Intussusception in Twins

(1)

suspected. Only one infant who had an intra

cranial

hemorrhage

at autopsy

(case

1, Table

I)

did not have a difference

in the percentages

of

fetal hemoglobin

in spinal

fluid and circulating

blood,

a

false-negative

test.

However,

the

patient's

clinical

course

suggested

the intraven

tricular

hemorrhage

occurred

after

his

initial

transfusion

and

that

the

lumbar

puncture

was

performed

prior to any subsequent

transfusions.

Mixing of hemorrhagic

spinal fluid and systemic

blood during lumbar

puncture

might also reduce

the difference

in the percentages

of fetal hemo

globin in blood and spinal fluid, also yielding

a

false-negative

test.

Changes in the percentage

of fetal hemoglobin

following several transfusions

were determined

in

one patient

(Fig. 2). By 18 hours of age, only 40%

of the circulating

hemoglobin

was fetal.

Since

96% of the spinal

fluid hemoglobin

obtained

at

this time was fetal, we concluded that the intra

ventricular hemorrhage occurred prior to 18

hours

of age. By collecting

samples

of blood

prior

to each transfusion

in a group of infants

at high

risk

of

having

intracranial

hemorrhage,

we

believe

this

technique,

together

with

clinical

signs,

might

establish

the

time

of

bleeding.

However,

the use of the

test

in this way

has

certain limitations.

Repeated

episodes of bleeding

into the spinal

fluid or mixing

of hemorrhagic

spinal

fluid and systemic

blood

at the time

of

lumbar

puncture

might

suggest

that

the

initial

bleeding

episode took place at a later age than it

actually

had occurred.

Despite

possible

limitations

of the

test

for

estimating the tinie intracranial hemorrhages

occur,

the

comparison

of the

percentages

in

samples

of blood

and

hemorrhagic

spinal

fluid

appears

to be a useful

way

to differentiate

a

subarachnoid

hemorrhage

from

a

traumatic

lumbar

puncture

in infants

who have

been

trans

fused.

EDWARD R. CHAPLIN, M.D.

MUREEN A. SCHLUETER, B.S.

RODERIC H. PHLBBS, M.D.

JOSEPH

A. KITTERMAN,

M.D.

WILLIAM H. TOOLEY, M.D.

Cardiovascular

Research

Institute

and the Depart

ments of Pediatrics

and

Neurology,

University

of California

San Francisco, California

Supported b@ NHL1 Pulmonary SCOR grant HL 14201

and Child Health and Human Development grant HD

00397. Dr. Chaplin is the recipient of a National Institutes of

Health Special Postdoctoral Fellowship Award (1 Fli

NS

02745) and Dr. Phibbs is the recipient of a National Institutes of Health Career Development Award (HD 18275).

ADDRESS FOR REPRINTS: (E.R.C.) Department of

Neurology, University of California, San Francisco, Cali fornia 94143.

REFERENCES

1. GrÃ¶ntoft 0: Intracerebral and meningeal haemorrhages in perinatally deceased infants: I. Intracerebral

haeniorrhages. Acta Obstet Gynecol Scand 32:303,

1953.

2. Larroche JC: HÃ©morragies cÃ©rÃ©bralesintraventricu laires chez le premature. I. Anatonlie et physiopa thologie. Biol Neonate 7:26, 1964.

3. Amid C: HÃ©niorragies cÃ©rÃ©bralesintraventriculaire chez Ic premature: II. Les elements du diagnostic

clinique. Biol Neonate 7:57, 1964.

4. Ross JJ, Dirnmette RM: Subependymal cerebral hemor

rhage in infancy. Am J Dis Child 110:531, 1965.

5. Towbin A: Cerebral intraventricular hemorrhage and

slll)ependvmal infarction in the fetus and prema

tore newborn. Am J Pathol 52:121, 1968. 6. Valdes-Dapena MA, Arey JB: The causes of neonatal

mortality: An analysis of 501 autopsies on newborn

infants. J Pediatr 77:366, 1970.

7. Fredrick J, Butler WR: Causes of neonatal death: II. Intraventricular haemorrhage. Biol Neonate

15:257, 1970.

8. Larroche JC: Post-haemorrhagic hydrocephalus in infancy: Anatomical study. Biol Neonate 20:287,

1972.

9. Oski FA, Naiman JL: Hematological Problems in the Newborn. Philadelphia, WB Saunders Co. 1972, p 135.

10. Chaplin ER, Schlueter MA, Phibbs RH, et al: The use of CSF fetal hemoglobin to differentiate pathological

subarachnoid bleeding from traumatic lumbar

punctures. Clin Res 23:158A, 1975.

11. Jonxis JHP, Huisman TH: The detection and estimation of fetal hemoglobin by means of the alkali denatu ration test. Blood 11:1006, 1956.

12. Lorber J, Bahat US: Post-hemorrhagic hydrocephalus. Arch Dis Child 49:751, 1975.

Intussusception

in Twins

The risk of intussusception

occurring

in the

siblings

of

patients

with

intussusception

is

thought

to be low, and there

is no previous

report

of the condition

occurring

in twins.

In our case the second twin was affected within

36 hours

of the onset

in the first. Adenovirus

was

cultured

from

the

one

twin

who

was

investi

gated.

754 PEDIATRICS

Vol. 58 No. 5 November1976

at Viet Nam:AAP Sponsored on September 8, 2020

www.aappublications.org/news

(2)

FIG. 1. Barium enema outlining intussusception in twin A.

FIG. 2. Barium enema outlining intussusception near hepatic flexure in twin B.

CASEREPORT

The twins were identical boys of West Indian descent, 25 months of age, born and bred in England. They were perfectly well until the early hours of May 26, 1972, when

twin A began to scream with acute abdominal pain. He was

admitted to hospital within 18 hours and by that time he had

vomited four times and had passed six loose stools, the last

two containing blood.

Examination revealed an ill-looking boy, with a tempera

ture of 39.6 C and a pulse rate of 100 beats per minute. There was a nontender ITLISSlying across the epigastriiim and on rectal examination blood was present. Hemoglobin was 10 gm/100 flll and the WBC was 5,500/cu mm with 58%

polymorphonuclear leukocytes, 1% eosinophils, 1% baso

phils, 34% lymphocytes, and 6% monocytes.

Shortly after admission a barium enema confirmed an

intussusception with the apex in the transverse colon (Fig. 1). The intussusception was partly reduced by the barium enema, but a filling defect was still present in the cecum, and at operation the intussusception was confirmed and reduced

easily, having started in the terminal ileum. It was noted that

there were masses of enlarged nodes in the mesentery. Twin B presented at 2 @sion May 27th within 36 hours of the onset of the condition in twin A. There was a five-hour

history of episodes of crying and drawing up his knees. He

had vomited twice and passed two stools consisting of mucus and blood only. Examination revealed a healthy-looking boy, with a temperature of 37.5 C, and a pulse rate of 100 beats

per minute.

He was tender on the right side of the abdomen with a

suspicion of a mass in the right hypochondrium. Rectal

examination revealed the presence of blood. Hemoglobin was 9.0 gm/100 ml and WBC 5,500/cu mm with 41% lynl phocytes and 12% monocytes.

A barium enema revealed a picture similar to that of the other twin, with the apex of the intussusception reaching the

transverse colon (Fig. 2). The intussusception was partly

reduced by the barium enema but still left a filling defect in the cecum and final reduction had to be undertaken by

operation, when it was found that the intussusception had

started in the terminal ileum.

The Peyer's patches were very enlarged, and one Peyer's

patch at the apex of the intusSu.sception felt almost like a foreign body. The mesenteric lymph nodes were also grossly enlarged. The appendix and one mesenteric node were re moved for virology studies.

Both twins made an uneventful recovery before discharge

EXPERIENCE AND REASON

755

(3)

together on June 5, 1972. A heavy growth of adenovirus was

obtained from the appendix and also from the mesenteric

lymph nodes. The antibody titers for twin B were less than 1:20 in all viruses tested (adenovirus, mumps virus V, mumps virus 5, herpes simplex virus, measles, varicella-zoster and

cox.sackie). It had been intended to undertake a study of

antibody titers in both twins: unfortunately the laboratory

staff thought a mistake had been made in sending a second

specimen with the same surname and discarded it, not realizing that this was from twin A, and there was a similar failure to undertake studies of convalescent serum.

DISCUSS1ON

Intussusception

occurring

in siblings has been

described.

MacMahon'

described

eight families in

which the siblings were affected

in a series of 296

confirmed cases of intussusception. Clos& found

five cases in siblings in 363 cases (a very low ratio)

and

Hogg

and

DonovanI

described

three

cases

arising

in

relatives

of

123 patients.

Ravitch4

concluded

that â€œ¿there

is no evidence

that there is

any increased likelihood of intussusception in a

sibling

once a child in a family

has been

affected,â€•

but MacMahon

estimated

the risk to siblings as 1

in 40, about

15 to 20 times the incidence

in the

general

population.

No reference

has been

found

relating

to intus

susception

in twins.

The lymph

nodes

and

Peyer's

patches

were

very

enlarged

ih

both

twins,

and

the

Peyer's

patches

seemed

to be the leading

points

in the

intussusception.

The

enlargement

of

this

lym

phoid tissue is likely to be associated

with a recent

infection

and many observers

have drawn

atten

tion

to the

role of viruses

in the

etiology

of

intussusception.3'

Indeed, Potter and Zachary'

were able to demonstrate

an adenovirus

in the

feces, throat

swabs, or mesenteric

nodes in more

than 50% of cases of intussusception,

compared

with 3% in children

admitted

to the hospital

with

various

other

conditions,

including

respiratory

infections.

It seems remarkable,

therefore,

that

Dennison

and Shakerâ€• were

unable

to find virus

infection

in children

with

intussusception

more

frequently

than

in a series

of controls.

The presence

of enlarged

lymphoid

tissue and

the positive culture

of virus from both these cases

is in itself suggestive that the infection was an

important

etiological

factor.

The

onset

of the

disease in the second twin within 36 hours of the

onset

in the first twin

strengthens

this view.

GARETH G. THOMAS, F.R.C.S.

R. B. ZACHARY, F.R.C.S.

Sub-Department

of Pediatric

Surgery,

University

of Sheffield,

and the Children's

Hospital

Sheffield, England

ADDRESS FOR REPRINTS: (R.B.Z.) The Children's Hospital, Western Bank, Sheffield 10, England.

REFERENCES

1. MacMahon B: Data on the etiology of acute intussuscep tion in childhood. Am J Hum Genet 7:430, 1955. 2. Close HG: Acute intussusception in children. Guy's

Hosp Rep 81:436, 1931.

3. Hogg BM, Donovan EJ: Acute intussusception in infants and children. Ann Surg 124:262, 1946.

4. Ravitch MM: Intussusceptiori in Infants and Children. Springfield, Illinois, Charles C Thomas, 1959. 5. Clarke EJ, Phillips IA, Alexander ER: Adenovirus infec

tion in intussusception in children in Taiwan.

JAMA 208:1671,1969.

6. Strang R: Intussusception in infancy and childhood. Br J Surg 46:484, 1959.

7. Bell TM, Steyn JH: Viruses in lymph nodes of children with mesenteric adenitis and intussusception. Br

Med J 2:700, 1962.

8. Gardiner PS, Knox EG, Court SDM, Green CA: Virus

infection and intussusception in childhood. Br Med

J 2:697, 1962.

9. PotterCW, Zachary RB: The etiology of intussuscep

tion: With particular attention to the adenovirus infectiolls. Surg Clin North Am 44:1509, 1964.

10. Dennison WM, Shaker M: Intussusception in infancy and childhood. Br J Surg 57:679, 1970.

756 INTUSSUSCEPTION IN TWINS

(4)

1976;58;754

Pediatrics

Gareth G. Thomas and R. B. Zachary

Intussusception in Twins

Services

Updated Information &

http://pediatrics.aappublications.org/content/58/5/754

including high resolution figures, can be found at:

Permissions & Licensing

http://www.aappublications.org/site/misc/Permissions.xhtml

entirety can be found online at:

Information about reproducing this article in parts (figures, tables) or in its

Reprints

http://www.aappublications.org/site/misc/reprints.xhtml

Information about ordering reprints can be found online:

(5)

1976;58;754

Pediatrics

Gareth G. Thomas and R. B. Zachary

Intussusception in Twins

http://pediatrics.aappublications.org/content/58/5/754

the World Wide Web at:

The online version of this article, along with updated information and services, is located on

been published continuously since 1948. Pediatrics is owned, published, and trademarked by the

Pediatrics is the official journal of the American Academy of Pediatrics. A monthly publication, it has