2 Clement PB, Scully RE. Mullerian adenosarcoma of the uterus; a clinicopathologic analysis of 100 cases with a review of the literature. Hum Pathol 1990; 21 (4): 363–381. 3 Judson PL. Vaginal adenosarcoma arising from endometriosis. Gynecol Oncol 2000; 76: 123–125. 4 Anderson J. Adenosarcoma in a patient with vaginal endometriosis. Obstet Gynecol 2001; 98: 964–966.
5 Liu L, Davidson S. Müllerian adenosarcoma of vagina arising in persistent endometriosis: report of a case and review of the literature. Gynecol Oncol 2003; 90 (2): 486–490. 6 Toyoshima M, Akahira J, Moriya T et al. Primary vaginal adenosarcoma with sarcomatous overgrowth. Gynecol Oncol 2004; 95 (3): 759–761.
A case of massive vulvar oedema due to septic pubic symphysitis complicating pregnancy Alice M. LAWFORD,1 Katherine SCOTT2,3 and Karin LUST4 1
University of Sheffield Medical School, Sheffield, UK, 2Royal Brisbane and Women’s Hospital, Herston, Queensland, 3Gold Coast Hospital – Endocrinology, Southport, Queensland, and 4Department of Internal Medicine, Royal Brisbane & Women’s Hospital, Brisbane, Queensland, Australia
We present a case of pyogenic pubic symphysitis presenting in the third trimester with progressive suprapubic pain, fever and massive vulvar oedema. This case demonstrates a rare, but important cause of sepsis in pregnancy, which, if not recognised and treated promptly, may result in significant morbidity. Key words: osteomyelitis, pregnancy, pregnancy complications, sepsis.
Case Report A 27-year-old nulliparous woman presented at 37 weeks gestation with a 2-day history of right hip and groin pain. Her antenatal course had been unremarkable barring mild swelling of her vulva from early pregnancy. She was previously well, and worked on a remote cattle station. At presentation at a rural hospital, she was febrile (39C), tachycardic (rate 130 beats ⁄ min) tachypneic (rate 32 ⁄ min) and normotensive (120 ⁄ 60 mmHg). Examination revealed tenderness to palpation in the right iliac fossa with signs of peritonism, and marked vulvar oedema. There was no regional lymphadenopathy. Electrolytes, liver function tests and full blood count were within normal limits, but C-reactive protein level was markedly raised (373 mg ⁄ L). Urinalysis revealed no abnormality. Provisional diagnosis was intra-abdominal sepsis and ceftriaxone, metronidazole and gentamicin were commenced prior to transfer to a regional hospital. Cardiotocography on arrival revealed fetal tachycardia with decreased variability and late decelerations. Emergency caesarean section was performed via a vertical incision to facilitate appendicectomy
and exploratory laparotomy. The appendix was normal and laparotomy was unremarkable. A live female infant was delivered weighing 2780 g. She had poor Apgar scores at 1 min and required resuscitation and cardiac massage, however, recovered rapidly, and blood cultures from the neonate were negative.
Correspondence: Dr Katherine Scott, Royal Brisbane and Women’s Hospital – Internal Medicine and Aged Care, Level 3 James Mayne Building, Butterfield St, Herston, Queensland 4029, Australia. Email: [email protected]
DOI: 10.1111/j.1479-828X.2010.01227.x Received 9 June 2010; accepted 29 July 2010.
Figure 1 MRI scan demonstrating massive vulvar oedema and a walled off pyogenic collection within the symphysis pubis.
2010 The Authors
Australian and New Zealand Journal of Obstetrics and Gynaecology 2010 The Royal Australian and New Zealand College of Obstetricians and Gynaecologists; 50: 573–581 The Australian and New Zealand Journal of Obstetrics and Gynaecology
Postoperatively, the patient remained febrile and was transferred to a tertiary referral centre. On arrival, she was febrile (39C) with rigours, suprapubic pain, peritonism and extensive bilateral labial swelling. Results from initial blood cultures revealed growth of Staphylococcus aureus from both bottles within 2 h. Magnetic resonance imaging demonstrated a 5 cm collection surrounding the symphysis pubis with extension into the soft tissues (Fig. 1). Under ultrasound guidance, 8 mL of purulent fluid was aspirated which also grew a fully sensitive Staphylococcus aureus. Antibiotic treatment was changed to flucloxacillin, and later to cephazolin following deranged liver function tests. One week post delivery, the patient was well enough to be discharged to a regional hospital for further 2 weeks of intravenous antibiotics followed by oral antibiotics to complete 6 weeks’ total therapy.
Our patient had no clear risk factors for staphylococcal sepsis. She was primiparous with no history of recent trauma or urinary tract instrumentation. The patient’s manual work during pregnancy may have contributed to inflammation of the tendon insertion to the symphysis pubis forming a possible nidus for infection.9 We postulate that she had a subacute infection, which caused mild vulvar oedema throughout pregnancy, and only manifest as sepsis at term. Imaging and culture from blood or tissue are the mainstays for diagnosis for osteomyelitis pubis. CT imaging of the pelvis antenatally is associated with radiation to the fetus and is therefore seldom performed. Early treatment with intravenous antibiotics and drainage is imperative to prevent adverse outcomes, and should not be delayed while awaiting imaging.10
1 Tam KS, Woods ML, Hill D. Staphylococcus septicaemia and massive vulvar oedema in pregnancy. Aust NZ J Obstet Gynaecol 2002; 42 (5): 554–556. 2 Saha KP, Rohilla M, Prasad G, Dhaliwal LK, Gupta I. Massive vulval edema in pregnancy. Internet J Gynaec Obs 2007; 6 (2). Available from: http://www.ispub.com/ostia/index. php?xmlFilePath=journals/ijgo/vol6n2/vulva.xml 3 Tsukahara SO, Momohara S, Ikari K et al. Disturbances of the symphysis pubis in rheumatoid arthritis: report of two cases. Mod Rheumatol 2007; 17: 344–347. 4 Sexton DJ, Heskestad L, Lambeth WR, McCallum R, Levin SL, Corey GR. Postoperative pubic osteomyelitis misdiagnosed as osteitis pubis: report of four cases and review. Clin Infect Dis 1993; 17 (4): 695–700. 5 Gamble K, Dardarian TS, Finstein J, Fox E, Sejdev H, Randall TC. Osteomyelitis of the pubic symphysis in pregnancy. Obstet Gynecol 2006; 107: 477–481. 6 Ikpeme IA, Archibong AE, Udoh JJ, Nya AB, Archibong GE. Chronic osteomyelitis of the pubis in the peri-partum period – a rare presentation: case report and a review of the literature. Internet J Third World Med 2008; 6 (2). Available from: http:// www.ispub.com/journal/the_internet_journal_of_third_world_ medicine/volume_6_number_2_20/article/chronic_osteomyelitis_ of_the_pubis_in_the_peri_partum_period_a_rare_presentation_ case_report_and_a_review_of_the_literature.html 7 Rosenthal RE, Anderson Spickard W, Markham RD, Rhamy RK. Osteomyelitis of the symphysis pubis: a separate disease from osteitis pubis. J Bone Joint Surg 1982; 64A (1): 123–128. 8 Ross JD, Hu LT. Septic arthritis of the pubic symphysis. Rev 100 Cases Med 2003; 82 (5): 340–345. 9 Pham DV, Scott KG. Presentation of osteitis and osteomyelitis pubis as acute abdominal pain. Perman J 2007; 11 (2): 65–68. 10 Eskridge C, Longo S, Kwark J, Robichaux A, Begneaud W. Osteomyelitis pubis occurring after spontaneous vaginal delivery: a case presentation. J Perinatol 1997; 17 (4): 321–324.
Massive vulvar oedema in pregnancy is an uncommon presentation. Physiological activation of the renin-angiotensin system and the compression of the inferior vena cava by the gravid uterus contribute to lower body oedema. The loose connective tissue of the vulva makes this area particularly prone to expansion.1,2 Differential diagnosis for the initial presentation included intra-abdominal sepsis, septic arthritis of the hip and discitis, and only later was osteomyelitis pubis considered. Osteitis pubis is a sterile inflammatory condition of the symphysis pubis, the exact aetiology of which is often unclear. Athletic exertion, childbirth, surgical trauma and some rheumatological conditions have been implicated in its initiation.3 It is a self-limiting condition and managed conservatively.4 In contrast, osteomyelitis pubis is caused by infection5 through either direct or haematogenous spread. Risk factors include recent urological surgery, direct or contiguous trauma, intravenous drug use, diabetes mellitus and an immunocompromised state.6,7 As in our patient, Staphylococcus aureus is the most common pathogen, although Pseudomonas aeruginosa and polymicrobial infection with faecal flora have also been implicated.8 It is important to remember that cephalosporins are ineffective against Enterococcus sp. therefore ampicillin is a better choice in ‘triple antibiotics’ for suspected intra-abdominal sepsis. Both osteitis and osteomyelitis pubis can result in long-term complications and morbidity.9 Timely diagnosis of infection is paramount as delayed treatment may result in bony destruction, diastasis of the symphysis, chronic disability and one case reported death in pregnancy.7 With early and adequate treatment with antibiotics and often surgical debridement, the prognosis is good.5
2010 The Authors
Australian and New Zealand Journal of Obstetrics and Gynaecology 2010 The Royal Australian and New Zealand College of Obstetricians and Gynaecologists; 50: 573–581