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Pneumorrhachis, pneumomediastinum, pneumopericardium and subcutaneous emphysema as complications of bronchial asthma.
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MedLine Citation:
PMID:  19641647     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
Pneumorrhachis (PR), or epidural emphysema, denotes the presence of air in the spinal epidural space. It can be associated with a variety of etiologies, including trauma; recent iatrogenic manipulations during surgical, anesthesiological and diagnostic interventions; malignancy and its associated therapy. It usually represents an asymptomatic epiphenomenon but also can be symptomatic by itself as well as by its underlying pathology. The pathogenesis and etiology of PR are varied and can sometimes be a diagnostic challenge. As such, there are no standard guidelines for the management of symptomatic PR, and its treatment is often individualized. Frequently, multidisciplinary approach and regimes are required for its management. PR associated with bronchial asthma is extremely rare, and only very few cases are reported in the literature. Here, we report a case of a 17-year-old Saudi male patient who is a known case of bronchial asthma; he presented with extensive subcutaneous emphysema, pneumomediastinum, pneumopericardium and pneumorrhachis as complications of an acute exacerbation of his primary ailment.
Authors:
Prasad K Manden; Almas H Siddiqui
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Annals of thoracic medicine     Volume:  4     ISSN:  1817-1737     ISO Abbreviation:  Ann Thorac Med     Publication Date:  2009 Jul 
Date Detail:
Created Date:  2009-07-30     Completed Date:  2011-07-14     Revised Date:  2013-05-23    
Medline Journal Info:
Nlm Unique ID:  101280721     Medline TA:  Ann Thorac Med     Country:  India    
Other Details:
Languages:  eng     Pagination:  143-5     Citation Subset:  -    
Affiliation:
King Saud Chest Hospital, Riyadh, Kingdom of Saudi Arabia. drkeyem@yahoo.co.in
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From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine

Full Text
Journal Information
Journal ID (nlm-ta): Ann Thorac Med
Journal ID (publisher-id): ATM
ISSN: 1817-1737
ISSN: 1998-3557
Publisher: Medknow Publications, India
Article Information
? Annals of Thoracic Medicine
open-access:
Received Day: 10 Month: 3 Year: 2009
Accepted Day: 30 Month: 4 Year: 2009
Print publication date: Season: Jul?Sep Year: 2009
Volume: 4 Issue: 3
First Page: 143 Last Page: 145
ID: 2714570
PubMed Id: 19641647
Publisher Id: ATM-04-143
DOI: 10.4103/1817-1737.53352

Pneumorrhachis, pneumomediastinum, pneumopericardium and subcutaneous emphysema as complications of bronchial asthma
Prasad K. MandenAF0001
Almas H. SiddiquiAF0001
King Saud Chest Hospital, Riyadh, Kingdom of Saudi Arabia
Correspondence: Address for correspondence: Dr. Prasad K. Manden King Saud Chest Hospital, P.B. No.: 7966, Riyadh : 11472, Kingdom of Saudi Arabia. E-mail: drkeyem@yahoo.co.in

The presence of air in the spinal epidural space is a rare radiological entity, mainly described in radiological literature. Various etiologies leading to the entrapment of air in the intra-spinal space, a condition called pneumorrhachis (PR), have been documented. Mainly, traumatic and iatrogenic incidents are reported in the development of this radiological epiphenomenon. PR associated with bronchial asthma is extremely rare, and only 13 cases are seen reported in the entire literature. Acute increase in the intra-alveolar pressure leads to rupture of alveoli resulting in air escape into the perivascular space. This air further moves through the facial planes into the posterior medias tinum and thus into the epidural space.

Here we present the case of a male patient who presented with an attack of bronchial asthma associated with subcutaneous emphysema, pneumomediastinum, pneumopericardium and pneumorrhachis.


Case Report

A 17-year-old Saudi male patient was presented to our ER with history of shortness of breath, nonproductive cough and wheezing since 48 hours prior to his visit. He was a known case of bronchial asthma from early childhood, hospitalized twice in the last 2 years due to worsening of his symptoms. He was never admitted to ICU nor did he ever receive assisted mechanical ventilation in the past. Since the last 9 months, he was taking beclomethasone MDI 200 ?g twice daily with p.r.n, beta-2 agonists.

There was neither any previous history of pulmonary tuberculosis or anti-tuberculous treatment nor any recent trauma or surgery. He has not been a smoker and denies any illicit drug usage.

On clinical examination, he was fully conscious and oriented, but dyspneic. He could complete a sentence at a stretch. Vitals showed PR- 104/ min, RR- 28/min, BP- 104/68 mm Hg, SPO2-89% in normal room air by pulse oxymetry and 92% with 4 L/ min oxygen delivered through nasal canula. Temperature was 38?C.

There was extensive subcutaneous emphysema involving bilateral upper chest, neck and axillary regions.

Auscultation revealed a systolic crunching sound over the left parasternal region, better heard during inspiration (Hamman' sign). The lung fields revealed bilateral extensive expiratory polyphonic wheezes with few inspiratory crackles on auscultation.

ABG on admission showed pH- 7.421, pCO2-31.1 mm Hg, pO2-54.6, SO2 85.6%, HCO3-25.8.

CBC: TLC- 24.3 K/?L, N- 84.4%, L- 4.7%, Hb- 17.2 gm%, Plat- 250 K/?L. All the baseline biochemical parameters were within normal limits. The initial chest x-ray revealed surgical emphysema involving the cervical, thoracic and axillary regions bilaterally without any evidence of rib fracture [Figure 1]. There was pneumonic consolidation involving the right lower lobe. It also revealed air in the mediastinum and pericardium.

For better delineation of the extent of air dissection, he was subjected to computed tomography (CT) of the chest. It confirmed air dissection into the subcutaneous, mediastinal and pericardial spaces [Figures 2 and 3]. High-resolution CT also revealed air in the posterior spinal epidural space at multiple cervical and thoracic levels [Figure 4].

The patient was admitted and was vigorously treated with broad-spectrum antibiotics, inhalation bronchodilators, systemic corticosteroids, high-flow oxygen and other supportive measures. Antibiotics were used since our patient had pneumonic consolidation involving right lower lobe. His hospital course was uneventful, and he showed progressive improvement.

He was subsequently shifted to cardiothoracic care, and follow-up at 2 months revealed an asymptomatic individual.


Discussion

PR is the presence of air in the spinal epidural space. It is an exceptional and often accidental radiological finding; it is caused by various traumatic and nontraumatic etiologies. Different pathways of air entry into the spinal canal are postulated. Only sporadic cases are reported in the literature, and no series of more than 3 cases of this condition could be found. Up to December 2005, only 71 articles were identified that reported on 86 cases. In association with bronchial asthma, only 13 cases are reported in the entire literature, caused by violent coughing and acute increase in the intra-thoracic pressure.[1]

Free air in the epidural space is an uncommon phenomenon that was first reported by Gordon et al. in 1977.[2] This condition was described under various terms ? intra-spinal pneumocele or pneumocele; spinal, epidural and subarachnoid pneumatosis; spinal and epidural emphysema; aerorachia; pneumosaccus; air myelogram; pneumomyelography; etc., by different authors. Newbold et al. introduced the term pneumorrhachis in 1987.[3]

PR usually constitutes an asymptomatic, often underdiagnosed, epiphenomenon of coincident underlying injuries and diseases. With the advent of more advanced imaging techniques, more cases are being reported.

For descriptive purposes, PR can be classified into internal, intradural (intra-spinal air within the subdural or subarachnoid space), external and extradural (intra-spinal, epidural air).[1, 2] External PR is usually asymptomatic and often an accidental radiological finding, whereas internal traumatic PR is often associated with fatal outcomes.[4]

Air can enter the epidural space from direct penetrating injuries of the spine. In bronchial asthma, probably, rupture of a peripheral pulmonary alveolus due to sudden increase in the intra-alveolar pressure could be the initial event. The leaked air into the pulmonary perivascular interstitium dissects the path of least resistance from the mediastinum to the facial planes of the neck. There are no facial barriers to prevent communications of the posterior mediastinum or the retro-pharyngeal space with the epidural space. Air thus freely communicates via the neural foramina and collects in the epidural space.[5] Because of the low resistance from the loose connective tissues as compared with the rich vascular network present anteriorly, the dissected air preferably collects in the posterior epidural space.[6]

Epidural PR may broadly be classified into iatrogenic, nontraumatic and traumatic.[7] Iatrogenic occurrences are the most common and result from the administration of epidural anesthesia. Nontraumatic occurrences have been described following spontaneous or non-trauma-related pneumomediastinum or pneumothorax.[5] The combination of pneumomediastinum with epidural pneumorrhachis without thoracic trauma has rarely been reported in the literature.

Almost exclusively, PR is found in combination with incidences of associated air distribution in other body cavities and facial compartments. These include pneumocephalus, pneumothorax, pneumomediastinum, pneumopericardium, subcutaneous emphysema, etc. In our patient, it was associated with the last three entities. We did not go for a CT head to diagnose a pneumocephalus.

As it is evident, PR is not a primary clinical diagnosis and is often detected accidentally. The diagnostic work-up of patients with PR should necessarily include plain roentgenograms and CT scanning of the spine.

In the differential diagnosis, intra-spinal gas collection due to degenerative,[8] malignant,[9] inflammatory[10] and infectious diseases, caused by gas-forming organisms, has to be considered. Furthermore, the coincidence of PR and intra-spinal gas also merits consideration.

Patients with pneumorrhachis are usually managed conservatively. Most of the times, PR is asymptomatic and constitutes a radiological curiosity. The intra-spinal air reabsorbs spontaneously and completely into the circulation. Rarely, symptomatic PR with features of neurological compression and consequent deficits is reported.[11]

Because of the rarity of the condition, no standard guidelines exist for the management of PR. It is thought to be associated with increased morbidity and mortality if associated with air dissection in other body cavities.[12] All the contributing factors leading to PR have to be appropriately addressed. Some mechanical deficits in the dura may warrant neuro-surgical intervention.[13, 14]

Bronchoscopy for the removal of foreign body, transient high-flow oxygen therapy to facilitate nitrogen washout, etc., are employed in individual cases. The use of broad-spectrum antibiotics remains controversial,[15, 16] and some schools believe that it may be beneficial in preventing possible mediastinitis; however, the decision has to be individualized. Thus, the management of patients with PR has to be decided on an individual basis and often requires inter- and multi-disciplinary approach.


Notes

Source of Support: Nil

Conflict of Interest: None declared.

References
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2. Gordon IJ,Hardman DR. The traumatic pneumomyelogram. A previously undescribed entityNeuroradiologyYear: 1997131078865671
3. Newbold RG,Wiener MD,Vogler JB,Martinez S. Traumatic pneumorrhachisAJR Am J RoentgenolYear: 19871483 rd61563492895
4. Goh BK,Ng KK,Hoe MN. Traumatic epidural emphysemaSpineYear: 200429E5283015543056
5. Drevelengas A,Kalaitzoglou I,Petridis A. Pneumorrhachis associated with spontaneous pneumomediastinumEur J RadiolYear: 19941812238055982
6. Eesa M,Kandpal H,Sharma R,Misra A. Spontaneous pneumorrhachis in bronchial asthmaAct RadiolYear: 2006476724
7. Atalar M,Do?an T,Cevit ?,G?m?? C. Epidural pneumorrhachis accompanying to spontaneous pneumomediastinum in a boy: A rare associationTurkish Respiratory JYear: 20078602
8. Ford LT,Gilula LA,Murphy WA,Gado M. Analysis of gas in vacuum lumbar discAJR Am J RoentgenolYear: 199712810567414544
9. Kennedy C,Phillips R,Kendall B. Epidural gas: AN unusual complication of metastatic oesophageal caNeuroradiologyYear: 1990326792333137
10. Burke V,Mall JC. Epidural gas: AN unusual complication of Crohn diseaseAJNR Am J NeuroradiolYear: 1984510566421117
11. Uemura K,Behr R,Roosen K. Symptomatic intra spinal air entrapmentBr J NeurosurgYear: 200014154610889894
12. Coniglio M,De Santis M,Pizzi G,Francioni F,Ricci P. Pneumorachide associato a pneumomediastino spontaneoDesrizione di un caso [pneumorrhachis associated with spontaneous pneumomediastinum. A Case report (in Italian)] Radiol Med (Torino)Year: 1997945312
13. Mangiardi JR,Brisson P,Weitzner I Jr,Bednar M. Traumatic intraspinal pneumoceleSurg NeurolYear: 19872758783576436
14. Oertel MF,Korinth MC,Reinges MH,Gilsbach JM. Pneumorrhachis of the entire spinal canalJ Neurol Neurosurg PsychiatryYear: 200576103615965224
15. Koelliker PD,Brannam LA. Epidural pneumatosis associated with spontaneous pneumomediastinum. Case report and review of literatureJ Emerg MedYear: 1999172475010195480
16. Krishnam,Mallick A. Air in the epidural space leading to a neurological deficitAnaesthesiaYear: 200358292312603477

Figures

[Figure ID: F0001]
Figure 1 

Chest x-ray showing extensive subcutaneous emphysema and pneumomediastinum



[Figure ID: F0002]
Figure 2 

CT chest showing pneumomediastinum, pneumopericardium and surgical emphysema



[Figure ID: F0003]
Figure 3 

CT chest showing extensive surgical emphysema and pneumomediastinum



[Figure ID: F0004]
Figure 4 

CT chest showing posterior pneumorrhachis



Article Categories:
  • Case Report

Keywords: Bronchial asthma, computed tomography, intra-spinal air, pneumorrhachis .

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