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Amnion nodosum
Amnion nodosum Report
of a case
.4LEX.
M.
JOHN
U.
M.
M
IO
N
tively to
N
to 6
H.41JST,
the
The
in
association
latter
in
with
renal
major ing
with is
have
been
an
in of
of
of
nodules
these
of
aspect
been
reported
urinary
large
threatened
in of
It
is
the
sum
blood
agenesis.
tiple
congenital
purpose
artery
leada fern
which
there
has
on
addition,
bronchial present
maternal to
pregnancy.”
this case
in pres-
related
early
of
was
fetus the
the
communication of amnion
associated
In
with
a dead
apparently in
was
renal umbilical
clots
an additional
which
other
tract,
and
placenta,
branes were intact and there was a breach presentation. Soft tissue films of the pelvis showed an anteriorly situated placenta and a relatively small uterus, closely applied to the fetus. The fetal heart sounds were heard at a rate of 130 to 140 per minute, until 40 minutes before delivery, at which time they could not be heard with certainty. Initially, the patient had mild contractions and she passed dark blood and blood clot per vaginam. Subsequently, she went into active labor and one hour later she was delivered of a stillborn male infant. The placenta followed spontaneously. No mention is made on the chart concerning the amount of amniotic fluid. The stillborn infant weighed 1,400 gram<. There were no external malformations and the typical “Potter facies,” found in renal anomalies, was not observed. There was bilateral rrnal agenesis associated with the absence of both renal arteries and ureters and with pronounced hypoplasia of the urinary bladder. The lungs were small and not expanded, the right weighing 8 Trams and the left 7 grams. Microscopically, numerous dilated spaces were seen (Fig. I) with some increase in interstitial bulk but without definite defect of the alveoli. These spaces contained a small amount of protein material and were lined by either cuboidal or columnar cpithelium. They wel-r considered to be of bronchial origin (Fig. 2). The bronchial walls were commonly devoid of supporting cartilage and muscle. No other abnormalities were noted.
found
of oligohydramnios
abortion
to report
lies
and
However,
retention
of the
plaThe
fetus
circummargination,”
ence
the
invariably the
spontaneous abortion in August of the same year. The third pregnancy, in 1954, was normal, and a healthy infant weighing 3,230 grams was delivered by forceps. On the present admission, the fetal mem-
(1
yellow
surface
the
micturition.
association
descripsmall
oligohydramnios.
of
postmaturity, uter0,3
refers
numerous
almost
agenesis
lack
M.D.
grayish
significance turn?
cases
of fetal
anomalies to
0 s u M’
D
circumscribed, on
centa. the
o
presence
mm.),
elevations
M.D.
Ontario
N
the
M.D
BALIS,
DARIA
Kirlgston,
A
BRYANS,
with there cysts, in
nodobilateral
were and the
only
mulone
cord.
A 28-year-old housewife was admitted to the Kingston General Hospital on Oct. 24, 1960, in the thirty-fourth week of her fourth pregnancy. She gave a history of vaginal bleeding and occasional cramps for 2 days. Previous obstetric history revealed that her first pregnancy had been uneventful, but it terminated in March, 1953, by forceps delivery of a stillborn infant weighting only 2,270 grams. She had a From the Departments of Paediatrics Pathology, Queen’s University and Kingston General Hospital.
and
582
Volunle Number
84 5
Amnion
The placenta weighed 510 grams. There was a large retroplacental blood clot covering approximately three fourths of the maternal surface. The amnion on the fetal surface was studded with numerous small, grayish yellow nodules varying in diameter from 1 to 6 mm. Some of them were confluent (Fig. 3). Microscopic examination showed that they were composed of conglomerates of squamous epithelium embedded in and held together by an amorphous acidophilic material (Fig. 4). The areas of the amnion on which these nodules lodged were devoid of their epithelial surface and, at some sites, connective tissue elements extended from the amnion to the nodules. The amniotic epithelium covered the deposited nodule either partly or completely (Fig. 5). Nowhere was squamous metaplasia of the amniotic epithelium seen. There was no evidence of inflammation in any of the placental tissue. The umbilical cord contained only two vessels, one artery and one vein, but was otherwise not remarkable (Fig. 6). Comment The term “amnion nodosum” was coined by Landing1 in 1950, a translation from the German ,“Amnionknotchen.” He encountered in the old literature 11 reported cases; of these, 3 cases were “doubtful,” in 5 there was no description of the fetus, in one case
nodosum
583
there was no malformation, and in remaining 2 only was there a major malformation of the urinary tract. Landing added 8 cases of his own, of which 5 were proved to be associated with urinary tract anomalies. Oligohydramnios was an almost constant feature in these cases. Fourteen further cases of amnion nodosum were reported by Scott and Bair? and Jeffcoate and Scott3 by 1959. 01 these, 10 were associated with oligohydramnios and renal agenesis, 3 were associated with oligohydramnios and a normal fetal urinary tract, and one case was associated with intrauterine death followed by retention of the fetus for 3 weeks. In the same year Blanc4 reported on a collection of 10 cases. He referred to this condition of the amnion as “vernix granulomas” and stated that oligohydramnios was actually observed or highly probable in 8 cases. Six patients in his series had severe genitourinary anomalies. The placentas in 2 other cases had severe circummargination, and in the remaining 2 cases there was prolonged retention of a dead fetus after rupture of the membranes. In 1960, Thompson added another case of amnion nodosum with renal agenesis and oligohydramnios.6 There are two possible pathways for the
Fig. 1. Section of lung. Multiple large spaces are present in lung parenchyma. (Hemalum-phloxine-saffron. x36.) Fig. 2. Detail of the eqithelium lining the spaces seen in the left lower corner Fig. 1. The epithelium is either cuboidal or columnar. (Hemalum-phloxine-saffron. X244.1
of
584
Bryans,
Balk,
and
Srptember
Hausi
1, 1962
Am. J. Obst. & Gynec.
Fig. 3. A section of the fetal surface of the placenta as seen in the gross. Multiple round to oval nodules cover the amnion. (~2.5.) Fig. 4. Microscopic appearance of an amniotic nodule. It is composed of amorphous acidophilic (pink in section, gray in photograph) material, embedded in which are squames. Note the desquamation of amniotic epithelium beneath the nodule and connective tissue extending from the amnion into the base of the nodule. (Hemalum-phloxine-saffron. x36.)
5 Fig. 5. Higher power view of a nodule to show amniotic segment and absent beneath it. (Hemalum-phloxine-saffron. Fig. 6. Umbilical cord contains a vein and only one (Hemalum-phloxine-saffron. x18.)
epithelium x102.) artery, at
genesis
Two
of
Landing. niotic cells
the
amnion
In
the
fluid stick
the to
degeneration
lium
upon
natively,
amnion of
which fetal
suggested
of
desquamated the
quent
nodosum
absence
they
movements
nios could traumatize areas whereon squames
fetal and
the
am-
epithelial
cause
subse-
amniotic
epithe-
precipitate. in
by
sufficient
Alter-
oligohydram-
the amnion precipitate.
in
focal
role
in
factors the
covering
it over
bottom
of
may
oligohydramnios
a short
photograph.
have
played
a causal
in
case:
our
the
absence of urine due to bilateral renal agenesis and the presence of a large blood clot adhering firmly to approximately three fourths of the maternal aspect of the placenta. Jeffcoate and Scott,3 when reviewing all the factors involved in secretion and absorption of amniotic fluid, considered blood
Volume 84 Number 5
clots over large areas of maternal aspect of the placenta as one of the causesof oligohydramnios. They referred to their 3 cases of oligohydramnios in which such was the finding, stating: “These lesions were such that, in these particular cases, the cause of oligohydramnios was a failure of the amnion to contribute its quota to the total volume of liquid.” It is known that bilateral agenesis of kidneys is associated with hypoplastic lungs.? It is of interest that our patient had multiple congenital bronchial cysts in the lungs. In addition, one umbilical artery was absent. Recently, Bourne and Benirschke* reviewed 113 cases of absent umbilical artery and the
REFEKENCES
1. 2.
3. 4. 5.
Landing, B. H.: AM. J. OBST. & GYNEC. 60: 1339, 1950. Potter, E. L.: Pathology of the Fetus and the Newborn, Chicago, 1952, Year Book Publishers, Inc. Jeffcoate, T. N. A., and Scott, J. S.: Canad. M. A. J. 80: 77, 1959. Blanc, W. A.: Am. J. Path. 35: 695, 1959. Scott, J. S., and Bain, A. D.: Proc. Roy. Sot. Med. 51: 512, 1958.
Amnion
nodosum
association of this anomaly with ditions, including oligohydramnios.
other
585
con-
Summary A case of amnion nodosum associated with bilateral renal agenesis, multiple bronchial cysts of the lungs, and an absent umbilical artery is reported and other cases from the literature are cited. While there is nothing in the chart referring to the amount of liquor amnii, it is assumed from the radiologic data that there was associated oligohydramnios. We wish to thank Dr. E. M. Robertson, Professor and Head of the Department of Obstetrics and Gynaecology, for his kind permission to use the obstetrical data concerning this case.
6. 7.
8.
Thompson, V. M.: J. Obst. & Gynaec. Brit. Emp. 67: 611, 1960. MacGregor, A. R.: Pathology of Infancy and Childhood, Edinburgh and London, 1960, E. & S. Livingstone, Ltd. Bourne, G. L., and Benirschke, K.: Arch. Dis. Childhood 35: 834, 1960.
Kingston Kingston,
General Hosjital Ontario, Canada
of a case
.4LEX.
M.
JOHN
U.
M.
M
IO
N
tively to
N
to 6
H.41JST,
the
The
in
association
latter
in
with
renal
major ing
with is
have
been
an
in of
of
of
nodules
these
of
aspect
been
reported
urinary
large
threatened
in of
It
is
the
sum
blood
agenesis.
tiple
congenital
purpose
artery
leada fern
which
there
has
on
addition,
bronchial present
maternal to
pregnancy.”
this case
in pres-
related
early
of
was
fetus the
the
communication of amnion
associated
In
with
a dead
apparently in
was
renal umbilical
clots
an additional
which
other
tract,
and
placenta,
branes were intact and there was a breach presentation. Soft tissue films of the pelvis showed an anteriorly situated placenta and a relatively small uterus, closely applied to the fetus. The fetal heart sounds were heard at a rate of 130 to 140 per minute, until 40 minutes before delivery, at which time they could not be heard with certainty. Initially, the patient had mild contractions and she passed dark blood and blood clot per vaginam. Subsequently, she went into active labor and one hour later she was delivered of a stillborn male infant. The placenta followed spontaneously. No mention is made on the chart concerning the amount of amniotic fluid. The stillborn infant weighed 1,400 gram<. There were no external malformations and the typical “Potter facies,” found in renal anomalies, was not observed. There was bilateral rrnal agenesis associated with the absence of both renal arteries and ureters and with pronounced hypoplasia of the urinary bladder. The lungs were small and not expanded, the right weighing 8 Trams and the left 7 grams. Microscopically, numerous dilated spaces were seen (Fig. I) with some increase in interstitial bulk but without definite defect of the alveoli. These spaces contained a small amount of protein material and were lined by either cuboidal or columnar cpithelium. They wel-r considered to be of bronchial origin (Fig. 2). The bronchial walls were commonly devoid of supporting cartilage and muscle. No other abnormalities were noted.
found
of oligohydramnios
abortion
to report
lies
and
However,
retention
of the
plaThe
fetus
circummargination,”
ence
the
invariably the
spontaneous abortion in August of the same year. The third pregnancy, in 1954, was normal, and a healthy infant weighing 3,230 grams was delivered by forceps. On the present admission, the fetal mem-
(1
yellow
surface
the
micturition.
association
descripsmall
oligohydramnios.
of
postmaturity, uter0,3
refers
numerous
almost
agenesis
lack
M.D.
grayish
significance turn?
cases
of fetal
anomalies to
0 s u M’
D
circumscribed, on
centa. the
o
presence
mm.),
elevations
M.D.
Ontario
N
the
M.D
BALIS,
DARIA
Kirlgston,
A
BRYANS,
with there cysts, in
nodobilateral
were and the
only
mulone
cord.
A 28-year-old housewife was admitted to the Kingston General Hospital on Oct. 24, 1960, in the thirty-fourth week of her fourth pregnancy. She gave a history of vaginal bleeding and occasional cramps for 2 days. Previous obstetric history revealed that her first pregnancy had been uneventful, but it terminated in March, 1953, by forceps delivery of a stillborn infant weighting only 2,270 grams. She had a From the Departments of Paediatrics Pathology, Queen’s University and Kingston General Hospital.
and
582
Volunle Number
84 5
Amnion
The placenta weighed 510 grams. There was a large retroplacental blood clot covering approximately three fourths of the maternal surface. The amnion on the fetal surface was studded with numerous small, grayish yellow nodules varying in diameter from 1 to 6 mm. Some of them were confluent (Fig. 3). Microscopic examination showed that they were composed of conglomerates of squamous epithelium embedded in and held together by an amorphous acidophilic material (Fig. 4). The areas of the amnion on which these nodules lodged were devoid of their epithelial surface and, at some sites, connective tissue elements extended from the amnion to the nodules. The amniotic epithelium covered the deposited nodule either partly or completely (Fig. 5). Nowhere was squamous metaplasia of the amniotic epithelium seen. There was no evidence of inflammation in any of the placental tissue. The umbilical cord contained only two vessels, one artery and one vein, but was otherwise not remarkable (Fig. 6). Comment The term “amnion nodosum” was coined by Landing1 in 1950, a translation from the German ,“Amnionknotchen.” He encountered in the old literature 11 reported cases; of these, 3 cases were “doubtful,” in 5 there was no description of the fetus, in one case
nodosum
583
there was no malformation, and in remaining 2 only was there a major malformation of the urinary tract. Landing added 8 cases of his own, of which 5 were proved to be associated with urinary tract anomalies. Oligohydramnios was an almost constant feature in these cases. Fourteen further cases of amnion nodosum were reported by Scott and Bair? and Jeffcoate and Scott3 by 1959. 01 these, 10 were associated with oligohydramnios and renal agenesis, 3 were associated with oligohydramnios and a normal fetal urinary tract, and one case was associated with intrauterine death followed by retention of the fetus for 3 weeks. In the same year Blanc4 reported on a collection of 10 cases. He referred to this condition of the amnion as “vernix granulomas” and stated that oligohydramnios was actually observed or highly probable in 8 cases. Six patients in his series had severe genitourinary anomalies. The placentas in 2 other cases had severe circummargination, and in the remaining 2 cases there was prolonged retention of a dead fetus after rupture of the membranes. In 1960, Thompson added another case of amnion nodosum with renal agenesis and oligohydramnios.6 There are two possible pathways for the
Fig. 1. Section of lung. Multiple large spaces are present in lung parenchyma. (Hemalum-phloxine-saffron. x36.) Fig. 2. Detail of the eqithelium lining the spaces seen in the left lower corner Fig. 1. The epithelium is either cuboidal or columnar. (Hemalum-phloxine-saffron. X244.1
of
584
Bryans,
Balk,
and
Srptember
Hausi
1, 1962
Am. J. Obst. & Gynec.
Fig. 3. A section of the fetal surface of the placenta as seen in the gross. Multiple round to oval nodules cover the amnion. (~2.5.) Fig. 4. Microscopic appearance of an amniotic nodule. It is composed of amorphous acidophilic (pink in section, gray in photograph) material, embedded in which are squames. Note the desquamation of amniotic epithelium beneath the nodule and connective tissue extending from the amnion into the base of the nodule. (Hemalum-phloxine-saffron. x36.)
5 Fig. 5. Higher power view of a nodule to show amniotic segment and absent beneath it. (Hemalum-phloxine-saffron. Fig. 6. Umbilical cord contains a vein and only one (Hemalum-phloxine-saffron. x18.)
epithelium x102.) artery, at
genesis
Two
of
Landing. niotic cells
the
amnion
In
the
fluid stick
the to
degeneration
lium
upon
natively,
amnion of
which fetal
suggested
of
desquamated the
quent
nodosum
absence
they
movements
nios could traumatize areas whereon squames
fetal and
the
am-
epithelial
cause
subse-
amniotic
epithe-
precipitate. in
by
sufficient
Alter-
oligohydram-
the amnion precipitate.
in
focal
role
in
factors the
covering
it over
bottom
of
may
oligohydramnios
a short
photograph.
have
played
a causal
in
case:
our
the
absence of urine due to bilateral renal agenesis and the presence of a large blood clot adhering firmly to approximately three fourths of the maternal aspect of the placenta. Jeffcoate and Scott,3 when reviewing all the factors involved in secretion and absorption of amniotic fluid, considered blood
Volume 84 Number 5
clots over large areas of maternal aspect of the placenta as one of the causesof oligohydramnios. They referred to their 3 cases of oligohydramnios in which such was the finding, stating: “These lesions were such that, in these particular cases, the cause of oligohydramnios was a failure of the amnion to contribute its quota to the total volume of liquid.” It is known that bilateral agenesis of kidneys is associated with hypoplastic lungs.? It is of interest that our patient had multiple congenital bronchial cysts in the lungs. In addition, one umbilical artery was absent. Recently, Bourne and Benirschke* reviewed 113 cases of absent umbilical artery and the
REFEKENCES
1. 2.
3. 4. 5.
Landing, B. H.: AM. J. OBST. & GYNEC. 60: 1339, 1950. Potter, E. L.: Pathology of the Fetus and the Newborn, Chicago, 1952, Year Book Publishers, Inc. Jeffcoate, T. N. A., and Scott, J. S.: Canad. M. A. J. 80: 77, 1959. Blanc, W. A.: Am. J. Path. 35: 695, 1959. Scott, J. S., and Bain, A. D.: Proc. Roy. Sot. Med. 51: 512, 1958.
Amnion
nodosum
association of this anomaly with ditions, including oligohydramnios.
other
585
con-
Summary A case of amnion nodosum associated with bilateral renal agenesis, multiple bronchial cysts of the lungs, and an absent umbilical artery is reported and other cases from the literature are cited. While there is nothing in the chart referring to the amount of liquor amnii, it is assumed from the radiologic data that there was associated oligohydramnios. We wish to thank Dr. E. M. Robertson, Professor and Head of the Department of Obstetrics and Gynaecology, for his kind permission to use the obstetrical data concerning this case.
6. 7.
8.
Thompson, V. M.: J. Obst. & Gynaec. Brit. Emp. 67: 611, 1960. MacGregor, A. R.: Pathology of Infancy and Childhood, Edinburgh and London, 1960, E. & S. Livingstone, Ltd. Bourne, G. L., and Benirschke, K.: Arch. Dis. Childhood 35: 834, 1960.
Kingston Kingston,
General Hosjital Ontario, Canada