Radiotherapy for Langerhans cell histiocytosis with paraplegia: A rare oncologic emergency case report in infancy and literature review |
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Authors: | Kentaro Nakashima Yuhki Koga Yasunari Sakai Hidetoshi Takada Katsumi Harimaya Saiji Ohga Tomoaki Taguchi Yoshinao Oda Hiroshi Honda Shouichi Ohga |
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Institution: | 1. Department of Pediatrics, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan;2. Department of Perinatal and Pediatric Medicine, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan;3. Department of Orthopedic Surgery, Kyushu University, Fukuoka, Japan;4. Department of Radiology, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan;5. Department of Pediatric Surgery, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan;6. Department of Pathology, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan |
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Abstract: | BackgroundLangerhans cell histiocytosis (LCH) is a clonal disease with focal or disseminated lesions that may compress the surrounding tissues, including the spinal cord. Because few reports have described the spinal symptoms as the first manifestation of pediatric LCH, the long-term neurological outcomes remain unclear.Case report and literature reviewWe report a 21-month-old boy who presented with sudden-onset paraplegia. Imaging analyses revealed that osteolytic lesions and epidural tumors compressing the spinal cord at the T7-9 vertebrae. Twelve days after he developed leg weakness, emergency radiotherapy was started after a tumor biopsy. During the course of radiotherapy, paralysis steadily ameliorated. After we excluded infections and determined the pathological diagnosis of LCH, multi-drug chemotherapy was started. Apparent improvement in his complete paraplegia was observed after a total 15?Gy of radiotherapy and subsequent chemotherapy, leaving no neurological sequelae at 4?years of age. Through a literature search of studies published from 1980 to 2017, we found that children with LCH showed a generally favorable recovery from neurological dysfunction after the acute phase of spinal symptoms.ConclusionThis report underscores the utility of emergency radiotherapy for the neurological recovery of spinal LCH in infants. Our long-term observation further denotes the value of this treatment in terms of the intact survival with preserved motor functions and physical growth. |
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Keywords: | Langerhans cell histiocytosis Spine Paraplegia Radiation therapy |
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