Diagnose and treatment of traumatic pleural-subarachnoid fistula in children: A case report and systematic review

Abstract

Background: Pleural-subarachnoid fistula (PSF) is a rare disease that is difficult to diagnose and treat. Secondary intracranial hypertension and the treatment are seldom mentioned previously among PSF. Case presentation: A 1-year-old boy diagnosed PSF developed into secondary intracranial hypertension after conservative treatment. He was finally cured by down-step treatment of mannitol, avoiding form ventricleperitoneal shunt. Then, we reviewed the literature of pleural-subarachnoid fistula. Fifty-six cases have been reported so far. Most of the cases (51.8%) were caused by surgery; only 17.9% were caused by car accidents. Regarding the treatment, half of the cases cured by surgery and the other by conservative measures. Our case is the first one involving secondary intracranial hypertension and cured by down-step treatment of mannitol. Conclusions: A comprehensive examination should be performed before the treatment to avoid any inappropriate medical strategies. Secondary acute intracranial hypertension may be cured by down step treatment of mannitol, evading from the long-term ventriculoperitoneal shunt.

Pneumocephalus resulting from chest infection complicated by pleural-subarachnoid fistula

“…Pleural-subarachnoid fistula is one of the rare complications of thoracic and spine surgery, especially when in the paravertebral gutter region or as a result of blunt or penetrating trauma [ 2 , 3 ]. Milloy et al in 1959 are the first ones who report a pleural-subarachnoid fistula [1] . It occurs when attract formed between pleural and meningeal tears [4] .…”

“…Nevertheless, the clinical manifestation is most commonly pleural effusion and pneumothorax respectively. patients could present with hemopneumothorax or widening of the mediastinum [1] . Accordingly, the Patient could present with a variety of symptoms passed on the clinical manifestation, including shortness of breath, chest pain, nausea, vomiting, fever, headache, altered mental status, the declining level of consciousness, or rarely neurological deficit [ 3 , 2 , 7 ].…”

“…Diagnosis of Pleural-subarachnoid fistula can be challenging, a combination of clinical, radiological, and laboratory data would help in reaching the diagnosis and preventing meningitis and progressive myelopathy [4] . Chest radiograph and chest CT will recognize the pleural effusion, but they are nonspecific, while pleural effusion analysis using β2 transferrin is a highly sensitive and specific test for detecting the presence of CSF [ 2 , 1 , 9 ]. However, this is not enough alone, since the exact location of the fistula needs further evaluation.…”

“…Different diagnostic modalities can be used and each carries a different specificity and sensitivity. For example, radioisotope myelography has a 100% positive rate, but on the other hand, it carries high radiation damage to the body comparing to other modalities [1] . CT myelography as obtained in our case is superior to the radioisotope myelography in terms of detailed anatomic location and can aid surgical planning.…”

“…There is no specific treatment or management of Pleural-subarachnoid fistula, as it depends on many factors such as the clinical background of the patient, the location and size of the fistula as well as its progressive course [ 10 , 7 ]. Both surgical repair and conservative management have been reported in the literature, including for example; bed rest, chest tube insertion, external lumbar drainage, positive pressure ventilation, epidural blood patch, or subcutaneous biological glue injection [ 1 , 2 , 7 ]. Interestingly, in our case, spontaneous closure of the fistula took place which is the least expected pathway as reported in the literature [4] , and follow-up CT scans of the brain showed a subsequent gradual decrease in the degree of pneumocephalus.…”