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Two Rare Cases of Laryngeal Intralymphatic Histiocytosis

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Date 2015 Apr 15
PMID 25870125
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Abstract

We report two rare cases of intralymphatic histiocytosis causing, respectively, recurrent and persistent episodes of upper airway swelling and breathing difficulties. Case 1 was a 39-year-old man who was referred with recurrent upper airway swelling causing difficulty in breathing. A direct laryngoscopy was performed under general anesthesia due to minimal effect from treatment with antibiotics and anti-oedema medication. On examination, the larynx was found to be swollen and oedematous but not inflamed. Biopsies from the aryepiglottic folds showed intralymphatic histiocytosis. The patient was extensively examined but the only abnormal finding was a low CD4 count. The breathing difficulties fluctuated during the diagnostic process and settled after a year. Case 2 was a 35-year-old man who presented with persistent laryngeal swelling. Biopsies from the epiglottis showed intralymphatic histiocytosis. Extensive investigations were performed but discovered no abnormal findings. He received CO2 laser treatment twice and the swelling decreased. Intralymphatic histiocytosis is extremely rare in upper airway pathology. It is an important differential diagnosis in patients with recurrent and chronic laryngeal swelling and dyspnoea.

References
1.
Classen C, Haffner D, Nizze H, Moller P, Prall F . Endolymphatic non-langerhans cell histiocytosis of the larynx: report of an uncommon disease manifestation. J Pediatr Hematol Oncol. 2010; 32(5):e173-6. DOI: 10.1097/MPH.0b013e3181d746eb. View

2.
de Unamuno Bustos B, Garcia Rabasco A, Ballester Sanchez R, Martinez Aparicio A, Alegre De Miquel V . Erythematous indurated plaque on the right upper limb. Intralymphatic histiocytosis (IH) associated with orthopedic metal implant. Int J Dermatol. 2013; 52(5):547-9. DOI: 10.1111/j.1365-4632.2012.05696.x. View

3.
Wang Y, Yang H, Tu P . Upper facial swelling: an uncommon manifestation of intralymphatic histiocytosis. Eur J Dermatol. 2013; 22(6):814-5. View

4.
Lee H, Kim S, Kim W, Lee K . Transoral CO(2) Laser Resection for Post-Radiation Arytenoid Edema. Clin Exp Otorhinolaryngol. 2011; 3(4):229-32. PMC: 3010544. DOI: 10.3342/ceo.2010.3.4.229. View

5.
Okazaki A, Asada H, Niizeki H, Nonomura A, Miyagawa S . Intravascular histiocytosis associated with rheumatoid arthritis: report of a case with lymphatic endothelial proliferation. Br J Dermatol. 2005; 152(6):1385-7. DOI: 10.1111/j.1365-2133.2005.06618.x. View