Article Type : Case Report
Authors : Kaur B and Fayyaz S
Keywords : Pyosalpinx; Hysterosalpingography (HSG); Infertility
We present a case where a young female in
an outside fertility clinic was worked up for infertility and underwent
hysterosalpingography. She subsequently landed up in distressed state and was
brought to our tertiary care hospital in as an emergency .We stabilized the
patient and then performed Diagnostic laparoscopy which revealed iatrogenic
pyosalpinx. The case is intended to make readers aware of the fact that every
procedure – diagnostic or therapeutic has inherent risk and therefore mandates
that the doctor makes a judicious decision weighing the advantages,
disadvantages, material risk involved in a case to case basis so that
iatrogenic complications are minimized in clinical practice if not eliminated.
The changing socioeconomic demographics of 21st
century have brought changes in life style, better awareness for women
education, career needs and have resulted in increased age of marriage. On the
other side of coin, late marriages with changing priorities and more focus on
career has resulted in increased pool of couples seeking advice for infertility
related issues. The added pressure from families for bearing children prompts
couples to approach infertility centers or gynaecologists to facilitate quick
conception. The mushrooming In vitro fertilization (IVF) clinics in fierce
competition with their peers broadcast their achievements by sharing reviews
and success stories of couples who received treatment resulting in quick
conception. The stake holders – patients, gynaecologists or IVF centers finally
are quick to resort to use of diagnostic modalities like Hysterosalpingography
(HSG) without assessing the pros and cones. The present case is a glaring
example where a young female undergoing work up for infertility underwent HSG
in an outside infertility clinic and eventually landed up in distressing
situation. She came to our tertiary care hospital in distressing situation and
after stabilized underwent Diagnostic laparoscopy at our centre revealing
pyosalpinx most likely an iatrogenic one. The case is intended to make readers
aware of the fact that every procedure – diagnostic or therapeutic has inherent
risk and therefore mandates that the doctor makes a judicious decision weighing
the advantages, disadvantages, material risk involved in a case to case basis
so that iatrogenic complications are minimized in clinical practice if not
eliminated.
A 27 year old married female PL1 presented to our
emergency with complaint of constant pain in lower abdomen (left side) associated with fever, nausea and vomiting for last 2 days.
She had undergone intrauterine insemination (IUI) at an outside center 3 days
back and thereafter had complaint of left sided abdominal pain. The pain was of
constant nature, non-radiating and was not relieved even by IV analgesics.
Prior to her visit in emergency she also had high grade fever 102 F0 associated
with chills and rigors. She was prescribed oral antibiotics and anti pyretics
by local physician. The medical history also revealed that she had an ovarian cyst 5 years back which was removed
by laparoscopic cystectomy , however no details of histopathology report was
available on record. She had a normal delivery a female child, now 7 years of
age. The history also revealed that she was seeking consultation from a
gynecologist for infertility. She underwent HSG a month back and thereafter had
abdominal pain. The HSG showed right cornual block with left side Fallopian
tube dilated, tortuous with minimal spill (Figure 1). Her gynaecologist
prescribed pain killers but there was no respite. Subsequently in her next
menstrual cycle she underwent IUI which further worsened her pain with no
respite even after taking IV analgesics and oral antibiotics, eventually landed
up in emergency service of our hospital. On examination she was alert, in pain,
febrile with temperature 1040 F, dehydrated, Pulse-120 /min, Respiratory rate
-20, Blood Pressure-110/70mmHg, with pain score of 7-8 (on Visual Analogue
scale). There was no pallor, icterus, clubbing, cyanosis or pedal edema. The
breast examination was normal and no lymphadenopathy noted. On examination
abdomen was soft without distention having normal bowel sounds but there was
tenderness on left lower quadrant of the abdomen. Per vaginum examination
revealed discharge from cervix, cervical motion tenderness was present with
fullness in left fornix. Her ultrasound report done a day back was suggestive
of left adnexal mass.
On examination she was having pain in abdomen, alert and dehydrated. Her vitals were temperature -1040 F, Pulse rate-120 /min, Respiratory rate -20/ min, BP-110/70mmHg and pain score - 7-8 (on Visual Analogue scale). There was no pallor, icterus, clubbing, cyanosis or pedal edema. The breast examination was unremarkable and there was no lymphadenopathy or hepatosplenomegaly. On P/A examination abdomen was soft without distension with normal bowel sounds however there was tenderness in left lower quadrant of the abdomen. Per vaginum examination revealed discharge from cervix; cervical motion tenderness was present and fullness in left fornix. She had an ultrasound report done a day back which was suggestive of left adnexal mass. The patient was admitted and preoperative work up was done. She had O+ve blood group. An automated complete blood count (CBC) demonstrated Hemoglobin- 112 g/L (reference range 130-170 g/L), white blood cell count 18.22 x 109 /L (reference range 4-10 x 109 /L) Platelet count 650 x 10 9/L (reference range 150-450 x 109/L) and ESR 30 mm per hour. The laboratory investigations revealed-BUN- 5 mg/dl, Creatinine -0.6 mg /dl, Sodium 136 m mol/liter, potassium 3.9 m mol/liter, chloride 99 m mol/lit, SGOT- 22U/L,SGPT 3 U/L and PTT 15.4 seconds (INR – 1.28). C reactive protein- 8 mg/dL, CA-125 -139 U/ml, AFP-<1.58 ng/ml, LDH -218U/L, B-HCG -<2.39 mIU/mL, CEA-3.58 ng/ml and thyroid profile was TSH-2.49 mcIu/ml, T3 -1.10 ng/ml & T4-16.7 mcg/dL. Test for COVID-19, HIV 1 & 2, Hepatitis B and C viral serology were non-reactive. Malarial smears and rapid malarial antigen test were negative. Routine urine examination did not detect any abnormality. ECG and chest X- ray were unremarkable. The urine culture and high vaginal swab reports were negative. The work up and battery of investigations led to provisional diagnosis of inflammatory pathology. The patient was given Intravenous antibiotics Inj Fortum 1 gm 8hourly, Inj Amikacin 500 mg IV 12 hourly and Inj Metrogyl 100ml IV 8 hourly, intravenous antacids and analgesics. The marker CA-125 was elevated but the values were lower to indicate any ovarian neoplasm. It was decided to perform diagnostic laparoscopy for assessment and deciding further course of operative management. An informed legally valid consent was taken from the patient and her husband explaining about the possibility of opening the abdomen, removal of left side tube and ovary.
Diagnostic laparoscopy was performed. A 5mm
laparoscope was inserted after creating pneumoperitoneum in supraumblical
region. It revealed a distorted pelvic anatomy with a big loop of intestines
engulfing left adnexal region which was densely adhered to left adnexa (Figure
2,3). The intestinal loops were bordered medially with left lateral surface of
uterus, superiorly with round ligament and laterally with lateral pelvic wall.
Another loop of intestine was densely adherent to the posterior surface of the
uterus restricting its mobility. A thick edematous right fallopian tube was
visible on the right side the remaining view of right side ovary was totally
obscured due to dense adhesions, slowly by sharp and blunt dissection right
fallopian tube was lifted up by putting a suction cannula in a space beneath
the right fallopian tube (Figure 4), lifting the tube made the visualization of
right ovary possible (Figure 5). The left round ligament was identified (Figure
3) and millimeter by millimeter dissection was done to remove the adherent
bowel loops adherent to left adnexa till copious amount of pus came out of
this left side pseudo sac engulfed by left side
tuboovarian tissue and intestinal loops
(Figure 6). The left side tube was swollen and was dilated about four times its
normal size, inflamed and partly necrotic
and copious amount of foul smelling pus started coming out from the tube and the sac formed
by intestinal loops around the left fallopian tube (Figure 7). The left side
tube was removed and sent for histopathological examination. Pus was seen all
around the field, the right side tube was also found swollen, enlarged and
inflamed .Removal of left fallopian tube cleared the view of the left ovary.
The left ovary though normal looking was embedded in the ovarian fossa and no
attempt was made to mobilize as the tissues were friable. The chromopertubation test was performed but
the right side tube had cornual block. As the patient was under the effect of
anesthesia and was not in a state to give legally valid consent for removal of
her right tube, it was retained and not removed. The pus was removed and sent
for culture. Repeated washing of the operative field was done there were no
intraoperative complications. At the end of the procedure Uterus was made free
from the adhesions to bowel loops and mobilized, right side tube and ovary
retained, left side ovary retained. Haemostasis was ensured and patient stood
the procedure well. Postoperatively IV Antibiotics were continued, antacids and
analgesics were continued. On day 2 of procedure the patient’s hydration
improved, she was a febrile and was relieved of pain (visual analogue score of
2). She was started on oral liquids on
day 2 and on semi solids on day 3 of the operative procedure. She passed urine
adequate urine and motion on day 3. There was no growth after 48 hours
incubation from the culture of pus the patient was discharged on 6th
postoperative day after giving her 7 days of IV antibiotics. On discharge she
was ambulatory, having regular bowel and bladder movements a febrile and free
of pain. She came for review after 7 days of her discharge and was apparently
happy about being managed appropriately. The histopathology report of left
fallopian tube showed an inflamed, swollen, necrotic edematous tube with evidence
of pus. She was advised to go for IVF and not attempt for natural conception
because her one tube left was also blocked.
The infection of the fallopian tubes also known as
Pelvic Inflammatory disease (PID) is associated with major health implications
[1]. PID frequently develops among sexually active women in reproductive age
group [2,3] When the genital organs of young females get infected by
either of these microorganism N.
gonorrhea, C trachomatis , mycoplasma genitalium, bacterial vaginosis and
anaerobes. The infection with these microorganisms have been implicated with
PID , which leads to irreversible damage to fallopian tubes and complications
like hydrosalpinx, pyosalpinx, ectopic pregnancy or infertility that may be
difficult to treat by medical or surgical methods [4,5]. The diagnosis of PID
and its sequel can pose challenge because the clinical manifestations may mimic
other pelvic or abdominal conditions. Non-invasive diagnostic modalities like
ultrasound, CT scan or MRI are sensitive but laparoscopy remains the modality
or choice and gold standard of diagnostic criteria [4]. A pyosalpinx is the
acute inflammation of the fallopian tube that fills up and swells with pus. It
is a serious complication from untreated or inadequately treated acute pelvic
inflammatory disease associated with high morbidity. Pyosalpinx may present
with very few specific symptoms. The most common presenting symptom is lower
abdominal pain [6]. Other symptoms include fever, nausea, vaginal discharge and
abnormal bleeding. On physical examination, patient may show tenderness over
the adnexal region with or without guarding or rebound. The absence of specific
symptoms and conclusive signs during the physical examination may delay a
proper diagnosis. This typical presentation was also given by index case.
Predisposing factors in the development of pyosalphinx
include sexual activity, multiple sexual partners, nulliparity, previous
episodes of PID, low socioeconomic status and use of intrauterine device [7,8].
In our case the cause of pyosalpinx is exacerbation of PID due to iatrogenic
procedures like HSG ( Hysterosalpingography)
and IUI (intra uterine
insemination) in the absence of treatment of ongoing PID. This is actually a
cause of concern as these procedures performed in the absence of treatment of
underlying PID lead to morbidity and complications. The dilemma in our case is
that the results of her HSG were indicative of proximal tubal obstruction on
left side and dilated, tortuous left fallopian tube with minimal spill (Figure
7). This implies that the left tube had hydrosalpinx with distal tubal
occlusion thereby explaining why the pyosalpinx manifested clinically after the
HSG and then subsequent IUI. The diagnosis of PID and its sequel can pose challenge
because the clinical manifestations may mimic other pelvic or abdominal
conditions. Non-invasive diagnostic modalities like ultrasound [9], CT scan or
MRI are sensitive but laparoscopy remains the modality or choice and gold
standard of diagnostic criteria [4]. There is often an overlap between
appearances of another diagnosis, including ovarian torsion, endometriosis,
hemorrhagic cyst or other ovarian cystic masses. In our case the CT report with
high CA-125 levels gave an erroneous perception of ovarian mass. Besides the
diagnostic modalities thorough history and clinical examination in combination
with laboratory investigations can help in clinching the diagnosis. Treatment
of pyosalpinx varies from conservative management with IV antibiotics and possibly
diagnostic laparoscopy followed by salphingectomy or salphingostomy [10].
Diagnostic laparoscopy followed by removal of pyosalpinx / salphingectomy
remains the gold standard treatment. In our case too diagnostic laparoscopy
followed by left pyosalpinx removal or salphingectomy with drainage of pus was
performed followed by intravenous antibiotics were sufficient for complete
resolution. A randomized clinical trial in 2008 demonstrated that drainage of
pus with IV antibiotics resolved the symptoms faster than IV antibiotics alone.
We have reported a rare case of pyosalpinx following
HSG which was suggestive of hydrosalpinx and partial occlusion followed by IUI
procedure in next cycle. Attempts should be made to get HSG done after proper
work up of the patient for PID. Vaginal culture followed by appropriate
antibiotics should be started prior to procedures like HSG. Diagnostic
laparoscopy instead should be encouraged as modalities for assessment of tubal
patency for Infertility workup.
Nil
Sources of
funding
Nil
The authors have taken written consent from patient
for publishing the case for academic purpose. Anonymity of patient
identification maintained.