Fetus in Fetu in Lesser Sac: A Case Report

Pandey P.¹*, Kar P.K.²

¹Assistant Professor, Surgery, Govt. Medical College, Jagdalpur (Bastar) India 494001

²Professor and H.O.D., Dermatology and S.T.D., Govt. Medical College, Jagdalpur (Bastar) India 494001

ABSTRACT:

History: Sporadic reports have appeared since 1950. Incidence is 1 in 500,000 live births. Less than one hundred cases have been reported in literature across the globe.

Material and method: Seven year old tribal girl attended O.P.D. with huge lump in upper abdomen.

Findings: On exploratory laparotomy a huge cyst in lesser sac, a fluid resembling pus was aspirated. On opening the cyst, hair seen after draining the fluid. On further exploration, a fetus found.

Conclusion: Fetus in fetu is a rare and interesting entity. Presents as abdominal mass during childhood or infancy. Using current imaging modalities, it can be diagnosed preoperatively. Complete excision is curative and confirms the diagnosis.

KEYWORDS: Fetus in fetu, Teratoma

INTRODUCTION:

It remains controversial whether fetus in fetu is distinct entity or a highly developed teratoma. It seems that really the both entities may be at separate points a spectrum of the processes of twining. At one end of the spectrum is a monozygotic twin, next conjoint twins, fetus in fetu and lastly teratoma. Arbitrarily presence of vertebral column has been accepted as the criteria for calling a tissue "Fetus in Fetu (FIF) ".Sporadic reports have appeared since 1950. Incidence is 1 in 500,000 live births, with fewer than 100 reported cases worldwide (1). Less than one hundred cases have been reported in literature across the globe so far. Keeping in mind the above facts and as per recommendations of the WHO study group in 1957, that in order to obtain a comprehensive picture of a health problem / disease, only vital statistics is not sufficient, more and more such studies have to be undertaken, Garg Narendra K.(2) and Garg Narendra K.and Sharma A.B.(3) , the authors have under taken this study.

Case Report:

A 7 year old tribal girl presented with abdominal pain and irritability with distended abdomen, vomiting, pressure effect on urinary system, dyspnoea was came in the Surgical Out Patient Department (OPD) of Maharani Hospital, Jagdalpur (Bastar). On physical examination, a huge lump in upper abdomen was found. On Ultra sonography and Transverse Computed tomographic (CT) Scan, a cyst in upper retroperitonium of abdomen with internal calcification was found (Figure-01 to 09). She was later on admitted in surgical ward of the Hospital for further follow up.

Routine investigation and as per requirement special investigation was done. On exploratory laparotomy, a huge cyst in lesser sac was seen. On aspiration of the cyst, fluid resembling thick pus was found. On exploring the cyst, hair seen after draining the fluid. On further exploration, a fetus was found.

It represents an aberration of monozygotic twinning in which unequal division of the totipotential inner cell mass of the developing blastocyst leads to the inclusion of a smaller cell mass within a maturing sister embryo. Later on, the outer embryo develops in to baby while the inner cell mass dies and is mummified after a variable time of development.

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DISCUSSION

It remains controversial whether fetus in fitu is distinct enticity or a highly developed teratoma. It seems that really the both rntitis may be at separate points a spectrum of the processes of twinning. At one end of the spectrum are monozygotic twins next conjoint twins, fetus in fitu and lastly teratoma. Arbitrarily presence of vertebral column has been accepted as the criteria for calling a tissue fetus in fetu. Although fetus in fetu is classically defined as an itraabdominal parasitic twin, it can be seen in locations other than its usual site, the upper retroperitoneal region. Fetus in fitu cases were reported in intracranial, thoracic, scrotal, sacroccygeal and facial regions in the literature. These regions are also those where terstoms mostly occur (4, 5, 6, 7)

The most common site is upper retroperitoneal, which also found in the present case. Other unusual sites where fetus in fetu found are Cranial Cavity, Oral Cavity, Sacrococcygeal region, Scrotum.

Robert M et al (8) found on CT Scans of the abdomen a large mass in the left upper quadrant extending to the lateral and posterior aspects of the abdomen. Several areas of markedly increased attenuation consistent with bon were noted. In addition, there were large areas of low attenuation within the mass. Statistical manipulation demonstrated that the lower attenuation areas were in the range of fat and the lesion was diagnosed as a large teratoma.

Grosfeld Jay L.et al (9) on Pathological examination, gross examination of the fetus showed it to anencephakic. Two upper limbs buds were noted. Finger like projections were seen coming off the superior limb buds. A patent omphalomesentric duct was noted and on an X-ray of the fetus, a contrast study of the bowel demonstrated relatively well-developed bowel in what appeared to be the mid-gut. The bowel contained soft green, inspissated material. A spleen, pancreas and adrenal gland were also identified. The lower part of the fetus demonstrated lower limb buds and radio graphically showed a boney pelvis. Each lower limb bud showed a primitive femur, tibia, fistula and primitive foot and phalanges. Hair was noted on the back of the inferior segment. There was an imperforate anus and it was obvious that the hind-gut had not fully developed. A complete vertebral column was present. A small area with what appeared to be brain was also identified. The authors on microscopic evaluation demonstrated normal histological features of Jejunum, ileum and proximal colon. A small fragment of pelvic tissue resembling primitive prostatic acini among fibro muscular stroma was also noted. Skin sections showed hyperkeratosis with follicular plugging. The authors further found, large number of hair follicles, sweat glands and sebaceous glands were seen in the dermis. The gastro duodenal region and adjacent pan creating tissue contain acini and islet cells were identified. The adrenal gland demonstrated normal cortex and few medullary cells. The vertebral column showed irregular cartilaginous arches of focal bone formation. The bone marrow active haemopoesis. A few ganglia, nerve segments and some pigment bearing choroids were identified. In the central canal of the vertebral area, neuroglial tissue contains nerve cells and ependymal lining was found. Nerve roots and telanectasis of local capillaries were seen. Section of the skull and brain showed vascular nervous tissue including glial areas, astrocytes, ependymal lining, meninges and a well-formed choroids plexus.

Chua JHY et al (1) on elective Laparotomy found a large retroperitoneal cystic mass that arose from the pelvis and extended superiorly to just below the bifurcation of the aorta. It was densely adherent to the sacrum. The bladder was displaced anteriorly and was floppy and distended. The mass represented a tense amniotic sac with a fetoid mass within. The sac contained clear fluid and the fetus within had grossly visible limbs. The blood supply was via multiple small posterior ally situated vessels. Both testes were intra-abdominal, each measuring about 0.5 ml.s. The authors further noted that at complete excision it is curative which is similar to the finding of the present study. Majhi A.K. et al (10) in an eight year old girl on X –ray, ultrasonography, and CT revealed that a fetiform mass containg long bones and vertebral bodies surrounded by soft tissue situated on right lumber region. On laparotomy, a retroperitoneal mass resembling a fetus of 585 gm was removed.It had a trunk and four limbs with fingers and toes, umbilical stump, intestinal loops, and abundant scalp hairs but was devoid of brain and heart. Histologically showed various well-differentiated tissues in respected sites. FIF is a mystery in reproduction and it is scarce in literature in such well developed stage.

Mohan H et al(11) noted in a three year old male child presented with a retroperitoneal mass, which on radiological examination showed retroperitoneal teratoma. Pathological examination revealed fetus in fetu with partially developed vertebral column.,

CONCLUSION:

From above observations and discussion the authors reached to the conclusion that –

(a) Fetus and fetu is a rare and interesting entity.

(b) Presents as abdominal mass during childhood or infancy.

(d) Complete excision is curative and confirms the diagnosis

ACKNOWLEDGEMENT:

The authors expresses their sense of gratitude to Dr. A.K. Bansal, Ex-Professor and H.O.D., Department of Community Medicine, Govt. Medical College, Jagdalpur (Bastar) 494 001, India, for his guidance in preparing this manuscript.

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Received on 02.04.2012

Modified on 20.05.2012

Accepted on 28.05.2012

Research J. Pharmacology and Pharmacodynamics. 5(2): March–April 2013, 119-122