Progressive form of pulmonary Langerhans’ cell histiocytosis in a female adult non‐smoker

Benčić, Dubravka; Smojver, Silvana; Boras, Zagorka; Potočki, Kristina; Batinić, Drago

doi:10.1046/j.1440-1843.2003.00492.x

Cited by 2 publications

(1 citation statement)

References 23 publications

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“…Hastalığın genellikle sigara içenlerde görüldüğü söylenmekteyse de sigara öyküsü olmayan olgular da bildirilmiştir. 6 Nitekim erkek olgumuz sigara kullanırken kadın olgumuzda sigara öyküsü yoktu. Diğer çevresel faktörlerle de arasında ilişki bulunamamıştır.…”

Section: Olguunclassified

Two Cases of Langerhans Cell Histiocytosis with Initial Diagnosis of Tuberculosis

Yıldırım¹,

Karalezli²,

Şentürk³

et al. 2012

Solunum

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ÖZETLangerhans hücreli histiyositozis (LHH) etiyolojisi bilinmeyen bir hastalık grubudur. Bu yazıda uzun süre tanı konulamayıp tüber-küloz düşünülen LHH tanılı iki olgu sunulmaktadır.Olgu 1: Tüberküloz lenfadenit ön tanısıyla kliniğimize sevk edilen 21 yaşındaki kadın hastanın efor dispnesi vardı. Sigara kullanımı yoktu. İki kez sağ aksiller lenfadenit nedeniyle lenf nodu (LN) diseksiyonu geçirmişti ancak lezyon nüks etmekteydi. Toraks BT'sinde yaygın kistik lezyonları, aksiller LN biyopsisinde CD1a ve S100 ile boyanan Langerhans hücreleri görüldü.Olgu 2: Efor dispnesi ve polidipsi şikâyeti olan 27 yaşında erkek hasta polikliniğimize başvurdu. On paket/yıl sigara kullanım öyküsü vardı ama 2 yıl önce sigarayı bırakmıştı. On üç yaşında 3 kez spontan pnömotoraks geçirmiş ve sonuncusunda dekortikasyon yapılmıştı. Sekiz yıl önce şüpheli bir akciğer tüberkü-lozu nedeniyle antitüberküloz tedavi görmüştü. Toraks BT'sinde yaygın kistik lezyonlar mevcuttu. Hastaya, sigara içen genç erkek olması, rekürren spontan pnömotoraks öyküsü, toraks BT'deki görünümü nedeniyle LHH tanısı konuldu.LHH, nadir bir hastalık olduğu için, tıpkı iki olgumuzda görüldüğü gibi ayırıcı tanıda akla gelmeyebilmekte ve ülkemizde yaygın bir hastalık olan tüberküloz yanlış tanısıyla hastalara gereksiz tedaviler uygulanabilmektedir. Özellikle tipik radyolojik görünüm bu hastalığı düşündürmelidir.Anahtar Kelimeler: Langerhans hücreli histiositozis, pulmoner LHH, tüberküloz ABSTRACTLangerhans cell histiocytosis (LCH) is a group of diseases with unknown etiology. In this article, two cases of LCH with initial diagnosis of tuberculosis were reported.Case 1: A 21-year-old woman who was admitted with dyspnea with the prediagnosis of tuberculosis lenfadenitis. She was not a smoker. Lymph node (LN) dissection was done twice because of right axillary lymphadenitis. Computed tomography (CT) revealed common cystic lesions. Axillary LN biopsy showed CD1 and S100 positive Langerhans cells.Case 2: A 27-year-old male admitted with and polydipsia. He is an ex-smoker with a history of 10 packs/year cigarette smoking. He had had spontaneous pneumothorax three times. He had spontaneous pneumothorax three times at 13 year-old and underwent decortication at the last time. He had received antituberculosis treatment eight years ago. Common cystic lesions were observed in his thorax CT. He was diagnosed as LCH because he was a smoking young man with a recurrent spontaneous pneumothorax history and a typical view in his thorax CT.If the possibility of LCH is not considered as a distinctive diagnosis, in patients like these two cases, patients could be treated incorrectly for diagnosis of tuberculosis, which is a common disease in our country. Especially typical radiological appearance should lead to consideration of this disease.Keywords: Langerhans cell histiocytosis, pulmonary LCH, tuberculosis

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Section: Olguunclassified

Two Cases of Langerhans Cell Histiocytosis with Initial Diagnosis of Tuberculosis

Yıldırım¹,

Karalezli²,

Şentürk³

et al. 2012

Solunum

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Multisystem Langerhans cell histiocytosis in an adult non‐smoker treated with steroid therapy

Ito

Kakuta

et al. 2020

Respirology Case Reports

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We describe the case of a 29‐year‐old female non‐smoker who was treated with steroid therapy for a subacute exacerbation of multisystem Langerhans cell histiocytosis (MS‐LCH) with worsening lung, skin, and oral mucosal lesions. The patient developed pneumonia, and computed tomography (CT) showed multiple thin‐walled cavities. Transbronchial lung cryobiopsy (TBLC) specimens revealed Langerhans cells, which were positive for CD1a and S‐100 expression. Similar histological findings were detected in the submandibular gland, skin, and tooth. On the basis of these findings, the patient was diagnosed with MS‐LCH and subsequently treated with steroid therapy. From the literature review, case reports of non‐smokers with pulmonary lesions that worsened and required treatment are rare. Almost all cases recurred and needed additional treatment. This case study contributes to our understanding of the potential role of steroid therapy in MS‐LCH treatment. Additionally, TBLC is a novel, potentially safer, diagnostic tool that has not been previously described for LCH.

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Progressive form of pulmonary Langerhans’ cell histiocytosis in a female adult non‐smoker

Cited by 2 publications

References 23 publications

Two Cases of Langerhans Cell Histiocytosis with Initial Diagnosis of Tuberculosis

Two Cases of Langerhans Cell Histiocytosis with Initial Diagnosis of Tuberculosis

Multisystem Langerhans cell histiocytosis in an adult non‐smoker treated with steroid therapy

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