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Open AcceCase reportMassive lung collapse with partial resolution after several years: a case reportElke Govaere1, Dirk Van Raemdonck2, Hugo Devlieger1, Maria-Helena Smet3, Eric Verbeken4, Marijke Proesmans1 and Kris De Boeck*1

Address: 1University Hospital of Leuven, Dept of Pediatrics, Herestraat 49, 3000 Leuven, Belgium, 2Dept Thoracic Surgery, Herestraat 49, 3000 Leuven, Belgium, 3Dept Radiology, Herestraat 49, 3000 Leuven, Belgium and 4Dept of Pathology, Herestraat 49, 3000 Leuven, Belgium

Email: Elke Govaere - Elke.Govaere@olvz-aalst.be; Dirk Van Raemdonck - Dirk.Vanraemdonck@uz.kuleuven.ac.be; Hugo Devlieger - Hugo.Devlieger@uz.kuleuven.ac.be; Maria-Helena Smet - Marleen.Smet@uz.kuleuven.ac.be; Eric Verbeken - Eric.Verbeken@uz.kuleuven.ac.be; Marijke Proesmans - Marijke.Proesmans@uz.kuleuven.ac.be; Kris De Boeck* - christiane.deboeck@uz.kuleuven.ac.be

* Corresponding author

AbstractBackground: Bronchitis obliterans is a severe and extremely rare complication of respiratorytract infections in children and is characterized by massive atelectasis and collapse of the affectedlung. Of the rare cases reported in the literature all surviving children underwent surgical resectionof the collapsed lung.

Case presentation: We report an infant with bronchitis obliterans that was treatedconservatively. 5 years after the initial event, partial lung re-expansion was documented.

Conclusion: This case therefore supports a conservative treatment whenever possible withpneumonectomy only as a last treatment option.

BackgroundIn children bronchiolitis obliterans occurs most com-monly secondary to infection [1]. Rarely the fibroblasticobliteration involves the more central, cartilage-contain-ing bronchi, leading to bronchitis obliterans, characterisedby massive atelectasis and collapse of the affected lung.There are only five case reports of bronchitis obliterans [2-6]. All patients underwent surgery except one who diedearly in the course. We report a case of obliterative bronchi-tis without bronchiolar involvement following bilateralbronchopneumonia in a 9-month-old boy. Massive lungcollapse occurred with partial spontaneous re-expansion5 years later. To our knowledge, this is the first case reportdocumenting such an evolution. Therefore, if possible,initial conservative treatment should be considered.

Case presentationAS was born by caesarean section at the postmenstrual ageof 38 weeks. At the age of nine months, he was admittedto a regional hospital with cough, high fever and moder-ate tachypnea. Chest X-ray revealed bilateral pulmonaryinfiltrates and the child was treated with intravenous anti-biotics. Ten days later he was referred to the universityhospital because of persistent cough, increasing respira-tory distress and atelectasis of the right lower and middlelobes on chest X-ray.

At referral we saw a pale and dystrophic child in moderaterespiratory distress. Weight was on the third percentile fora height on the 75th percentile. The child was afebrile. Hehad chest hyperinflation, tachypnea (60 per minute) and

Published: 21 October 2005

BMC Pediatrics 2005, 5:39 doi:10.1186/1471-2431-5-39

Received: 15 March 2005Accepted: 21 October 2005

This article is available from: http://www.biomedcentral.com/1471-2431/5/39

© 2005 Govaere et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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intercostal retractions. Fine rales were heard over the righthemithorax. Transcutaneous oxygen saturation was 92%when awake but dropped below 90% during sleep. Whiteblood cell count was 22900 per microliter (42% neu-trophils), C-reactive protein was slightly elevated at 25mg/L (normal value below 5 mg/L). Bronchoscopy dem-onstrated patent central airways: no mucus plug, no for-eign body, no external airway compression. There wassome erythema of the mucosa and scanty secretions in theright bronchial tree. Neutrophils (90%; normal less than20%) predominated in the bronchoalveolar lavage fluid.Haemophilus influenzae and Moraxella cattharalis were iso-lated from the bronchial aspirate; no fungi nor mycobac-teria were isolated. Viral cultures and/or paired serologyfor respiratory syncytial virus, cytomegalovirus, adenovi-rus, influenza, parainfluenza and Mycoplasma pneumoniaeremained non-diagnostic. Serum immunoglobulins werewithin the normal range for age. The sweat chloride was13 mmol/l. (normal

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associated with radiological changes. The necessity of lungbiopsy to make the diagnosis is under debate [8,10].

Relative to obliterative bronchiolitis, obliterative bronchi-tis due to obliteration of the segmental and subsegmentalbronchi without bronchiolar involvement is rare. Unilat-eral pulmonary collapse is the most frequent presenta-tion. The pathogenesis is uncertain [11]. Only five reportsdescribe bronchitis obliterans [2-6]. All children under-went pneumonectomy except one patient who died earlyin the course. Two cases were presumed to be of viral eti-ology [4,6], one case was related to Mycoplasma pneumo-niae [2] infection. Two other cases were associated withStevens-Johnson syndrome [3] andhypogammaglobulinemia [5]. We describe a first case ofbronchitis obliterans treated conservatively in whom par-tial lung re-expansion occurred five years after the onset ofdisease.

Our case, a previous healthy 9-month-old boy, developeda severe respiratory distress with collapse of the right lung,following a bilateral bronchopneumonia. Microscopicexamination demonstrated normal collapsed lung tissuethat was histologically normal and slightly thickened vis-ceral pleura and interlobular septa. No abnormalities ofthe membranous and respiratory bronchioles were identi-

fied, so the diagnosis of bronchiolitis obliterans can beexcluded. The central cartilaginous bronchi were notpresent in the biopsy, so the diagnosis of bronchitis oblit-erans was made on clinical and radiological ground butcould not be confirmed histologically since no lung resec-tion was performed. However, what other diagnosiswould be possible when bronchiolitis obliterans isexcluded, a foreign body, local airway compression andmucus plug are ruled out and the lung collapse is total andresistant to selective intubation and ventilation (even sup-plemented with surfactant instillation and steroidtreatment).

Chest X-ray shows persisting total atelectasis of the right lungFigure 1Chest X-ray shows persisting total atelectasis of the right lung. Note the marked loss of volume on the right, pro-nounced shift of the mediastinum to the right, and compensa-tory overexpansion of the left lung.

A High resolution computed tomography demonstrates par-tial re-expansion of the right lung with very moderate bronchiectasisFigure 2A High resolution computed tomography demonstrates par-tial re-expansion of the right lung with very moderate bron-chiectasis. B The right lower lobe remains collapsed and shows more extensive bronchiectasis.

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Because of relatively mild respiratory symptoms and therisk of a post-pneumonectomy syndrome [12], a conserv-ative approach was maintained. To our surprise, five yearsafter the initial diagnosis, a partial re-expansion of theaffected right lung was noted on high-resolution com-puted tomography. This is the first case ever describedwith a comparable evolution. Could recanalization oflarger airways be the cause? Reversibility of pulmonaryinvolvement is documented in bronchiolitis obliteransorganising pneumonia (BOOP) and other diseases like idi-opathic interstitial pneumonia but has not been describedin bronchitis obliterans.

ConclusionThis case illustrates that for children with bronchitis oblit-erans, surigical resection of the collapsed lung may notalways be necessary. In our opinion, conservative treat-ment should be considered when possible. Pneumonec-tomy or lobectomy should only be reserved as a lastoption.

List of abbreviationsBOOP: bronchiolitis obliterans organising pneumonia

Competing interestsThe author(s) declare that they have no competinginterests.

Authors' contributionsClinicians responsible for patient management: M. Proes-mans, E. Govaere, K. De Boeck

Surgeon who performed surgery: D. Van Raemdonck

Intensivist: H. Devlieger

Radiologist: M.H. Smet

Pathologist: E. Verbeken

AcknowledgementsWritten consent was obtained from the patient's parents for publication of the study

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10. Estenne M, Maurer JR, Boehler A, Egan JJ, Frost A, Hertz M, MalloryGB Jr, Snell GI, Yousem S: Bronchiolitis Obliterans Syndrome2001: an update of the diagnostic criteria. J Heart LungTransplant 2002, 21(3):297-310.

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Pre-publication historyThe pre-publication history for this paper can be accessedhere:

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AbstractBackgroundCase presentationConclusion

BackgroundCase presentationDiscussionConclusionList of abbreviationsCompeting interestsAuthors' contributionsAcknowledgementsReferencesPre-publication history