Acute gonococcal Fitz-Hugh–Curtis syndrome: a case report

INTRODUCTION

In 1930 the violin-string appearance between the anterior hepatic surface and the anterior abdominal wall was first described in gonorrhoea patients. ¹ Later in 1934 gonococcal peritonitis accompanied by pain in the right upper abdomen was reported. ² This established the involvement of hepatic capsules in gonococcal peritonitis or gonococcal pelvic inflammatory disease, which came to be known as Fitz-Hugh–Curtis syndrome (FHCS) in honour of the two authors who established the aetiological relationship of gonococci in the clinical presentation of such patients. Neisseria gonorrhoeae remained the major cause of FHCS for nearly 50 years, but it was realized in the 1980s that Chlamydia trachomatis is in fact the most common cause of FHCS. ³ This may be attributed to more improved diagnostic facilities for C. trachomatis in recent decades. However, N. gonorrhoeae remains the other major aetiological agent responsible for FHCS. The syndrome usually occurs as a complication of pelvic inflammatory disease.

Cases of FHCS are largely under-diagnosed. Even gonococcal peritonitis without involvement of hepatic capsules is infrequently diagnosed. In a recent report from Japan, only six cases of gonococcal peritonitis were diagnosed in 20 years. ⁴ Recently, FHCS is more frequently diagnosed using radiological/imaging techniques. ⁵ However, such techniques have their own limitations. The gold standard is still the culture and isolation of the causative agent from FHCS relevant specimens to establish the diagnosis, so that the specific treatment may be instituted.

CASE HISTORY

A 32-year-old South Asian women working in Kuwait was admitted to the casualty department of Al-Adan-Hospital in January 2009 with complaining of acute abdominal pain, most severe in the right iliac fossa (RIF) of one day's duration. There was no history of vomiting, change of bowel habits and no urinary complaints. The pain was aggravated on coughing and body movements. There was no past history of diabetes or hypertension. She had been married one year four months previously but had never conceived. She had a past history of regular periods but these had been scanty and irregular after marriage. On clinical examination, she was conscious, oriented, pulse 120/minute and regular, temp = 38.2°C and BP = 100/64 mmHg. Generalized abdominal rigidity and tenderness were present but more marked in the RIF. Pelvic examination was normal. There was no vaginal discharge and the cervix was clear. Transvaginal sonography was also normal. A diagnosis of acute appendicitis was made and the patient was transferred to the ward and treated with intravenous and cefuroxime metronidazole and gentamicin intravenously. The patient did not improve and so underwent open appendectomy by gridiron incision. Operative findings suggested acute suppurative appendicitis and suppurative peritonitis. The right ovary was inflamed, suggesting acute oophoritis. Peritoneal pus was sent for culture and sensitivity and appendix for histopathological examination. Pus was drained and the peritoneum was washed and mopped with saline.

N. gonorrhoeae was isolated in pure culture from peritoneal pus on standard media, identified by standard techniques and confirmed by Gonogen 11 (New Horizons Diagnostics Corp., Columbia, MD, USA).The strain was found to be positive for beta lactamase production by the starch paper technique. It was sensitive to cefuroxime, cefotaxime, ceftriaxone, chloramphenicol, ciprofloxacin and erythromycin but resistant to penicillin G, ampicillin and tetracycline. Histopathological gross examination showed a swollen congested appendix and cut surface with obliterated lumen. Microscopic examination revealed normal appendicular architecture with reactive hyperplasia and wall showing diffuse infiltration by neutrophils and eosinophils with no evidence of neoplasm. Other laboratory results were as follows: haemoglobin 13.4 g/dL, leukocytes 13.7 (unit 10⁹/L, ref: 3.98–10.04), neutrophils 94% (ref: 34–70%), alanine transaminase 129 U/L (ref: 0–35), aspartate transaminase 103 IU/L (ref: 0–31) and bilirubin total 24 µmol/L (ref: 2–22). The serum pregnancy test was negative. High vaginal swab did not grow any bacterial pathogens. Blood culture (both aerobic and anaerobic) was sterile after five days incubation in the Bactec 9240 series blood culture system (Becton Dickinson, Sparks, MD, USA). After four days of treatment, the liver enzymes and bilirubin returned to normal levels. Serum mycoplasma IgG was positive but mycoplasma IgM was negative. Serum anti-HIV 1 and 2 and chlamydia IgG and IgM were all negative.

The patient was treated with ceftriaxone 1 g intravenous daily, which was continued for eight days. On follow-up after three months, her endocervical, urethral and Bartholin swabs did not culture any N. gonorrhoeae or other bacterial pathogens and she was asymptomatic. Advice was also conveyed to her husband to seek treatment for gonorrhoea.

DISCUSSION

There are a large number of clinical conditions in which a patient may present with acute abdominal pain, such as duodenal ulcer, gall stones, acute pancreatitis, acute cholecystitis, renal stones, acute appendicitis and a host of others. In women of reproductive age, the possibility of sexually transmitted infections, especially N. gonorrhoeae and C. trachomatis, and their complications such as acute salpingitis, acute oophoritis, peritonitis and acute FHCS should also be considered. In the present case, the patient is a young women who presented with acute abdominal pain which localized more in the RIF but had generalized abdominal tenderness and rigidity. The characteristic pain in the right upper quadrant (RUQ) of the abdomen was, however, missing in the patient, and the pain increased on body movement and cough reflexes. This indicated that the hepatic capsule was likely involved in the disease process. It is equally possible that severe pain due to acute appendicitis and acute oophoritis in RIF could have masked the RUQ pain. Moreover, in some cases, patients with FHCS present with pain in the lower abdomen only without the characteristic RUQ pain. ^5,6 Hepatic enzymes are normal or slightly raised in FHCS. ^7,8 In our case they were raised, and returned to normal levels soon after specific therapy was instituted.

CONCLUSION

The possibility of FHCS in a young woman presenting with acute abdomen, with or without pain in the RUQ should be kept in mind. Specific microbiological diagnosis is important in guiding the appropriate therapy. Follow-up of the patients is equally important so that there is no residual pelvic infection or re-infection to avoid future complications.