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A case report of a misdiagnosed cesarean scar pregnancy in a hemodynamically compromised patient
Human Pathology: Case Reports 18 (2019) 200344
Contents lists available at ScienceDirect
Human Pathology: Case Reports journal homepage: www.elsevier.com/locate/ehpc
Case Report
A case report of a misdiagnosed cesarean scar pregnancy in a hemodynamically compromised patient
T
A.J. Rivera-Rodrígueza,b, O. Jiménez-Zarazúab,c, L.A. Castaldi-Bermúdeza,b, L.N. Vélez-Ramírezb,d, O. Camacho Coronel-Cioccaa,b, M.A. Romero-Francesa,b, ⁎ A.M. Becerra-Baezab, JD. Mondragóne,f, a
Hospital General León, Department of Gynecology and Obstetrics, Mexico Universidad de Guanajuato, Department of Medicine and Nutrition, Mexico c Hospital General León, Department of Internal Medicine, Mexico d Hospital General León, Department of Radiology, Mexico e University of Groningen, University Medical Center Groningen, Department of Neurology, The Netherlands f University of Groningen, University Medical Center Groningen, Alzheimer Research Center Groningen, The Netherlands b
A R T I C LE I N FO
A B S T R A C T
Keywords: Cesarean scar pregnancy Hemodynamic instability Multiple cesarean deliveries Satisfied reproductive plans Total abdominal hysterectomy
Differential diagnosis between a cesarean scar pregnancy and a cervical pregnancy is difficult. Prompt and accurate diagnosis of an ectopic pregnancy is essential, thus localization of the implanted gestational sac and associated comorbidities are necessary to efficiently provide management. A case of a 30-year-old woman with three previous cesarean deliveries and satisfied reproductive plans developed hemodynamic instability (i.e. onset of anemia, tachycardia, and hypotension within 12 h) secondary to transvaginal blood loss, even after fluid replacement and vaginal tamponade. After considering expectant, medical, and surgical options, a shared decision between the patient and the treating team to perform a total hysterectomy was reached. This case was initially misdiagnosed as a cervical pregnancy but after histopathological evaluation and integration of the past gynecological history, a cesarean scar pregnancy was diagnosed. This case provides an example of the interaction between past medical history, current clinical picture, and future reproductive plans that can help obstetrics and gynecology specialist’s decision-making in regards to hysterotomy scar pregnancies. Although a recent increase in publications regarding cesarean scar pregnancies in the past decade, this case also addressed the need for more publications of cesarean scar pregnancy cases from countries with high cesarean delivery rates.
1. Introduction An ectopic pregnancy (EP) is a pregnancy that occurs outside of the uterine cavity, occurring in approximately 1% to 2% of all pregnancies [1]. Although EPs occur at higher frequencies at the uterine tubes, other less common locations are the cervix, ovaries, within the myometrium (intramural), abdominal cavity, and fibrous tissue from cesarean scars. Cervical pregnancies (CPs) account for less than 2% of EPs and are estimated to occur in 1:2000 to 1:18,000 pregnancies [2], while cesarean (hysterotomy) scar pregnancies (CSPs) account for approximately 1:1800 to 1:2216 [3]. Awareness about CSP has increased in recent years, as only 19 articles reporting CSP cases were published in the 1990s, 48 between 2000 and 2005 and 104 between 2006 and 2011, accounting for 751 CSP cases in the medical literature by 2012
⁎
[4]. A more recent systematic review, reported a total of 2037cases by 2015 [5]. However, this pathology could be underreported in developing countries due to misdiagnoses and lower publication rates. We present the case report in line with the SCARE criteria [6] of a patient with transvaginal bleeding and abdominal pain treated in a university hospital, who after physical, laboratory, and ultrasonographic examination was initially diagnosed with a cervical pregnancy. Total hysterectomy was performed after the patient’s hemodynamic stability was compromised. After a careful histopathological evaluation and clinical integration of the gynecological history (i.e. three previous cesarean deliveries), the final diagnosis was a cesarean scar pregnancy. This case contributes to the existing literature with a successfully treated case involving a complicated CSP, that was initially misdiagnosed and upon further review was correctly classified. A
Corresponding author at: University Medical Center Groningen, Department of Neurology, PO Box 30001, 9700 RB Groningen, The Netherlands. E-mail address: [email protected] (J. Mondragón).
https://doi.org/10.1016/j.ehpc.2019.200344 Received 26 August 2019; Received in revised form 11 October 2019; Accepted 16 October 2019 2214-3300/ © 2019 The Author(s). Published by Elsevier Inc. This is an open access article under the CC BY license (http://creativecommons.org/licenses/BY/4.0/).
Human Pathology: Case Reports 18 (2019) 200344
A.J. Rivera-Rodríguez, et al.
6 × 6 cm right ovarian cyst was performed. No post-surgical complications, signs of infection or low cardiac output syndrome were reported. The patient was released after seven days of hospitalization due to clinical improvement and the conclusion of the antibiotic management for the urinary tract infection; additionally, the patient was reevaluated by the OBGYN Department 60 days after being discharged, without reporting any morbidities associated with the ectopic pregnancy.
discussion on the misdiagnosis of CSPs and the management of CSPs with hemodynamic instability is included. 2. Clinical presentation A 30-year-old Hispanic female arrived at the Emergency Department complaining of scant bright-red transvaginal bleeding of 24 h onset, associated with progressive localized pain in the left pelvic region reaching an intensity of 6/10 in the visual analog scale for pain (VAS) at the time of admission. The patient reported a pregnancy of 8 weeks and 3 days (i.e. gestational age by the last menstrual period), gravida IV, para III, and three cesarean deliveries (i.e. last cesarean delivery 7 years earlier) with a normal evolution. The patient’s family history included a mother with type 2 diabetes and arterial hypertension; other relevant aspects of family history were questioned and denied. Among the patient’s personal history the patient denied the use of controlled substances, allergies, past blood transfusions, traveling to regions with endemic diseases within the last three months, tattoos, and body piercings. Upon initial physical exploration, we found a recumbent patient with a freely chosen body position, Glasgow coma score of 15, without focal neurologic deficits, aware of her environment, with reference to place, time, and people. The patient’s integumentary system was hydrated and without alterations. Upon inspection, palpation, auscultation, and percussion the cardio-respiratory system had no abnormal findings. The abdominal exploration presented pain in the left pelvic region upon light and deep palpation, without signs of peritoneal irritation or any other alterations. Gynecological examination yielded a closed external cervical os, mild pain upon cervical manipulation, limited transvaginal hemorrhage, and the uterus below the pubic symphysis. Upon admission to the Obstetrics and Gynecology (OBGYN) Department, the patient had the following vital signs: blood pressure of 110/70 mmHg; a heart rate of 80 bpm; a respiratory rate of 20 rpm; a body temperature of 36.5 °C; a body-weight of 68 kg; the height of 165 cm.
4. Discussion The case presented above is an example of a CSP complicated by profuse hemorrhage thus leading to acute hemodynamic instability. Although CSP case reports and case series have been previously reviewed in the literature, CSPs are infrequent; furthermore, complicated cases involving hemodynamic compromise secondary to uncorrected blood loss are rare but require prompt and coordinated management. Another added value to the medical literature regarding CSPs that this case provides, is the importance of incorporating the patient’s gynecological history (i.e. history of three previous cesarean deliveries is a major risk factor for CSP) to integrate an accurate diagnosis. This patient was initially misdiagnosed, however upon further discussion and careful assessment of the histopathological findings, a cesarean scar pregnancy was diagnosed. Transvaginal ultrasound seems to be the best diagnostic tool to establish the diagnosis of CSP [4], having a sensitivity of 84.6% [3]; Doppler examination and magnetic resonance imaging could aid diagnosing complicated cases [7]. The current case was diagnosed using conventional clinical and sonographic findings, identifying a 7-weekold product through transvaginal ultrasonography (Fig. 1). However, after histopathological assessment of the surgical piece and integration of the patient’s gynecological history, a CSP was diagnosed. CSPs are difficult to diagnose, often being misdiagnosed as a low uterine pregnancy, cervical pregnancy, or miscarriage in progress [4]. Misdiagnosis of a CSP has been previously reported. Liu and colleagues (2019), report three cases with ambiguous transvaginal ultrasound findings of which, one was misdiagnosed as a cornual pregnancy [8]. One way that has been suggested to easily differentiate between a CSP and an intrauterine pregnancy is the location of the gestational sac relative to the midpoint axis of the uterus; as a gestational sac implanted low in a uterus at 5–10 weeks of gestation highly predicts a CSP [9]. Based upon case series, five treatment modalities are recommended for a CSP: 1) transvaginal resection; 2) laparoscopic resection; 3) uterine artery embolization plus uterine dilatation and curettage combined with hysteroscopy; 4) uterine artery embolization combined with uterine dilatation and curettage without methotrexate; and 5) hysteroscopy alone [5]. Since the patient was initially diagnosed with a cervical pregnancy, surgical curettage and cervical cerclage were dismissed, as surgical cutterage had a high risk for excessive hemorrhage and cervical cerclage did not provide an optimal outcome due to the continuous hemorrhagic condition. Angiographic embolization of the uterine artery (UAE) was considered; however, due to the recurrent vaginal bleeding associated with this procedure and the fact that the patient no longer wished to have offspring, the surgical team decided that UAE was not the optimal management for this case. Finally, total abdominal hysterectomy was performed as it is the treatment of choice in EPs with unstable vital signs, excessive vaginal bleeding, or have completed their families [10]. The patient had β-hCG levels of 10,500 mIU/mL upon admission; however, the patient’s β-hCG levels did not favor surgical treatment over medical management. Dior and colleagues (2018) correlate β-hCG levels and conversion to surgical management from medical treatment, reporting a 40% conversion for levels > 400,000 IU, 35.3% for levels > 20,000 IU, and only 6.3% in patients with levels at presentation < 10,000 IU [11]. Hysterectomy is a rare but necessary treatment option in cases where severe bleeding compromises the
3. Clinical evolution Transvaginal ultrasonography was performed, where the uterus cavity presented no contents and presumably the cervix contained a gestational sac at approximately 7 weeks of gestational age (Fig. 1) with a heart rate of 150 bpm; compatible with a probable cervical ectopic pregnancy. Serum human chorionic gonadotropin (β-hCG) levels upon admission were 10,500 mIU/mL. The only abnormal laboratory findings upon admission were the serum leukocyte count (i.e. 13,300 per microliter), the urinalysis (i.e. abundant bacteria and 15 leukocytes per high power field), and the urine culture test which reported growth of Escherichia coli greater than 100,000 colony forming units (CFU)/mL sensitive to multiple antibiotics, which resolved after treatment (i.e. intravenous ceftriaxone 1 g every 12 h for 7 days). Serum procalcitonin levels of 0.3 ng/mL were reported. A double-balloon cervical ripening catheter was used as a vaginal tamponade strategy. The patient developed sudden hemodynamic compromise (i.e. onset of anemia with a serum hemoglobin decrease from 14.2 g/dL to 10.3 g/dL after 12 h, tachycardia, and blood pressure of 96/66 mmHg) due to the profuse blood loss even after vaginal tamponade. No instrumentation was performed on the cervical canal. Intravenous fluid reposition was performed to hemodynamically stabilize the patient. Total hysterectomy was performed with approximately 300 mL of blood loss during the surgical procedure. The pathological findings were a uterus measuring 9.5 × 6.5 × 3.8 cm with the presence of a non-ruptured gestational sac at 6 weeks of gestation implanted in the endometrium above the cervical canal, expanding into the lower uterine cavity (Fig. 2). Upon further review, deliberation, and integration of the patient’s gynecological history, the diagnosis resulting was cesarean scar pregnancy. Additionally, resection of a 2
Human Pathology: Case Reports 18 (2019) 200344
A.J. Rivera-Rodríguez, et al.
Fig. 1. Transvaginal ultrasonography. Transvaginal ultrasonography. A) Longitudinal view of gestational sac (*) above the cervical canal (**) observed. B) Transversal view of gestational sac (*) above the cervix and expanding into the lower uterine cavity. C) Longitudinal augmented view with gestational sac (*) above the cervical canal (**) observed. D) Transversal augmented view with gestational sac (*) and cervical canal (**) observed.
mortality associated with this state; as well as, the patient’s satisfaction with her reproductive plans.
patient’s hemodynamic stability. Thus, considering that the patient’s serum hemoglobin levels had decreased from 14.2 g/dL upon admission to 10.3 g/dL after 12 hr observation and intravenous fluid reposition, hysterectomy was considered as a viable option. Previously, emergency hysterectomy, for 4 out of 57 cases in a 12-year retrospective study, was a second-line treatment option for excessive bleeding in one case, severe bleeding with disseminated intravascular coagulation during dilatation and curettage in a second case, fetal death in another case, and severe placental adhesion in the last case [12]. In the same study, one patient elected hysterectomy as the first-line treatment option since the patient had satisfied her future reproductive plans and had a previous history of adenomyosis and multiple uterine manipulations [12]. The case presented above describes a woman with her fourth pregnancy who underwent a total hysterectomy as the treatment option to correct for the hemodynamic instability. In this case, the treatment decision was justified based on two factors, acute blood loss and the increased
5. Limitations Although a rare pathology, we have reported only presented one case of a hysterotomy scar pregnancy. Over two thousand CSP cases have been reported to date; nonetheless, more cases from developing countries are needed to address the reporting bias present in all previously published reviews and systematic reviews. Underreporting from developing countries is another problem. Before 2012, most cases were reported from China (i.e. 483 out of 751 cases), the United States (i.e. 44 cases of 751), the United Kingdom (i.e. 107 cases of 751) [4]. Recently, more cases continue to be published out of China, with four randomized controlled trials assessing management strategies for CSPs from 2009 to 2015 [5]. However, there is a growing need for reports Fig. 2. Uterus with hysterotomy scar pregnancy. Surgical piece. Coronal cut of the uterus. A and B) uterus measuring 9.5 × 6.5 × 3.8 cm and weighing 136 g, with the presence of a non-ruptured gestational sac at 6 weeks of gestation implanted in the endometrium at the lower portion of the uterine cavity. Embryonic sac measuring 2.8 × 2.0 cm, with a sac wall measuring 10 to 20 mm. The content of the embryonic sac with presence of embryo measuring 6 mm longitudinally. Ocular orbits, nasal cavities, and palmar plaques present in the embryo.
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Human Pathology: Case Reports 18 (2019) 200344
A.J. Rivera-Rodríguez, et al.
References
coming from developing countries, especially those with high cesarean delivery rates like Brazil (i.e. 45.9%), Iran (i.e. 41.9%), Dominican Republic (41.9%), Italy (i.e. 38.2%), and Mexico (i.e. 37.8%) [13].
[1] J. Chang, L.D. Elam-Evans, C.J. Berg, J. Herndon, L. Flowers, K.A. Seed, C.J. Syverson, Pregnancy related mortally surveillance – United States, 1991–1999, MMWR Surveill. Summ. 52 (2) (2003) 1–8 PMID: 12825542. [2] J. Yankowitz, J. Leake, G. Huggins, P. Gazaway, E. Gates, Cervical ectopic pregnancy: review of the literature and report of a case treated by single-dose methotrexate therapy, Obstet. Gynecol. Surv. 45 (7) (1990) 405–414 PMID: 2195408. [3] M.A. Rotas, S. Haberman, M. Levgur, Cesarean scar ectopic pregnancies: etiology, diagnosis, and management, Obstet. Gynecol. 107 (6) (2006) 1373–1381, https:// doi.org/10.1097/01.AOG.0000218690.24494.ce PMID: 16738166. [4] I.E. Timor-Tritsch, A. Monteagudo, Unforeseen consequences of the increasing rate of cesarean deliveries: early placenta accreta and cesarean scar pregnancy. A review, Am. J. Obstet. Gynecol. 207 (1) (2012) 14–29, https://doi.org/10.1016/j. ajog.2012.03.007 PMID: 22516620. [5] K. Birch Petersen, E. Hoffmann, C. Rifbjerg Larsen, Nielsen H. Svarre, Cesarean scar pregnancy: a systematic review of treatment studies, Fertil. Steril. 105 (4) (2016) 958–967, https://doi.org/10.1016/j.fertnstert.2015.12.130 PMID: 26794422. [6] R.A. Agha, M.R. Borrelli, R. Farwana, K. Koshy, A. Fowler, D.P. Orgill, For the SCARE Group. The SCARE, Statement: updating consensus surgical CAse REport (SCARE) guidelines, Int. J. Surgery 2018 (60) (2018) 132–136, https://doi.org/10. 1016/j.ijsu.2018.10.028 PMID: 30342279. [7] P. Pędraszewski, E. Wlaźlak, W. Panek, G. Surkont, Cesarean scar pregnancy – a new challenge for obstetricians, J. Ultrason. 18 (72) (2018) 56–62, https://doi.org/ 10.15557/JoU.2018.0009 PMID: 29844942. [8] D. Liu, X. Gu, F. Liu, F. Shi, M. Yang, Q. Wu, Application of contrast-enhanced ultrasound for scar pregnancy cases misdiagnosed as other diseases, Clin. Chim. Acta 496 (2019) 134–139, https://doi.org/10.1016/j.cca.2019.06.002 PMID: 31173731. [9] I.E. Timor-Tritsch, A. Monteagudo, G. Cali, H. El Refaey, A. Kaelin Agten, A.A. Arslan, Easy sonographic differential diagnosis between intrauterine pregnancy and cesarean delivery scar pregnancy in the early first trimester, Am. J. Obstet. Gynecol. 215 (2) (2016), https://doi.org/10.1016/j.ajog.2016.02.028 225. e1-7, PMID: 26899908. [10] M.M. Hosni, R.P. Herath, R. Mumtaz, Diagnostic and therapeutic dilemmas of cervical ectopic pregnancy, Obstet. Gynecol. Surv. 69 (5) (2014) 261–276, https:// doi.org/10.1097/OGX.0000000000000062 PMID: 25101692. [11] U.P. Dior, R. Palma-Dias, K.L. Reidy, C. Cheng, M. Healey, Cesarean scar pregnancies: incidence and factors associated with conversion to surgery from medical management, J. Minim. Invasive Gynecol. 26 (5) (2019) 919–927, https://doi.org/ 10.1016/j.jmig.2018.09.771 PMID: 30243684. [12] S.Y. Kim, S.R. Yoon, M.J. Kim, J.H. Chung, M.Y. Kim, S.W. Lee, Cesarean scar pregnancy; diagnosis and management between 2003 and 2015 in a single center, Taiwan J. Obstet. Gynecol. 57 (5) (2018) 688–691, https://doi.org/10.1016/j.tjog. 2018.08.013 PMID: 30342652. [13] L. Gibbons, J.M. Belizan, J.A. Lauer, A.P. Betran, M. Merialdi, F. Althabe, Inequities in the use of cesarean section deliveries in the world, Am. J. Obstet. Gynecol. 206 (4) (2012), https://doi.org/10.1016/j.ajog.2012.02.026 331.e1-19 PMID: 22464076.
6. Conclusion Although CSPs are rare, management must consider the patient’s previous gynecological history, clinical evolution, and future family plans. The case presented here involves a woman with three previous cesarean deliveries, with acute onset of hemodynamic instability secondary to continuous blood loss despite management to attain hemostasis, and reference to have completed her family. This case provides an example of the interaction between the past medical history, current clinical picture, and future reproductive plans that can help OBGYN specialist’s decision-making in regards to CSPs; as well as a case initially misdiagnosed as a cervical pregnancy but upon histopathological inspection, a hysterotomy scar pregnancy was diagnosed. 7. Patient consent statement Upon hospital admission, the patient signed an informed consent permitting the use of her clinical file information for didactic, research, and publication purposes. Signed consent (Statement in Spanish) can be made available upon request. Acknowledgments This study was supported by CONACyT (Consejo Nacional de Ciencia y Tecnología) Grant #440591. Disclosure of interest: this research did not receive any specific grant from funding agencies in the commercial sector, the authors report no conflict of interest. Data availability and deposition: the clinical data supporting the conclusions of this article is included in the article; data availability can be provided on a need basis. Approval from the ethical committee was not required due to the nature of this case report. Abiding by the Declaration of Helsinki, patient anonymity was guaranteed. Upon hospital admission, the patient signed an informed consent permitting the use of her clinical file information for didactic, research, and publication purposes. We would like to commend the work of the medical staff (i.e. specialists, medical residents, and nursing staff) of the Obstetrics and Gynecology Department, as well as the Pathology department at our institution.
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Contents lists available at ScienceDirect
Human Pathology: Case Reports journal homepage: www.elsevier.com/locate/ehpc
Case Report
A case report of a misdiagnosed cesarean scar pregnancy in a hemodynamically compromised patient
T
A.J. Rivera-Rodrígueza,b, O. Jiménez-Zarazúab,c, L.A. Castaldi-Bermúdeza,b, L.N. Vélez-Ramírezb,d, O. Camacho Coronel-Cioccaa,b, M.A. Romero-Francesa,b, ⁎ A.M. Becerra-Baezab, JD. Mondragóne,f, a
Hospital General León, Department of Gynecology and Obstetrics, Mexico Universidad de Guanajuato, Department of Medicine and Nutrition, Mexico c Hospital General León, Department of Internal Medicine, Mexico d Hospital General León, Department of Radiology, Mexico e University of Groningen, University Medical Center Groningen, Department of Neurology, The Netherlands f University of Groningen, University Medical Center Groningen, Alzheimer Research Center Groningen, The Netherlands b
A R T I C LE I N FO
A B S T R A C T
Keywords: Cesarean scar pregnancy Hemodynamic instability Multiple cesarean deliveries Satisfied reproductive plans Total abdominal hysterectomy
Differential diagnosis between a cesarean scar pregnancy and a cervical pregnancy is difficult. Prompt and accurate diagnosis of an ectopic pregnancy is essential, thus localization of the implanted gestational sac and associated comorbidities are necessary to efficiently provide management. A case of a 30-year-old woman with three previous cesarean deliveries and satisfied reproductive plans developed hemodynamic instability (i.e. onset of anemia, tachycardia, and hypotension within 12 h) secondary to transvaginal blood loss, even after fluid replacement and vaginal tamponade. After considering expectant, medical, and surgical options, a shared decision between the patient and the treating team to perform a total hysterectomy was reached. This case was initially misdiagnosed as a cervical pregnancy but after histopathological evaluation and integration of the past gynecological history, a cesarean scar pregnancy was diagnosed. This case provides an example of the interaction between past medical history, current clinical picture, and future reproductive plans that can help obstetrics and gynecology specialist’s decision-making in regards to hysterotomy scar pregnancies. Although a recent increase in publications regarding cesarean scar pregnancies in the past decade, this case also addressed the need for more publications of cesarean scar pregnancy cases from countries with high cesarean delivery rates.
1. Introduction An ectopic pregnancy (EP) is a pregnancy that occurs outside of the uterine cavity, occurring in approximately 1% to 2% of all pregnancies [1]. Although EPs occur at higher frequencies at the uterine tubes, other less common locations are the cervix, ovaries, within the myometrium (intramural), abdominal cavity, and fibrous tissue from cesarean scars. Cervical pregnancies (CPs) account for less than 2% of EPs and are estimated to occur in 1:2000 to 1:18,000 pregnancies [2], while cesarean (hysterotomy) scar pregnancies (CSPs) account for approximately 1:1800 to 1:2216 [3]. Awareness about CSP has increased in recent years, as only 19 articles reporting CSP cases were published in the 1990s, 48 between 2000 and 2005 and 104 between 2006 and 2011, accounting for 751 CSP cases in the medical literature by 2012
⁎
[4]. A more recent systematic review, reported a total of 2037cases by 2015 [5]. However, this pathology could be underreported in developing countries due to misdiagnoses and lower publication rates. We present the case report in line with the SCARE criteria [6] of a patient with transvaginal bleeding and abdominal pain treated in a university hospital, who after physical, laboratory, and ultrasonographic examination was initially diagnosed with a cervical pregnancy. Total hysterectomy was performed after the patient’s hemodynamic stability was compromised. After a careful histopathological evaluation and clinical integration of the gynecological history (i.e. three previous cesarean deliveries), the final diagnosis was a cesarean scar pregnancy. This case contributes to the existing literature with a successfully treated case involving a complicated CSP, that was initially misdiagnosed and upon further review was correctly classified. A
Corresponding author at: University Medical Center Groningen, Department of Neurology, PO Box 30001, 9700 RB Groningen, The Netherlands. E-mail address: [email protected] (J. Mondragón).
https://doi.org/10.1016/j.ehpc.2019.200344 Received 26 August 2019; Received in revised form 11 October 2019; Accepted 16 October 2019 2214-3300/ © 2019 The Author(s). Published by Elsevier Inc. This is an open access article under the CC BY license (http://creativecommons.org/licenses/BY/4.0/).
Human Pathology: Case Reports 18 (2019) 200344
A.J. Rivera-Rodríguez, et al.
6 × 6 cm right ovarian cyst was performed. No post-surgical complications, signs of infection or low cardiac output syndrome were reported. The patient was released after seven days of hospitalization due to clinical improvement and the conclusion of the antibiotic management for the urinary tract infection; additionally, the patient was reevaluated by the OBGYN Department 60 days after being discharged, without reporting any morbidities associated with the ectopic pregnancy.
discussion on the misdiagnosis of CSPs and the management of CSPs with hemodynamic instability is included. 2. Clinical presentation A 30-year-old Hispanic female arrived at the Emergency Department complaining of scant bright-red transvaginal bleeding of 24 h onset, associated with progressive localized pain in the left pelvic region reaching an intensity of 6/10 in the visual analog scale for pain (VAS) at the time of admission. The patient reported a pregnancy of 8 weeks and 3 days (i.e. gestational age by the last menstrual period), gravida IV, para III, and three cesarean deliveries (i.e. last cesarean delivery 7 years earlier) with a normal evolution. The patient’s family history included a mother with type 2 diabetes and arterial hypertension; other relevant aspects of family history were questioned and denied. Among the patient’s personal history the patient denied the use of controlled substances, allergies, past blood transfusions, traveling to regions with endemic diseases within the last three months, tattoos, and body piercings. Upon initial physical exploration, we found a recumbent patient with a freely chosen body position, Glasgow coma score of 15, without focal neurologic deficits, aware of her environment, with reference to place, time, and people. The patient’s integumentary system was hydrated and without alterations. Upon inspection, palpation, auscultation, and percussion the cardio-respiratory system had no abnormal findings. The abdominal exploration presented pain in the left pelvic region upon light and deep palpation, without signs of peritoneal irritation or any other alterations. Gynecological examination yielded a closed external cervical os, mild pain upon cervical manipulation, limited transvaginal hemorrhage, and the uterus below the pubic symphysis. Upon admission to the Obstetrics and Gynecology (OBGYN) Department, the patient had the following vital signs: blood pressure of 110/70 mmHg; a heart rate of 80 bpm; a respiratory rate of 20 rpm; a body temperature of 36.5 °C; a body-weight of 68 kg; the height of 165 cm.
4. Discussion The case presented above is an example of a CSP complicated by profuse hemorrhage thus leading to acute hemodynamic instability. Although CSP case reports and case series have been previously reviewed in the literature, CSPs are infrequent; furthermore, complicated cases involving hemodynamic compromise secondary to uncorrected blood loss are rare but require prompt and coordinated management. Another added value to the medical literature regarding CSPs that this case provides, is the importance of incorporating the patient’s gynecological history (i.e. history of three previous cesarean deliveries is a major risk factor for CSP) to integrate an accurate diagnosis. This patient was initially misdiagnosed, however upon further discussion and careful assessment of the histopathological findings, a cesarean scar pregnancy was diagnosed. Transvaginal ultrasound seems to be the best diagnostic tool to establish the diagnosis of CSP [4], having a sensitivity of 84.6% [3]; Doppler examination and magnetic resonance imaging could aid diagnosing complicated cases [7]. The current case was diagnosed using conventional clinical and sonographic findings, identifying a 7-weekold product through transvaginal ultrasonography (Fig. 1). However, after histopathological assessment of the surgical piece and integration of the patient’s gynecological history, a CSP was diagnosed. CSPs are difficult to diagnose, often being misdiagnosed as a low uterine pregnancy, cervical pregnancy, or miscarriage in progress [4]. Misdiagnosis of a CSP has been previously reported. Liu and colleagues (2019), report three cases with ambiguous transvaginal ultrasound findings of which, one was misdiagnosed as a cornual pregnancy [8]. One way that has been suggested to easily differentiate between a CSP and an intrauterine pregnancy is the location of the gestational sac relative to the midpoint axis of the uterus; as a gestational sac implanted low in a uterus at 5–10 weeks of gestation highly predicts a CSP [9]. Based upon case series, five treatment modalities are recommended for a CSP: 1) transvaginal resection; 2) laparoscopic resection; 3) uterine artery embolization plus uterine dilatation and curettage combined with hysteroscopy; 4) uterine artery embolization combined with uterine dilatation and curettage without methotrexate; and 5) hysteroscopy alone [5]. Since the patient was initially diagnosed with a cervical pregnancy, surgical curettage and cervical cerclage were dismissed, as surgical cutterage had a high risk for excessive hemorrhage and cervical cerclage did not provide an optimal outcome due to the continuous hemorrhagic condition. Angiographic embolization of the uterine artery (UAE) was considered; however, due to the recurrent vaginal bleeding associated with this procedure and the fact that the patient no longer wished to have offspring, the surgical team decided that UAE was not the optimal management for this case. Finally, total abdominal hysterectomy was performed as it is the treatment of choice in EPs with unstable vital signs, excessive vaginal bleeding, or have completed their families [10]. The patient had β-hCG levels of 10,500 mIU/mL upon admission; however, the patient’s β-hCG levels did not favor surgical treatment over medical management. Dior and colleagues (2018) correlate β-hCG levels and conversion to surgical management from medical treatment, reporting a 40% conversion for levels > 400,000 IU, 35.3% for levels > 20,000 IU, and only 6.3% in patients with levels at presentation < 10,000 IU [11]. Hysterectomy is a rare but necessary treatment option in cases where severe bleeding compromises the
3. Clinical evolution Transvaginal ultrasonography was performed, where the uterus cavity presented no contents and presumably the cervix contained a gestational sac at approximately 7 weeks of gestational age (Fig. 1) with a heart rate of 150 bpm; compatible with a probable cervical ectopic pregnancy. Serum human chorionic gonadotropin (β-hCG) levels upon admission were 10,500 mIU/mL. The only abnormal laboratory findings upon admission were the serum leukocyte count (i.e. 13,300 per microliter), the urinalysis (i.e. abundant bacteria and 15 leukocytes per high power field), and the urine culture test which reported growth of Escherichia coli greater than 100,000 colony forming units (CFU)/mL sensitive to multiple antibiotics, which resolved after treatment (i.e. intravenous ceftriaxone 1 g every 12 h for 7 days). Serum procalcitonin levels of 0.3 ng/mL were reported. A double-balloon cervical ripening catheter was used as a vaginal tamponade strategy. The patient developed sudden hemodynamic compromise (i.e. onset of anemia with a serum hemoglobin decrease from 14.2 g/dL to 10.3 g/dL after 12 h, tachycardia, and blood pressure of 96/66 mmHg) due to the profuse blood loss even after vaginal tamponade. No instrumentation was performed on the cervical canal. Intravenous fluid reposition was performed to hemodynamically stabilize the patient. Total hysterectomy was performed with approximately 300 mL of blood loss during the surgical procedure. The pathological findings were a uterus measuring 9.5 × 6.5 × 3.8 cm with the presence of a non-ruptured gestational sac at 6 weeks of gestation implanted in the endometrium above the cervical canal, expanding into the lower uterine cavity (Fig. 2). Upon further review, deliberation, and integration of the patient’s gynecological history, the diagnosis resulting was cesarean scar pregnancy. Additionally, resection of a 2
Human Pathology: Case Reports 18 (2019) 200344
A.J. Rivera-Rodríguez, et al.
Fig. 1. Transvaginal ultrasonography. Transvaginal ultrasonography. A) Longitudinal view of gestational sac (*) above the cervical canal (**) observed. B) Transversal view of gestational sac (*) above the cervix and expanding into the lower uterine cavity. C) Longitudinal augmented view with gestational sac (*) above the cervical canal (**) observed. D) Transversal augmented view with gestational sac (*) and cervical canal (**) observed.
mortality associated with this state; as well as, the patient’s satisfaction with her reproductive plans.
patient’s hemodynamic stability. Thus, considering that the patient’s serum hemoglobin levels had decreased from 14.2 g/dL upon admission to 10.3 g/dL after 12 hr observation and intravenous fluid reposition, hysterectomy was considered as a viable option. Previously, emergency hysterectomy, for 4 out of 57 cases in a 12-year retrospective study, was a second-line treatment option for excessive bleeding in one case, severe bleeding with disseminated intravascular coagulation during dilatation and curettage in a second case, fetal death in another case, and severe placental adhesion in the last case [12]. In the same study, one patient elected hysterectomy as the first-line treatment option since the patient had satisfied her future reproductive plans and had a previous history of adenomyosis and multiple uterine manipulations [12]. The case presented above describes a woman with her fourth pregnancy who underwent a total hysterectomy as the treatment option to correct for the hemodynamic instability. In this case, the treatment decision was justified based on two factors, acute blood loss and the increased
5. Limitations Although a rare pathology, we have reported only presented one case of a hysterotomy scar pregnancy. Over two thousand CSP cases have been reported to date; nonetheless, more cases from developing countries are needed to address the reporting bias present in all previously published reviews and systematic reviews. Underreporting from developing countries is another problem. Before 2012, most cases were reported from China (i.e. 483 out of 751 cases), the United States (i.e. 44 cases of 751), the United Kingdom (i.e. 107 cases of 751) [4]. Recently, more cases continue to be published out of China, with four randomized controlled trials assessing management strategies for CSPs from 2009 to 2015 [5]. However, there is a growing need for reports Fig. 2. Uterus with hysterotomy scar pregnancy. Surgical piece. Coronal cut of the uterus. A and B) uterus measuring 9.5 × 6.5 × 3.8 cm and weighing 136 g, with the presence of a non-ruptured gestational sac at 6 weeks of gestation implanted in the endometrium at the lower portion of the uterine cavity. Embryonic sac measuring 2.8 × 2.0 cm, with a sac wall measuring 10 to 20 mm. The content of the embryonic sac with presence of embryo measuring 6 mm longitudinally. Ocular orbits, nasal cavities, and palmar plaques present in the embryo.
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Human Pathology: Case Reports 18 (2019) 200344
A.J. Rivera-Rodríguez, et al.
References
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6. Conclusion Although CSPs are rare, management must consider the patient’s previous gynecological history, clinical evolution, and future family plans. The case presented here involves a woman with three previous cesarean deliveries, with acute onset of hemodynamic instability secondary to continuous blood loss despite management to attain hemostasis, and reference to have completed her family. This case provides an example of the interaction between the past medical history, current clinical picture, and future reproductive plans that can help OBGYN specialist’s decision-making in regards to CSPs; as well as a case initially misdiagnosed as a cervical pregnancy but upon histopathological inspection, a hysterotomy scar pregnancy was diagnosed. 7. Patient consent statement Upon hospital admission, the patient signed an informed consent permitting the use of her clinical file information for didactic, research, and publication purposes. Signed consent (Statement in Spanish) can be made available upon request. Acknowledgments This study was supported by CONACyT (Consejo Nacional de Ciencia y Tecnología) Grant #440591. Disclosure of interest: this research did not receive any specific grant from funding agencies in the commercial sector, the authors report no conflict of interest. Data availability and deposition: the clinical data supporting the conclusions of this article is included in the article; data availability can be provided on a need basis. Approval from the ethical committee was not required due to the nature of this case report. Abiding by the Declaration of Helsinki, patient anonymity was guaranteed. Upon hospital admission, the patient signed an informed consent permitting the use of her clinical file information for didactic, research, and publication purposes. We would like to commend the work of the medical staff (i.e. specialists, medical residents, and nursing staff) of the Obstetrics and Gynecology Department, as well as the Pathology department at our institution.
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