ORIGINAL ARTICLE

The Clinical Respiratory Journal

Pneumomediastinum as a complication of critical pertussis Francesco Monaco1, Mario Barone1, Valeria G. Manfredi1, Rosario Marando1, Flavia Nunnari1, Antonio David2, Maurizio Monaco1 and Antonio Cascio3 1 Thoracic Surgery Unit, Cardiovascular and Thoracic Department, Policlinic University Hospital, University of Messina, Messina, Italy 2 Department of Neurosciences, Psychiatric and Anesthesiological Sciences, University of Messina, Messina, Italy 3 Infectious Diseases Unit, Department of Human Pathology, Policlinic University Hospital, University of Messina, Messina, Italy

Abstract Background and Aims: Pertussis is a common and potentially serious disease affecting mainly infants and young children. In its non-classic presentation, pertussis can be clinically indistinguishable from other respiratory illnesses. Pertussis today often remains underdiagnosed in adults. Our aims was to report a complicated cases of pertussis. Results: A case of serologically confirmed pertussis occurred in an 18-year-old man presenting with pneumomediastinum, subcutaneous emphysema in the neck and chest, and persistent attacks of coughing with apnea that required treatment in the intensive care unit. Conclusion: Pneumomediastinum and subcutaneous emphysema have never been described in adult patients with pertussis. Physicians should be aware that patients presenting with persistent cough and pneumomediastinum may have pertussis and include this in their differential diagnosis.

Key words Bordetella pertussis – cough – pertussis – pneumomediastinum Correspondence Antonio Cascio, MD, PhD, UOC Malattie Infettive, Policlinico ‘G. Martino’, Via Consolare Valeria n. 1, 98125 Messina, Italy. Tel: +39 090 2213680 Fax: +39 090 692610 email: [email protected] Received: 11 April 2014 Revision requested: 07 February 2015 Accepted: 01 March 2015 DOI:10.1111/crj.12285

Please cite this paper as: Monaco F, Barone M, Manfredi VG, Marando R, Nunnari F, David A, Monaco M and Cascio A. Pneumomediastinum as a complication of critical pertussis. Clin Respir J 2015; ••: ••–••. DOI:10.1111/crj.12285.

Authorship and contributorship Literature search: Valeria G. Manfredi, Rosario Marando and Antonio Cascio; Data collection: Francesco Monaco, Mario Barone, Valeria G. Manfredi, Rosario Marando, Maurizio Monaco, Antonio Cascio and Flavia Nunnari; Analysis and interpretation: Valeria G. Manfredi, Rosario Marando and Antonio Cascio; Manuscript preparation: Valeria G. Manfredi, Francesco Monaco, Mario Barone, Antonio Cascio and Antonio David; Review of manuscript: Flavia Nunnari, Antonio Cascio, Antonio David and Maurizio Monaco. Ethics This work was created in accordance with the ethical standards of the Declaration of Helsinki. Conflict of interest The authors have stated explicitly that there are no conflicts of interest in connection with this article.

Introduction Pertussis is a highly contagious bacterial disease caused by Bordetella pertussis. It is recognized to be a

The Clinical Respiratory Journal (2015) • ISSN 1752-6981 © 2015 John Wiley & Sons Ltd

significant cause of morbidity and mortality in young children worldwide. Its incidence may have been underestimated in recent years, as immunized children and adults may have only catarrhal symptoms or mild

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Pneumomediastinum and pertussis

Monaco et al.

bronchitis (1). Most healthy subjects will make a full recovery from pertussis, although infants and individuals with significant comorbidity may experience a higher risk of complications and mortality. A clinically severe form of pertussis can lead to critical conditions, with symptoms mimicking other respiratory diseases (2–4). Infants and adolescents with critical pertussis may require treatment in the intensive care unit (ICU) if they experience prolonged and recurring episodes of apnea and coughs, leading to cardiorespiratory imbalances necessitating sedation treatment (5). Here we report the case of an 18-year-old male who presented to our observation with pneumomediastinum and subcutaneous emphysema, and who required mechanical ventilation in ICU.

Case report An 18-year-old male was admitted to our University Hospital complaining of a cough for 2 weeks, and of dyspnea for 6 h. He had come from a peripheral hospital where he had already received medical therapy with corticosteroids, antihistamines and bronchodilators for a week, without benefit. Personal history was negative for respiratory diseases, critical care admission, prematurity and recreational drug use. Family history was also negative for bronchial asthma or other respiratory diseases. On admission, he appeared severely ill; vital signs were quite unstable: body temperature, 40°C; blood pressure, 130/70 mmHg; heart rate, 140 beats/min.; respiratory rate, 25 breaths/min and PO2/FiO2 ratio was 350. WBC count was 25 900 cells/mm3 (lymphocytes, 42%); other blood tests were in the normal range. A crackling feel to the touch of the upper part of the chest and neck, up to the mandibles, bilaterally, was noted and a clinical diagnosis of subcutaneous emphysema was made. Computed tomography (CT) scan showed pneumomediastinum and soft tissue emphysema of the chest wall, supraclavicular regions and neck, up to the mandibles. Irregularities of the posterior tracheal wall in correspondence of T1 vertebra were also seen (Fig. 1). No changes were documented by CT in the lung parenchyma. Flexible optical fibre bronchoscopy documented intense mucosal hyperemia of the entire tracheobronchial tree and the presence of a large quantity of thick sticky clear mucus. A more careful analysis highlighted a tracheal tear at the level of the 4th tracheal ring extending longitudinally for about 2.5 cm (imaging not available). Bronchoalveolar lavage was performed with samples sent for cytological, microbio-

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Figure 1. Computed tomography (CT) scan. The white arrow indicates the irregularities of the posterior tracheal wall in correspondence of T1 vertebra.

logical and mycobacterium analysis, which all returned negative. The patient was soon started on fluid therapy and intravenous ceftriaxone 2 g/day. After consultation for infectious disease, a diagnosis of pertussis was considered. The patient’s mother referred that her son had never been vaccinated against pertussis. Serological tests for B. pertussis and nasopharyngeal swab cultures were performed, and clarithromycin 500 mg twice daily was added to ceftriaxone. The diagnosis of pertussis was then confirmed once NovaLisa (NovaTec Immundiagnostica GmbH, Dietzenbach, Germany) enzyme-linked immunosorbent assays showed: IgG anti B. pertussis toxin (PT) and filamentous hemagglutinin (FHA) = 31.00 NovaTec Units (NTU) (positive: >11); IgA anti-PT and FHA = 14.00 NTU (positive: >11); and IgM anti purified lysate antigens of B. pertussis = 16.00 NTU (positive: >11); even if nasopharyngeal swab cultures were negative. Once the positive result of pertussis serology was obtained, ceftriaxone was stopped. During hospitalization, despite therapy, the patient continued to present numerous coughing episodes and much chest pain, treated with nonsteroidal antiinflammatory drugs. On day 10, the patient experienced a severe episode of post-tussive apnea followed by cardiac arrest. He was intubated and a chest tube positioned, then transferred to ICU where he stayed for 3 days. The patient was subjected to a pressurecontrolled ventilation for 24 h, with an inspiratory pressure of 14 cmH2O (and related tidal volume between 6 and 8 mL/kg), respiratory rate of 10 breaths per minute, Ti/Ttot 20%, levels of FiO2 of 30%, with

The Clinical Respiratory Journal (2015) • ISSN 1752-6981 © 2015 John Wiley & Sons Ltd

Monaco et al.

improvement of clinical, radiological and blood gas parameters. General condition and respiratory symptoms improved with gradual reduction and then disappearance of subcutaneous emphysema and pneumomediastinum. Ten days after intubation, bronchoscopy was performed to assess for healing. Antibiotic therapy was discontinued after 21 days and the patient was finally discharged home, symptom free, 30 days after admission. The patient had outpatient follow-up for 1 year, without presenting respiratory symptoms.

Discussion Whooping cough is typically characterized by three phases: catarrhal, paroxysmal and convalescent. The first phase lasts for 1–2 weeks during which patients experience coryza and intermittent cough, high fever is rare. During the paroxysmal phase, which lasts 4–6 weeks, classic symptoms are spasmodic cough, posttussive vomiting and inspiratory whoop. During the recovery phase, which usually lasts 2–6 weeks, symptoms slowly improve. In adults, B. pertussis infection covers a range from asymptomatic (in patients with some level of immunity) and mild cough illness to classic pertussis. In its non-classic presentation, pertussis can be clinically indistinguishable from other respiratory illnesses (6). Pertussis is often underdiagnosed in adults, and complications because of severe paroxysmal coughing have been reported in adults and adolescents. These include subconjunctival hemorrhages, rib fractures, herniated lumbar disk, pneumothorax, inguinal hernia, hearing loss, cough syncope and carotid artery dissection (7–14). Pneumomediastinum or mediastinal emphysema may be spontaneous, caused by pulmonary barotrauma; iatrogenic, usually because of endoscopic maneuvers that can cause perforation of the viscera examined; or pathologic, as a complication of injury to the organs that are located in the mediastinum, e.g. spontaneous perforation of the esophagus in the neoplastic processes (15, 16). Spontaneous pneumomediastinum is most commonly seen in men in their 20s (17), and tends to occur after intense episodes of increased intrathoracic pressure (e.g. coughing, straining, vomiting, strenuous crying), especially in people with asthma or chronic obstructive pulmonary disease. Risk factors include inhalation drug use (e.g. marijuana, cocaine), athletic exertion and mechanical ventilation. Most patients with spontaneous pneumomediastinum present with chest pain, and up to half experience dyspnea (18, 19).

The Clinical Respiratory Journal (2015) • ISSN 1752-6981 © 2015 John Wiley & Sons Ltd

Pneumomediastinum and pertussis

The occurrence of pneumomediastinum and subcutaneous emphysema in patients with pertussis is extremely rare and has never been described in adults. In international literature, there is only one Italian report by Gandullia et al., dated 1964, describing a case that occurred in a child (20). No other cases have been described thereafter. In our case, pneumomediastinum was caused by a lesion of the trachea secondary to the violence of coughing. In adult patients with pertussis, a treatment with active antitussive drugs should be recommended (21). There are no ventilator strategies codified and universally accepted for the management of mechanical ventilation in patients with primary or secondary pneumomediastinum. Certainly, it is necessary to practice the appropriate therapy to the resolution of underlying respiratory disease. In addition, for those patients in severe respiratory failure, it is advisable to perform a protective mechanical ventilation, aimed to minimizing the risk of developing a ventilationinduced lung injury. Although mechanical ventilation may cause air leaks, including pneumomediastinum, continuing the mechanical ventilation and even escalating respiratory support may be necessary depending on the severity of the underlying respiratory distress and the degree of compromise caused by the air leak. Principle objectives include the use of the lowest pressures (maintaining a plateau pressure

Pneumomediastinum as a complication of critical pertussis.

Pertussis is a common and potentially serious disease affecting mainly infants and young children. In its non-classic presentation, pertussis can be c...
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