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Early-onset multisystem Langerhans Cell Histiocytosis with pulmonary and cutaneous involvement in a 3.5-month-old
Narges Khazaei1, Ruhollah Shirzadi2, Seyed Hossein Mirlohi2
1Pediatric Pulmonology, Pediatric Respiratory and Sleep Medicine Research Center, Hakim Children's Hospital, Tehran University of Medical, Sciences, Tehran, Iran.
Background:
Langerhans cell histiocytosis (LCH) is a rare clonal proliferative disorder characterized by the accumulation of Langerhans-type dendritic cells in various tissues, including the lungs and skin [1]. One of the major challenges in this disease is its early diagnosis, the most important reason being that pulmonary involvement is rare in this disease. [2].
Case Presentation:
A 3.5-month-old male infant presented to the emergency department of a children's hospital with symptoms of respiratory distress, mild fever, and vesicular-pustular skin lesions. Imaging studies revealed diffuse interstitial lung disease with bronchiectasis and cystic changes. Skin biopsy was positive for immunohistochemical markers CD1a and S-100, confirming the diagnosis of LCH. The patient initially required invasive mechanical ventilation and was subsequently managed with noninvasive mechanical ventilation and systemic corticosteroids, resulting in clinical improvement.
Conclusion:
Considering the patient presented, attention should be paid to Langerhans Cell Histiocytosis in infants who present with respiratory distress and skin lesions to enable early diagnosis and timely intervention.
