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Bilateral Pyosalpinx as A Rare Cause Of Pelvic Cystic Lesions In A 12-Year Old Girl

Carolin Schmieg¹, Markus Ebert², Bertram Reingruber¹, Christian Knorr¹

¹Clinic for pediatric surgery, Klinik St. Hedwig, Barmherzige Brüder Regensburg, Regensburg, Germany

²Clinic for pediatric radiology, Klinik St. Hedwig, Barmherzige Brüder Regensburg, Regensburg, Germany

 

Abstract

Pyosalpinx is a rare condition in a 12-year old virgin female patient. We report a case caused by Escherichia coli infection. A history of previous laparotomy may have contributed to the salpinges dysmotility. As possible causes we discuss infective and postsurgical complications. Reports of association with Hirschsprung’s disease are reviewed focusing on similarities to our case.

Keywords: pyosalpinx, adolescent, laparoscopy, surgical complication, pelvic cyst

 

Correspondence

Christian Knorr
Klinik St. Hedwig
Steimnetzstr. 1-3
93049 Regensburg
Germany
Email: This email address is being protected from spambots. You need JavaScript enabled to view it.

 

Introduction

Pelvic inflammatory disease (PID) can affect the inner genital tract entirely or only parts of it, as for example in pyosalpinx. Pyosalpinx is an ascending inflammation of the adnexa with retention of purulent fluid. Most common causing germs are Neisseria gonorrhoea, Chlamydia trachomatis and enteral bacterias which commonly ascend through vagina and cervix in a sexually active patient. The typical clinical presentation consists of lower abdominal pain and clinical signs of infection.

Case report

A 12-year old girl was referred to our hospital by her family physician suspecting bowel obstruction due to postoperative adhesions as the cause for her acute abdominal pain. On admission she presented with a 2 day history of lower abdominal pain and nausea. Vomiting has occurred only once; last bowel movements were present 2 days ago and she had no fever. The girl had the debut of her menses half a year ago. Upon admission she had her regular period. No previous sexual activity was stated and the hymen was intact.

At the age of 18 months open appendectomy had been performed through a low median-line incision due to a perforated appendicitis. Furthermore, occasional slight abdominal pain and a disposition to constipation were related by the patient.

On physical examination the girl appeared in relative good condition. Pain on palpation was present in the lower part of the abdomen with no muscular guarding. There was no palpable abdominal mass or hernia of the abdominal wall. Bowel sounds were present. The rest of her physical exam was unremarkable.

On abdominal ultrasound, minimal amount of free fluid was seen in the right lower abdomen next to an inhomogeneous echogenic 7cm x 9cm size cystic structure without signs of inflammation. This tubular structure was directly adherent to the dorsal aspect of the ovaries, with no peristaltic movements, although a fluid level could be seen. The presumed imagistic diagnosis of this structure was a fixed bowel loop or a teratoma. Figure 1 shows the ultrasound image of the structure with no evident fluid levels.

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Figure 1. Ultrasound of lower right abdomen showing an inhomogeneous cystic structure

Magnetic resonance imaging with contrast substance confirmed the previously described image: a well-defined tubular structure, with inhomogeneous filling and fluid levels without signs of inflammation as shown in Figure 2.

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Figure 2. MRI aspect in sagittal section

Furthermore, initial lab results showed a slightly elevated white blood cell count of 14.4 103/ μl. C-reactive protein was elevated up to 151.1mg/l. Plain abdominal X-ray excluded ileus. The patient started to have spontaneous bowel movements with enemas. Abdominal pain subsided thereafter. Intravenous treatment was started and the patient was kept NPO for re-evaluation while on standby for surgery.

Bilateral ovarian teratoma was the suspected preoperative diagnosis. Tumor markers: alpha-feto-protein and beta-HCG were negative. After an interdisciplinary discussion of the case (pediatric oncology, pedatric surgery, radiology, gynecology) we decided to perform diagnostic laparoscopy and to explore the lesion. Laparoscopy showed adhesions in the upper abdomen possibly due to former appendectomy. Upon exploration of the lower abdomen, adnexa were adherent to the sigmoid colon. Solid structures of unclear anatomical correlation were found on both sides.

At this time, conversion to laparotomy was decided for reasons of surgical (anatomical structures were distorted) and oncological safety. Figure 3 shows the intraoperative finding in the right lower abdomen. 

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Figure 3. Tubular-cystic structure in the right lower abdomen.

Intraoperatively, during mobilisation, left adnexa discharged purulent fluid. The findings were demonstrated to the attending gynaecologist, who confirmed the diagnosis of bilateral pyosalpinx. Microbiological sampling, incision and lavage of the adnexa, adhesiolysis, intraabdominal drain placement and abdominal closure were performed.

Escherichia coli was detected in the microbiological fluid samples. The patient was treated with intravenous antibiotics for 5 days and then continued with oral treatment for further 5 days. The patient was discharged on the sixth postoperative day after removal of surgical drains, having normal GI function and negative infectious parameters.

At gynaecological examination 10 days post-surgery the patient presented in good general health without any complaints and no noticeable problems. On 10 month follow-up the patient states a good physical condition without any further abdominal complaints and a regular menstrual period.

Differential diagnosis of pelvic cystic lesions

In general, tumors of the female genital tract can be classified into true cystic lesions and solid tumors; however, solid tumors may have a cystic appearance. Follicular cysts are the most common tumor lesions with a frequency of 35% of the total tumor lesions. Out of the further 65%, one third are malignant [1]. Table 1 shows the most common cysts. Massive tumors are divided into benign and malignant neoplasms as shown in table 2. They may have cystic and solid parts. Dilemma of cystic lesions is that ovarian cysts especially those with diameters above 5 cm have a tendency to cause torsion of the ovary or adnexa.

Table 1. Cystic lesions of the female genital tract and their distribution of frequency

type of cyst

distribution of frequency

  • Follicular cyst 

50%

  • Corpus luteum cyst
  • Endometrioid cyst
  • Paraovarian cyst

50% combined

Table 2. Neoplasms of the ovary, their dignity and frequency. (modified after [2])

 

type of tumor

Malignancy

Frequency

germ cell tumors

  • mature teratoma
  • immature teratoma
  • dysgerminoma
  • mixed tumors

 

benign

malignant

malignant

malignant

67%

stromal sex-chord tumors

 

13%

epithelial tumors

benign/malignant

17% (adults 65-80%, not seen in prepubescence)

gonadoblastoma

malignant

rare, but risk of to 25% of development in dysgenetic gonads

Imaging and the appropriate surgical approach

Any suspicion of an ovarian torsion requires immediate surgical inspection and derotation to prevent possible further damage to the ovary. Chronic or gradual onset symptoms require further investigations such as ultrasound and lab tests with hormone analysis to distinguish the most probable causes.

Ultrasound is superior to MRI/CT in cases of ovarian torsion. Laparoscopy is the preferred surgical approach in painful simple ovarian cysts larger than 5cm in diameter. Lesions with multiple cysts and solid parts are highly suspicious for neoplasms. Exploratory laparoscopy may give useful information but in case of resection, it must meet the safety standards in case of malignancy and the threshold to conversion should be low.

Causes

The somewhat unexpected intraoperative finding of a bilateral pyosalpinx is extremely rare in sexually inactive patients such as ours. If sexual activity is ruled out, what else could be the cause for this condition? Taking into consideration infection as a possible cause, there have been a few reported cases of pyosalpinx in virgins caused by pneumococcus, streptococcus or shigella pathogens [3-6]. Urogenital malformations are a known cause for pyo- and hydrosalpinx at menarche in the affected females [7].

In the literature, an association of Hirschsprung’s disease with pyosalpinx has been previously mentioned. Merlini et al. report about three cases of adolescent girls with a bilateral hydrosalpinx and a Hirschsprung’s disease in their prior medical history [8]. The author suggests as possible causes either postsurgical complications or congenital defects of innervation in the context of aganglionosis which may also affect the fallopian tube. Desai et al.[9] also report about this association in one case.

Stavroulis et al. [10] found endometriosis as a cause for bilateral pyosalpinx in a girl. In our case, endometriosis was ruled out by MRI and the intraoperative findings and she did not have a history of Hirschsprung’s disease.

A common feature with the previously described Hirschsprung patients is that our patient also underwent surgery in early childhood. May adhesions be the predisposing common factor?

The reported Hirschsprung patients underwent a Duhamel, Soave (2 patients) and a Martin’s procedure [8,9]. One of the patients with Soave had incidental appendectomy [9] while our patient had surgical indication for open appendectomy.

As Merlini et al. already stated, it is unlikely to be a direct postoperative complication [8], but undeniably it is a possible common condition in these rare cases. In rabbits, loss of adrenergic innervation causes hydrosalpinx [11]. In humans, the fallopian tube is innervated by a nerve complex of the mesenteric, renal, uterovaginal and inferior hypogastric plexus. Intraoperative damage of its innervation might result in diminished activity of the moving ciliated epithelia causing an accumulation of secretion resulting in hydrosalpinx.

Contradicting the hypothesis of an exclusively postsurgical complication is its frequent association with innervation anomalies as cited in the literature. Thus, the combination of preexisting malformations together with a history of surgical intervention, may be necessary to set off the so-called postsurgical Hirschsprung complications [12,13]; this is further supported by the fact that pyosalpinx so far has not been reported in girls with imperforated anus [8].

Conclusion

In conclusion, the pyosalpinx in the case of our patient was caused by an E.coli infection. Whether a hypothetic dysmotility of the fallopian tubes secondary to early childhood appendectomy may have contributed to the pathology is unclear. However, it has to be considered as a possible coexisting condition in the rare appearance of bilateral pyosalpinx in a sexually inactive female.




References

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