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Intussusception During Pregnancy: A Case Report and Review of the Literature
""'"
CASE
REPORT"""
,
INTUSSUSCEPTION DURING PREGNANCY: A CASE REPORT AND REVIEW OF THE LITERATURE P. Mahungu, MD, 1 F. Galerneau, MD, FRCSC,2 D. Pugash, MD, FRCPC,3 K. Williams, MB BS, FRCSC, 4 1Perinatal
Fellow, 2Clinical Associate Professor, 3Radiologist, 4Associate Professor, 1-4Department of Obstetrics and Gynaecology, B.C. Women's Hospital, University of British Columbia ABSTRACT
Intussusception is an extremely rare occurrence during pregnancy. The nan-specific presenting symptoms may be attributed to the pregnancy itself, making the diagnosis difficult. We report a case in which the use of ultrasound led to the prompt suspicion of the diagnosis.
RESUME
L'intussusception est un phen.amene rarissime pendant Ia grossesse. Les sympt&nes revelateurs sont nan-specifiques et peuvent etre attribuables
alagrossesse elle-meme, ce qui complique le diagnostic. Nous decrivons un cas ou l'echographie a vite permis de presumer ce diagnostic.
J SOGe 1996;18:77-80 KEY WORDS
Intussusception, pregnancy, ultrasound, intestinal obstruction, laparotomy. Received on March 16th, 1995. Revised and accepted on July 31st, 1995.
JOURNAL SOGe
77
JANUARY 1996
, , , A repeat white count was elevated at seventeen thousand seven hundred. Electrolyte levels were normal. The possibility of torsion of an ovary, or partial or complete obstruction of the bowel was raised. Abdominal and obstetrical ultrasounds were requested and showed: • A single live fetus in frank breech presentation with normal growth and anatomy. The placenta was fundal and the amniotic fluid volume was normal. • The gallbladder and biliary ducts were normal. The visualized portions of the liver, spleen, and pancreas were normal. The proximal aorta was not dilated. There was mild fullness of the right renal collecting system, consistent with physiologic hydronephrosis of pregnancy. The left kidney was normal. • In the left upper quadrant, a moderate amount of free fluid was present around the bowel loops. There was an echogenic mass measuring approximately ten centimetres in diameter which was surrounded by a crescent-shaped fluid collection. The appearance of this did not change during the scan. • (Figures 1,2, and 3). The differential diagnosis now included intussusception. The possibility of an adnexal mass that had undergone torsion was also considered, although this was thought to be unlikely in view of the high location of the mass. The patient was transferred to Grace Hospital (British Columbia's tertiary maternity care centre) some forty-eight hours after the onset of her symptoms. She appeared acutely ill. There was acute tenderness on palpation of the left upper quadrant, with an obvious mass palpable in that area.
INTRODUCTION
Intussusception, and intestinal obstruction in general, are uncommon complications during pregnancy. Thirteen cases were reviewed in the literature from 1914 to 1975 by Bourque and Gibbons, and they added two cases from their centre. l Since then, three more cases have been reported by Holbert,2 Seidman,3 and Jansen. 4 We are presenting the nineteenth case to be published as an addition to the small series of intussusception in pregnancy. This case serves as a reminder that all nausea and vomiting in pregnancy is not hyperemesis gravidarum and that the index of suspicion for less common but more severe gastro-intestinal (GI) problems must always be high. 2 CASE REPORT
Mrs. C.M., a 33-year-old, G3, A2, at twenty-nine weeks gestation, presented with a clinical picture of bowel obstruction. She had been admitted to her local hospital the day before transfer, with a two-hour history of acute abdominal pain and vomiting. The pain was colicky in nature but constant. There was no haematemesis. She had had her last bowel movement shortly following the onset of pain. Her past medical history was negative. Her pregnancy had been progressing normally. Her vital signs were stable, but she was in distress. She was restless and unable to find any position to relieve the epigastric pain that she was experiencing. Examination of the abdomen revealed pain on palpation of the epigastric region. There was no costovertebral angle tenderness and no rebound. Peristalsis was decreased. The uterus was soft and not tender. No contractions were palpated. The fundal height was thirty-two centimetres. The fetal heart rate was reactive. A vaginal examination showed a long, closed, posterior, and thick cervix. A complete blood count, plasma electrolytes, and amylase levels were normal. At this point, gastritis or oesophagitis, or oesophageal spasm were suspected. The patient was given an antacid and analgesics. The pain settled for twenty-four hours, but returned as severe cramps. Her abdomen was now distended and no bowel sounds were present. A mass was palpable in the left upper quadrant. The mass was tender and was situated just above and slightly to the left of the dome of the uterus. The uterus remained soft and not tender. The fetal heart rate was unchanged.
JOURNAL SOGC
Coronal views of the left upper quadrant show a central echogenic mass surrounded by a crescent-shaped or concentric hypoechoic area.
78
JANUARY 1996
, , , of the surgical specimen showed the apex of the intussusception to contain a small intramural lesion, which appeared to be either small bowel lipoma, leiomyoma or possibly even a carcinoid. Pathological examination confirmed the presence of a lipoma in the mesentery, which was most likely the aetiology for the intussusception. In the recovery room, the patient was given indocid one hundred milligrams in an intra-rectal suppository for prevention of premature labour. Her postoperative course was uncomplicated. She was discharged home on day ten. Her pregnancy progressed uneventfully. The fetus stayed in a breech presentation until term. A trial of external version at thirty-seven weeks was unsuccessful. The delivery was by Caesarean section at thirty-nine weeks after spontaneous rupture of the membranes and lack of spontaneous labour. The baby girl weighed 3,840 grams and had Apgar scores of nine at the first minute of life, and nine at five minutes.
A sagittal demonstrates the fixed echogenic mass surrounded by a fluid collection. This is contained by a clearly defined wall. representing the intussuscipiens.
DISCUSSION
No peristalsis was heard. Symphysis fundal height was appropriate for gestational age. Vaginal examination showed the cervix to be long, closed, and with the presenting part high. An abdominal x-ray showed non-specific findings, describing a single fetus in a longitudinal lie, paucity of gas within the bowel, and minimal distention of small bowel loops. These features were more in keeping with a paralytic ileus than a mechanical intestinal obstruction. A consultation was obtained with a general surgeon. In view of the clinical picture of acute abdomen, the patient was taken to the operating room for an exploratory laparotomy. The abdomen was opened through an upper midline incision. Greenish, turbid fluid was noted when the peritoneal cavity was entered. Swabs were obtained for culture and sensitivity. The peritoneal fluid was then suctioned. Further examination of the abdomen demonstrated markedly distended loops of small bowel which were blackish green in colour and appeared to be undergoing early gangrene. The initial thought was of a volvulus but the attempt to reduce the small bowel after aspiration was unsuccessful. The distended bowel loop was then opened after isolating it from the peritoneal cavity. Upon opening the bowel, another loop of bowel was noted within the first one and, thus, the diagnosis of intussusception was obvious. The ischaemic portion of small bowel was resected with primary end-to-end anastomosis. The type of intussusception was jejuno-jejunal. Examination
JOURNAL SOGC
Intestinal obstruction is an uncommon complication of pregnancy and is rarely due to intussusception. According to some authors, intussusception is said to account for between four and six percent of obstruction in pregnant womenY It is a rare condition in adults, whereas in children, it accounts for approximately 80 to 90 percent of obstruction.
A transverse view shows marked thickening of the small bowel wall and valvulae. This leads up to and is included within the echogenic mass. representing the intussusceptum.
79
JANUARY 1996
, , , Intussusception during pregnancy is recognized as one of the most dangerous forms of bowel obstruction. 7 Maternal mortality of up to 75 percent and infant survival of only 28 percent have been reported, when current diagnostic and therapeutic facilities were not available. 8 More recently, considerable improvement in feto-maternal surveillance has resulted in a higher rate of both maternal and fetal survival. A high index of clinical suspicion needs to be maintained. The diagnosis of intestinal obstruction during pregnancy may be delayed because most of the clinical manifestations may be attributed to the pregnancy. According to most authors, a demonstrable cause is usually found in adults,9,lo with ileocaecal intussusception being the most common type and Meckel's diverticulum, the most common precipitating factor. ll ,12 In children, over 90 percent of the cases are idiopathic. The causes of intussusception in pregnant women are usually Meckel's diverticulum, or as in the present case, an initiating neoplasm.iJ,1O While symptoms can clearly lead to the diagnosis in children, the clinical picture associated with intussusception in pregnant women is usually atypical and a correct pre-operative diagnosis is rarely made. The major cause of morbidity reported in these patients, is delay from two days to fifteen weeks in making the correct diagnosis. The chief complaint in adults is of abdominal pain which is usually colicky in nature. 1O Vomiting is usually mild, abdominal tenderness is present in almost all patients, and abnormal bowel sounds are present in only one-third of patients. Routine laboratory studies are of little value. The role of plain abdominal radiography is limited. Occasionally on abdominal x-rays, gaseous distension of the bowel may be seen with bowel displacement related to the gravid uterus. These findings are non-specific and have not in previous reports aided in the diagnosis of the condition.14 Considering the difficulty in clinical diagnosis, and the relative contra-indication of antenatal barium studies ultrasound or MRI appears to offer the best hope for early diagnosis. The MRI may have as yet an undefined role in the investigation, and has been successful in making the diagnosis in one case. 3 Whether it adds anything to the diagnosis after ultrasound examination has not been evaluated. Certainly at present, it is not accessible in the majority of centres and, subsequently, ultrasound should be the primary diagnostic
JOURNAL SOGC
method. Early use of non-invasive diagnostic studies such as ultrasound and MRI, both considered harmless to the fetus, may prevent the erroneous attribution of the clinical manifestation to the pregnancy itself and may provide important information. Previous reports of intussusception diagnosed by ultrasound, describe a "single or double concentric ring of sonolucency surrounded by a coarse central echogenic focus," as in our patient. 1S .16 This provides an early index of suspicion to the correct diagnosis, and with these findings, early surgical exploration of the abdomen is indicated. REFERENCES 1. 2. 3.
4. 5. 6. 7. 8. 9. 10. 11.
12.
13.
14.
15. 16.
80
Bourque MR, Gibbons JM. Intussusception causing intestinal obstruction in pregnancy. Conn Med J 1979;43:130-3. Holbert T. Intussusception in pregnancy. A case report. J Tenn Med Assoc 1987;80:409-10. Seidman DS, Heyman Z, Ben-Ari GY, Maschiach S, Barkai G. Use of magnetic resonance imaging in pregnancy to diagnose intussusception induced by colonic cancer. Obstet GynecoI1992;79(5 Pt 2):822-3. Jansen GR. Two simultaneous emergencies in one patient [letter]. J Tropical Doctor 1982;12(4 Pt 1):189. Goldthorp WOo Intestinal obstruction during pregnancy and the puerperium. Br J Clin Pract 1966;20:367-76. Beck WW. Intestinal obstruction in pregnancy. Obstet Gynecol 1974;43:374-8. Svesko VS, Pisani BJ. Intestinal obstruction in pregnancy. Am J Obstet Gynecol 1960;79: 157 -61. Chaffin L, Mason VR, Slemons JM. Intussusception during pregnancy. Surg Gynecol Obstet 1937;64:811-19. Donhauser JL, Kelly EC. Intussusception in the adult. Am J Surg 1950;79:673-7. Weilbaecher D, Bolin JA, Hearn D, Ogden W. Intussusception in adults. Am J Surg 1971;121:531-5. Gay BB, Leigh TF, Rogers JV. Meckel's diverticulum as a cause of intussusception. J Med Assoc Georgia 1953;42: 15-18. Gibbins RE, Symmers WS. Chronic intussusception due to inversion of a Meckel's diverticulum containing a chronic peptic ulcer, heterotopic tissues, and a lipoma. Br J Surg 1953;40:500-5. Sanders GB, Hogan WH, Kinnard DW. Adult intussusception and carcinoma of the colon. Ann Sur 1958;147:796-803. Foulds DM, Beamish WE. The plain film diagnosis of acute ileo-ileal intussusception. J Can Assoc Radio 1970;21 :178-80. Uhland H, Parshley PF. Obscure intussusception diagnosed by ultrasound. JAMA 1978;239:224. Morin ME, Blumenthal DH, Tan A, Li YP. The ultrasonic appearance of ileocolic intussusception. J Clin Ultrasound 1981;9:516-18.
JANUARY 1996
CASE
REPORT"""
,
INTUSSUSCEPTION DURING PREGNANCY: A CASE REPORT AND REVIEW OF THE LITERATURE P. Mahungu, MD, 1 F. Galerneau, MD, FRCSC,2 D. Pugash, MD, FRCPC,3 K. Williams, MB BS, FRCSC, 4 1Perinatal
Fellow, 2Clinical Associate Professor, 3Radiologist, 4Associate Professor, 1-4Department of Obstetrics and Gynaecology, B.C. Women's Hospital, University of British Columbia ABSTRACT
Intussusception is an extremely rare occurrence during pregnancy. The nan-specific presenting symptoms may be attributed to the pregnancy itself, making the diagnosis difficult. We report a case in which the use of ultrasound led to the prompt suspicion of the diagnosis.
RESUME
L'intussusception est un phen.amene rarissime pendant Ia grossesse. Les sympt&nes revelateurs sont nan-specifiques et peuvent etre attribuables
alagrossesse elle-meme, ce qui complique le diagnostic. Nous decrivons un cas ou l'echographie a vite permis de presumer ce diagnostic.
J SOGe 1996;18:77-80 KEY WORDS
Intussusception, pregnancy, ultrasound, intestinal obstruction, laparotomy. Received on March 16th, 1995. Revised and accepted on July 31st, 1995.
JOURNAL SOGe
77
JANUARY 1996
, , , A repeat white count was elevated at seventeen thousand seven hundred. Electrolyte levels were normal. The possibility of torsion of an ovary, or partial or complete obstruction of the bowel was raised. Abdominal and obstetrical ultrasounds were requested and showed: • A single live fetus in frank breech presentation with normal growth and anatomy. The placenta was fundal and the amniotic fluid volume was normal. • The gallbladder and biliary ducts were normal. The visualized portions of the liver, spleen, and pancreas were normal. The proximal aorta was not dilated. There was mild fullness of the right renal collecting system, consistent with physiologic hydronephrosis of pregnancy. The left kidney was normal. • In the left upper quadrant, a moderate amount of free fluid was present around the bowel loops. There was an echogenic mass measuring approximately ten centimetres in diameter which was surrounded by a crescent-shaped fluid collection. The appearance of this did not change during the scan. • (Figures 1,2, and 3). The differential diagnosis now included intussusception. The possibility of an adnexal mass that had undergone torsion was also considered, although this was thought to be unlikely in view of the high location of the mass. The patient was transferred to Grace Hospital (British Columbia's tertiary maternity care centre) some forty-eight hours after the onset of her symptoms. She appeared acutely ill. There was acute tenderness on palpation of the left upper quadrant, with an obvious mass palpable in that area.
INTRODUCTION
Intussusception, and intestinal obstruction in general, are uncommon complications during pregnancy. Thirteen cases were reviewed in the literature from 1914 to 1975 by Bourque and Gibbons, and they added two cases from their centre. l Since then, three more cases have been reported by Holbert,2 Seidman,3 and Jansen. 4 We are presenting the nineteenth case to be published as an addition to the small series of intussusception in pregnancy. This case serves as a reminder that all nausea and vomiting in pregnancy is not hyperemesis gravidarum and that the index of suspicion for less common but more severe gastro-intestinal (GI) problems must always be high. 2 CASE REPORT
Mrs. C.M., a 33-year-old, G3, A2, at twenty-nine weeks gestation, presented with a clinical picture of bowel obstruction. She had been admitted to her local hospital the day before transfer, with a two-hour history of acute abdominal pain and vomiting. The pain was colicky in nature but constant. There was no haematemesis. She had had her last bowel movement shortly following the onset of pain. Her past medical history was negative. Her pregnancy had been progressing normally. Her vital signs were stable, but she was in distress. She was restless and unable to find any position to relieve the epigastric pain that she was experiencing. Examination of the abdomen revealed pain on palpation of the epigastric region. There was no costovertebral angle tenderness and no rebound. Peristalsis was decreased. The uterus was soft and not tender. No contractions were palpated. The fundal height was thirty-two centimetres. The fetal heart rate was reactive. A vaginal examination showed a long, closed, posterior, and thick cervix. A complete blood count, plasma electrolytes, and amylase levels were normal. At this point, gastritis or oesophagitis, or oesophageal spasm were suspected. The patient was given an antacid and analgesics. The pain settled for twenty-four hours, but returned as severe cramps. Her abdomen was now distended and no bowel sounds were present. A mass was palpable in the left upper quadrant. The mass was tender and was situated just above and slightly to the left of the dome of the uterus. The uterus remained soft and not tender. The fetal heart rate was unchanged.
JOURNAL SOGC
Coronal views of the left upper quadrant show a central echogenic mass surrounded by a crescent-shaped or concentric hypoechoic area.
78
JANUARY 1996
, , , of the surgical specimen showed the apex of the intussusception to contain a small intramural lesion, which appeared to be either small bowel lipoma, leiomyoma or possibly even a carcinoid. Pathological examination confirmed the presence of a lipoma in the mesentery, which was most likely the aetiology for the intussusception. In the recovery room, the patient was given indocid one hundred milligrams in an intra-rectal suppository for prevention of premature labour. Her postoperative course was uncomplicated. She was discharged home on day ten. Her pregnancy progressed uneventfully. The fetus stayed in a breech presentation until term. A trial of external version at thirty-seven weeks was unsuccessful. The delivery was by Caesarean section at thirty-nine weeks after spontaneous rupture of the membranes and lack of spontaneous labour. The baby girl weighed 3,840 grams and had Apgar scores of nine at the first minute of life, and nine at five minutes.
A sagittal demonstrates the fixed echogenic mass surrounded by a fluid collection. This is contained by a clearly defined wall. representing the intussuscipiens.
DISCUSSION
No peristalsis was heard. Symphysis fundal height was appropriate for gestational age. Vaginal examination showed the cervix to be long, closed, and with the presenting part high. An abdominal x-ray showed non-specific findings, describing a single fetus in a longitudinal lie, paucity of gas within the bowel, and minimal distention of small bowel loops. These features were more in keeping with a paralytic ileus than a mechanical intestinal obstruction. A consultation was obtained with a general surgeon. In view of the clinical picture of acute abdomen, the patient was taken to the operating room for an exploratory laparotomy. The abdomen was opened through an upper midline incision. Greenish, turbid fluid was noted when the peritoneal cavity was entered. Swabs were obtained for culture and sensitivity. The peritoneal fluid was then suctioned. Further examination of the abdomen demonstrated markedly distended loops of small bowel which were blackish green in colour and appeared to be undergoing early gangrene. The initial thought was of a volvulus but the attempt to reduce the small bowel after aspiration was unsuccessful. The distended bowel loop was then opened after isolating it from the peritoneal cavity. Upon opening the bowel, another loop of bowel was noted within the first one and, thus, the diagnosis of intussusception was obvious. The ischaemic portion of small bowel was resected with primary end-to-end anastomosis. The type of intussusception was jejuno-jejunal. Examination
JOURNAL SOGC
Intestinal obstruction is an uncommon complication of pregnancy and is rarely due to intussusception. According to some authors, intussusception is said to account for between four and six percent of obstruction in pregnant womenY It is a rare condition in adults, whereas in children, it accounts for approximately 80 to 90 percent of obstruction.
A transverse view shows marked thickening of the small bowel wall and valvulae. This leads up to and is included within the echogenic mass. representing the intussusceptum.
79
JANUARY 1996
, , , Intussusception during pregnancy is recognized as one of the most dangerous forms of bowel obstruction. 7 Maternal mortality of up to 75 percent and infant survival of only 28 percent have been reported, when current diagnostic and therapeutic facilities were not available. 8 More recently, considerable improvement in feto-maternal surveillance has resulted in a higher rate of both maternal and fetal survival. A high index of clinical suspicion needs to be maintained. The diagnosis of intestinal obstruction during pregnancy may be delayed because most of the clinical manifestations may be attributed to the pregnancy. According to most authors, a demonstrable cause is usually found in adults,9,lo with ileocaecal intussusception being the most common type and Meckel's diverticulum, the most common precipitating factor. ll ,12 In children, over 90 percent of the cases are idiopathic. The causes of intussusception in pregnant women are usually Meckel's diverticulum, or as in the present case, an initiating neoplasm.iJ,1O While symptoms can clearly lead to the diagnosis in children, the clinical picture associated with intussusception in pregnant women is usually atypical and a correct pre-operative diagnosis is rarely made. The major cause of morbidity reported in these patients, is delay from two days to fifteen weeks in making the correct diagnosis. The chief complaint in adults is of abdominal pain which is usually colicky in nature. 1O Vomiting is usually mild, abdominal tenderness is present in almost all patients, and abnormal bowel sounds are present in only one-third of patients. Routine laboratory studies are of little value. The role of plain abdominal radiography is limited. Occasionally on abdominal x-rays, gaseous distension of the bowel may be seen with bowel displacement related to the gravid uterus. These findings are non-specific and have not in previous reports aided in the diagnosis of the condition.14 Considering the difficulty in clinical diagnosis, and the relative contra-indication of antenatal barium studies ultrasound or MRI appears to offer the best hope for early diagnosis. The MRI may have as yet an undefined role in the investigation, and has been successful in making the diagnosis in one case. 3 Whether it adds anything to the diagnosis after ultrasound examination has not been evaluated. Certainly at present, it is not accessible in the majority of centres and, subsequently, ultrasound should be the primary diagnostic
JOURNAL SOGC
method. Early use of non-invasive diagnostic studies such as ultrasound and MRI, both considered harmless to the fetus, may prevent the erroneous attribution of the clinical manifestation to the pregnancy itself and may provide important information. Previous reports of intussusception diagnosed by ultrasound, describe a "single or double concentric ring of sonolucency surrounded by a coarse central echogenic focus," as in our patient. 1S .16 This provides an early index of suspicion to the correct diagnosis, and with these findings, early surgical exploration of the abdomen is indicated. REFERENCES 1. 2. 3.
4. 5. 6. 7. 8. 9. 10. 11.
12.
13.
14.
15. 16.
80
Bourque MR, Gibbons JM. Intussusception causing intestinal obstruction in pregnancy. Conn Med J 1979;43:130-3. Holbert T. Intussusception in pregnancy. A case report. J Tenn Med Assoc 1987;80:409-10. Seidman DS, Heyman Z, Ben-Ari GY, Maschiach S, Barkai G. Use of magnetic resonance imaging in pregnancy to diagnose intussusception induced by colonic cancer. Obstet GynecoI1992;79(5 Pt 2):822-3. Jansen GR. Two simultaneous emergencies in one patient [letter]. J Tropical Doctor 1982;12(4 Pt 1):189. Goldthorp WOo Intestinal obstruction during pregnancy and the puerperium. Br J Clin Pract 1966;20:367-76. Beck WW. Intestinal obstruction in pregnancy. Obstet Gynecol 1974;43:374-8. Svesko VS, Pisani BJ. Intestinal obstruction in pregnancy. Am J Obstet Gynecol 1960;79: 157 -61. Chaffin L, Mason VR, Slemons JM. Intussusception during pregnancy. Surg Gynecol Obstet 1937;64:811-19. Donhauser JL, Kelly EC. Intussusception in the adult. Am J Surg 1950;79:673-7. Weilbaecher D, Bolin JA, Hearn D, Ogden W. Intussusception in adults. Am J Surg 1971;121:531-5. Gay BB, Leigh TF, Rogers JV. Meckel's diverticulum as a cause of intussusception. J Med Assoc Georgia 1953;42: 15-18. Gibbins RE, Symmers WS. Chronic intussusception due to inversion of a Meckel's diverticulum containing a chronic peptic ulcer, heterotopic tissues, and a lipoma. Br J Surg 1953;40:500-5. Sanders GB, Hogan WH, Kinnard DW. Adult intussusception and carcinoma of the colon. Ann Sur 1958;147:796-803. Foulds DM, Beamish WE. The plain film diagnosis of acute ileo-ileal intussusception. J Can Assoc Radio 1970;21 :178-80. Uhland H, Parshley PF. Obscure intussusception diagnosed by ultrasound. JAMA 1978;239:224. Morin ME, Blumenthal DH, Tan A, Li YP. The ultrasonic appearance of ileocolic intussusception. J Clin Ultrasound 1981;9:516-18.
JANUARY 1996