The THIRD OF THREE ORIGINAL OBSERVATIONS

That coeliac disease in the Yorkshire region was disappearing as judged by the numbers of children referred to me for jejunal biopsy.   

Littlewood JM, Crollick AJ, Richards IDG. Childhood coeliac disease is disappearing. Lancet 1980; ii: 1359.

During the Seventies paediatric consultants in the Yorkshire Region increasingly referred children to Seacroft for our biochemist’s (Mr Alan Steel) reliable sweat tests and my paediatric jejunal biopsy service which I had started in the late Sixties. 

When I returned to Leeds from Great Ormond Street in 1964, having observed the new iontophoresis sweat tests there, I persuaded our Prof. Craig to approve the purchase of Gibson and Cooke sweat testing apparatus. Alan Steel used this equipment to perform very accurate sweat tests from 1964 that soon became a well-established service that most paediatricians from Leeds and surrounding general hospitals used for their patients.

Per oral jejunal biopsy was the other new major gastrointestinal investigation. Prof. John Walker Smith regards per oral intestinal biopsy as being the basis of paediatric gastroenterology developing as a speciality. In 1968 I obtained a paediatric sized Crosby intestinal biopsy capsule (Watson capsule) and after a steep personal learning curve, started a paediatric jejunal biopsy service; this facility soon attracted referrals from paediatricians in the region to confirm or exclude coeliac disease (CD) in their children. We eventually changed to the twin port Kilby modification of the Crosby capsule to use one specimen for  histology and the other for biochemical (disaccharide) estimation (Kilby, 1976).

         

PAEDIATRIC INTESTINAL BIOPSY CAPSULES – KILBY AND WATSON

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    Normal small intestinal mucosa
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    Subtotal villous atrophy of untreated coeliac disease
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    Biopsy capsule in the small bowel
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              Dr Margot Shiner
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Prof. Charlotte Anderson

The characteristic histological small bowel appearance of subtotal villous atrophy of gluten induced coeliac disease was first identified in 1954 by Paulley, a physician in Ipswich, in laparotomy specimens from four adults with idiopathic steatorrhoea (coeliac disease). In 1957 Margot Shiner was the first clinician to obtain specimens by per oral small bowel biopsy from an 8-year old child; soon a series of small bowel biopsies in children was reported by Charlotte Anderson.
In contrast to the subtotal villous atrophy in coeliac disease, in people with CF the intestinal villi are of normal height – in some children with CF whom we biopsied the intestinal villi appeared to be even taller than normal.

Paulley J W. Observations on the aetiology of idiopathic steatorrhoea. Jejunal and lymph node biopsies. BMJ 1954; 173:1318-1321. [PubMed]
Sakula J, Shiner M. Coeliac disease with atrophy of the small intestine mucosa. Lancet 1957; ii: 876-877. [PubMed]
Anderson CM. Histological changes in the duodenal mucosa in coeliac disease. Reversibility during treatment with a wheat gluten free diet. Arch Dis Child 1960; 35:419-427. [PubMed]
Kilby A. Pediatric small intestinal biopsy capsule with two ports. Gut 1976; 17:158-15

In this venture I had the invaluable help of my friend and colleague Dr. Sidney Smith, our paediatric radiologist, who was responsible for the rapid X-ray screening of the capsule into the duodenum. Sydney was a good friend of paediatrics and helped us with many studies and clinical problems over the years. Dr Mike Mason, again avery helpful pathologist from St James’s, provided histology reports for many years.

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Dr Avril Crollick

Later Dr. Avril Crollick was appointed as my Clinical Assistant and she eventually performed many of the biopsies until I started using the paediatric endoscope for the purpose during the Eighties.                                                                      The ability to obtain jejunal mucosa for histological and biochemical examination from children was a relatively recent but one of the really major advances in the Seventies.

Jejunal biopsy with a paediatric sized Crosby capsule was a relatively new technique in the Sixties. Initially Sir Wilfred Sheldon (the senior consultant paediatrician at Great Ormond Street) and a senior registrar at GOS at the time, Dr. Eddie Tempany, (who was subsequently Professor of Paediatrics in Dublin), considered the investigation to be “outside the scope of routine investigations in children” due to the increased difficulty and risk of perforation reported in small children.

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Eddie Tempany

It is true there were a number of reports of intestinal perforation. On one occasion, when I was working at GOS and observing Eddie performing a biopsy, the capsule could not be retrieved until the next day. So I admit it was with some trepidation that I started the jejunal biopsy service at Seacroft.  However, as with many practical procedures when restricted to a few experienced individuals complications were less likely.

Subsequently, elsewhere, I have published in detail our experience with over 1000 jejunal biopsies and 108 children with coeliac disease in my friend Professor Peter Howdle’s book (Howdle PD, 1997). Peter, was a major friend of paediatrics in St James’s, eventually succeeded Monty Losowsky as professor  of medicine at St James’s. He was an excellent and helpful colleague in many areas of gastroenterology where his expertise and kind approach benefited many of our young patients including those with cystic fibrosis