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The Dalkon syndrome — a rare condition?
European Journal of Obstetrics & Gynecology and Reproductive Biology 57 (1994) 51-53
ELSEVIER
Case report
The Dalkon Syndrome I. Currie*, University Department
J. Onwude,
a rare condition? A.C. Crompton
of Obstetrics and Gynaecology, St. JamesS Beckett Street. Leeds 159 7TF, UK
University Hospital,
Accepted I July 1994
Abstract
A patient who presented acutely with severe pelvic sepsis related to Dalkon shield is reported. Although the Dalkon syndrome has been described (Tatum HJ, Connell EB. Intrauterine contraception. Duvant: Creative Informatics, 1985) the term is not widely used and controversy surrounds this condition. However, the condition may still present to the modern day gynaecologist. Keywords: Dalkon;
Intrauterine
contraceptive
device; Actinomycosis
1. Intraduction
The Dalkon syndrome may be associated with longterm use of the Dalkon shield intrauterine contraceptive device (IUD). After many years of trouble-free use the patient may present with an overwhelming pelvic sepsis which can be life-threatening. 2. Case report A 45-year-old woman presented acutely with a 4-day history of lower abdominal pain associated with fever and vomiting. The patient had noticed a yellow vaginal discharge. An intrauterine contraceptive device had been in situ for 21 years. There was no previous history of pelvic inflammatory disease. On admission the patient looked unwell, her temperature was 38.5X, her pulse was 97 beats/min and her blood pressure was 130/80 mmHg. There was tenderness in the lower abdomen and a non-tender mass was palpable in the suprapubic region. Bowel sounds were present. On vaginal examination there was a pelvic mass obliterating the pouch of Douglas and restricting the * Corresponding author. 0028-2243/94/.$07.00 0 1994 Elsevier SSDI 0028-2243(94)01917-V
Science Ireland
mobility of the uterus. Attempted removal of the IUD by pulling on the thread was unsuccessful. A full blood count showed a leucocytosis of 16.7 x 109. A diagnosis of pelvic inflammatory disease secondary to an IUD was made. Intravenous broad spectrum antibiotics were commenced; 750 mg of cefuroxime and 500 mg of metronidazole were administered intravenously every 8 h. Pelvic ultrasound examination showed a bulky uterus containing an IUD and a 4-cm fundal fibroid. A 9-cm right-sided complex cystic mass was also noted. The possibilty of actinomycosis related to an inert IUD was raised and so the administration of 1.2 g of intravenous benzylpenicillin was started. Over the next 48 h her general condition improved, although her temperature continued to spike to 39°C. A laparotomy was planned. Examination under anaesthesia revealed a purulent cervical discharge. Braided IUD threads were seen. These threads snapped at attempted removal thus requiring cervical dilatation to remove the IUD. The IUD removed was a Dalkon shield. Laparotomy through a vertical midline incision showed an enlarged uterus containing a fundal fibroid. There were omental and bowel adhesions particularly to the right adnexa. Both adnexae obliterated the pouch of Douglas, the right side being grossly enlarged and containing
Ltd. All rights reserved
52
1. Currie et al. /Eur. J. Obstet. Gynecol. Reprod. Biol. 57 (1994) 51-S
pockets of purulent fluid. A bilateral salpingoophorectomy was performed and the pelvis was drained by two abdominal suction drains. A hysterectomy was not possible due to dense vascular adhesions. Postoperatively the patient’s general condition improved rapidly. Antibiotics were continued and her temperature gradually returned to normal. She was allowed home on the eighth postoperative day. Cultures from the IUD and cervical swab grew coliforms whilst the pelvic swab showed profuse bacteroides. No actinomycosis was isolated from culture. Histology showed non-specific inflammation and abscess formation in both tubes and ovaries. No acid-fast bacilli were seen following staining and no actinomycosis organisms were seen. 3. Discussion To many junior gynaecologists the Dalkon shield will most likely be just another name of an intrauterine contraceptive device. However, the older gynaecologist will not be able to forget quite so easily the name of a device that was thought to be responsible for several maternal deaths and created much adverse publicity in the USA highlighted following the findings of the Women’s Health Study published in 1980 [ 11.The hostility against the device resulted in the company, A.H. Robins, withdrawing the product and requesting removal of all Dalkon shields. This same association and public reaction was not seen in Europe. The Dalkon shield intrauterine contraceptive device was developed in the late 1960s. A tail fixed to the base of the device extended down the endocervical canal. The tail was fixed to the lower portion of the shield by means of a double knot. There was a single knot approximately 2 cm above the end of the dependent tail, which was used to ascertain the position of the IUD within the uterine cavity. It was thought to be unique among IUDs in that its transcervical tail consisted of a bundle of many monofilaments enclosed within a sheath. This tail had the physical characteristics of a wick, harbouring fluid and bacteria and transporting them into the uterine cavity [2]. The Dalkon syndrome has recurred over the years [3], and is characterised by prolonged problem-free use of the shield. Then, without obvious predisposing events, the patient presents with an overwhelming acute episode of pelvic inflammatory disease. Attention towards the Dalkon shield initially resulted from its higher rate of pregnancy-related complications, although the increased incidence of serious complications was most likely related to both an expected higher failure rate with its early insertion experience, and a higher rate of spontaneous abortion among pregnancy failures associated with this device [4]. Association of the Dalkon shield with pelvic inflammatory disease
(PID) in subsequent case-control studies was due to referral bias and Kessel concluded that the device did not differ from other IUDs in PID risk 151. Since then a complete reappraisal of the literature on IUDs in general has been undertaken and all have concluded that pelvic inflammatory disease is not an important complication of IUD usage 161. There is only a slightly increased risk of infection in the first 20 days following insertion of an IUD, and thereafter there is little difference in infection rates when compared with the background population rate [7]. This initial transient increase is presumably related to the insertion procedure and highlights the importance of assessing patients before insertion and also the adoption of strict aseptic technique at insertion. IUDs offer no protection from PID (unlike barrier methods of contraception) and the risk of infection in IUD users is related to sexual lifestyle and not to the IUD itself. Microbial culture of specimens obtained either at surgery (purulent material from tubes and ovaries) or directly from the IUD itself usually produces many organisms that are predominantly anaerobic species. Pelvic inflammatory disease where actinomycosis is the primary pathogen is uncommon and so caution must be exercised in making this diagnosis, which requires pathological verification from tissue extirpated at the time of surgery. If, however, actinomycosis is isolated, intensive management is required with massive doses of penicillin for several weeks and a wide surgical excision is necessary [8]. The mortality from true actinomycosis is lo-20% and so it must be emphasised that this infection must always be considered in anyone who presents with infection and is a long-term IUD user. Management of our patient was initially medical with the administration of broad spectrum antibiotics and as there was an inadequate response, surgical intervention was necessary. Surgery is usually limited to drainage of purulent fluid and removal of any devitalised tissue which would otherwise hinder antibiotic penetration. Formal and definitive surgery may be performed as an interval procedure. This case, although not complicated in its management, serves to emphasise to the younger gynaecologist that the Dalkon syndrome as described may arise in present day gynaecology. This should be suspected in cases of pelvic inflammatory disease associated with prolonged use of an IUD, where the tail is blue, braided and difficult to remove. References 111 (21 [3]
Burkman RT. Intrauterine device use and the risk of pelvic inflammatory disease. Am J Obstet Gynecol 1980; 138: 861-863. Tatum HJ et al. Morphological studies of Dalkon shield tails recovered from patients. Contraception 1975; 11:465-477. Tatum HJ, Connell EB. Intrauterine contraception. Durant. Oklahoma: Creative Informatics, 1985.
I. Currie et al. /Eur. J. Obste!. Gynecol. Reprod. Biol. 57 (1994) 51-53 [4]
[5] [6]
Kessel E. Pelvic inflammatory disease with intrauterine device use: a reassessment. Fertil Steril 1989; 51: l-l I. Kessel E. Uterine devices and pelvic inflammatory disease [letter] Lancet 1992; 339: 1306. Kronmal RA. The intrauterine device and pelvic inflammatory disease: the Women’s Health Study reanalyzed. J Clin Epid 1991; 44: 109-122.
]7]
[8]
53
Farley TM. Intrauterine devices and pelvic inflammatory disease: an international perspective. Lancet 1992; 339: 785-788. Charles D. Infection in obstetrics and gynecology. Major Prob. Obstet Gynaecol 1980; 12: 80.
ELSEVIER
Case report
The Dalkon Syndrome I. Currie*, University Department
J. Onwude,
a rare condition? A.C. Crompton
of Obstetrics and Gynaecology, St. JamesS Beckett Street. Leeds 159 7TF, UK
University Hospital,
Accepted I July 1994
Abstract
A patient who presented acutely with severe pelvic sepsis related to Dalkon shield is reported. Although the Dalkon syndrome has been described (Tatum HJ, Connell EB. Intrauterine contraception. Duvant: Creative Informatics, 1985) the term is not widely used and controversy surrounds this condition. However, the condition may still present to the modern day gynaecologist. Keywords: Dalkon;
Intrauterine
contraceptive
device; Actinomycosis
1. Intraduction
The Dalkon syndrome may be associated with longterm use of the Dalkon shield intrauterine contraceptive device (IUD). After many years of trouble-free use the patient may present with an overwhelming pelvic sepsis which can be life-threatening. 2. Case report A 45-year-old woman presented acutely with a 4-day history of lower abdominal pain associated with fever and vomiting. The patient had noticed a yellow vaginal discharge. An intrauterine contraceptive device had been in situ for 21 years. There was no previous history of pelvic inflammatory disease. On admission the patient looked unwell, her temperature was 38.5X, her pulse was 97 beats/min and her blood pressure was 130/80 mmHg. There was tenderness in the lower abdomen and a non-tender mass was palpable in the suprapubic region. Bowel sounds were present. On vaginal examination there was a pelvic mass obliterating the pouch of Douglas and restricting the * Corresponding author. 0028-2243/94/.$07.00 0 1994 Elsevier SSDI 0028-2243(94)01917-V
Science Ireland
mobility of the uterus. Attempted removal of the IUD by pulling on the thread was unsuccessful. A full blood count showed a leucocytosis of 16.7 x 109. A diagnosis of pelvic inflammatory disease secondary to an IUD was made. Intravenous broad spectrum antibiotics were commenced; 750 mg of cefuroxime and 500 mg of metronidazole were administered intravenously every 8 h. Pelvic ultrasound examination showed a bulky uterus containing an IUD and a 4-cm fundal fibroid. A 9-cm right-sided complex cystic mass was also noted. The possibilty of actinomycosis related to an inert IUD was raised and so the administration of 1.2 g of intravenous benzylpenicillin was started. Over the next 48 h her general condition improved, although her temperature continued to spike to 39°C. A laparotomy was planned. Examination under anaesthesia revealed a purulent cervical discharge. Braided IUD threads were seen. These threads snapped at attempted removal thus requiring cervical dilatation to remove the IUD. The IUD removed was a Dalkon shield. Laparotomy through a vertical midline incision showed an enlarged uterus containing a fundal fibroid. There were omental and bowel adhesions particularly to the right adnexa. Both adnexae obliterated the pouch of Douglas, the right side being grossly enlarged and containing
Ltd. All rights reserved
52
1. Currie et al. /Eur. J. Obstet. Gynecol. Reprod. Biol. 57 (1994) 51-S
pockets of purulent fluid. A bilateral salpingoophorectomy was performed and the pelvis was drained by two abdominal suction drains. A hysterectomy was not possible due to dense vascular adhesions. Postoperatively the patient’s general condition improved rapidly. Antibiotics were continued and her temperature gradually returned to normal. She was allowed home on the eighth postoperative day. Cultures from the IUD and cervical swab grew coliforms whilst the pelvic swab showed profuse bacteroides. No actinomycosis was isolated from culture. Histology showed non-specific inflammation and abscess formation in both tubes and ovaries. No acid-fast bacilli were seen following staining and no actinomycosis organisms were seen. 3. Discussion To many junior gynaecologists the Dalkon shield will most likely be just another name of an intrauterine contraceptive device. However, the older gynaecologist will not be able to forget quite so easily the name of a device that was thought to be responsible for several maternal deaths and created much adverse publicity in the USA highlighted following the findings of the Women’s Health Study published in 1980 [ 11.The hostility against the device resulted in the company, A.H. Robins, withdrawing the product and requesting removal of all Dalkon shields. This same association and public reaction was not seen in Europe. The Dalkon shield intrauterine contraceptive device was developed in the late 1960s. A tail fixed to the base of the device extended down the endocervical canal. The tail was fixed to the lower portion of the shield by means of a double knot. There was a single knot approximately 2 cm above the end of the dependent tail, which was used to ascertain the position of the IUD within the uterine cavity. It was thought to be unique among IUDs in that its transcervical tail consisted of a bundle of many monofilaments enclosed within a sheath. This tail had the physical characteristics of a wick, harbouring fluid and bacteria and transporting them into the uterine cavity [2]. The Dalkon syndrome has recurred over the years [3], and is characterised by prolonged problem-free use of the shield. Then, without obvious predisposing events, the patient presents with an overwhelming acute episode of pelvic inflammatory disease. Attention towards the Dalkon shield initially resulted from its higher rate of pregnancy-related complications, although the increased incidence of serious complications was most likely related to both an expected higher failure rate with its early insertion experience, and a higher rate of spontaneous abortion among pregnancy failures associated with this device [4]. Association of the Dalkon shield with pelvic inflammatory disease
(PID) in subsequent case-control studies was due to referral bias and Kessel concluded that the device did not differ from other IUDs in PID risk 151. Since then a complete reappraisal of the literature on IUDs in general has been undertaken and all have concluded that pelvic inflammatory disease is not an important complication of IUD usage 161. There is only a slightly increased risk of infection in the first 20 days following insertion of an IUD, and thereafter there is little difference in infection rates when compared with the background population rate [7]. This initial transient increase is presumably related to the insertion procedure and highlights the importance of assessing patients before insertion and also the adoption of strict aseptic technique at insertion. IUDs offer no protection from PID (unlike barrier methods of contraception) and the risk of infection in IUD users is related to sexual lifestyle and not to the IUD itself. Microbial culture of specimens obtained either at surgery (purulent material from tubes and ovaries) or directly from the IUD itself usually produces many organisms that are predominantly anaerobic species. Pelvic inflammatory disease where actinomycosis is the primary pathogen is uncommon and so caution must be exercised in making this diagnosis, which requires pathological verification from tissue extirpated at the time of surgery. If, however, actinomycosis is isolated, intensive management is required with massive doses of penicillin for several weeks and a wide surgical excision is necessary [8]. The mortality from true actinomycosis is lo-20% and so it must be emphasised that this infection must always be considered in anyone who presents with infection and is a long-term IUD user. Management of our patient was initially medical with the administration of broad spectrum antibiotics and as there was an inadequate response, surgical intervention was necessary. Surgery is usually limited to drainage of purulent fluid and removal of any devitalised tissue which would otherwise hinder antibiotic penetration. Formal and definitive surgery may be performed as an interval procedure. This case, although not complicated in its management, serves to emphasise to the younger gynaecologist that the Dalkon syndrome as described may arise in present day gynaecology. This should be suspected in cases of pelvic inflammatory disease associated with prolonged use of an IUD, where the tail is blue, braided and difficult to remove. References 111 (21 [3]
Burkman RT. Intrauterine device use and the risk of pelvic inflammatory disease. Am J Obstet Gynecol 1980; 138: 861-863. Tatum HJ et al. Morphological studies of Dalkon shield tails recovered from patients. Contraception 1975; 11:465-477. Tatum HJ, Connell EB. Intrauterine contraception. Durant. Oklahoma: Creative Informatics, 1985.
I. Currie et al. /Eur. J. Obste!. Gynecol. Reprod. Biol. 57 (1994) 51-53 [4]
[5] [6]
Kessel E. Pelvic inflammatory disease with intrauterine device use: a reassessment. Fertil Steril 1989; 51: l-l I. Kessel E. Uterine devices and pelvic inflammatory disease [letter] Lancet 1992; 339: 1306. Kronmal RA. The intrauterine device and pelvic inflammatory disease: the Women’s Health Study reanalyzed. J Clin Epid 1991; 44: 109-122.
]7]
[8]
53
Farley TM. Intrauterine devices and pelvic inflammatory disease: an international perspective. Lancet 1992; 339: 785-788. Charles D. Infection in obstetrics and gynecology. Major Prob. Obstet Gynaecol 1980; 12: 80.