Isolated skeletal Langerhans cell histiocytosis in adults: A case report.

INTRODUCTION AND IMPORTANCE: LCH in adults is rarely encountered, with the preference in children and axial skeleton as predilection site. Limited understanding of adult LCH causes frequent misdiagnosis, as our experience in an adult case of LCH threw off our differential diagnosis. CASE PRESENTATION: A 21-year-old male was referred to our hospital due to pain in his right shoulder. Plain radiograph and MRI showed a solitary well-marginated lytic lesion on the distal third of the clavicle. Together with a clear history and physical exam, the benign bone cyst was suspected and we performed an open biopsy simultaneously with curettage followed by internal fixation using a bone graft. Pathology and immunohistochemistry dismissed our suspicion and confirmed LCH as the main diagnosis. At six months post-surgery, no signs of recurrence were seen on the fixated site nor complained by the patient. DISCUSSION: Diagnosing LCH involves considering imaging appearances and patient demographics as initial clues. However, confirming the diagnosis requires a biopsy with proven CD1 expression. Currently, the majority of studies recommend confirming the diagnosis before initiating therapy. This precaution is necessary due to the unclear pathophysiology of LCH, which complicates the implementation of specific therapies. Based on benign features of skeletal lesions found from imaging, invasive treatment before biopsy confirmation still gave a satisfactory outcome despite not being in line with the current recommendation. CONCLUSION: Excisional biopsy and curettage in solitary LCH yield satisfactory outcomes. However, further studies are needed with larger sample sizes and interventional designs.

Longstanding Volkmann ischemic contracture of the forearm following traditional bonesetter practice successfully treated with combined flexor origin slide and tendon transfer: A case report.

INTRODUCTION AND IMPORTANCE: The Volkman ischemic contracture (VIC) of the forearm is a sequel of the compartment syndrome of the forearm. There were no studies reported the outcome of surgical treatment for VIC, particularly the muscle origin slide procedure, in treating the VIC that had been developed more than 20 years. CASE PRESENTATION: We reported a 34 years old right-hand dominant painter with fingers flexion contracture, decreased hand mobility and sensation on her right hand. She had history of closed right forearm fracture when she was 10 years old that was treated by a traditional bonesetter. A week afterward, she experienced clinical presentation of compartment syndrome. Not long thereafter, the pain subsided but she started to have stiffness and discomfort in her forearm and hand condition. Twenty-four years later, she wants to get treatment for her hand. We diagnosed her with VIC, moderate degree based on Tsuge Classification. We then performed flexor origin slide procedure and tendon transfer. In 1.5 year of follow-up, a satisfactory functional outcome was noted. CLINICAL DISCUSSION: Flexor origin slide was the preferred treatment if the flexors still retain adequate strength. During the surgery, severe degeneration of FPL and FDP was found. Therefore, we performed tendon transfer to increase the grip strength. CONCLUSION: VIC is not uncommon following fracture treated in the traditional bonesetter. For moderate VIC, the flexor origin slide procedure and tendon transfer could still give benefit, even in the case of treatment delay of more than 20 years from onset.