CHRONIC
ULCERATIVE
COLITIS
Case
Report
in a Newborn
Infant
By SAMUEL
L.
BERANBAUM,M.D.,
AND ROBERTJ.
WALDRON,M.D.
. New
York
City
C
HRONIC
ulcerative
colitis
in children
is not uncommon. Of 871 cases at the Mayo Clinic reviewed by Jackman et al.,’ 10.9/c were children. Of these 95 cases, three had an onset of the disease within the first year of life. Hart’ reported a case in the new-born with evidence of perforation, which was later shown by postmortem examination tobe due
to chronic
ulcerative
colitis.
This
case
report
is presented
as
another
beginning
soon
after
birth,
with
roentgeno-graphic studies of the colon. Although other reports of the disease in infancy are well
documented,
this
report
is
the
first
in
the
newborn
with
complete
roentgenographic
studies.
REPORT OF CASE
At the age of 2 1 days, this white Italian male was admitted for the first time to University Hos-pital with the chief complaint of vomiting.
The present illness began 3 days after birth when it was noticed in the nursery that the bfant began to vomit part of or all of its feedings. At the same time the infant became constipated so that enemata were necessary to obtain bowel movements. On one occasion, a small amount of blood was present in the stool. The infant was discharged from the nursery on the 5th day after birth on an evaporated milk, boiled water, dextrin-maltose-dextrose formula, but again at home he proceeded to have vomiting spells. The vomiting was never projectile. After the use of an antispasmodic before each feeding, the vomiting was controlled. Two days before admission date, the infant began to vomit again and now it was noticed that abdominal distension was present.
The family history is noncontributory.
Past history was unrevealing. The pregnancy was uneventful. The infant was a full term spontaneous delivery after a 2 hr. labor period and weighed 3943 gm. at birth.
Admission physical examination revealed a temperature of 38.4#{176}C., pulse 100/mm., respiration 44/mm. The admission weight was 3.5 kg.
l’he infant did not appear acutely ill and the only positive physical findings at this time were abdominal distension and some prominence of the superficial veins of the abdominal wall.
Laboratory work on this admission revealed: RBC 3.45 million/cmm. ; Hgb. 10.8 gm./100 cc.; WBC 17 thousand/cmm. with 34 neutrophiles; 36 lymphocytes, 12 monocytes, 15 metamyelocytes
and 3 myelocytes of the neutrophilic type. An admission CO, combining power was 16.8 mEq./l. Seven stool cultures for enteric pathogens were negative. The erythrocyte sedimentation rate was 33 mm/hr. corrected.
Roentgenographic Examination (see Figs. 1 and 2) : 1. Roentgenographic examination of the
thorax showed no cardiac or pulmonary abnormalities and scout films of the long bones showed no
gross abnormalities.
2. An initial scout film of the abdomen showed moderate gaseous distension of the intestines.
3. Colonic studies by barium enema and meal by mouth showed multiple mucosal irregularities and ulcerations throughout ascribable to ulcerative colitis. The colon was tubular in its entire course with absent haustrations.
Course in the Hospital: In the first part of the infant’s stay in the hospital, it was considered that the infant had a small bowel obstruction. The infant passed small amounts of stool, consisting
From the Departments of Radiology and Pediatrics, University Hospital, New York University-Bellevue Medical Center, New York City.
(Received for publication Dec. 26, 1951.)
774 SAMUEL L.
BERANBAUM
AND
ROBERT
J.
WALDRONFIG. 1A and B. Colon shows multiple irregularities of mucosal pattern with multiple small ulcera-tion. Patient was 24 days old at this time.
FIG. 2A and B. Colon visualized by meal by mouth shows tubularity throughout with absence of
CHRONIC
ULCERATIVE
COLITIS
775
of black slimy material, which gave a negative reaction to the guaiac test. The abdomen remained distended and fl() bowel sounds were heard. Since the consensus of surgical opinion was that this was a small bowel obstruction, a laparotomy was performed on the 6th hospital day.
At operation, the terminal ileum, cecum, ascending colon and part of the transverse colon showed a thick gut wall which was extremely friable and considerably inflamed. Following minimal manipu-lation of the intestine at operation, a perforation was inadvertently made into the cecum and just superior to that point in the ascending colon. The cecal perforation was closed and the ascending colon perforation was utilized for drainage.
Postoperatively the infant was given antimicrobial agents and supportive therapy, including
FIG. 3. Gross specimen of colon showing multiple tiny ulcerations from ileocecal valve to midportion of rectum, ulcerations averaging approximately 0.2 cm. in greatest diameter.
whole blood transfusions and plasma. However, in spite of the initial improvement of the child, abdominal distension slowly increased. The colostomy opening was constantly irrigated and was felt to be draining well. On the 4th postoperative day, the infant was doing better. The abdomen was now soft and the colostomy was closed. The infant was placed on an evaporated milk formula.
On the 10th postoperative day, the symptoms of abdominal distension, vomiting and paucity of stools recurred. A WBC revealed 29,000/cmm. with a differential of 78% neutrophiles, 20 lympho-cytes and 2 monocytes.
776
SAMUEL
L.
BERANBAUM
AND
ROBERT
J.
WALDRONFIG. 4. Photomicrograph of section of colon showing typical ulcer with exudate.
The infant was readmitted to University Hospital at the age of 8 mo. for closure of the transverse colostomy.
Admission physical examination revealed a well developed infant, now weighing 9.1 kg. The only positive physical finding was a colostomy opening on the abdominal wall.
On the 12th hospital day, a closure of the transverse colostomy was performed. The infant did well after the operation and was taking feedings by mouth. On the 8th postoperative day, gradual abdominal distension developed and the temperature rose to 39.4#{176}C. The infant then developed a diarrhea and had 5 large loose to watery brown stools within 12 hr. In addition, the infant began to vomit his feedings.
Oral feedings were discontinued and parenteral feedings started. In spite of adequate intravenous fluids and electrolytes, as well as antimicrobial agents, the infant’s condition grew steadily worse.
A scout film of the abdomen at this time revealed dilated loops of transverse colon on the right side with gas in the descending and sigmoid colon.
In spite of all efforts at supportive therapy, the infant died on the 23rd hospital clay. Pathology: Only the pertinent pathologic findings are outlined.
Alimentary Tract: Esophagus: Measures 11.0 X 1.5 cm. The mucosa has a greyish-white appear-ance and its surface is covered by small flecks of a blackish-brown material. Stomach: Measures 20 cm. along the greater curvature, 8 cm. along the lesser curvature and 10 cm. in maximal fundal circumference. The rugal pattern is maintained and the mucosa is covered by a mucoid material which is generally clear but contains some areas of dark reddish-black material. The pylorus is patent. The duodenum does not appear unusual. The jejunum is not remarkable but the most of the ileum shows marked congestion of the serosal vessels with some thickening of the wall and greenish-yellow patchy diphtheroid membrane on the mucosal aspect. The appendix is present and measures 3 cm. in length and 0.8 cm. in diameter. It is bound down to the retrocecal portion.
CHRONIC
ULCERATIVE
COLITIS
777is pale and smooth and there are numerous punctate areas of the mucosa which show a dark reddish or blackish-brown appearance. These areas average approximately 0.4 to 5 cm. in size. In the midportion of the transverse colon, there is an indurated area containing numerous suture materials. Apparently this area represents the end-to-end anastomosis following the transverse colostomy. There is no evidence of leakage at this point nor is there evidence of abscess along the entire bowel. Distal to the midpoint of the rectum the mucosa does not appear unusual. No stric-tures nor valvelike obstructive areas are observed in any other portion of the bowel. The wall of the large intestine averages 0.4 cm. in thickness. Cultures from the intestine at postmortem were negative for enteric pathogens.
Microscopic examination revealed:
lleum: These sections are from areas grossly covered with a friable yellowish-grey membrane. The membrane is seen to be composed of a layer of fibrin in which are enmeshed innumerable polymorphonuclear leukocytes and lymphocytes overlying a congested and somewhat fragmented mucosa which also shows infiltration by inflammatory cells and what appear to be myelocytes.
The submucosa is also infiltrated by these cells. The muscularis does not appear unusual but the
serosa is thickened and infiltrated by polymorphonuclear leukocytes, lymphocytes and plasma cells. Some areas of the mucosa show a simplified epithelium which suggests regeneration.
Large Intestine: Multiple sections of large intestine taken from the grossly involved areas show
the mucosa to be generally intact. However, there are scattered areas which show necrosis and ulceration of the mucosa characterized by the appearance of small areas of fibrin containing numerous polymorphonuclear Ieukocytes, red blood cells and nuclear dust which replace the mucosal ele-ments. Many of these areas are seen overlying hyperplastic lymph follicles in the submucosa.
Be-tween these ulcerated areas the mucosa does not appear altered. The submucosa in some of the
sections shows edema and thickening while the muscularis mucosa is clearly demarcated. The muscularis and serosa generally show no unusual features. Meissner’s and Auerback’s plexuses show no changes of interest. Stains for bacteria reveal gram-negative rods on the superficial portions of the exudate but not in the bowel wall. Section of rectosigmoid junction shows a transitional thickening of the submucosa by edema fluid in the sigmoid area.
Final Diagnosis: 1. Acute ulcerative colitis; 2. Acute ileitis.
CONCLUSION
A case
of
ulcerative
colitis
in the newborn infant is presented with roentgenographic and postmortem data. This is the earliest case on record with roentgenographic studies of the colon, the latter being made at 24 days of age.REFERENCES
1. Jackman, R., Bergen, A. J., and Helmholz, H., Life histories of 95 children with chronic
ulcera-tive colitis, Am. J, Dis. Child. 59:459, 1940.
2. Hart, J. A., Ulcerative colitis with perforation in newborn, Texas State J. Med. 42:286, 1946-47.
SPANISH ABSTRACT
Colitis
Cronica
Ulcerosa
; Un
caso
en un
reclen
nacido
Solamente existen tres casos de colitis de este tipo en Ia literatura m#{233}dicainiciados en el primer a#{241}ode la vida y uno en un reclen nacido que a la autopsia mostrO ser una colitis ulcerosa crOnica perforada.
El de estos autores es el primero en que se hace un estudio radiolOgico completo.
778
SAMUEL
L.
BERANBAUM
AND
ROBERT
J.
WALDRON
demostraran distension moderada de los intestinos y ulceraciones e irregularidades miiltiples de Ia mucosa del colon que era de aspecto tubular en toda su extensiOn y sin pliegues. La impresi#{243}n radiolOgica fu#{233}de colitis ulcerosa. Pens#{225}ndose en una oclusiOn intestinal se hizo laparotomla pero el diagnOstico no fu#{233}comprobado encontr#{225}ndose engrosamiento de las paredes del Ileo, ciego, colon ascendente y parte del transverso el cual estaba muy friable y considerablemente inflamado, practic#{225}ndose una colostomla del colon ascendente. Debido a Ia recidiba de los sIntomas despu#{233}s de un perlodo transitorio de mejorla se realizO posteriormente una colostomIa transversa a partir de
la cual hubo mejorla franca, y el enfermo se enviO a su domicilio.
A los ocho meses se admitiO nuevamente para cerrar Ia colostomla ya que el ni#{241}opesaba alrededor de 9 kgs., pero el postoperatorio se complicO con distensiOn abdominal, temperatura y
diarrea profusa terminando con la vida del paciente en las prOximas dos semanas. El estudio
postmortem reporO fundamentalmente numerosas ulceraciones diminutas en todo el colon que
microscOpicamente correspondlan a areas diseminadas de necrosis y ulceraci#{243}n de la mucosa cubiertas de fibrina e infiltradas con gran nOmero de leucocitos polimorfonucleares y eritrocitos.
Muchas de estas areas de fibrina se encontraban cubriendo follculos linf#{225}ticoshiperpl#{225}sticos de la submucosa. El yeyuno presentaba zonas cubiertas de una membrana difteroide verde amarillenta
y algo de engrosamiento parietal, rnembrana que estaba constituida por una capa de fibrina infiltrada profusamente por polinucleares y linfocitos, infiltraciOn que se extendla a la mucosa, submucosa y serosa, respetanto la muscularis.