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PubMedDate not supplied

[Intrahepatic cholestasis with situs inversus of the liver].

Intrahepatic cholestasis with situs inversus of the liver has not been reported in the literature. PURPOSE--A follow-up case for approximately 14 years (by one of the authors, MM). METHODS--Clinical aspects were studied and several laboratory exams as well as three ultrasonography and one computerized tomography exams were performed. Percutaneous and endoscopic cholangiographies were attempted. RESULTS--Four days after the patient was submitted to a surgical replacement of the aortic valve (with extracorporeal circulation), she developed jaundice, severe pruritus, dark urine, and pale stools. Serum levels of bilirubin, alkaline phosphatase, gama-glutamil transferase, and cholesterol were very high. Ultrasonography and computerized tomography exams did not reveal any evidence of extrahepatic obstruction. The tomography exam confirmed the existence of situs inversus in the stomach and liver. We were not successful in performing the cholangiography exams. Clinical and laboratory data as to cholestasis returned to normal after 4 months with no recurrence or complication during the follow-up period (14 years). CONCLUSION--The differential diagnosis between intra and extra-hepatic cholestasis in patients with situs inversus of the liver may be difficult. Therefore it will be necessary to collect clinical data and various complementary exams such as endoscopic retrograde cholangiopancreatography which is very difficult to perform in these patients.

Cholestasis, Intrahepatic↗
PubMed1991

Function and morphology of respiratory cilia in situs inversus.

The activity and ultrastructure of respiratory cilia were studied in 13 individuals with situs inversus but without serious respiratory complaints, in 8 patients with Kartagener's syndrome and in 50 healthy controls without situs inversus. In 3 individuals with situs inversus abnormal ciliary activity was found, comparable in every aspect to the ciliary activity in patients with Kartagener's syndrome. In 4 individuals with situs inversus a dynein arm deficiency was present in the cilia, with abnormal ciliary activity in 3 of them. The number of outer dynein arms per cilium differed significantly between the 3 groups (P less than 0.02). Apparently, situs inversus without serious respiratory complaints can be associated with primary ciliary dyskinesia. Based on the findings in this study, the incidence of primary ciliary dyskinesia in the Caucasian population appears to be 1:12,500.

Adolescent↗
PubMed2003

Situs inversus totalis with malignant lymphoma of the stomach: report of a case.

Situs inversus totalis is a rare congenital anomaly that often occurs concomitantly with other disorders. We report a case of situs inversus totalis with malignant lymphoma of the stomach, which was successfully treated by surgery followed by chemotherapy and irradiation. The patient was a 51-year-old woman who present with colicky pain in the left upper quadrant of her abdomen. Chest X-ray showed a right-sided heart, and ultrasonography and computed tomography (CT) of the abdomen showed a situs inversus totalis with multiple gallstones in the gallbladder. Tree-dimensional reconstructed CT of the abdomen showed no other malformations coexisting with situs inversus totalis, but a barium upper gastrointestinal series found an inverted stomach and an elevated tumor with ulceration in the center, localized in the antrum of the stomach. First, we performed a cholecystectomy, followed by a total gastrectomy with dissection of the lymph nodes and splenectomy, and Roux-en-Y reconstruction. Histopathological examination confirmed a diagnosis of malignant lymphoma of the stomach (diffuse large B-cell type) with metastasis to the regional lymph nodes. Chemotherapy using the CHOP regimen was given three times, starting 1 month postoperatively. A followup CT scan showed enlargement of one lymph node around the abdominal aorta and irradiation was delivered to the area of the inverted Y in the abdomen. At the time of writing, 10 months after surgery, the patient is well with no signs of recurrence and leading a normal life. Careful preoperative assessment is very important for determining the most appropriate surgical procedure in patients with situs inversus totalis associated with a malignancy.

Combined Modality Therapy↗
PubMedDate not supplied

Laparoscopic cholecystectomy in a patient with situs inversus totalis: feasibility and technical difficulties.

Situs inversus is a rare anomaly characterized by transposition of organs to the opposite side of the body. In patients with this anomaly, cholelithiasis is observed with a frequency similar to that in the normal population. Herein, we report on a patient with situs inversus totalis who underwent laparoscopic cholecystectomy for mucocele of the gallbladder. Diagnostic pitfalls and technical difficulties of the operation with technical options are discussed in the context of the available literature. Difficulty is encountered particularly in skeletonizing the structures in Calot's triangle, which consumes extra time and is more demanding than in patients with a normally located gallbladder. A summary of an additional 32 similar cases reported in the medical literature is also presented.

Aged↗
PubMed1979

Situs inversus and appendicitis.

Two cases of left lower quadrant appendicitis associated with situs inversus totalis are presented. Historic, genetic, and embryologic aspects of situs inversus, as well as recent theories regarding etiology are discussed. The clinical recognition of appendicitis in situs inversus is often difficult because of the unusual location of the appendix and abnormal pain localization. The differentiation of situs inversus totalis and abnormal rotation of the abdominal viscera (situs indeterminus) is important surgically because fixation of the midgut loop may be indicated in patients with situs indeterminus.

Adult↗
PubMed1994

Receptor subtype and intracellular signal transduction pathway associated with situs inversus induced by alpha 1 adrenergic stimulation in rat embryos.

In previous studies, we have demonstrated that stimulation of alpha 1 but not alpha 2 or beta adrenergic receptors in rat embryos grown in culture interferes with normal development of the left/right body axis leading to situs inversus. In the present study, we aimed to determine the alpha 1 adrenergic receptor subtype and signal transduction pathway involved in this phenomenon. Rat embryos at Stage 11a by a modified Theiler's staging system were cultured for 50 hr in medium containing various compounds which are known to activate or inhibit different sites of the signal transduction pathways associated with alpha 1 adrenergic receptors. They were then examined to determine the sidedness of asymmetric body structures. WB4101, a selective antagonist of alpha 1A adrenergic receptor subtype, but not chlorethylclonidine, a selective antagonist of alpha 1B adrenergic receptor subtype, inhibited phenylephrine (an alpha 1 adrenergic agonist)-induced situs inversus. Neither the protein kinase C (PKC) activators phorbol 12-myristate 13-acetate and SC-9 nor the PKC inhibitor calphostin C caused situs inversus. Furthermore, calphostin C did not block phenylephrine-induced situs inversus. A23187, a Ca2+ ionophore, induced situs inversus; nifedipine, a L-type Ca2+ channel blocker, partially blocked phenylephrine-induced situs inversus. The calmodulin antagonists trifluoperazine, W-7, and W-13 blocked phenylephrine-induced situs inversus, although they did not cause situs inversus by themselves. KN-62, a Ca2+/calmodulin-dependent protein kinase II (CaM kinase II) inhibitor, dose-dependently blocked phenylephrine-induced situs inversus. However, higher concentrations of this compound produced no block in the presence of phenylephrine and in its absence produced a 50% incidence of situs inversus. These results indicate that alpha 1 adrenergic stimulation-induced situs inversus is mediated by the alpha 1A adrenergic receptor subtype and that activation of CaM kinase II but not PKC may be involved.

1-(5-Isoquinolinesulfonyl)-2-Methylpiperazine↗
PubMed1995

Evaluation, operative management, and outcome after liver transplantation in children with biliary atresia and situs inversus.

INTRODUCTION: Biliary atresia, a common indication for liver transplantation, can be associated with situs inversus. Our experience with liver transplantation in children (n = 6) was reviewed retrospectively. PATIENTS AND METHODS: Preoperative duplex sonography, computerized tomography, and visceral angiography were obtained. Vascular anomalies identified included preduodenal portal vein (6/6), interrupted inferior vena cava (5/6), and aberrant hepatic artery (4/6). RESULTS: The liver graft was placed in a midline position. Venous continuity was achieved by donor suprahepatic inferior vena cava to recipient hepatic cloaca and direct end-to-end portal anastomosis. The donor infrahepatic inferior vena cava was oversewn. Arterial continuity was restored using either a direct branch-patch anastomosis (3/6) or a supraceliac aortic interposition graft (3/6). In retrospect, preoperative diagnostic work-up was noncontributory and outcome was not complicated by pre-existing situs inversus. CONCLUSION: Situs inversus in liver recipients requires operative technical modifications, but does not change outcome. Furthermore, extensive preoperative work-up should be avoided.

Biliary Atresia↗
PubMed2000

Renal dysplasia and situs inversus totalis: an autopsy case report and literature review.

Renal dysplasia has rarely been reported to be associated with situs inversus. Only 4 cases were reported previously with a spectrum of anomalies including bilateral renal dysplasia, situs inversus totalis, and pancreatic and hepatic fibrosis. Recently we encountered another case. A 22-year-old mother, gravida 1 para 0, was in her 23rd week of gestation when she was found to have oligohydramnios. A dead baby was delivered by extraovular induction. On autopsy, Potter's sequences including flattened, low-set ears, receding chin, upturned nose, and club hands and feet were noted. The posterior fontanel was obliterated. Internal examination showed situs inversus totalis, bilateral renal dysplasia, and agenesis of bilateral ureters. There was increased interstitial fibrosis in the pancreas. None of the family members was known to have any renal anomalies. Pinar and Rogers suggested a new syndrome when situs inversus totalis was combined with bilateral renal dysplasia as well as multisystem fibrosis. In our case, 2 major abnormalities, i.e., situs inversus totalis and bilateral renal dysplasia, were present though the increase of interstitial fibrosis within the pancreas was not as significant. This case is likely to be within the spectrum of anomalies mentioned by Pinar and Rogers.

Adult↗
PubMed1996

[In utero diagnosis of situs abnormalities. A case of prenatal diagnosis of visceral situs inversus with corrected transposition of great vessels].

A case of situs inversus with discordant atrio-ventricular and ventriculo-arterial structures was diagnosed at the ultrasonography examination performed at 34 weeks gestation. This observation underscores the importance of recognizing the position of the viscerae and discordant segments of the heart, atria, ventricles and greater vessels. Overall frequency of situs inversus is estimated at 1/10,000 births. It is often associated with an inborn cardiopathy, especially corrected transposition of the greater vessels resulting from abnormal rotation of the cardiac tube. The anatomic, pathologic, embryologic, etiologic and and sonographic features of situs inversus and related anomalies of the cardiac structures is presented.

Abnormalities, Multiple↗
PubMed2003

[First orthotopic liver transplantation in patient with biliary atresia and situs inversus in spain].

Biliary atresia is the most common indication for liver transplantation in the pediatric age group. The Kasai portoenterostomy has become established as the primary treatment for biliary atresia. If portoenterostomy fails, death before 2 years of age is likely without liver transplantation. The most common multiple malformation syndrome associated with biliary atresia is polysplenia syndrome, which forms a constellation of defects of body symmetry, splenic development and vascular anomalies, including situs inversus, polysplenia and others. The situs inversus was formerly considered an absolute contraindication for liver transplantation. Recently however, several case reports have been published suggesting that neither situs inversus nor this particular subset of vascular abnormalities should be considered contraindications to liver transplantation. We present one case of liver transplantation performed in patient with biliary atresia, situs inversus and polysplenia. This is the first report described in Spain for a liver transplant in a child with biliary atresia plus situs inversus.

Abnormalities, Multiple↗
PubMed2006

Laparoscopic Nissen fundoplication in a patient with situs inversus totalis: an ergonomic consideration.

We report a laparoscopic Nissen fundoplication for gastroesophageal reflux disease in a patient with situs inversus totalis. The 65-year-old man was previously diagnosed with situs inversus totalis and presented with chronic gastroesophageal reflux disease inadequately controlled by medications. The laparoscopic procedure was performed with 5 ports placed in a mirror-image configuration and with the patient in the lithotomy position. Few technical difficulties were encountered during the operation. The position of the primary surgeon, working between the lower limbs of the patient, was considered critical to the success of this case. In situs inversus totalis, this position provides the least visual disorientation from the reversed abdominal organs. We recommend this position for all upper abdominal laparoscopic procedures in patients with situs inversus totalis, including laparoscopic cholecystectomy.

Aged↗
PubMedDate not supplied

[Surgical treatment of complex forms of congenital heart malformations associated with situs inversus (nine cases) (author's transl)].

Although are, situs inversus is nevertheless encountered in the surgical treatment of heart malformations. This series concerns 1 000 patients who where operated with heart-lung by-pass between 1974 and 1977. In nine cases, the heart was in the situs inversus position and the cardiac malformations associated with this condition, were complex. Yet the results were comparable to those obtained when the heart was in the normal position.

Abnormalities, Multiple↗
PubMed2002

Implantation of a left ventricular assist device in situs inversus.

A 42-year-old man with situs inversus was referred to our institution because of end-stage ischemic cardiomyopathy. Cardiac arrest occurred after admission, and the patient underwent cardiopulmonary resuscitation. An extracorporeal left ventricular assist device (Berlin Heart) was implanted as a bridge to transplantation. This is one of the first reports of left ventricular assist device implantation in a patient with situs inversus and ischemic cardiomyopathy.

Adult↗
PubMed1998

Abdominal situs inversus with congenital duodenal stenosis: rare association.

Abdominal situs inversus carries a significant mortality because the majority of cases (95 %) have associated cardiac and splenic defects. A review of all cases reported in the English literature confirms in addition the presence of significant gastro-intestinal pathologies, mainly annular pancreas, midgut volvulus and duodenal atresia. We report on an additional case of abdominal situs inversus without cardiac or splenic abnormalities, who had partial duodenal obstruction secondary to a mucosal diaphragm. This seems to be the thirteenth case associated with duodenal obstruction and the sixth case secondary to a mucosal duodenal diaphragm, to date, in this setting.

Duodenal Obstruction↗
PubMed1992

Coarctation of inferior vena cava in situs inversus totalis.

A woman aged 28 years with situs inversus totalis presented with chronic Budd-Chiari syndrome with symptoms of 3 years' duration. The left sided inferior vena cava was occluded below the diaphragm above a patent morphologic right hepatic vein. A dorsal cavoatrial bypass was attempted but abandoned due to bleeding from extensive collaterals. The occurrence of coarctation of the inferior vena cava in situs inversus totalis lends support to the view that it is a congenital condition and occurs due to disturbance in fusion of the hepatic segment of the inferior vena cava and the hepatocardiac channel.

Adult↗
PubMed2004

The management of esophageal cancer with situs inversus totalis by simultaneous hand-assisted laparoscopic gastric mobilization and thoracoscopic esophagectomy.

Situs inversus is a rare disorder. We present a case of esophageal cancer with situs inversus totalis, which was successfully managed by laparoscopic gastric mobilization and thoracoscopic esophagectomy. A 57-year-old man presented to our hospital with intermittent epigastric and retrosternal pains. X-ray and computed tomography demonstrated situs inversus totalis. Upper gastrointestinal endoscopy and barium swallow showed esophageal cancer of the lower thoracic esophagus. After neoadjuvant chemotherapy, he underwent surgical intervention. Under general anesthesia, laparoscopic gastric mobilization using hand-assisted laparoscopic surgery technique was performed. The locations of the port sites were all the mirror image of the regular fashion, and the right hand of the surgeon was inserted into the peritoneal cavity. Reconstruction of the digestive tract using a gastric tube via the retrosternal route was then achieved. Finally, thoracoscopic esophagectomy with the patient in the right decubitus position was successfully completed. Postoperative recovery was uneventful.

Esophageal Neoplasms↗
PubMed2001

Feasibility of laparoscopic cholecystectomy in situs inversus.

PURPOSE: To address the feasibility and safety of laparoscopic cholecystectomy in situs inversus and highlight the necessary modifications in the surgical technique. PATIENTS AND METHODS: We present our experience in two patients with situs inversus and symptomatic gallstones who were treated successfully by laparoscopic cholecystectomy. The surgeon stood on the right side with the video monitor above the patient's left shoulder. Two 10-mm ports were placed in the epigastric and subumbilical positions. Two 5-mm ports were placed in the left mid-clavicular and left anterior axillary lines. The two procedures were carried out uneventfully after reorientation of the visual-motor skills of the surgeon and cameraman to the left upper quadrant. A summary of a further similar 13 cases so far treated in the English-language medical literature is also presented. RESULTS: Skeletonizing the structures in Calot's triangle consumed extra time and was more difficult than in patients with a normally sited gallbladder. However, the hospital stay and postoperative complications were similar. CONCLUSION: Laparoscopic cholecystectomy in situs inversus seems to be feasible and safe provided it is performed by an expert laparoscopic surgeon who takes time in clearly demonstrating the extrahepatic mirror image anatomy of the biliary tree with its right-to-left shift.

Adult↗
PubMedDate not supplied

Orthotopic liver transplantation in an adult with situs inversus: an easy way to fit the liver.

Situs inversus (SI) is a rare congenital disorder with a complete mirror image of thoracic and abdominal organs. In adults with SI and decompensated cirrhosis experience with liver transplantation is limited. Orthotopic liver transplantation (OLT) in an adult with cirrhosis using a technique where the recipient liver was placed using a 90-degree rotation of the graft was previously reported by Klintmalm et al, however no other reports using this technique have been described. We report a case of a 41 year-old man with situs inversus and decompensated cirrhosis who successfully underwent OLT using this technique. The donor liver was rotated 90-degrees towards the left and easily fitted into the recipients'fossa with the left lobe pointing toward the left lower quadrant. The patient had an uneventful recovery and has been followed for 21 months without any complications. This technique has the advantage of preventing compromise of the size of the donor liver, permits an easy reconstruction of vascular and biliary tree and in this case was associated with an excellent outcome.

Adult↗