HE PATIENT wasafull-termfemaleinfantborn to a mother with genital condylomas. The infant had a hoarse cry from birth. At 5 months of age, she was brought to the emergency department with respiratory distress and aphonia. Her condition was diagnosed and she was treated for a pertussis-like illness. At 6 months of age, she returned with respiratory distress and stridor. She required intubation. Multiple nodular lesions in the subglottic area were resected by laser. The gross and microscopic appearances are shown in Figure 1, Figure 2, and Figure 3.The lesions were recurrent. She required a tracheostomy at 10 months of age to maintain airway patency. The lesions had spread to her bronchi and lungs. She undergoes laryngoscopy and excision of the lesions every 2 to 3 weeks. There are multicavitary cystic lesions in the lungs bilaterally as seen on the computed tomographic scan (Figure 4).
Multicentric reticulohistiocytosis (MRH) is a rare, class IIb non-Langerhans cell histiocytosis associated with skin and joint involvement. It is more prevalent (80%) in Caucasian females in their fifth to sixth decade of life. Patients usually demonstrate symptoms and signs of symmetric polyarthritis and papulonodular cutaneous lesions. In addition to skin and joints, multiple organs can be involved, such as the lung (pleural effusion, interstitial fibrosis, hilar lymphadenopathy), heart (pericardial effusion, myocarditis), gastrointestinal system, and urogenital system (genital tract and kidney). Pericardial involvement is a rare manifestation, and around three cases have been reported in the literature so far. Our case report is a valuable contribution to the literature, which aids clinicians in contemplating MRH as one of the differentials among patients presenting with pericardial effusion. We described the characteristics of MRH along with its differentiating features from other autoimmune conditions and management.
scite is a Brooklyn-based organization that helps researchers better discover and understand research articles through Smart Citations–citations that display the context of the citation and describe whether the article provides supporting or contrasting evidence. scite is used by students and researchers from around the world and is funded in part by the National Science Foundation and the National Institute on Drug Abuse of the National Institutes of Health.