Two rare cases of laryngeal intralymphatic histiocytosis

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.

Intralymphatic Histiocytosis of the Appendix and Fallopian Tube Associated With Primary Peritoneal High-Grade, Poorly Differentiated Adenocarcinoma of Müllerian Origin

“…In 2014, a group of Korean authors described a case of ILH involving the buccal mucosa of a 35-year-old Korean woman, which clinically mimicked oral lymphangioma circumscriptum. 20 Two additional ILH cases of noncutaneous ILH in the head and neck area were reported in 2014 by Reznitsky et al 21 Both patients were male, 39 and 35 years old and were found to have swelling of the laryngeal area. Biopsies in both cases revealed aggregates of histiocytes in the lumen of submucosal dilated blood vessels consistent with ILH.…”

Persistent Laryngeal Swelling Caused by Primary Intralymphatic Histiocytosis

“…Discussion | Primary ILH of the larynx has, to our knowledge, only been described in 2 (male) patients before. 6 These patients had very similar clinical presentations, with persistent swelling of the epiglottis and aryepiglottic folds, and without cutaneous manifestations. Likewise, exacerbations of laryngeal swelling were described, 6 which even required 1 patient to be admitted to the intensive care unit.…”

“…Primary ILH of the larynx has, to our knowledge, only been described in 2 (male) patients before . These patients had very similar clinical presentations, with persistent swelling of the epiglottis and aryepiglottic folds, and without cutaneous manifestations.…”

“…These patients had very similar clinical presentations, with persistent swelling of the epiglottis and aryepiglottic folds, and without cutaneous manifestations. Likewise, exacerbations of laryngeal swelling were described, which even required 1 patient to be admitted to the intensive care unit. Importantly, intralymphatic histiocytes may also be found on histopathologic analysis as part of inflammatory changes caused by an (ILH-associated) inflammatory or malignant disease, which is referred to as secondary ILH .…”