Bochdalek hernia: A rare case report of adult age.

INTRODUCTION: Bochdalek hernia is the most common type of congenital diaphragmatic hernia and constitutes 85% of cases. Bochdalek hernia (BH) in adults is extremely rare. We present a BH case in an adult patient and discuss the literature.
PRESENTATION: 22-year-old female patient with abdominal pain, occasional cramps, dysphagic problems, constipation, shortness of breath and choking for about 2 years applied to our clinic. DIAGNOSIS: A defect about 5 cm in the left hemidiaphragm posterior area and herniation of intra-abdominal fat plan in the left hemithorax was seen in intravenous and oral whole abdominal CT. TREATMENT: Patient was operated laparoscopically. Transverse colon and a large portion of the omentum entering into hemidiaphragm were pulled in to intraperitoneal area carefully. Approximately 10 × 8 cm intraabdominal mesh was fixed to the defect area with the help of laparoscopic tacker.
CONCLUSION: Adult BH is very rare and when confronted laparoscopic treatment with mesh fixation can be performed safely.

Introduction

Congenital diaphragmatic hernia (CDH) characterized by protruding abdominal organs into the thoracic cavity through the posterolateral diaphragmatic defect and has high mortality [1]. The incidence of CDH is 1 in 2500 births and left congenital diaphragmatic hernias are more common than right-side hernias (85%–12%) [1]. Although CDH are diagnosed prenatally or in the immediate postnatal period, diagnosis in 5–25% of cases can be late, and could be detected during routine examinations or examination because of respiratory or gastrointestinal problems [1].

Bochdalek hernia (BH) is a congenital diaphragmatic hernia caused by the failure of the posterolateral diaphragmatic foramina to fuse properly; it results in the displacement of abdominal components into the thoracic cavity [2]. This occurs mainly during the ninth or tenth week of fetal life [2]. Bochdalek first described this anomaly in 1848 [2]. The incidence is reportedly 1 in 2200–12 500 live births, and Bochdalek hernia (BH) usually occurs (80%–90%) on the left side [2]. Bochdalek hernia in adults is extremely rare; with less than 100 published cases in the literature. Larger defects in BH are associated with pulmonary hypoplasia on the affected side and respiratory distress syndrome after birth [3]. Minor defects are not associated with a deficit in lung development and may be asymptomatic until herniation of abdominal contents into the thoracic cavity with respiratory consequences [3]. Colon is the most common intra-abdominal organs migrating through the diaphragmatic defect and may cause large bowel obstruction [3].

In our case, there was no bowel obstruction although a big part of transverse colon and omentum were in thorax.

Surgical repair of the defect is the recommended therapy for all patients with BH, regardless of the presence of symptoms.

Management of a BH includes reducing the abdominal contents and repairing the defect through laparatomy or thoracotomy [2]. Surgical repair has been done by laparatomy, traditionally, however since laparoscopy was used, laparoscopic treatment was started to be used more often due to less hospital stay and early work starting time [4]. Both laparoscopic and thoracoscopic repairs of BH have been reported [2]. The procedure of choice depends on the surgeon's experience [2]. Small defects are easier to repair, but larger defects may involve a reduction of the intra-abdominal contents [2].

In this article we aimed to report a 22 years old patient operated and followed up for Bochdalek hernia in our clinic. We believe this report is going to add important valuable information to the medical literature. Although it is not the first report discussing Bochdalek hernias with laparoscopic repair BH in adults is extremely rare and could be misdiagnosed. Bochdalek hernia should be in mind in case of patient presenting with intestinal and pulmonary symptoms.

Case report

22-year-old female patient had complaints such as abdominal pain, occasional cramps, dysphagic problems, constipation, shortness of breath and choking for about 2 years. There was no history of thoracic or abdominal trauma. There was no relevant past medical history. Patient and family psychosocial history were normal. Physical examination was unremarkable. The bowel sounds were audible on the left side of the chest. CT was used as diagnostic methods because of its acceptable diagnostic rate and easy availability. Routine blood test were done and were non significant. A defect about 5 cm in the left hemidiaphragm posterior area and herniation of intra-abdominal fat plan with transverse colon in the left hemithorax was seen in intravenous and oral whole abdominal CT (Fig. 1). In the differential diagnosis other thoracic pathology were considered such as tension pneumothorax, left middle lobe collapse, air space consolidation, pericardial fat pad, sequestration of the lung, mediastinal lipoma, or anterior mediastinal mass. They were ruled out by plain film chest radiography and CT. After all no challenges met when attempting to diagnose the Bochdalek hernia.

Fig. 1

Computerized tomography view of defect (arrow).

Therapeutic intervention

Patient was operated laparoscopically. Cefazolin IV 1 gr antibiotic was given within 1 h before the surgery for prophylaxia and one more dose was given in 24 h postoperatively. The abdomen was insflulated from umbilical trocar. Exploration showed about one third of the transverse colon and a large portion of the omentum majus entering to left diaphragm in a 5 × 6 centimeter defect area in posterior lateral left hemidiaphragm (Fig. 2). The adhesions were carefully released. The herniated contents were carefully reduced to the peritoneal cavity through the hernia defect. There were no ischemic changes of the bowel, and omentum. Structures entering into hemidiaphragm were pulled in to intraperitoneal area carefully. Approximately 10 × 8 cm intraabdominal mesh was fixed to the defect area with the help of laparoscopic tacker (Fig. 3). There were no significant events during the procedure. We opt to tackle the hernia laparascopically because of its being easy and practical. One flat drain was placed under the left diaphragm at the end of the surgery. Post-surgical pain was treated with one pain medication Nonsteroidal anti-inflammatory drugs (NSAIDs) such as diclofenac or with a combination of contramal if necessary. No complication was observed postoperatively and patient was discharged. This case report was written with the patient's full, informed, written consent, available if requested.

Fig. 2

Laparoscopic view of defect.

Fig. 3

Mesh fixation of defect.

Follow up

The patient's postoperative recovery was uneventful there was no pneumothorax and lung fields were clear. The patient was discharged on the 3rd day in satisfactory. No post-operative CT scan was performed. At 6 month follow-up, patient was well having no problem and is now on regular follow-up with us in the out-patient department. Patients symptoms were all improved in the follow up. The procedure was accepted helpful.

Discussion

Diaphragmatic hernia through the posterolateral foremen of Bochdalek is the commonest type of CDH and more common on the left side (85%) than on the right. Left sided hernia include spleen, stomach, small intestine and colon [5]. Right sided hernia includes liver and intestine. The colon as hernia content is relatively rare compared to other contents. Late presentation may be due to delayed rupture of peritoneal sac containing the viscera, or plugging of hernia defect by solid organ due to raised abdominal pressure in severe strain, obesity, pregnancy and during labour [5].

The diagnosis of congenital diaphragmatic hernias typically occurs prior to delivery, but 5–25% of cases are late presenting, after the neonatal period. The most frequently encountered type of CDH is posterolateral hernia, through the foramen of Bochdalek [1].

Many patients may remain asymptomatic until adulthood [3]. Most hernias are asymptomatic And found incidentally. Patients may present with chronic symptoms such as recurrent chest or abdominal pain and postprandial fullness or vomiting [6]. Brown SR et al. [7] searched the literature yielded 141 articles containing 173 cases from 31 countries. Only 14% of patients were symptomatic at the time of presentation. Presenting symptoms were pain/pressure (chest or abdominal discomfort that was not related to a bowel obstruction) 69%, obstruction 39%, pulmonary symptoms (dyspnea, cough, and shortness of breaths) 37%, strangulated 28%, dysphagia 3%, bleeding 4%, GERD 4%, and other (HTN, fatigue, indigestion) 9%.In our case, patient has symptoms such as shortness of breath, chest pain, choking, and dysphagic symptoms.

The diagnosis of a BH in adults is not easy and it is commonly misdiagnosed. Unlike infants who show with respiratory distress early, the most frequent symptom in adults is mild discomfort and 25% of adult patients are asymptomatic. Consequently, many patients are merely treated according to their symptoms [8]. In the diagnosis of BH, direct chest and abdominal radiography, fluoroscopy, barium examinations, ultrasound, computed tomography, magnetic resonance imaging is available. But among them, multislice computed tomography of the sagittal and coronal images of diaphragmatic hernia was most commonly used. Contrast-enhanced CT is the most accurate imaging modality for detection of BH. It provides detailed information regarding the herniated viscera and the diaphragmatic defect [9]. The presence of a soft tissue contour in the chest CT, in addition to opaque, filled, dilated bowel segments above the diaphragm, establishes definitive diagnosis [4].

The management of Bochdalek hernia includes reduction of hernial contents to the peritoneal cavity and repair of the diaphragmatic defect [9]. Although laparatomy was the most widely used surgical approach (38%), minimally invasive surgical techniques have gained popularity since their first report in 1995. Laparoscopic repair can be performed with a low complication rate (7%) and short hospital stay (4 days) [7]. Regardless of the type of surgical procedure, Suturing the defect is likely important for the restoration of the anatomy between the thoracic and abdominal cavities [4]. Many surgeons prefer a prosthetic graft because of the continuing stress on the diaphragm that results from respiratory movements and cardiac motions. Nevertheless, a tensionless type of repair has been validated as an option for BHs, which is similar to the type of repair used for all other hernia repairs [4]. Many types of meshes are available for use in these types of repairs. Although polypropylene mesh has the benefit of support and excellent tissue growth, erosion of the mesh into the gastrointestinal organs represents a theoretical risk [4]. The decreased tendency for adhesion formation of polytetrafluoroethylene and other dual prostheses makes them more desirable [4]. Surgeons should take great care during the fixation of grafts with a laparoscopic tacker where the diaphragm is relatively thin [4].

Conclusion

Adult BH is very rare and when confronted laparoscopic treatment with mesh fixation can be performed safely.

Ethical approval

There is no ethical approval. The study does not require ethical approval.

Sources of funding

There is no source of funding.

Author contribution

Yusuf Yagmur operated the patient and contributed [1] the conception and design of the study, or acquisition of data, or analysis and interpretation of data [2], drafting the article or revising it critically for important intellectual content [3], final approval of the version to be submitted.

Ebral Yigit contributed [1] the conception and design of the study, or acquisition of data, or analysis and interpretation of data [2], drafting the article or revising it critically for important intellectual content [3], final approval of the version to be submitted.

Mehmet Babur contributed [1] the conception and design of the study, or acquisition of data, or analysis and interpretation of data [2], drafting the article or revising it critically for important intellectual content [3], final approval of the version to be submitted.

Serdar Gumus contributed [1] the conception and design of the study, or acquisition of data, or analysis and interpretation of data [2], drafting the article or revising it critically for important intellectual content [3], final approval of the version to be submitted.

Conflict of interest

We have no conflicts of interest.

Guarantor

We all accept there is no Guarantor for this study.

Consent

We accept that written and signed consent to publish this case report from the patient prior to submission was obtained. Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Research registry

This case presentation is not ‘first-in-man studies’, the first time it has been performed/reported in the literature.