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Congenital fetal lymphangioma causing shoulder dystocia and uterine rupture
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Congenital fetal lymphangioma causing shoulder dystocia and uterine rupture Prabhat C. Mondal ⁎, Debdutta Ghosh, Anirban Mondal, Arup K. Majhi Department of Gynecology and Obstetrics, Bankura Sammilani Medical College, Bankura, India
a r t i c l e
i n f o
Article history: Received 10 August 2010 Received in revised form 3 September 2010 Accepted 26 November 2010 Keywords: Cystic hygroma Lymphangioma McRoberts maneuver Shoulder dystocia Uterine rupture Woods maneuver
spontaneously aborted fetuses [2]. Based on the autopsy findings, the lesions in the present case were considered to be cystic lymphangiomas. Lymphangiomas result from a defect in the connection between the lymphatic channels and the venous system, or from abnormal development of the lymphatic vessels [5]. Burke et al. [6] described a similar case in which cystic lymphangioma caused a difficult breech delivery. The shoulders and head of the dead fetus were delivered after multiple perforations of the lesion with a curette. Prenatal diagnosis of lymphangioma helps to plan the mode of delivery and intrapartum/postpartum care, and may decrease the morbidity of both mother and infant. Intrapartum ultrasound, if available, may be useful in cases of significant, unexpected shoulder dystocia caused by fetal lymphangioma. Conflict of interest
Lymphangiomas are rare hamartomatous malformations of skin lymphatics and subcutaneous tissues, and can occur anywhere in the body. Approximately 50% of lymphangiomas are present at birth and up to 90% are visible by the age of 2 years [1]. Approximately 70%–80% occur in the neck [2] and the remaining 20%–30% occur in the axillary region [3]. Rare locations include the mediastinum, extremities, trunk, retroperitoneal area, abdominal viscera, pelvis, and chest wall [4]. In the present case, previously undetected fetal lymphangiomas were discovered during delivery at Bankura Sammilani Medical College, Bankura, India. A 24-year-old woman, gravida 2, was referred to the hospital's tertiary center at 39 weeks of pregnancy owing to nonprogress of labor. Her first infant had been delivered vaginally 2.5 years previously. On admission, vaginal examination revealed 4-cm cervical dilation, 80% effacement of cervix, vertex presentation, and 0 station. Oxytocin infusion was started. After 4 hours, the head of the fetus was delivered; however, delivery of the shoulders proved difficult and shoulder dystocia was diagnosed. The McRoberts, Woods, and Zavanelli maneuvers were unsuccessful. At laparotomy, the anterolateral uterine segment was ruptured. The dead fetus was delivered abdominally with assistance from below and the rupture was repaired. A 10 ×10-cm swelling was found on the right side of the anterior chest of the fetus (Fig. 1) and nonpitting edema was found on the right upper limb. The fetus weighed 4.5 kg. Multiple fluid-filled spaces were found via ultrasonogram. The presence of lymphangioma and hemangioma was suspected; this was supported by computed tomography scan and confirmed via histopathology. Lymphangiomas are classified into 3 groups: lymphangioma simplex, consisting of capillary-sized channels; cavernous lymphangioma, consisting of dilated lymphatic channels; and cystic lymphangioma (hygroma), comprising multiple cysts of varying sizes [3]. Although the incidence of cystic lymphangioma is estimated to be 1 per 6000 pregnancies, it is a relatively common anomaly (1 per 875) in
⁎ Corresponding author. Department of Gynecology and Obstetrics, Bankura Sammilani Medical College, Bankura, West Bengal, India. Tel.: + 91 9434438618; fax: + 91 3242256417. E-mail address: [email protected] (P.C. Mondal).
The authors have no conflicts of interest. References [1] Marchese C, Savin E, Dragone E, Carozzi F, De Marchi M, Campogrande M. Cystic hygroma: prenatal diagnosis and genetic counselling. Prenat Diagn 1985;5(3):221–7. [2] Song TB, Kim CH, Kim SM, Kim YH, Byun JS, Kim EK. Fetal axillary cystic hygroma detected by prenatal ultrasonography: a case report. J Korean Med Sci 2002;17(3):400–2. [3] Kosir MA, Sonnino RE, Gauderer MW. Pediatric abdominal lymphangiomas: a plea for early recognition. J Pediatr Surg 1991;26(11):1309–13. [4] Rasidaki M, Sifakis S, Vardaki E, Koumantakis E. Prenatal diagnosis of a fetal chest wall cystic lymphangioma using ultrasonography and MRI: a case report with literature review. Fetal Diagn Ther 2005;20(6):504–7. [5] Hayashi A, Kikuchi A, Matsumoto Y, Tatematsu M, Horikoshi T, Ogiso Y, et al. Massive cystic lymphangiomas of a fetus. Congenit Anom (Kyoto) 2005;45(4):154–6. [6] Burke MF, Curry EM, King JM. Fetal axillary lymphangioma complicating breech delivery. BJOG 1975;82(7):585–7.
Fig. 1. Mass in the right side of the chest.
0020-7292/$ – see front matter © 2010 International Federation of Gynecology and Obstetrics. Published by Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijgo.2010.09.018
BRIEF COMMUNICATIONS
Congenital fetal lymphangioma causing shoulder dystocia and uterine rupture Prabhat C. Mondal ⁎, Debdutta Ghosh, Anirban Mondal, Arup K. Majhi Department of Gynecology and Obstetrics, Bankura Sammilani Medical College, Bankura, India
a r t i c l e
i n f o
Article history: Received 10 August 2010 Received in revised form 3 September 2010 Accepted 26 November 2010 Keywords: Cystic hygroma Lymphangioma McRoberts maneuver Shoulder dystocia Uterine rupture Woods maneuver
spontaneously aborted fetuses [2]. Based on the autopsy findings, the lesions in the present case were considered to be cystic lymphangiomas. Lymphangiomas result from a defect in the connection between the lymphatic channels and the venous system, or from abnormal development of the lymphatic vessels [5]. Burke et al. [6] described a similar case in which cystic lymphangioma caused a difficult breech delivery. The shoulders and head of the dead fetus were delivered after multiple perforations of the lesion with a curette. Prenatal diagnosis of lymphangioma helps to plan the mode of delivery and intrapartum/postpartum care, and may decrease the morbidity of both mother and infant. Intrapartum ultrasound, if available, may be useful in cases of significant, unexpected shoulder dystocia caused by fetal lymphangioma. Conflict of interest
Lymphangiomas are rare hamartomatous malformations of skin lymphatics and subcutaneous tissues, and can occur anywhere in the body. Approximately 50% of lymphangiomas are present at birth and up to 90% are visible by the age of 2 years [1]. Approximately 70%–80% occur in the neck [2] and the remaining 20%–30% occur in the axillary region [3]. Rare locations include the mediastinum, extremities, trunk, retroperitoneal area, abdominal viscera, pelvis, and chest wall [4]. In the present case, previously undetected fetal lymphangiomas were discovered during delivery at Bankura Sammilani Medical College, Bankura, India. A 24-year-old woman, gravida 2, was referred to the hospital's tertiary center at 39 weeks of pregnancy owing to nonprogress of labor. Her first infant had been delivered vaginally 2.5 years previously. On admission, vaginal examination revealed 4-cm cervical dilation, 80% effacement of cervix, vertex presentation, and 0 station. Oxytocin infusion was started. After 4 hours, the head of the fetus was delivered; however, delivery of the shoulders proved difficult and shoulder dystocia was diagnosed. The McRoberts, Woods, and Zavanelli maneuvers were unsuccessful. At laparotomy, the anterolateral uterine segment was ruptured. The dead fetus was delivered abdominally with assistance from below and the rupture was repaired. A 10 ×10-cm swelling was found on the right side of the anterior chest of the fetus (Fig. 1) and nonpitting edema was found on the right upper limb. The fetus weighed 4.5 kg. Multiple fluid-filled spaces were found via ultrasonogram. The presence of lymphangioma and hemangioma was suspected; this was supported by computed tomography scan and confirmed via histopathology. Lymphangiomas are classified into 3 groups: lymphangioma simplex, consisting of capillary-sized channels; cavernous lymphangioma, consisting of dilated lymphatic channels; and cystic lymphangioma (hygroma), comprising multiple cysts of varying sizes [3]. Although the incidence of cystic lymphangioma is estimated to be 1 per 6000 pregnancies, it is a relatively common anomaly (1 per 875) in
⁎ Corresponding author. Department of Gynecology and Obstetrics, Bankura Sammilani Medical College, Bankura, West Bengal, India. Tel.: + 91 9434438618; fax: + 91 3242256417. E-mail address: [email protected] (P.C. Mondal).
The authors have no conflicts of interest. References [1] Marchese C, Savin E, Dragone E, Carozzi F, De Marchi M, Campogrande M. Cystic hygroma: prenatal diagnosis and genetic counselling. Prenat Diagn 1985;5(3):221–7. [2] Song TB, Kim CH, Kim SM, Kim YH, Byun JS, Kim EK. Fetal axillary cystic hygroma detected by prenatal ultrasonography: a case report. J Korean Med Sci 2002;17(3):400–2. [3] Kosir MA, Sonnino RE, Gauderer MW. Pediatric abdominal lymphangiomas: a plea for early recognition. J Pediatr Surg 1991;26(11):1309–13. [4] Rasidaki M, Sifakis S, Vardaki E, Koumantakis E. Prenatal diagnosis of a fetal chest wall cystic lymphangioma using ultrasonography and MRI: a case report with literature review. Fetal Diagn Ther 2005;20(6):504–7. [5] Hayashi A, Kikuchi A, Matsumoto Y, Tatematsu M, Horikoshi T, Ogiso Y, et al. Massive cystic lymphangiomas of a fetus. Congenit Anom (Kyoto) 2005;45(4):154–6. [6] Burke MF, Curry EM, King JM. Fetal axillary lymphangioma complicating breech delivery. BJOG 1975;82(7):585–7.
Fig. 1. Mass in the right side of the chest.
0020-7292/$ – see front matter © 2010 International Federation of Gynecology and Obstetrics. Published by Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijgo.2010.09.018