Main Text:
Introduction: 30-years-primigravida-patient came in emergency
of our tertiary care Institution with ‘on and off’ per-vaginal (P/V)
bleeding for 1-week following amenorrhea for 2-months duration with
positive urine pregnancy test. She was mildly anemic and her vitals were
stable. Per-speculum (P/S) examination, cervix was bluish in color,
ballooned-up with dilated cervical Os and minimal bleeding from the
cervix was seen. P/V-examination revealed soft, bulky cervix and 8-weeks
size uterus. Hemoglobin was 9.5g/dL and β-HCG was 45667 mI U
/mL.Thyroid, liver and renal functions tests were within normal limits.
Ultrasonography revealed multi-cystic mass measuring~
55x48x44 mm size involving lower uterine segment and cervix with
surrounding increased vascularity suggesting molar pregnancy (Fig 1,2).
Materials and methods: Injectable methotrexate 1mg/kg IM with
Injection leucovorin 0.1mg/kg IM on alternate day was given for 1 week.
Her β-HCG levels decreased upto 157mIU/mL after 1-week but bleeding P/V
persisted. Patient had heavy bleeding for 3-days after completion of
chemotherapy, so emergent suction and evacuation was done but brisk
bleeding persisted.20 mL diluted vasopressin was injected
intra-cervically but bleeding did not cease. 4-units-PRBC was transfused
and intra-cervical foley’s balloon was inflated with 30mL of normal
saline and placed for tamponade effect but bleeding still continued.
Histology section showed diffuse villous enlargement with marked
hydropic changes and proliferating extra-villous trophoblast, consistent
with complete hydatidiform mole. After approval by institutional ethics
committee and obtained written informed consent from patient and her
attendants finally, trans arterial bilateral uterine arteries
embolization (UAE) was planned with modification of conventional UAE
technique called as trans-arterial-chemo-embolization in gestational
trophoblastic diseases (gTACE). Injectable methotrexate soaked, mixed
and agitated with gelfoam “slurry” as temporary embolic agents were
supereselectively injected into offending branches of bilateral uterine
arteries to manage immediate ongoing bleeding and to traet cervical
molar pregnancy simultaneously. This modified novel technique has
successfully managed the ongoing torrential life threatening hemorrhage
and preserved the uterus for menstruation and future fertility. Digital
subtraction angiography (DSA) of pelvis was done on either side through
® trans-femoral route to identify the offending vessels on each side
(Fig 3A, 3B).We super-selectively cannulated each uterine artery with
4F/5F SIM-1 catheters; and if needed progreat microcatheter (Terumo
corporation, Tokyo, Japan). Gelfoam “slurry” was prepared after
slicing the gelfoam pad into small particles with 11 no. surgical blade
and gelfoam particles were mixed with 3mL of iodinated contrast media.
After that, the gelfoam “slurry” was mixed and agitated with
injectable 2mL of methotrexate drug (50mg). Vigorous mixing of all three
materials were done through triway attached with two leur-lock syringes
of 10mL capacity and injection made through previously placed 4F-SIM-1
catheter alone or progreat microcatheter co-axially placed through
5F-SIM1 catheter at intended site of embolization till adequate
thrombosis of offending vessels were achieved in bilateral uterine
arteries (Fig 4A, 4B).
Results: Ongoing bleeding was ceased. Procedure was uneventful.
Immediate post procedure Duplex-US revealed maintained slow flow
vascularity within uterine myometrium in fundal and body regions, and
bilateral adenexae and ruled out possibilities of uterine infarction.
Prophylactic broad-spectrum antibiotics, antiemetic, H2-blocker and
analgesic medications were advised for 5 days to avoid post embolization
syndrome and sepsis.Patient was discharged on day 6thin satisfactory condition with β-HCG value of 39.23 mIU/mL and
Hemoglobin of 10.7 gm/dl. Patient was advised to avoid pregnancy for
1-year and was followed-up with serial β-HCG weekly monitoring till
normal level (<5 mIU/mL) which took 5 weeks.Cervical mass
which was significantly reduced in size after procedure and was
gradually disappeared after 5-months (Fig 5).
Discussion: First time, arteriography and infusional
chemotherapy with methotrexate / dactinomycin drug in 8 cases of
gestational trophoblastic diseases was reported by Marqulis GB et al in
19751.Cervical molar pregnancy is very rare
entity2.Fertility preserving management is difficult
in presence of torrential life threatening
haemorrhage3. Usually, vascular recanalization occurs
between 2-weeks to 4-months duration with gelfoam as temporary
embolic-agent hence this procedure effectively maintains immediate
hemostasis, normal menstruation and future fertility4.
After extensive literature search and our knowldege,
trans-arterial-chemo-embolization (TACE) was first time modified and
used in gestational trophoblastic diseases (GTD) hence named as
“gTACE” which is a minimally invasive procedure performed under
DSA-guidance to cause mechanical thrombosis of offending vessels,
ischemic necrosis and cytocidal effect on tumor cells simultaneously for
longer duration. Gelfoam sponge as temporary embolic-agent attracts
platelet aggregation and causes acute panarteritis and mechanical
thrombosis. Injectable methotrexate drug soaked and mixed with gelfoam
sponge “slurry” releases slowly for longer duration of its targetted
cytocidal action within the tumor mass as compared to methotrexate drug
infusion alone hence a chance of systemic side effects of methotrexate
drug is also markedly reduced and abnormally high plasma level of
Beta-HCG secreted by abnormal cytotrobhoblast and synciotrophoblastic
cells were ceased.Beta-HCG level falls within normal limits in shorter
duration. “Slurry” made by mixing 3mL of non-ionic iodinated contrast
media (350mg/dL) was able to visualize flow of injectable methotrexate
mixed embolic materials on DSA-fluoroscopy.
Conclusions :This modified novel technique was succesful in
management of rare cervical molar pregnancy and its related
complications.We can termed this technique preferably as ‘gTACE’ as
promising alternative management technique in gestational trophoblastic
diseases and its related complications as life saving, future fertility
and uterine preserving measures.
Acknowledgement: We sincerely acknowledge our thanks to the
patient and her attendants for giving written and informed consent.
Conflicts of interests: All authors have no relevant financial,
personal, political, intellectual or religious conflict of interests.
Contribution to authorships : Kumar manoj and Neera Kohli have
conceptualised, carried out and planned this procedure and helped in
writing the manuscript.Manju LATA Verma, Uma Singh, Rekha Sachan and
Pushp Lata Sankhwar have done pre and post procedure clinical work-up
and searched the literature.
Details of ethical approval: There is no need of ethical
approval for a single case as per our institutional ethics
committee/IRB.However, written informed consent has been taken from the
patient and her husband.
Fundings: Nil.