Abstract
A 36-year-old female (gravida 1, para 0) had persistent anemia since early pregnancy in an otherwise normal course of pregnancy. She visited our department with a complaint of abdominal pain at 37 weeks 2 days of gestation. Her pain was initially accompanied by uterine contractions but later appeared independently. Fever and fetal tachycardia with decelerations were also observed. Abdominal ultrasound revealed fluid accumulation in the abdominal cavity, and an emergency cesarean section was performed with suspicion of placental abruption and imminent uterine rupture. A small amount of intestinal fluid was found in the abdominal cavity, leading to a diagnosis of peritonitis due to small intestinal perforation. A female infant weighing 2,560 g was delivered (Apgar score: 9; umbilical artery pH: 7.278). The patient underwent resection of the perforated portion of the small intestine and ileostomy. Postoperative pathological examination revealed Crohn’s disease. The patient was discharged after surgery and is undergoing remission induction therapy and nutritional therapy for Crohn’s disease.
Abbreviations and acronyms
CD: Crohn’s disease
CT: computed tomography
HELLP syndrome: hemolysis, elevated liver enzymes, and low platelet count syndrome
IBD: inflammatory bowel disease
IDA: iron deficiency anemia
MRI: magnetic resonance imaging
US: ultrasound
5-ASA: 5-aminosalicylic acid
Introduction
Crohn’s disease (CD) is a chronic inflammatory bowel disease that affects the entire gastrointestinal tract from the oral cavity to the anus. It is characterized by discontinuous and generalized inflammation, and is thus considered an inflammatory bowel disease (IBD) along with ulcerative colitis. The number of patients with CD has been on the rise in Japan. With a high incidence among women in their 20s and 30s who are experiencing pregnancy and childbirth, the risk of pregnancy complicated by CD is also increasing. Although perforation of the gastrointestinal tract is a relatively rare complication of CD, its occurrence during pregnancy is often difficult to differentiate from other obstetric emergencies such as imminent uterine rupture, placental abruption, and HELLP (hemolysis, elevated liver enzymes, and low platelet count) syndrome. We report a case of a patient with small bowel perforation due to CD who, after presenting to our hospital with a chief complaint of lower abdominal pain during pregnancy, underwent an emergency cesarean section with suspicion of uterine rupture and placental abruption.
Case report
The patient was a 36-year-old woman (gravida 1, para 1) with no medical history of CD. Persistent low Hb (8.3 g/dl; microcytic anemia) was present from the first trimester; however, there were no additional complications. At 37 weeks 2 days of gestation, she visited our obstetrics outpatient clinic with a chief complaint of lower abdominal pain. On arrival at the hospital, her temperature was 36.2°C, blood pressure was 108/60 mmHg, and heart rate was 82 beats/min. She had pain in the entire lower abdomen, consistent with uterine contractions, with a Bishop score of 3 on internal examination. The fetal heart rate chart showed a baseline of 150 bpm, transient tachycardia, and no bradycardia by cardiotocography, with a diagnosis of reassuring fetal status. Uterine contractions with pain were observed at 4–10 minute intervals (Figure 1A). Blood examination data included white blood cell count 5,030/mm3, Hb level 9.7 mg/dl, and C-reactive protein level 2.4 mg/dl. The patient was diagnosed with labor onset and admitted for delivery management. During hospitalization, lower abdominal pain gradually developed independently of uterine contractions; this pain intensified, with an increase in body temperature to 38.4°C. Cardiotocography revealed a baseline fetal heart rate of 170 bpm, tachycardia, mildly variable decelerations, and frequent uterine contractions (Figure 1B). Abdominal ultrasound (US) showed fluid accumulation in the abdominal cavity with the absence of abnormal findings related to the fetus and placenta. An emergency cesarean section was performed with suspicion of placental abruption and imminent uterine rupture. While there were no abnormal findings of the uterus and placenta, the presence of a small amount of intestinal fluid and food residue in the abdominal cavity suggested peritonitis due to small intestinal perforation. There were multiple longitudinal ulcers and stenosis in the ileum around the ileocecal area, which could be attributed to CD. Ileal resection, involving 40 cm of the small intestine where perforation and multiple ulcers were present (Figure 2A, B), and functional end-to-end anastomosis were performed, followed by ileostomy. Total blood loss was 1,250 ml (including amniotic fluid), and operation time was 319 minutes. Histopathological examination revealed inflammation in all layers of the gastrointestinal tract, comprising lymphocytic aggregates and scattered non-caseating granulomas in the serous membrane. Postoperatively, the patient had persistent fever, a high inflammatory response, and a liver abscess (Figure 2C), which resolved with antibiotics. After nutritional therapy with total parenteral nutrition, the patient’s condition stabilized, and dietary therapy and oral treatment with 5-aminosalicylic acid (5-ASA) were initiated. The patient was discharged on postoperative day 120 (Figure 2D) and is undergoing treatment with 5-ASA and component nutrition therapy at the gastroenterology department of our hospital.
Discussion
Immune abnormalities are considered the cause of CD, but the underlying mechanism is still unknown. Some studies have reported that pregnancy may be associated with an increased risk of CD deterioration. Indeed, deterioration was noted in 50–60% of patients in the active phase of CD, while only 20–25% of patients in the remission phase experienced CD deterioration.1) It is generally accepted that the level of CD activity during pregnancy is related to the level of CD activity at the time of conception. In terms of the effects of CD on pregnancy, associations with an increased frequency of preterm birth, low-birth-weight babies, and cesarean section have been reported.2,3,4) In particular, planned cesarean section is recommended for patients with active CD who have anorectal lesions such as hemorrhoids, rectovaginal fistulas, fissures, abscesses, and anal stenosis, because these symptoms are aggravated by damage to the anal sphincter during vaginal delivery.5) If possible, pregnancy should be avoided during the active stage of CD, and until a remission period of at least 6 months has elapsed.6)
When CD is not diagnosed before pregnancy and symptoms such as abdominal pain appear as in the present case, differential diagnosis becomes important. In the present case, we suspected placental abruption or imminent uterine rupture and performed an emergency cesarean section due to the presence of uterine contractions. We did not anticipate gastrointestinal disease, much less the presence of CD. This experience reaffirms the importance of differentiation between non-pregnancy-related and pregnancy-related conditions as the cause of abdominal pain.7) To differentiate from pregnancy-related diseases, it is important to consider the stage of pregnancy, the presence or absence of uterine contractions, and findings from blood analysis, ultrasonography, and cardiotocography. In addition, medical history and physical examination findings should be taken into consideration. Non-pregnancy-related diseases are often gastrointestinal in nature and include uterine myoma, ovarian cysts, gastrointestinal perforation, mesenteric ischemia, acute appendicitis, diverticulitis, gastroenteritis, cholelithiasis, cholecystitis, gastroduodenal ulcer, acute pancreatitis, ileus, urolithiasis, and cystitis.7)
In the present case, the patient had a medical history of microcytic anemia that had persisted since early pregnancy. The mean corpuscular volume can be used to diagnose microcytic anemia. In addition, iron levels, total iron binding capacity, and ferritin levels should be checked to determine whether patients have iron deficiency anemia (IDA), secondary iron deficiency anemia, or thalassemia. IDA can often be detected during pregnancy due to increased iron demand. Our patient continued to take iron supplements throughout the course of her pregnancy, but no improvement in anemia was observed. In such cases, it should not be simply assumed that IDA is a result of pregnancy; rather, other causes, such as gastrointestinal diseases, should be suspected. Gastrointestinal causes of IDA include gastrointestinal bleeding or absorption disorders other than pregnancy, and gastrointestinal endoscopy is useful in these cases. If anemia persists and gastrointestinal bleeding is suspected, clinicians should not hesitate to perform endoscopy, even during pregnancy.
Abdominal radiography is simple and minimally invasive, but when used to search for the cause of acute abdomen, it has a lower diagnostic sensitivity of 30.0% compared to 96.8% for plain computed tomography (CT). Abdominal radiography also has a lower specificity and positive diagnosis rate compared to CT, making it less useful in the diagnosis of urgent acute abdomen.8) US is simple and minimally invasive, and does not expose patients to radiation. However, its diagnostic ability is highly dependent on the skills of the operator, making objective evaluation difficult. In the case of acute abdomen caused by gastrointestinal diseases, the sensitivity of US is lower than that of plain CT (70% vs. 89%, respectively). However, since there is little difference in sensitivity for acute appendicitis,9) US is the first choice especially during pregnancy. Although CT requires radiation exposure, results are not influenced by operator skills, images can be evaluated objectively, and the diagnostic ability for acute abdomen is very high. In particular, contrast-enhanced CT has a sensitivity higher than 90% for gastrointestinal perforation and mesenteric ischemia, and it is relatively easy to identify the site of damage.10) Therefore, if a definitive diagnosis cannot be reached using US, CT is recommended as a second choice. On the other hand, magnetic resonance imaging (MRI) is superior to CT in diagnosing biliary and gynecological diseases such as uterine myoma and ovarian tumors, and although time-consuming, it is useful for investigating acute abdomen during pregnancy with a low risk of radiation exposure. Depending on the clinical findings and urgency, MRI should be considered the investigative third option, after US and CT.
In the present case, our patient visited the hospital with a chief complaint of lower abdominal pain and underwent an emergency cesarean section, as impending uterine rupture and placental abruption were suspected. Intraoperatively, a diagnosis of small intestinal perforation was made and surgical assistance was requested. Prompt surgical attention enabled us to save the lives of both the mother and child. This case highlights the need to consider not only obstetric diseases but also gastrointestinal diseases, and to establish an environment where comprehensive care can be provided, including gynecology, surgery, and neonatology, when pregnant women present with acute abdomen. Transporting patients to a high-order medical institution may also need to be considered depending on the situation.
With the increase in the prevalence of CD, we expect to encounter more pregnancies with such complications; however, as in the present case, the disease is often undiagnosed. It is thus important to consider the possibility of diseases other than obstetric diseases, even during pregnancy.
Acknowledgments
We thank the pregnant woman for taking part in this study.
Disclosure
The authors declare no conflicts of interest in connection with this article.
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