5-Year Activity and Participation Outcomes of the First Successful Pediatric Bilateral Hand Transplantation: A Case Report.

2022

1-16

2022 Apr 04

1541-3144

<p><strong>AIMS: </strong>Describe the 5-year outcomes of the first successful pediatric bilateral hand transplantation.</p>

<p><strong>METHODS: </strong>The child underwent quadrimembral amputation at age two and received bilateral hand allografts at age eight. Rehabilitation included biomechanical, neurorehabilitation, and occupational approaches in acute and outpatient settings. Therapist observed outcomes, patient-reported measures, and parent-reported measures were repeated over a 5-year period.</p>

<p><strong>RESULTS: </strong>Observation assessments revealed functional dexterity skills and modified independence to full independence with self-care activities. The parent reported the child had moderate difficulty with upper extremity functioning 25-, 41-, and 48-months post-transplantation, and mild difficulty at 60-months; the child reported no difficulties in this domain at 41 months. Five years post-transplantation the child reported enjoying many age-appropriate activities, and high-quality peer relations were endorsed by both parent and child.</p>

<p><strong>CONCLUSION: </strong>The child developed hand movements for daily activities and was completing daily activities with improved efficiency. Health-related quality of life outcomes were favorable.</p>

10.1080/01942638.2022.2057210

Phys Occup Ther Pediatr

35379065

The current outcomes and future challenges in pediatric vascularized composite allotransplantation.

2020

576-583

2020 Dec

1531-7013

<p><strong>PURPOSE OF REVIEW: </strong>We review the outcomes and future challenges associated with pediatric vascularized composite allotransplantation, including follow-up data from our bilateral pediatric hand-forearm transplantation.</p>

<p><strong>RECENT FINDINGS: </strong>In 2015, the first heterologous pediatric upper extremity hand-forearm transplant was performed at the Children's Hospital of Philadelphia, and in 2019, the first pediatric neck reconstructive transplantation was performed in Poland. The 5-year follow-up of the pediatric upper extremity recipient demonstrates similar growth rates bilaterally, an increase in bone age parallel to chronologic age, and perhaps similar overall growth to nontransplant norms. The pediatric upper extremity recipient continues to make gains in functional independence. He excels academically and participates in various extracurricular activities. Future challenges unique to the pediatric population include ethical issues of informed consent, psychosocial implications, limited donor pool, posttransplant compliance issues, and greater life expectancy and therefore time to inherit the many complications of immunosuppression.</p>

<p><strong>SUMMARY: </strong>Currently, we recommend pediatric vascularized composite allotransplantation (VCA) for bilateral upper extremity amputees, preferably on immunosuppression already, and those patients who would have the most potential gain not available through standard reconstructive techniques while being able to comply with postoperative immunosuppression protocols, surveillance, rehabilitation, and follow-up.</p>

10.1097/MOT.0000000000000809

Curr Opin Organ Transplant

33044345

Pediatric Hand Transplantation: A Decision Analysis.

2019

1558944719890041

2019 Dec 17

1558-9455

<p>The first successful bilateral pediatric hand transplant was performed in 2015. Previous hand transplant decision analysis models have focused on the adult population. This model principally aimed to determine whether adverse outcomes associated with immunosuppression outweigh the benefits of performing bilateral hand transplant surgery in a pediatric candidate. The model also conceptualized the valuation of losing years of life and sought to determine the impact of that valuation on the surgical decision. A decision model compared undergoing bilateral hand transplant surgery with using prosthetics for an 8-year-old patient. The outcome measure used was quality adjusted life years (QALYs), and sensitivity analysis was performed on the immunosuppressive risks associated with the surgical decision, as well as the perceived valuation of aversion to life years lost. The decision to perform surgery was marginally optimal compared to the prosthetic decision (50.11 QALY vs. 47.95 QALY). A Monte Carlo simulation revealed that this difference may be too marginal to detect an optimal decision (50.14 ± 8.28 QALY vs. 47.95 ± 2.12 QALY). Sensitivity analysis identified decision thresholds related to immunosuppression risks ( = 29% vs. = 33% modeled), and a trend of increasing risk as a patient is more averse to losing life years. The marginally optimal treatment strategy currently is bilateral hand transplant, compared to prosthetics for pediatric patients. Key determinants of the future optimal strategy will be whether immunosuppressive regimens become safer, with a reduced risk of losing life years due to immunosuppressive complications, and whether prosthetics become more acceptable and enable higher functioning.</p>

10.1177/1558944719890041

Hand (N Y)

31847578

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

Pediatric and congenital hand transplantation.

2017

2017 Jul 14

1531-7013

<p><strong>PURPOSE OF REVIEW: </strong>We review the approach and challenges associated with pediatric hand transplantation, including new knowledge gleaned from our recent case of bilateral hand-forearm transplantation in an 8-year old child.</p>

<p><strong>RECENT FINDINGS: </strong>Bilateral heterologous hand-forearm transplantation was performed in a child with a prior kidney transplant in July 2015. The initial surgery necessitated a large team of experts in microvascular surgery, transplant surgery, orthopedics and plastic surgery as well as pediatric anesthesia. Medical management has highlighted the need for extensive multidisciplinary support with pediatric expertise in transplant medicine, occupational therapy and rehabilitation, neurology, and neuroradiology. The clinical course has been complicated by the need for robust immunosuppression to control ongoing, even though low-grade, and intermittent rejection. Neurological findings have demonstrated cortical neuroplasticity with reorganization of the somatosensory cortex.</p>

<p><strong>SUMMARY: </strong>Heterologous hand-forearm transplantation in a child is feasible and has the potential for functional benefit to improve quality of life; however, immunological and ethical concerns warrant proceeding with caution. More data are needed to inform patient and family selection to achieve optimal functional and quality of life outcomes.</p>

10.1097/MOT.0000000000000453

Curr Opin Organ Transplant

28719391