Nonspecific Infectious Bilateral Chylothorax and Cyhloabdomen with Symptoms of Acute Abdomen

Co-existence of chylothorax and chyloabdomen is a rare clinical entity. Apart from surgery and extra-surgical trauma, malignancies, liver cirrhosis, nephrotic syndrome, thrombosis in the superior vena cava and acute pancreatitis play roles in the etiology. The case presented in this article was a 35-year-old woman, and the chronic infectious pathology in the cytology, plus the other supportive nonspecific infection parameters prompted us in establishing this diagnosis. Obstruction in the abdominal lymphatics leads to chylous ascites and chylothorax develops in due course. Similar to our case, the clinical entities of chylothorax and chyloabdomen of nonspecific infectious origin may co-exist in the same patient.


Introduction
Chylothorax is a rare clinicopathological condition in thoracic surgery.It is characterized by the presence of fluid and chylomicrons rich in triglycerides in the pleural cavity.Tumors, trauma, idiopathic factors and other causes may be stated in the etiology [1].It is primarily observed following thoracic and heart surgery with a rate of 0.5%-2.5% [2].Apart from trauma, another leading cause of chylothorax is malignancy.Chylothorax related to extra-malignant conditions constitutes a very small percentage and in 15% of the cases, the actual cause cannot be clarified [3,4].
Following confirmation of the diagnosis through biochemical examinations, treatment should be initiated promptly.Oral nutrition must be replaced with parenteral nutrition and the chylous fluid must be drained.In cases where drainage is persistent despite 2-4 weeks of treatment, the ductus thoracicus may be ligated surgically.Additional surgical and medical approaches may also be utilized.
Our patient was examined in detail and diagnosed as a case of nonspecific-infectious chylothorax, based on our findings.

Case Report
A thirty-five-year-old woman presented to the general surgery department with the complaint of severe abdominal pain and she was hospitalized for evaluation and treatment with the diagnosis of acute abdomen.Abdominal ultrasonography and computerized tomography of the thorax and the abdomen that were performed prior to laparotomy revealed the presence of free fluid in the abdomen and bilateral pleu-ral cavities (Figure 1, 2).The case was explored by a general surgeon.No signs were observed during the operation, apart from diffuse chylous fluid.She was referred to our department on the first postoperative day with bilateral pleurisy.
The patient was hospitalized in our clinic and chylous fluid was aspirated through bilateral pleural puncture.Analysis of the drained fluid revealed the following: Total protein: 3.6% gr, Total lipids: 3.7% gr, Cholesterol: 270.8 mg/dl, Triglycerides: 1354.11mg/dl, Glucose: 69.21 mg/dl, Na: 116 mEq/lt, K: 4.4 mEq/L, and Cl: 97 mEq/L.The percentage of lymphocytes in the fluid was over 80%.The diagnosis of chylothorax was confirmed upon microscopic determination of fat globules in the pleural fluid with Sudan III. Biochemical blood parameters were within normal limits.On initial presentation, the patient had dyspnea.Blood gas analysis revealed an oxygen saturation of 89%.The initial complete blood count showed normal values of hemoglobin, hematocrit and leucocytes, but a significant change was observed in favor of lymphopenia and neutrophilia.
Following thoracentesis and other examinations, bilateral tube thoracostomy was performed, in addition to central venous catheterization.Oral nutrition was stopped and total parenteral nutrition was commenced.Respiratory support was provided.Drainage continued for 2-3 days after tube thoracostomy, but on day 4, the drainage from right tube stopped completely.Drainage from the left thorax tube continued for an additional duration of 3-4 days and completely stopped on the 7. Day following tube thoracostomy.After evaluating the drainage and pulmonary expansion, the tubes were removed under control.On day 6 of hospitalization, the C-Reactive Protein (CRP) value was 54.9 and the sedimentation rate was 32 mm/h.The Tuberculin test results and all the tumor markers were within normal limits.Since the patient had occasional symptoms of hypoglycemia, evaluation of folate, vitamin B-12, thyroid markers, somatomedin, growth hormone and cortisol values were requested as a result of the endocrinological consultation; the results were regarded as normal.All the parameters requested by the infectious diseases consultant were within normal limits.There was no growth in the culture of pleural fluid; induration was measured as 4 mm in the Tuberculin test.There was no growth in the pleural fluid culture and the bacteria were negative.Cytological examination of the pleural fluid was reported as active chronic inflammation.
On day 8 of referral to our clinic, oral nutrition was reinitiated with a high-calorie diet, rich in proteins and medium chain triglycerides with low fat.Following oral nutrition, the clinical status of the patient improved with no signs of drainage and the patient was discharged from the hospital.The findings at the control visit 4 months after discharge was regarded as completely normal.

Discussion
Co-existence of chylothorax and chyloabdomen is a relatively rare condition.It develops by accumulation of chylous fluid in the pleural cavity and the abdomen, associated with obstruction of the ductus thoracicus or obliteration in one of its branches due to tumoral infiltration, inflammation or trauma [5].Obstruction in the abdominal lymphatics related  to any cause leads to chylous ascites and this condition causes cylothorax [6].The most common causes of chylothorax are surgery and extra-surgical trauma [2].Apart from trauma and malignancies (especially lymphoma), chylothorax and chyloabdomen may develop in filariasis, lymphangioleiomyomatosis, amyloidosis, cirrhosis, thrombosis in the jugulo-subclavian region, pericarditis, nephrotic syndrome, sarcoidosis, tuberculosis and in the post-radiotherapy stage in malignancies [5].
In a series of 203 patients reported by Doerr et al., surgery or trauma was present in 101 patients, lymphoma and lymphatic disorders were detected in 89 patients, and idiopathic chylopathology was determined in 13 patients [7].
In the diagnosis of cyhlothorax and chyloabdomen, macroscopic appearance of chylous fluid is important.Diagnosis is confirmed based on biochemical examination of the fluid with triglycerides of >110 mg/dl and a ratio of cholesterol/ triglycerides of <1, and detection of fat globules with Sudan III staining in the microscopic examination.The triglyceride value in our patient was 1354.11mg/dl, the cholesterol value was 270.8 mg/dl, and the ratio of cholesterol/triglycerides was much lower than 1.Furthermore, lymphocyte domination was prominent (80% lymphocyte domination, 4050 cells/µL).In our case, the diagnosis of chylothorax was confirmed based on the 2 parameters stated above, in addition to the macroscopic appearance.
In the diagnosis of chylothorax, assessment of abdominal and thoracic tomographies and evaluation of the fluid are critical in terms of eliminating a possible malignancy.Furthermore, lymphangiography may also play an important role in locating the obstructed region.In the lymphangiography of our patient, multiple abnormalities and lymphangiectasia were observed in the lymphatic system, but the leakage could not be located.Possible causes were eliminated based on the tomography and other examinations and consultations.According to the supportive results of cytology and laboratory analyses and the quick recovery of the clinical state, the patient was regarded as a case of nonspecific infectious chylopathology.The patient promptly responded to treatment and oral nutrition was re-initiated on day 8 of tube thoracostomy.
In cases of chylothorax and chyloabdomen, various disorders such as nutritional deficiency, disorders of immunological function and dehydration develop due to leakage of calories, fluids and proteins.Cardiopulmonary functions are disturbed due to chylothorax.
There is no consensus on the treatment of chyothorax and chyloabdomen.However, daily loss of chylous fluid and the resulting metabolic disorder, age and etiology play critical roles in the treatment.The surgical treatment approach should be considered in cases where conservative treatment is unsatisfactory.In our case, central venous catheterization was performed following establishment of the diagnosis and oral nutrition was discontinued, followed by hyperalimentation.Biochemical values were monitored daily.Chylous drainage was also monitored.Chylous drainage significantly diminished in a few days and then fully stopped.
In chylothorax and chyloabdomen, lymphatic ducts generally close spontaneously with conservative treatment.In cases where drainage is persistent and increased, surgical intervention should not be disregarded.The main principle in the surgical treatment is repairing the chylous leakage.Ligation of the ductus and primary repair comprises the main surgical method.The surgical methods include direct ligation of the ductus, suturing with the peripheral soft tissues, pleuro-peritoneal shunt procedures, pleurectomy and chemical pleurodesis [8].The success rate of surgical intervention (thoracotomy and thoracoscopy) is higher than 90%.In recent years, fluoroscopic embolization has also performed on the ductus thoracicus with platinum coils [9].
In conclusion, co-existence of bilateral chylothorax and chyloabdomen is occasionally seen, although it is a rare condition.Especially in cases where trauma, surgery and malignancy are eliminated, an infectious etiology should be considered.In cases with no response to treatment in two weeks, surgical treatment should not be disregarded due to increase in the risk of complications.