18-month outcomes of heterologous bilateral hand transplantation in a child: a case report.

2017

35-44

2017 Sep

2352-4650

<p><strong>BACKGROUND: </strong>Although heterologous vascular composite allotransplantation has become a burgeoning treatment option for adult amputees, there have been no successful cases previously reported in children. Here, we describe the surgical, immunological, and neurorehabilitation details with functional outcomes 18 months after heterologous bilateral hand and forearm transplantation in an 8-year-old child with quadrimembral amputations and a previous kidney transplant.</p>

<p><strong>METHODS: </strong>2 years of extensive preparation by medical and surgical teams preceded the hand-forearm transplantation of this child. The initial immunosuppressive protocol included thymoglobulin, tacrolimus, prednisone, and mycophenolate mofetil. In July, 2015, our vascularised composite allotransplantation team did the first bilateral hand and forearm transplantation in a child, an 8-year-old boy with previous living-related kidney transplantation. The surgery included four teams working simultaneously on the donor and recipient limbs, aided by customised cutting guides that aimed to reduce ischaemia time. Following an extended length of time in hospital, skin biopsies and close monitoring of renal function and drug concentrations occurred weekly for the first 3 months and were slowly tapered to monthly, and then quarterly. Skin biopsies were also done when tissue rejection was suspected. Paediatric-specific rehabilitation techniques were applied to promote patient engagement during rehabilitation. Progress was assessed by monthly sensory and motor function tests during routine clinic visits and with serial functional brain imaging studies, including structural brain MRI, magnetoencephalography and transcranial magnetic stimulation.</p>

<p><strong>FINDINGS: </strong>The surgery lasted 10 h and 40 min. Vascular revision of the ulnar artery was required a few hours postoperatively. There were no further immediate postsurgical complications. Rejection episodes occurred throughout the first year but were reversed. An increase in serum creatinine led to the addition of sirolimus at 3 months after transplantation with concomitant reduction in tacrolimus targets. Sensibility to light touch was present by 6 months after transplantation. Intrinsic hand muscle innervation was present by 7-10 months after transplantation. At 18 months, the child had exceeded his previous adapted abilities. As of 18 months after transplantation surgery he is able to write and feed, toilet, and dress himself more independently and efficiently than he could do before transplantation. He remains on four immunosuppressive medications and functional neuroimaging studies have shown motor and somatosensory cortical reorganisation.</p>

<p><strong>INTERPRETATION: </strong>Hand transplantation in a child can be surgically, medically, and functionally successful under carefully considered circumstances. Long-term data on the functional trajectory, neurological recovery, psychological sequelae, and the potential late effect of immunosuppression are still needed to support broader implementation of paediatric vascular composite allotransplantation.</p>

<p><strong>FUNDING: </strong>The Children's Hospital of Philadelphia.</p>

10.1016/S2352-4642(17)30012-3

Lancet Child Adolesc Health

30169225

The Treatment of Juvenile Fibromyalgia with an Intensive Physical and Psychosocial Program.

2015

731-7

2015 Sep

1097-6833

<p><strong>OBJECTIVE: </strong>To assess the short-term and 1-year outcomes of children with fibromyalgia treated with intensive physical and occupational therapy (PT/OT) and psychotherapy.</p>

<p><strong>STUDY DESIGN: </strong>Children with fibromyalgia seen at a tertiary care hospital were treated with 5-6 hours of intensive PT/OT daily and at least 4 hours of psychosocial services weekly. All medications used for fibromyalgia were discontinued. Children underwent standardized testing, including a visual analog scale for pain; the Bruininks-Oseretsky Test of Motor Performance, Second Edition; the Bruce treadmill protocol; the Functional Disability Inventory; the Pain Stages of Change Questionnaire, adolescent version; and the Pediatric Quality of Life Inventory, Teen Report, at 3 time points: at program entry, at the end of the intensive program, and 1 year after the end of the program.</p>

<p><strong>RESULTS: </strong>Sixty-four children (median age, 16 years; 95% Caucasian; 94% female; median duration of symptoms, 21 months) were studied. The mean pain score decreased significantly from program entry to the end of the program (from 66 of 100 to 25 of 100; P = .001). At the 1-year follow-up, 33% reported no pain. All measures of function on the Bruininks-Oseretsky Test of Motor Performance, Second Edition improved significantly and remained at that level or continued to improve over the subsequent year. The mean Bruce treadmill protocol time first increased from 588 seconds to 801 seconds (P &lt; .001) and then dropped to 750 seconds (P = .005), which is at the 90th percentile for age and sex. All Pain Stages of Change Questionnaire, adolescent version subset scores improved significantly initially and were stable or improved at 1 year, as did the Pediatric Quality of Life Inventory, Teen Report total score.</p>

<p><strong>CONCLUSION: </strong>Children with fibromyalgia can be successfully treated without medications with a very intensive PT/OT and psychotherapy program. They have significantly improved pain and function by subject report and objective measures of function.</p>

10.1016/j.jpeds.2015.06.036

J. Pediatr.

26209526