necrotising enterocolitis5 but the significance of this scanty nucleated cells seen in the blood film were infiltrate is unknown. No reference to disseminated either lymphocytes or normoblasts with no increase eosinophilic infiltration in the neonate followed by in eosinophils. intestinal perforation has been found in a search of Septicaemia was diagnosed and treatment with the recent literature. intravenous gentamicin and cloxacillin was started. Arch Dis Child: first published as 10.1136/adc.56.1.66 on 1 January 1981. Downloaded from Disseminated eosinophilic infiltration ofa newborn infant 67 Oral feeding was stopped and intravenous dextrose be patent even a fine probe could not be passed along saline started. Blood culture subsequently grew the cystic duct. The lungs were severely congested. Escherichia coli sensitive to gentamicin. The other organs appeared normal. At age 53 hours the infant had a severe apnoeic attack requiring endotracheal intubation and inter- Histological examination. The perforated segment of mittent positive pressure ventilation. 30-ml of blood terminal ileum showed intense congestion and were transfused and metabolic acidosis corrected oedema with infiltration of the entire thickness of the with intravenous sodium bicarbonate. However, at wall by large numbers of eosinophils together with age 72 hours abdominal distension developed and some lymphocytes and plasma cells (Figs 1 and 2). the limbs showed sclerematous changes. Refractory The serosal surface showed a fibrino-purulent exu- hypoxaemia and acidosis ensued and despite con- date containing, in addition to polymorphonuclear tinued mechanical ventilation the infant died. He was leucocytes, numerous clumps of Gram-negative aged 90 hours. bacilli and moderate numbers of eosinophils. The gall bladder showed mild focal infiltration of its wall Necropsy findings by mononuclear cells and eosinophils while the bile ducts, particularly the cystic duct, showed severe Perforation of a severely congested short segment of congestion and oedema with cellular infiltration terminal ileum had caused acute peritonitis. The gall resembling that of the ileum. The liver showed bladder was distended with clear mucus and although haematopoiesis of normal extent for a preterm infant the common bile duct and hepatic ducts appeared to of this size but eosinophils were absent from these copyright. http://adc.bmj.com/ on September 30, 2021 by guest. Protected Fig. 1 Full thickness section of - terminal ileum near point of l_- N perforation showing cellular infiltration. Prominent dilated blood vessels are seen in the submucosa. (H and E x 178). Arch Dis Child: first published as 10.1136/adc.56.1.66 on 1 January 1981. Downloaded from 68 Murray and Woods Fig. 2 Greater magnification of boxed area ofFig. 1. 49 % ofthe cells in this field are eosinophils. copyright. (H and E x 1650). http://adc.bmj.com/ haematopoietic foci. The portal tracts contained normal. In all the tissues except bone marrow the numerous eosinophils but there was no significant eosinophils were of mature form. Mast cells were not increase in portal fibrous tissue. greatly increased in any of the tissues. The subcapsular region of the kidney showed on September 30, 2021 by guest. Protected infiltration by eosinophils which extended deeply into the cortex in a linear manner associated with the Discussion nephrogenic formations at the margins of persistent fetal lobules (Figs 3 and 4). The lungs showed The sequence of events in this infant appeared to be intense congestion and intra-alveolar haemorrhage eosinophilic infiltration of various organs, particu- but there was no inflammatory cell infiltration and no larly the terminal ileum, followed by perforation of eosinophil infiltration. Extra medullary haemato- the terminal ileum and death from E. coli peritonitis. poiesis was not present in the lung. Bone marrow The main interest is in the nature of the eosinophilic from sternum and upper end of femur was modera- infiltrate and this suggests an inflammatory rather tely cellular and contained large numbers of mature than a neoplastic process. The abnormal histiocytic and immature eosinophils. The subcapsular sinuses cells characteristic of Letterer Siwe's disease are not of a lymph node from the neck were infiltrated by seen and the findings do not support a diagnosis of eosinophils but these were not seen elsewhere in the eosinophilic leukaemia. node. Salivary gland and myocardium appeared The infant lived for only 4 days and it is unlikely Arch Dis Child: first published as 10.1136/adc.56.1.66 on 1 January 1981. Downloaded from Disseminated eosinophlilic infiltration ofa newborn infant 69 Fig. 3 Renal cortex showing band-like cellular infiltrate at interface between 2 persistent fetal lobules. (H and E x 143). copyright. that such a widespread and severe eosinophilia et al.11 described eosinophilic infiltration of the could be an allergic response to the reconstituted oesophagus in a 3-month-old infant. Eosinophilic milk with which he had been fed for 48 hours before infiltration of the pancreas in infants born to diabetic http://adc.bmj.com/ the sudden collapse. The membranes had been mothers is well documented.12 ruptured for several hours before delivery but in the Eitzman and Smith'3 have produced evidence that absence of inflammatory cell infiltration of the lungs some infants between ages 2 and 21 days respond to this is an unlikely cause of the abnormal cellular nonspecific stimuli with a pronounced eosinophilia. infiltration of other tissues. Careful search of all the Perhaps this is the explanation for the widespread fetal tissue taken at necropsy showed no fungi or eosinophilia seen
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