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Nonrotation of gut with nutcracker syndrome: a rare presentation

Nutcracker syndrome caused by left renal vein entrapment in acute aorto-mesenteric angle usually presents in second or third decade of life with haematuria, abdominal pain in adult age group both the conditions may present with chronic abdominal pain hence causing a diagnostic and therapeutic dilemma as mere presence of Nutcracker anatomy doesn’t constitute presence of Nutcracker syndrome. Presence of both entities in same patient has been rarely reported in literature.


INTRODUCTION
Intestinal rotational abnormalities usually present in infancy or paediatric age group, however patients may remain quiescent during childhood and present with symptoms in adulthood or incidentally diagnosed when investigated for other pathologies.
Nutcracker syndrome caused by left renal vein entrapment in acute aorto-mesenteric angle usually presents in second or third decade of life with haematuria, abdominal pain in adult age group both the conditions may present with chronic abdominal pain hence causing a diagnostic and therapeutic dilemma as mere presence of Nutcracker anatomy doesn't constitute presence of Nutcracker syndrome. Presence of both entities in same patient has been rarely reported in literature. 1

CASE REPORT
38 year old female presented with dull aching left lower quadrant pain since 2 years with orthostatic exacerbation and no associated symptoms. The severity of pain was debilitating to hinder patient's daily routine work. No previous history of any major illness or surgery could be elicited. Patient's appetite was normal with no fever, nausea or vomiting. No history of Koch's contact was there. Patient was married with two children. Patient was lean and thin built, other than this the general survey was unremarkable. Per abdominal examination was unremarkable except for mild left iliac fossa tendreness. Patient had undergone ultrasound examination of abdomen at other institute which showed presence of uncomplicated non rotation of gut. Patient was investigated and diagnosed as having non rotation of gut with nutcracker syndrome. Orthostatic exacerbation of pain suggested nutcracker syndrome as the likely cause of pain. Patient underwent exploratory laparotomy with left renal vein transposition and Ladd's procedure. Post operatively patient was kept on anticoagulant therapy postoperatively and discharged after 1 week.

Investigation
Patient was investigated over 2 years for chronic left sided pain abdomen, underwent CECT abdomen twice which revealed nonrotation of gut with appendix and larger bowel on the left and small bowel on the right side of abdomen. Presence of few Ladd's bands and absence of retromesenteric third segment of duodenum which was not passing to the left of the spine. Reduced aortomesenteric angle of 32.9 degrees was noted on CT. Superior mesenteric artery and vein axis was slightly inverted with vein anterior to artery.
Renal Doppler ultrasound revealed a dialated left renal vein of 9.8 mm diameter and dilated left gonadal vein of 6 mm diameter. Aorto-mesenteric angle on renal Doppler was 26 degrees.
All other investigations including routine blood investigations and urine routine examination were normal.

Differential diagnosis
Symptomatic non rotation of gut with Ladd's band, nonrotation of gut with subacute appendicitis and nutcracker syndrome were considered as differential diagnosis after excluding pelvic inflammatory syndrome or other gynaecological disorders by clinical examination and ultrasonography.

Treatment
Patient underwent exploratory laparotomy with left renal vein transposition and Ladd's procedure.

Outcome and follow up
Patient tolerated procedure well and was kept on anticoagulant therapy for 6 weeks postoperatively, discharged on postoperative day 7.
The patient was symptom free on 12 weeks follow up.

DISCUSSION
Incidence of rotational abnormalities of gut diagnosed in adults was 0.2%, however symptomatic presentation was very rare. 5

CONCLUSION
Both Nutcracker syndrome and malrotation in adults are rare diagnosis and require high index of suspicion to diagnose.
Definitive clinical diagnosis is difficult due to presence of non-specific signs and symptoms and high incidence of clinical suspicion is required for the diagnosis and management of such cases.
Radiological investigations are of utmost importance in deciding upon the course of management of such cases.