Unusual presentation of more common disease/injury

Pregnancy complicated by abdominopelvic

hydatid disease
Shakun Tyagi, Chanchal Singh, Reva Tripathi, Yedla Mala

Department of Obstetrics and SUMMARY was taken and the patient was put on tablet
Gynaecology, Maulana Azad A 22-year-old second gravida presented with Albendazole 400 mg daily for 6 weeks. However it
Medical College, New Delhi,
India
asymptomatic abdominal and pelvic hydatid disease required discontinuation after 4 weeks due to neu-
at 16 weeks gestation. She opted for conservative tropenia. Neutropenia resolved spontaneously on
Correspondence to management and was treated with oral Albendazole. discontinuation and albendazole was restarted after
Dr Chanchal Singh, She underwent elective caesarean along with cyst 2 weeks for another 6 weeks’ duration. During this
[email protected]
excision at term as the large pelvic cyst precluded period she received routine antenatal care. Her
vaginal delivery. A healthy baby girl weighing 2600 g leukocyte counts and biochemical profile remained
with Apgar of 9, 9 at 1 and 5 min was delivered. normal throughout. The fetus showed normal
growth and she did not develop any obstetric
complications.
BACKGROUND
Hydatid disease caused by Echinococcus granulosus OUTCOME AND FOLLOW-UP
is common in tropical countries but it is rare in Near term the patient was reassessed to decide the
pregnancy with a reported incidence of 1 in 20 000 mode of delivery. Abdominal examination indicated
to 1 in 30 000.1–3 The presentation may range longitudinal lie with free-floating fetal head.
from asymptomatic disease to acute complications Bimanual examination revealed whole of pelvis
like cyst rupture, anaphylaxis and obstruction of occupied by cystic mass which was preventing
labour.1 2 4 Since the condition is rarely encoun- descent of fetal head. Repeat MRI (figure 1) was
tered in pregnancy, there are no standard guidelines done to assess the response of the hydatid cysts to
available for its treatment. medical therapy and to plan excision of resectable
cysts. MRI revealed disappearance of one of the
CASE PRESENTATION two cysts located anterior to kidney and calcifica-
A 22-year-old second gravida presented to the ante- tion of the subphrenic cyst. The pelvic cyst showed
natal clinic at 16 weeks of gestation for a routine a marginal decrease in size. The findings were dis-
antenatal visit. She had no significant medical or cussed with the patient and an elective lower
surgical history. On abdominal examination gravid segment cesarian section (LSCS) along with exci-
uterus of 16-week size appeared to be pushed ante- sion of hydatid cysts was planned in consultation
rolaterally to the right by a separate non-mobile with surgical team at 38 weeks gestation.
mass arising from the pelvis. On vaginal examin- A healthy female baby weighing 2600 g with an
ation the same cystic mass was felt posterior to Apgar of 9, 9 at 1 and 5 min was delivered by elect-
uterus as a bulge in the posterior fornix. Rectal ive LSCS at 38 weeks as planned. The uterine inci-
examination confirmed the mass to be anterior to sion was closed in double layer as per routine and
rectum. the uterus was exteriorised to improve visibility
and access to pelvic cyst in the pouch of Douglas
INVESTIGATIONS (figure 2). Packs soaked in scolicidal agent (10%
An ultrasound revealed a single live fetus corre- povidone iodine) were placed around the cyst
sponding to gestation. There was a large cystic during the procedure. The cyst was adherent to
lesion in the pelvis pushing the uterus anterolater- right ureter and posterior surface of the uterus and
ally. MRI confirmed multiple well-encapsulated required ureteric dissection in the ureteric canal.
multicystic lesions with internal daughter cysts and The cyst was removed intact without spillage.
matrices suggestive of hydatid cysts. The largest Exploration of the abdomen revealed another
cyst was midline in the pelvis (18×15×10 cm). flaccid cyst 6×8 cm on the under surface of liver.
Another cyst was found in right subphrenic region One more pedunculated right subhepatic cyst
(16×14×8 cm) and two in right lumbar region arising from greater omentum and adherent to the
(8×8×5 cm and 5×6 cm). Indirect haemagglutin- colon was excised. The lumbar cyst anterior to
ation test was positive for cystic echinococcosis. right kidney was left in situ due to its proximity to
The patient’s haemogram, blood sugar, liver and renal vessels. Postoperative period was uneventful
kidney function tests were normal. and the mother recovered well. Postpartum ultra-
sonography revealed calcified subphrenic
TREATMENT (12×12×6 cm) cyst and the cyst anterior to right
To cite: Tyagi S, Singh C,
Tripathi R, et al. BMJ Case
Management options including surgical removal of kidney (9×6 cm) in situ. Histopathology confirmed
Reports Published online: the cysts in second trimester, medical management the diagnosis of hydatid cyst. Patient was dis-
10 December 2012 vis-á-vis expectant management were discussed charged on tablet albendazole 15 mg/kg body
doi:10.1136/bcr-2012- with the patient. The patient declined surgery and weight daily for a further period of 6 weeks. She is
007880 opted for medical management. Physician opinion on follow-up and continues to be asymptomatic.

Tyagi S, et al. BMJ Case Reports 2012. doi:10.1136/bcr-2012-007880 1

 Unusual presentation of more common disease/injury

and may cause problems during labour. Management thus needs

to be individualised.
In this case the patient opted for conservative management.
The role of medical management in hydatid disease during preg-
nancy is at best limited. Albendazole is the drug of choice for
hydatid disease and is a useful adjunct to surgical removal.
Decrease in size and disappearance of daughter cysts with
medical therapy has been documented.6 Although albendazole
has been reported to be embryotoxic and teratogenic in animals,
inadvertent exposure of pregnant women to Albendazole during
mass drug administration for lymphatic filariasis showed no
increase in risk for gross congenital anomalies.7 As the period of
gestation was already 18 weeks when treatment was started,
teratogenicity was not a concern in our patient. Medical man-
agement did prove beneficial with disappearance of one cyst,
regression in the size of the other cysts and calcification of one
cyst. Moreover, there was no increase in the size of any of the
Figure 1 MRI showing multiple abdominal and pelvic hydatid cysts.
cysts.
To conclude, asymptomatic hydatid disease complicating
pregnancy presents with a management dilemma. Medical man-
DISCUSSION
agement with albendazole after the first trimester along with
Surgical treatment is the mainstay in management of hydatid
close monitoring for adverse drug reactions should be consid-
disease and surgery is individualised according to the number
ered in patients not suitable or not willing to undergo surgery.
and location of cysts, physiological condition of the patient and
Surgical excision of the cysts can be postponed and performed
the presence of complications such as cyst infection and
either postdelivery or simultaneously at the time of caesarean
rupture. Frequently the site of hydatid cyst may be such as not
delivery. This approach optimises both obstetric and perinatal
to have any impact on pregnancy, for example, liver, kidney and
outcomes along with definitive management of the hydatid
spleen.1 If the cyst is located in the pelvis, problems are likely to
disease.
manifest at the time of labour and delivery. There are earlier
reports of pelvic hydatid cysts presenting with symptoms requir-
ing urgent intervention.4
When patient presents with asymptomatic pelvic hydatid Learning points
disease during antenatal period controversy arises whether to
manage these patients solely on pharmacological therapy or ▸ Hydatid disease complicating pregnancy is rare with an
perform immediate cyst excision.5 Surgery during pregnancy incidence of 1 in 20 000 to 1 in 30 000.
may be associated with increased intraoperative morbidity due ▸ The disease has variable presentation ranging from
to poor manoeuvrability due to the gravid uterus and also poses asymptomatic disease to acute complications like rupture,
risk to the pregnancy in terms of miscarriage or preterm labour. anaphylaxis and obstructed labour.
On the other hand, if left in situ, the cyst may increase in size ▸ Treatment should be individualised taking into consideration
the site, size, number of cyst(s), the period of gestation and
the patient’s wishes.

Competing interests None.

Patient consent Obtained.

REFERENCES
1 Can D, Oztekin O, Oztekin O, et al. Hepatic and splenic cyst during pregnancy:
a case report. Arch Gynecol Obstet 2003;268:239–40.
2 Dede S, Dede H, Caliskan E, et al. Recurrent pelvic hydatid cyst obstructing labor,
with a concomitant hepatic primary: a case report. J Reprod Med 2002;47:164–6.
3 McManus DP, Zhang W, Li J, et al. Echinococcosis. Lancet 2003;362:1295–304.
4 Goswami D, Tempe A, Arora R, et al. Successful management of obstructed labor in
a patient with multiple hydatid cysts. Acta Obstet Gynecol Scand 2002;83:600–3.
5 Rodrigues G, Seetharam P. Management of hydatid disease (echinococcosis) in
pregnancy. Obstet Gynecol Surv 2008;63:116–23.
6 Morris DL, Dykes PW, Marriner S, et al. Albendazole—objective evidence of response
in human hydatid disease. J Am Med Assoc 1985;253:2053–7.
7 Gyapong JO, Chinbuth MA, Gyapong M. Inadvertent exposure of pregnant women
Figure 2 Intraoperative picture showing the large pelvic hydatid cyst to ivermectin and albendazole during mass drug administration for lymphatic
posterior to uterus. filariasis. J Rep Med Int Health 2003;8:1093–101.

2 Tyagi S, et al. BMJ Case Reports 2012. doi:10.1136/bcr-2012-007880

 Unusual presentation of more common disease/injury

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