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American Journal of Public Health Research
DOI: 10.12691/ajphr-1-1-3

The Implementation of a Tele-Homecare System with Service Oriented Architecture and HL7 Message Transmission Standard
Ching-Sung Wang

American Journal of Medical and Biological Research
DOI: 10.12691/ajmbr-1-1-2

The Influence of Ethanol on Pyruvate Kinases Activity in Vivo, in Vitro, in Silico
Lelevich Sergey Vladimirovich, Khrustalev Vladislav Victorovich, Barkovsky Eugene Victorovich, Shedogubova Tatyana Aleksandrovna

American Journal of Medical Sciences and Medicine
DOI: 10.12691/ajmsm-1-1-1

Clinical and Histological Features in Wegener Granulomatosis
Li Cavoli Gioacchino, Passantino Rita, Bono Luisa, Tortorici Calogera, Ferrantelli Angelo, Giammarresi Carlo, Rotolo Ugo

American Journal of Infectious Diseases and Microbiology
DOI: 10.12691/ajidm-1-1-2

The Curse of Lymphatic Filariasis: Would the Continual Use of Diethylcarbamazine Eliminate this Scourge in Papua New Guinea?
Anne Lanham, Lillian Mwanri

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American Journal of Infectious Diseases and Microbiology
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Tension Pneumothorax as the Presentation of Pulmonary Langerhans Histiocytosis

Conor McCartney*, Andrew Freeman, Ravi Nayak*

Department Internal Medicine, St. Louis University, United StatesInternal Medicine, Program Director Division of Pulmonary, Critical Care and Sleep Medicine, St. Louis University, United States

Abstract: A 36 year old male smoker presented to the emergency department with shortness of breath. A chest x-ray showed a tension pneumothorax. Chest tube was placed urgently. CT scan showed upper lobe predominant cystic lung lesions. Biopsy confirmed the diagnosis of pulmonary Langerhans Histiocytosis.

Keywords: tension pneumothorax, pneumothorax, langerhans, histiocytosis, lagerhans histiocytosis

Citation: Conor McCartney, Andrew Freeman, Ravi Nayak, Tension Pneumothorax as the Presentation of Pulmonary Langerhans Histiocytosis. American Journal of Medical Case Reports, 4 (5). 173-174. January 2016. doi:10.12691/ajmcr-4-5-8

1. Introduction

Langerhaan’s histiocytosis is rare disease affecting 1-2 cases per million [1]. Langerhans is characterized by a proliferation of bone marrow derived dendritic cells [2] that cause localized destruction at sites of proliferation. Gold standard for diagnosis is positive stain for CD1a cells [1,2,3,4]. The disease is classified based on organ involvement. Langerhans can be either multi organ system or single organ system. Pulmonary Langerhans histiocytosis can occur as part of a multisystem disease or isolated entity. Isolated pulmonary Langerhans accounts for 20% of adult disease [1].

2. Case

A 36 year old male smoker with no past medical history presented to the emergency department. He had first noted feeling ill five days prior with nasal drainage, night sweats and non-productive cough. Two days prior to admission he began experiencing right sided chest pain that was sharp, sudden in onset and unrelenting. When the chest pain wouldn’t go away he came into the emergency department.

A chest X-ray in the emergency department showed a tension pneumothorax. A chest tube was immediately placed; repeat chest X-ray showed resolution of the tension pneumothorax and interstitial prominence and cystic changes. CT of the chest with contrast showed upper lobe predominance of cystic lesions.

The patient was admitted to the hospital for management of his pneumothorax and further work up of his underlying condition. A quantiferon gold, PPD and AFB smear x1 were all negative. Bronchoscopy was considered but thought to be relatively low yield. Cardiothoracic surgery took the patient to the OR for a video-assisted thoracoscopic surgery (VATS) biopsy.

Figure 1. Portable Chest X-ray in the Emergency Department

Lung biopsy showed on three of three samples the specimen was stained with S-100 that showed few scattered individual Langerhans histiocytes. Cd1a stain was positive showing few scattered focal collections of Langerhans cells.

Figure 2. High Resolution CT scan done one day after admission shows an upper lobe predominant cystic disease

Further work up for extra pulmonary manifestations was planned as an outpatient. Unfortunately the patient was rehospitalized three times in the following 8 months for recurrent pneumothorax. He missed scheduled appointments and was briefly hospitalized for suicidal ideation. In total he only made one scheduled pulmonary appointment before being lost to follow up.

3. Importance

Pulmonary Langerhans is an upper lobe predominant nodular and cystic lung disease [3]. It is strongly associated with smoking [5] and smoking cessation is the critical [1,3] therapy for patients. Outcomes are variable and range from spontaneous resolution to progression to respiratory failure [3]. For those that progress to respiratory failure lung transplantation is a viable option [5].

The diagnosis of Pulmonary Langerhans Histiocytosis should be strongly considered in smokers presenting with pneumothorax and bilateral indeterminate infiltrates [3]. Definitive diagnosis is made by biopsy and stain with for CD1a cells [1].

References

[1]   | Stockschlaeder M, Sucker C. Adult Langerhans cell histiocytosis. European journal of haematology. 2006; 76(5):363-368.
[2]   | Abla O, Egeler RM, Weitzman S. Langerhans cell histiocytosis: Current concepts and treatments. Cancer treatment reviews. 2010;36(4):354-359.
[3]   | Suri HS, Yi ES, Nowakowski GS, Vassallo R. Pulmonary langerhans cell histiocytosis. Orphanet journal of rare diseases. 2012;7:16.
[4]   | Hoover KB, Rosenthal DI, Mankin H. Langerhans cell histiocytosis. Skeletal radiology. 2007;36(2):95-104.
[5]   | Dauriat G, Mal H, Thabut G, et al. Lung transplantation for pulmonary langerhans' cell histiocytosis: a multicenter analysis. Transplantation. 2006;81(5):746-750.