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Ruptured cystic teratoma masquerading as a pelvic abscess
European Journal of Obstetrics & Gynecology and Reproductive Biology 132 (2007) 237–244 www.elsevier.com/locate/ejogrb
LETTERS TO THE EDITOR—BRIEF COMMUNICATION Ruptured cystic teratoma masquerading as a pelvic abscess Dear Editors, Spontaneous rupture is an uncommon complication of benign cystic teratoma (dermoid cyst). Here, we report a rare presentation of this condition as a pelvic abscess without the characteristic chemical peritonitis. A 28-year-old, para two presented with high-grade fever since 15–20 days, pain in the lower abdomen since 6 days and loose motions since 2 days. She gave no history of tenesmus, abdominal distention or urinary symptoms. Her last menstrual period was 15 days earlier. On general examination, she was pale, febrile and cachexic. The pulse was 120/min, regular; blood pressure 130/ 70 mmHg. Respiratory and cardiovascular systems were unremarkable on examination. The abdomen was tender with mild distention, guarding and rigidity with bowel sounds in all four quadrants. On gynecological examination, the vaginal mucosa was pale with evidence of vaginitis and an ill-defined fullness was felt through all the fornices and particularly in the pouch of Douglas. The uterus was of normal size. Pregnancy test was negative. On ultrasound, there was free fluid in the abdomen with internal echoes and a large loculated collection in cul-de-sac. There was a heterogeneous complex cystic lesion in the right adnexa. On culdocentesis, 50 cm3 of pus was drained. At laparotomy, abundant pus was drained from the abdominal cavity and the pouch of Douglas. Multiple flimsy adhesions were present between the omentum and bowel loops except at the site of the sigmoid colon, which was densely adherent to a complex tuboovarian mass of about 6 in. 6 in. on the right side. Uterus and left fallopian tube were normal. During separation of the dense adhesions, the mass accidentally opened with a spread of cheesy and hairy material. No evidence of torsion or gangrene was seen and the mass was excised. The remaining abdominal viscera and bowel were normal, as examined by the surgeons, with no evidence of infective focus or perforation. Culture of the pus showed presence of pseudomonas. Microscopic examination of the ovarian mass was suggestive of a benign cystic teratoma with areas of chronic inflammation. Sections from
the omentum showed features of chronic inflammatory infiltrate. The patient had an uneventful postoperative recovery. The most common complication of teratoma is torsion, while rupture is very rarely reported (<1%) probably due to the thick cystic wall [1–3]. Ruptured teratoma can present as acute abdomen due to sudden and severe chemical peritonitis or as progressive abdominal distention with gastrointestinal disturbances due to slow leakage from a breach in the cyst wall [3]. If the perforation is small, the signs and symptoms maybe minimal and the patient may complain only of vague low abdominal pain with or without increase in the abdominal girth [2]. Intraperitoneal rupture of teratoma results in spillage of sebaceous material, which causes chemical irritation and granulomatous peritonitis, forming extensive adhesions and granulomas involving the entire peritoneal cavity [4]. The presentation of a ruptured teratoma as a pelvic abscess along with the absence of the characteristic granulomatous peritonitis are very unusual features which were present in our case. The likely etiology of rupture of the thick walled dermoid cyst into the peritoneal cavity is the combined softening and traumatic effects of inflammation [5]. This explains the rupture of cyst wall in our patient which must have been a slow leakage causing vague symptoms. As our patient had vaginitis, we feel the source of infection could have been an episode of ascending pelvic inflammatory disease, which led to the subsequent secondary infection of the already spilled sebaceous contents in the pelvic cavity. The breach in the cyst wall was sealed off leading to the dense adhesions with the sigmoid colon at the ruptured site. In the literature, the only two previously reported cases of cystic ovarian teratoma presenting as a pelvic abscess were associated with pregnancy termination [6,7]. Roman et al. [8] also recently reported a case of ruptured teratoma in advanced pregnancy which was managed laproscopically. Abscess formation in presacral (nonovarian) teratoma though can sometimes mimic a pararectal abscess [9]. To conclude, the presentation of a ruptured benign cystic teratoma as a pelvic abscess without any associated granulomatous reaction is very unusual and deserves a special mention in the literature.
0301-2115/$ – see front matter # 2006 Elsevier Ireland Ltd. All rights reserved.
238
Letters to the Editor / European Journal of Obstetrics & Gynecology and Reproductive Biology 132 (2007) 237–244
References
Successful use of insulin glargine during entire pregnancy until delivery in six Type 1 diabetic women
[1] Scully RF, Young RH, Clement PB. Atlas of tumor pathology. Tumors of the ovary, maldeveloped gonads, fallopian tube, and broad ligament. Third series. AFIP 1998;274. [2] Waxman M, Boyce JG. Intraperitoneal rupture of benign cystic ovarian teratoma. Obstet Gynaecol 1976;48(1 Suppl.):9–13. [3] Bhatla N, Khanna R, Bhargava VL. Intraperitoneal rupture of benign cystic teratoma. Int J Gynecol Obstet 1993;(40):163–4. [4] Kistner RW. Intraperitoneal rupture of benign cystic teratoma. Review of the literature with a report of two cases. Obstet Gynecol Surv 1952;7(October (5)):603–17. [5] Ranney B. Iatrogenic spillage from benign cystic teratoma causing severe peritoneal granulomas and adhesions. Report of a case. Obstet Gynaecol 1970;35(April (4)):562–4. [6] Giustini FG, Sohn S, Khosrani H. Pelvic abscess and perforation of the sigmoid colon by a segment of benign cystic teratoma: an unusual complication of induced abortion. J Reprod Med 1978;20(5): 291–2. [7] Lichtenberg S. Cystic teratoma-provoked peritonitis after pregnancy termination. J Obstet Gynaecol Can 2004;26(September (9)): 823–5. [8] Roman H, Accoceberry M, Bolandard F, Bourdel N, Lenglet Y, Canis M. Laparoscopic management of a ruptured benign dermoid cyst during advanced pregnancy. J Minim Invasive Gynecol 2005;12(July–August (4)):377–8. [9] Saygun O, Avsar FM, Topaloglu S, et al. Retrorectal benign cystic teratoma misdiagnosed with high lying pararectal abscess. Hepatogastroenterology 2003;50(December (Suppl. 2)). cclii–ccliii.
Sudipta Banerjee* FR 1 Sri Narayan Appartments, 72 Khare Town, Dharampeth, Nagpur 440010, India Rajeshri Patil Department of Obstetrics and Gynaecology, Government Medical College, Nagpur 440003, India Rupan Sanyal Department of Radiology, Government Medical College, Nagpur 440003, India Rekha Sapkal Department of Obstetrics and Gynaecology, Government Medical College, Nagpur 440003, India *Corresponding author. Tel.: +91 9822574098 E-mail address: [email protected] (S. Banerjee) Tel.: + 91 9823101738. E-mail address: [email protected] (R. Sanyal) 6 November 2005 doi:10.1016/j.ejogrb.2006.06.003
Dear Editors, Insulin glargine is a long-acting insulin analogue which is recommended in Type 1 diabetes mellitus (T1DM). It is given by injection once a day. When optimally combined with rapid-acting insulin analogues (RA-IA) at meal-time, glargine decreases inter-prandial blood glucose and lowers the risk of nocturnal hypoglycaemia versus Neutral Protamine Haghedorn (NPH) insulin [1]. Few cases of pregnancy conceived during glargine treatment have been reported [2–4], and in most cases glargine was withdrawn within a few weeks from the onset of pregnancy [2,3]. With the exception of a recent retrospective analysis of hospital files from three different clinics in the Netherlands [5], and a report of one patient with Addison’s disease [4] there have been no reports of uninterrupted use of glargine until delivery in T1 diabetic women. We report six cases of Type 1 diabetics followed in our department from the conception to the delivery and who attended the first diabetes evaluation at our outpatient clinic between September 2002 and December 2004. Clinical characteristics of the patients are shown in Table 1. None of them had planned the pregnancy. They were all informed about the lack of indication for use of glargine in pregnancy, however, refused continuous subcutaneous insulin infusion (CSII) and decided to continue/initiate glargine to improve glycaemic control. Four patients were on glargine before pregnancy and two patients started glargine during the first trimester because of frequent hypoglycaemia and unstable glycaemic control (Table 1). In all patients, glycaemic control improved during pregnancy, with an average HbA1c of 6.0 0.2% at the end of pregnancy. In two patients the gynaecologist, after induction of lung maturity, proceeded with caesarean section because of preeclampsia (respectively at 33 and 36 weeks). No progression in diabetic complications was detected at the follow-up. All babies (three males and three females) were delivered at a mean gestational age of 36.6 0.8 weeks by caesarean section in five out of the six women. Mean birth weight was 3160 270 g, with one baby >4 kg, whereas the other babies’ weights were adequate for gestational age. They were healthy and none of them had perinatal complication. This is the first report on conception, pregnancy and delivery of women with T1DM using glargine in combination with RA-IA under the personal supervision of the authors. The insulin regimen resulted in an improvement of blood glucose control in pregnancy and delivery of healthy babies. In a recent report [5], similar observations were made from ‘‘a posteriori’’ examination of hospital files in seven women, although the kind of prandial insulin was not specified (regular? analogue?). No insulin analogue has officially received specific approval for use in pregnancy, but lispro appears safe in pregnancy versus human regular
LETTERS TO THE EDITOR—BRIEF COMMUNICATION Ruptured cystic teratoma masquerading as a pelvic abscess Dear Editors, Spontaneous rupture is an uncommon complication of benign cystic teratoma (dermoid cyst). Here, we report a rare presentation of this condition as a pelvic abscess without the characteristic chemical peritonitis. A 28-year-old, para two presented with high-grade fever since 15–20 days, pain in the lower abdomen since 6 days and loose motions since 2 days. She gave no history of tenesmus, abdominal distention or urinary symptoms. Her last menstrual period was 15 days earlier. On general examination, she was pale, febrile and cachexic. The pulse was 120/min, regular; blood pressure 130/ 70 mmHg. Respiratory and cardiovascular systems were unremarkable on examination. The abdomen was tender with mild distention, guarding and rigidity with bowel sounds in all four quadrants. On gynecological examination, the vaginal mucosa was pale with evidence of vaginitis and an ill-defined fullness was felt through all the fornices and particularly in the pouch of Douglas. The uterus was of normal size. Pregnancy test was negative. On ultrasound, there was free fluid in the abdomen with internal echoes and a large loculated collection in cul-de-sac. There was a heterogeneous complex cystic lesion in the right adnexa. On culdocentesis, 50 cm3 of pus was drained. At laparotomy, abundant pus was drained from the abdominal cavity and the pouch of Douglas. Multiple flimsy adhesions were present between the omentum and bowel loops except at the site of the sigmoid colon, which was densely adherent to a complex tuboovarian mass of about 6 in. 6 in. on the right side. Uterus and left fallopian tube were normal. During separation of the dense adhesions, the mass accidentally opened with a spread of cheesy and hairy material. No evidence of torsion or gangrene was seen and the mass was excised. The remaining abdominal viscera and bowel were normal, as examined by the surgeons, with no evidence of infective focus or perforation. Culture of the pus showed presence of pseudomonas. Microscopic examination of the ovarian mass was suggestive of a benign cystic teratoma with areas of chronic inflammation. Sections from
the omentum showed features of chronic inflammatory infiltrate. The patient had an uneventful postoperative recovery. The most common complication of teratoma is torsion, while rupture is very rarely reported (<1%) probably due to the thick cystic wall [1–3]. Ruptured teratoma can present as acute abdomen due to sudden and severe chemical peritonitis or as progressive abdominal distention with gastrointestinal disturbances due to slow leakage from a breach in the cyst wall [3]. If the perforation is small, the signs and symptoms maybe minimal and the patient may complain only of vague low abdominal pain with or without increase in the abdominal girth [2]. Intraperitoneal rupture of teratoma results in spillage of sebaceous material, which causes chemical irritation and granulomatous peritonitis, forming extensive adhesions and granulomas involving the entire peritoneal cavity [4]. The presentation of a ruptured teratoma as a pelvic abscess along with the absence of the characteristic granulomatous peritonitis are very unusual features which were present in our case. The likely etiology of rupture of the thick walled dermoid cyst into the peritoneal cavity is the combined softening and traumatic effects of inflammation [5]. This explains the rupture of cyst wall in our patient which must have been a slow leakage causing vague symptoms. As our patient had vaginitis, we feel the source of infection could have been an episode of ascending pelvic inflammatory disease, which led to the subsequent secondary infection of the already spilled sebaceous contents in the pelvic cavity. The breach in the cyst wall was sealed off leading to the dense adhesions with the sigmoid colon at the ruptured site. In the literature, the only two previously reported cases of cystic ovarian teratoma presenting as a pelvic abscess were associated with pregnancy termination [6,7]. Roman et al. [8] also recently reported a case of ruptured teratoma in advanced pregnancy which was managed laproscopically. Abscess formation in presacral (nonovarian) teratoma though can sometimes mimic a pararectal abscess [9]. To conclude, the presentation of a ruptured benign cystic teratoma as a pelvic abscess without any associated granulomatous reaction is very unusual and deserves a special mention in the literature.
0301-2115/$ – see front matter # 2006 Elsevier Ireland Ltd. All rights reserved.
238
Letters to the Editor / European Journal of Obstetrics & Gynecology and Reproductive Biology 132 (2007) 237–244
References
Successful use of insulin glargine during entire pregnancy until delivery in six Type 1 diabetic women
[1] Scully RF, Young RH, Clement PB. Atlas of tumor pathology. Tumors of the ovary, maldeveloped gonads, fallopian tube, and broad ligament. Third series. AFIP 1998;274. [2] Waxman M, Boyce JG. Intraperitoneal rupture of benign cystic ovarian teratoma. Obstet Gynaecol 1976;48(1 Suppl.):9–13. [3] Bhatla N, Khanna R, Bhargava VL. Intraperitoneal rupture of benign cystic teratoma. Int J Gynecol Obstet 1993;(40):163–4. [4] Kistner RW. Intraperitoneal rupture of benign cystic teratoma. Review of the literature with a report of two cases. Obstet Gynecol Surv 1952;7(October (5)):603–17. [5] Ranney B. Iatrogenic spillage from benign cystic teratoma causing severe peritoneal granulomas and adhesions. Report of a case. Obstet Gynaecol 1970;35(April (4)):562–4. [6] Giustini FG, Sohn S, Khosrani H. Pelvic abscess and perforation of the sigmoid colon by a segment of benign cystic teratoma: an unusual complication of induced abortion. J Reprod Med 1978;20(5): 291–2. [7] Lichtenberg S. Cystic teratoma-provoked peritonitis after pregnancy termination. J Obstet Gynaecol Can 2004;26(September (9)): 823–5. [8] Roman H, Accoceberry M, Bolandard F, Bourdel N, Lenglet Y, Canis M. Laparoscopic management of a ruptured benign dermoid cyst during advanced pregnancy. J Minim Invasive Gynecol 2005;12(July–August (4)):377–8. [9] Saygun O, Avsar FM, Topaloglu S, et al. Retrorectal benign cystic teratoma misdiagnosed with high lying pararectal abscess. Hepatogastroenterology 2003;50(December (Suppl. 2)). cclii–ccliii.
Sudipta Banerjee* FR 1 Sri Narayan Appartments, 72 Khare Town, Dharampeth, Nagpur 440010, India Rajeshri Patil Department of Obstetrics and Gynaecology, Government Medical College, Nagpur 440003, India Rupan Sanyal Department of Radiology, Government Medical College, Nagpur 440003, India Rekha Sapkal Department of Obstetrics and Gynaecology, Government Medical College, Nagpur 440003, India *Corresponding author. Tel.: +91 9822574098 E-mail address: [email protected] (S. Banerjee) Tel.: + 91 9823101738. E-mail address: [email protected] (R. Sanyal) 6 November 2005 doi:10.1016/j.ejogrb.2006.06.003
Dear Editors, Insulin glargine is a long-acting insulin analogue which is recommended in Type 1 diabetes mellitus (T1DM). It is given by injection once a day. When optimally combined with rapid-acting insulin analogues (RA-IA) at meal-time, glargine decreases inter-prandial blood glucose and lowers the risk of nocturnal hypoglycaemia versus Neutral Protamine Haghedorn (NPH) insulin [1]. Few cases of pregnancy conceived during glargine treatment have been reported [2–4], and in most cases glargine was withdrawn within a few weeks from the onset of pregnancy [2,3]. With the exception of a recent retrospective analysis of hospital files from three different clinics in the Netherlands [5], and a report of one patient with Addison’s disease [4] there have been no reports of uninterrupted use of glargine until delivery in T1 diabetic women. We report six cases of Type 1 diabetics followed in our department from the conception to the delivery and who attended the first diabetes evaluation at our outpatient clinic between September 2002 and December 2004. Clinical characteristics of the patients are shown in Table 1. None of them had planned the pregnancy. They were all informed about the lack of indication for use of glargine in pregnancy, however, refused continuous subcutaneous insulin infusion (CSII) and decided to continue/initiate glargine to improve glycaemic control. Four patients were on glargine before pregnancy and two patients started glargine during the first trimester because of frequent hypoglycaemia and unstable glycaemic control (Table 1). In all patients, glycaemic control improved during pregnancy, with an average HbA1c of 6.0 0.2% at the end of pregnancy. In two patients the gynaecologist, after induction of lung maturity, proceeded with caesarean section because of preeclampsia (respectively at 33 and 36 weeks). No progression in diabetic complications was detected at the follow-up. All babies (three males and three females) were delivered at a mean gestational age of 36.6 0.8 weeks by caesarean section in five out of the six women. Mean birth weight was 3160 270 g, with one baby >4 kg, whereas the other babies’ weights were adequate for gestational age. They were healthy and none of them had perinatal complication. This is the first report on conception, pregnancy and delivery of women with T1DM using glargine in combination with RA-IA under the personal supervision of the authors. The insulin regimen resulted in an improvement of blood glucose control in pregnancy and delivery of healthy babies. In a recent report [5], similar observations were made from ‘‘a posteriori’’ examination of hospital files in seven women, although the kind of prandial insulin was not specified (regular? analogue?). No insulin analogue has officially received specific approval for use in pregnancy, but lispro appears safe in pregnancy versus human regular