Recurrent Pyogenic Cholangitis
A 59-year-old South Asian female presented with the complain of pain
upper abdomen and recurrent fever for one year. There was no history of
vomiting, jaundice, anorexia or weight loss. Family history was
unsupportive of diagnosis. On laboratory examination, alkaline
phosphatase was elevated by two times. Serum bilirubin was normal and
her leukocytes count was 11200/mm3. Rest of the
systemic examination was normal. Contrast enhanced computed
tomography(CECT) revealed multiple hypodense/hyperdense lesion in
segment 3 of liver. Magnetic resonance imaging(MRI) showed
hypo-hyperdense focal lesions with conglomerate appearance on segment 3
of liver suggestive of hepatolithiasis (Figure 1). Left lateral
segmental resection (Figure 2) and intrahepatic choledochoscopy from
dilated left hepatic duct and stone clearances was done. On
histopathological examination, definitive diagnosis of recurrent
pyogenic cholangitis was established with no evidence of malignancy. At
a 3-year follow-up, the patient is free of recurrence.
The incidence of hepatolithiasis is variable. It is highly prevalent in
parts of Asia, such as China, Japan, and South Korea, with a reported
incidence between 3.1% and 21.2%.1 However, with an
incidence of about 1%, hepatolithiasis is rare in Western countries.The
mechanism of development of hepatolithiasis is yet to be fully
elucidated. Cholestasis, cholangitis, an anatomical abnormality of the
bile ducts, abnormal bile metabolism, malnutrition, and low
socio-economic status are significant risk factors for hepatolithiasis.
According to several studies, indications of hepatectomy for
hepatolithiasis include the following instances: (i ) unilobar
hepatolithiasis, and particularly that on the left; (ii ) atrophy
or severe fibrosis of the affected liver segments or lobe; (iii )
presence of a liver abscess; (iv ) cholangiocarcinoma; and
(v ) multiple intrahepatic stones causing marked biliary stricture
or dilation.2 Globalization, apart from changing the
socioeconomic status of regions, has additionally altered disease
dynamics globally. Hepatolithiasis, as a result of recurrent pyogenic
cholangitis, although still rare, is becoming progressively evident in
the West because of immigration from the Asia-Pacific region, where the
disease is endemic. Such rare but emerging disease has imposed
significant challenges to the physicians and surgeons. Uenishi et
al.3 presented outcomes for 86 patients who underwent
a hepatectomy from 1998 to 2012. Seventy-six patients (88%) had
immediate stone clearance whereas 82 patients (95%) had final stone
clearance.3 A multi-disciplinary team approach
involving radiologists, internists, pathologists, oncologists, and
surgeons is deemed necessary for the timely diagnosis and safe,
affordable treatment, thus ensuring good prognosis to the patients.
Low socio-economic status of our patient could have been the attributing
factor. Also, unilobar hepatolithiasis, and particularly that on the
left was the main indication for left lateral segmental hepatectomy in
our case. With this image, we would like to recommend that the
differential diagnosis of hepatolithiasis should be thought in the back
of mind if an elderly person from endemic region presents with the
complain of pain upper abdomen and recurrent fever. Surgical
intervention is deemed necessary with the aforementioned indications and
it can provide good prognosis to the patient with an excellent stone
clearances.
Conflict of interests: None
Consent: Written informed consent was obtained from the patient
for the accompanying images.