Obituary PBC Dr Pat Morris JonesDr Morris Jones who was President of SIOP in 1980/81 died on March 16 2020 at the age of 87. She was one of the first doctors in the UK to specialise in the care of children with cancer and over her 40 year career from 1953 to 1993 she saw survival rates improve from cancer being mainly a fatal disease to one where there was a real chance of cure with more than half of children surviving. She was an only child ,born in Oswestry ,close to the border with Wales. She trained in medicine at the Royal Free Medical School in London and found her way to Manchester to specialise in paediatrics. There she came under the influence of Basil Marsden, a paediatric pathologist and Dr Dorothy Pearson a radiotherapist. In 1954 Marsden had established the world’s first population based children’s malignant disease registry which became a prototype for registries all over the world. Dr Pearson was a founding member of SIOP and its second president. In the late 50s and early 60s the only real treatments available were surgery and radiotherapy but the emerging success of chemotherapy pioneered in the US, France and Germany led to a need for paediatricians to sub specialise in paediatric oncology. By the time Pat became involved the antagonism towards giving children chemotherapy had largely dissipated but how to use it to best advantage remained to be determined. She took up this challenge and built up one of the largest units in the UK. For many years she was single handed and absolutely dedicated to the care of children. She rarely took a day off. . Before people were talking about evidence based medicine she was adamant that treatment should be given in trials where we could learn what was best for the future. She was especially interested in Wilms’ tumour and very involved in the Medical Research Council embryonal tumour group and in the MRC UKALL trials. In 1977 she helped found the UK Children’s Cancer Study Group (UKCCSG) which eventually ensured that all children in the UK with cancer had access to the most up to date treatment. The early involvement of Manchester in the pre chemotherapy era using radiotherapy, and the registry, led her to take a real interest in the late effects of treatment. She had a collection of slides which she used to illustrate the late effects of radiotherapy when given to young children. Failure of skeletal growth could ensue from radiotherapy to growing bones and second malignancies occurred within radiation fields. She became heavily involved with Anna Meadows in Philadelphia in setting up the Late Effects Study Group which received substantial funding from NIH. This work was extended and has been perpetuated by Les Robison and his colleagues and has hugely influenced the design of clinical treatment protocols in recent years. She wrote an influential paper in 1990 entitled Childhood Cancer; Cure at what cost? In which she described the evolution of treatment for childhood cancer from one of cure at any cost to cure at least cost. Her fervent hope was that we would eventually get to a stage of cure at no cost. Pat was an inspirational teacher and leader and many of the young aspiring paediatric oncologists in the 70s turned to her for career advice and then once appointed to a post used her as a source of support and advice on patient management. She was most people’s “phone a friend”.The psychosocial problems faced by both children and parents became much more obvious once potentially curative treatments were being used. Paradoxically parents seemed to be able to cope better with the near certainty of death of their child than with the possibility, but by no means certainty, of long term survival. She worked closely with Peter McGuire, a psychiatrist, and with social workers to define the problems faced by children and their families and devised interventions to try and help. It was clear from the early leukaemia trials, which involved cranial irradiation and brain tumours that endocrine insufficiency was a real problem and she worked closely with Professor Steve Shalet, a paediatric endocrinologist, to not only define what these problems were but also how to follow up and screen survivors. She was always much in demand for conferences once causing consternation in Bruges when, invited to take a shot at the men only archery club she hit the bull’s eye first time. The local tradition was that anyone who did this was invited to become a member.Her retirement at the age of 60 was marked by an enormous party in Manchester Town Hall attended by many of the patients whom she had successfully treated and the parents of many of those who had died. She remembered all of their names. She was short in stature but big in personality, loved clothes and is remembered for her leather skirts and red shoes. In later years her hair was often a shade of blue and always well coiffeured . She was always a straight talker and had lots to say in meetings. She was direct and always had strong opinions. She was once quoted in a national newspaper that money was being wasted on sending children to Disneyland.Throughout her career she loved foreign travel which was often associated with scientific meetings. She built up a wide circle of friends including Anna Meadows, Dan D’Angio, Mark Nesbit and Audrey Evans whom she would call upon to offer training places to her aspiring young colleaguesWhen she retired from clinical work in 1993 she decided to move to London. “As a single lady why would I want to move into a country cottage? Soon after her move to central London she met and married her Italian hairdresser, Alfonso Cassarini, and enjoyed 20 blissful years travelling widely and exploring the capital’s culture.Pat Morris Jones was a forceful and formidable pioneer who always had the best interests of children and their families at the forefront of everything that she did. The incredible survival rates for children with cancer in the present era using treatments which are designed to minimise late effects are built on the shoulders of giants like her who dared to try.Alan CraftTim Eden

The coagulopathy of the novel severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) is well documented in adults, with increases in D-dimer and prothrombin time strong predictors of mortality and anticoagulation shown to decrease this mortality. Viscoelastic parameters such as elevations in maximum clot firmness (MCF) on rotational thromboelastometry (ROTEM) have correlated with a hypercoagulable state in adults with SARS-CoV-2. We report our experience in children infected with SARS-CoV-2, with noted elevations in D-dimer and MCF on ROTEM (indicating hypercoagulability). Exploration of viscoelastic testing to provide additional laboratory-based evidence for pediatric-specific risk-assessment for thromboprophylaxis in SARS-CoV-2 is warranted.

As we celebrate 2020 as the Year of the Nurse and the Midwife and recognize the Global Initiative for Childhood Cancer, members of the International Society of Pediatric Oncology (SIOP) Baseline Nursing Standards Taskforce would like to highlight advocacy efforts promoting the baseline nursing standards.1, 2 Your published article, An ethical imperative: safety and specialization as nursing priorities of WHO Global Initiative for Childhood Cancer(Pergert and colleagues) reveals the importance of ongoing efforts to support implementation of the Baseline Nursing Standards.3 Given that the majority of hospitals are not meeting the standards in low- and middle-income countries (LMIC), as well as some high-income countries (HIC),4, 5 advocacy initiatives are required to raise awareness of the need to meet these standards. During the COVID-19 pandemic, health facilities face new challenges in meeting the standards. To achieve the WHO global initiative’s goal to save one million children’s lives by 2030, it is important to continue efforts to address baseline nursing standards.Pediatric oncology as a subspecialty requires a nursing workforce with specialized education and clinical skills to achieve optimal patient outcomes. Knowledge itself is not enough if nurses lack the resources and support to practice or implement appropriate nursing care in their work settings. The six Baseline Nursing Standards focus on key elements essential to delivering quality and safe care (Table 1). Collectively, they serve as a framework and foundation for positive pediatric oncology nursing practice environments internationally.Advocacy efforts to disseminate the baseline standards are well established. To date, fourteen organizations have endorsed the Standards. Members of the SIOP PODC Nursing Working Group hosted a “Leadership and Advocacy Workshop: Disseminating the Baseline Nursing Standards” prior to the SIOP Conference in October 2017. Twenty-two pediatric hematology/oncology nurse leaders and four stakeholder-group representatives (parent, physicians, advocates) from 14 countries met and established goals and strategic priorities for advocacy of the standards. As a result, the Baseline Nursing Standards Advocacy Toolkit was developed and can be found on the SIOP Nursing Website https://siop-online.org/baseline-nursing-standards-advocacy-toolkit. The toolkit contains practical advocacy resources, including a PowerPoint presentation, an endorsement letter template, publications, podcasts, a social media campaign and examples of elevator speeches for each standard. Furthermore, the Standards have been featured in international presentations, such as a keynote presentation (S. Day) in SIOP Lyon, an award session and nursing abstract presentations at SIOP congresses and continental meetings.To reach the WHO target of doubling the global childhood cancer survival rate to 60%, achievement of baseline nursing standards for pediatric oncology must be prioritized and appropriately resourced by hospital administrators, governments and other stakeholders. Amid a global pandemic where nursing resources are stretched, creative ways to support and advocate for implementation of the standards is needed. In recognition of the recent publication by the Nurse Specialists of the Global Initiative for Childhood Cancer noting the baseline standards, now is the time to act and improve childhood and adolescent cancer outcomes through raising the standard of pediatric oncology nursing practice around the world.Linda Abramovitz, Rehana Punjwani, Glenn M. Afungchwi and Courtney Sullivan and the SIOP PODC Baseline Standards Nursing Task Force.A special thank you to Rachel Hollis for her commitment and ongoing advocacy efforts focused on the baseline nursing standards.ReferencesDay S, Hollis R, Challinor J, Bevilacqua G, Bosomprah E, SIOP PODC Nursing Working Group. Baseline standards for paediatric oncology nursing care in low to middle income countries: position statement of the SIOP PODC Nursing Working Group. Lancet Oncol. 2014; 15(7):681-682 PMID: 24872097.Day S, Challinor J, Hollis R, Abramovitz L, Hanaratri Y, Punjwani R. Paediatric Oncology nursing care in low-and middle-income countries: a need for baseline standards. Cancer Control. 2015;2015:111-116Pergert P, Sullivan CE, Adde M, et al. An ethical imperative: Safety and specialization as nursing priorities of WHO Global Initiative for Childhood Cancer. Pediatr Blood Cancer. 2019;e28143. https://doi.org/ 10.1002/pbc.28143Morrissey L, Lurvey M, Sullivan C, et al. Disparities in the delivery of pediatric oncology nursing care by country income classification: international survey results. Pediatr Blood Cancer. 2019;66(6):e27663.Sullivan CE, Morrissey L, Day SW, Chen Y, Shirey M, Landier W. Predictors of Hospitals’ Nonachievement of Baseline Nursing Standards for Pediatric Oncology. Cancer Nurs. 2019 Mar 29;

As the effects of cancer and its treatment have long-lasting negative impacts on the health and quality of life of survivors, there is a need to explore new avenues to optimize long-term patient outcomes in pediatric oncology. Therefore, this scoping review aims to report on the state of the evidence on the use and effects of behavioural interventions targeting physical activity and diet behaviours in pediatric oncology. Fourteen quantitative studies were included. Studies evaluated a combination of two or three different modalities, including education (n=11), physical activity (n=6), psychosocial support or training (n=6), reward system (n=2) and adventure-based activities (n=1).  Overall, behavioural interventions in pediatric oncology appear beneficial; however, no conclusive evidence favouring specific interventions were identified.

There are no proven safe and effective therapies for children who develop life-threatening complications of SARS-CoV-2. Convalescent plasma (CP) has demonstrated potential benefit in adults with SARS-CoV-2 but has theoretical risks. We report on the first use of CP in children with life-threatening COVID-19, providing data on four pediatric patients with acute respiratory distress syndrome. We measured donor antibody levels and recipient antibody response prior to and following CP infusion. Infusion of CP was not associated with antibody-dependent enhancement (ADE) and did not suppress endogenous antibody response. We found CP was safe and possibly efficacious. Randomized pediatric trials are needed.

The song from my radio filled the car, and floated out into the air and mingled with the cool breeze. Today the chirping of birds were clear and I could hear the rustling leaves. When I stopped at the traffic light, I realized that I never saw how green the trees were and how beautiful the sky with a few wispy clouds passing. Nature seemed to be at peace even when the world around me groaned due to this virus. By whatever name you call it, it is a symbol of tear and fear.Just as I step out of my car, I remember to don my mask and quickly walk along the walkway to the entrance. I look to the branches of a tree on the right of the walkway and noticed the little robin who sings daily without fail. I know the screening questions by heart now and the thermometer scans my forehead. The little sticker placed on my badge feels like a gold star that I received in kindergarten for good behavior. I start another day at the hospital.Everything has changed since this pandemic began. I miss seeing the smiles of people and hearing their voices now muffled through the masks. How this invisible thing has visibly altered the world, as we know it. How every touch and every breath became calculated. You really want to hug a patient as their silent tears soak their masks and you just have to hold back. Nevertheless, I remember that our presence and words can embrace people around us, to comfort and strengthen them.We zoom in for meetings and you may or may not see the face behind the voices or the silent listeners. Birthdays, graduations and send off parties are on hold. Patients lie alone on their beds as their families see and talk to them from a screen. A stranger holds their hand and watch their breathing as the machines chime. Many lives are lost and funerals held quietly and far from homes.I long for the days when we could meet friends in the park or stroll through the mall. Will the days come back when we ate in a restaurant or enjoyed a walk along the beach? I wish those moments could be frozen and kept in a glass jar that could never be shattered.As the orange glow of the sun drenches everything around me, I slowly walk back to my car. A silent prayer floats in the evening breeze; the drive is quite, the sky an impossible hue of gold and purple. The leaves gently rustle and believe it, the birds are still chirping!

Background: Pediatric palliative care (PPC) for oncology patients improves quality of life and the likelihood of goal-concordant care. However, barriers to involvement exist. Objectives: We aimed to increase days between PPC consult and death for patients with refractory cancer from a baseline median of 13.5 days to ≥30 days between March 2019 and March 2020. Methods: Outcome measure was days from PPC consult to death; process measure was days from diagnosis to PPC consult. The project team surveyed oncologists to identify barriers. Plan-Do-Study-Act cycles included establishing target diagnoses, offering education, standardizing documentation, and sending reminders. Results: The 24-month baseline period included 30 patients that died and 25 newly diagnosed patients. The yearlong intervention period included 6 patients that died and 16 newly diagnosed patients. Interventions improved outcome and process measures. Targeted patients receiving PPC ≥ 30 days prior to death increased from 43% to 100%; median days from consult to death increased from 13.5 to 159.5. Targeted patients receiving PPC within 30 days of diagnosis increased from 28% to 63%; median days from diagnosis to consult decreased from 221.5 to 14. Of those without PPC consult within 30 days after diagnosis, 17% had template documentation of the rationale. Conclusion: Interventions utilized met the global aim, outcome and process measures. Use of QI methodology empowered providers to involve PPC. Poor template use was a barrier to identifying further drivers. Future directions for this project relate to expanding the target list, creating long-term sustainability, formalizing standards, and surveying patients and families.

A document by Jacqueline Montoya. Click on the document to view its contents.

In the survivorship setting, adolescent and young adult (AYA) cancer survivors frequently demonstrate little knowledge of infertility risk, are unclear regarding their fertility status and may under- or over-estimate their treatment-related risk for infertility. In female AYA survivors, ovarian function usually parallels fertility, and can be assessed with serum hormone levels and ultrasonography. Post-treatment fertility preservation may be appropriate for survivors at risk for primary ovarian insufficiency. In male AYA survivors, fertility and gonadal function are not always equally affected, and can be assessed with a semen analysis and serum hormones, respectively. As reproductive health issues are commonly cited as an important concern by survivors of AYA cancer, multidisciplinary care teams including oncology, endocrinology, psychology, and reproductive medicine are advocated, with the aim of optimal provision of fertility advice and care for AYA cancer survivors.

Background The International Soft Tissue Sarcoma Consortium (INSTRuCT) was founded as an international collaboration between different pediatric soft tissue sarcoma cooperative groups (COG, EpSSG, CWS). Besides other tasks, a major goal of the INSTRuCT is to develop consensus expert opinions for best clinical treatment. This consensus paper for patients with rhabdomyosarcoma of the female genital tract (FGU-RMS) provides treatment recommendations for local treatment, long term follow up and fertility preservation. Methods Review of the current literature was combined with recommendations of the treatment protocols of the appropriate clinical trials. Additionally, opinions of international FGU-RMS experts were incorporated into recommendations. Results The prognosis of FGU-RMS is favorable with an excellent response to chemotherapy. Initial complete surgical resection is not indicated, but diagnosis should be established properly. In patients with tumors localized at the vagina or cervix demonstrating incomplete response after induction chemotherapy, local radiotherapy (brachytherapy) should be carried out. In patients with persistent tumors at the corpus uteri, hysterectomy should be performed. Fertility preservation should be considered in all patients. Conclusion For the first time, an international consensus for the treatment of FGU-RMS patients could be achieved, which will help to harmonize the treatment in different study groups.

Background: Cancer among infants (<1 year old) has unique epidemiologic, clinical and genetic characteristics compared with cancer in older children. Nonetheless, data on secular trends in infant cancer incidence and survival in the US is sparse. Methods: Population-based data from Surveillance Epidemiology and End Results (SEER 9) was used to estimate the incidence, average annual percentage change (APC) for trends and survival of malignant neoplasm among infants from 1975-2014. Data were stratified by gender, race, registry and cancer type. Results: There were 3,437 new infant cancer cases with an overall incidence of 23.6/100,000. Neuroblastoma was the most common infant malignancy (6.5/100,000), followed by leukemia (3.8/100,000), and brain and central nervous system tumors (3.3/100,000). The incidence rate increased significantly over the observational period (APC 0.68; 95%CI 0.30-1.06; p<0.05). Variations in overall incidence rates were uneven across SEER registry geographic areas, with the lowest rates among both males and females in New Mexico. Relative to other racial distribution, rates were highest among whites. The relative survival rates improved over time for all tumors except for renal, sarcomas and germ cell and were not significantly different by gender or race. Conclusions: Cancer incidence among infants increased over time largely driven by leukemia, germ cell and sarcoma mainly among male infants. The overall survival for infant cancer has improved over the years especially since 1990 for hepatic tumors, lymphoma and leukemia. Further research is needed to explore the potential impacts of genetic, environmental, and perinatal factors for possible explanations for these increased cancer incidence trends.

1 Division of Pediatric Hematology and Oncology, Department of Pediatrics, Rainbow Babies and Children’s Hospital, Case Western Reserve University, Cleveland, OH.* CorrespondenceJignesh Dalal, Pediatric Hematology Oncology, Rainbow Babies and Children’s Hospital, Case Western Reserve University, 11100 Euclid Avenue, Cleveland OH 44106, Tel: 216 844 3345, Email: Jignesh.dalal@uhhospitals.orgText word count: 951Brief running title: Challenges in HLH transplantKey words: primary hemophagocytic lymphohistiocytosis, hematopoietic stem cell transplant, mixed chimerismTables: 1

Introduction.Despite progress obtained with current treatments,the event-free survival of high-risk neuroblastoma(HR-NB)patients does not exceed 40-50% and the prognosis in refractory or relapsed patients is poor,still representing a challenge for pediatric oncologist.Therapeutic Iodine-131 meta-iodobenzylguanidine(Th-131I-MIBG) is a recognized safe and potentially effective treatment in NB.Materials.In this retrospective study,we report outcome of 28 MIBG-avid NB patients with advanced disease,because refractory or relapsed,underwent,from 1996 to 2014,to Th-131I-MIBG administered shortly before(median of 17 days) high-dose chemotherapy with Busulfan and Melphalan(HD-BuMel) and autologous stem cell transplantation(ASCT)at Gaslini Institute in Genoa,with the aim to analyze feasibility,safety and efficacy of this approach.Results.Engraftment occurred in all patients after a median of 14(11-29)and 30 days(13-80)from ASCT for neutrophil and platelet respectively.No treatment-related deaths were observed.The main high grade(3-4)toxicity observed was oral and gastrointestinal mucositis in 78.6% and 7.1% of patients respectively,while high grade hepatic toxicity was observed in 10.7%;two patients developed veno-occlusive-disease(7.1%),completely responsive to defibrotide.Hypothyroidism was the main late complication occurred in 9 patients(31.1%).After Th-131MIBG and HD-BuMel, 19 patients(67.8%) showed an improvement of disease status.Over a median follow-up of 15.9 years,the 3-year and 5-year overall survival(OS)probability were 53%(CI 0.33-0.69)and 41%(CI 0.22-0.59)and the 3-year and 5-year rates of cumulative risk of progression/relapse were 64%(CI 0.47-0.81)and 73%(CI 0.55-0.88),respectively.MYCN amplification emerged as the only risk factor significantly associated with OS(HR 3.58;p0.041).Conclusion.Th-131I-MIBG administered shortly before HD-BuMel turned out to be a safe and effective regimen,suggesting it should be included in a sequential approach in patients with advanced MIBG-avid NB.These patients could be benefit to be manage in centers with proven expertise in these treatments.

Treatment of refractory Evans syndrome (ES) remains a challenge in Hematology practice. Due to rarity of this condition, evidence-based approaches are limited and often treatment choices stem from small case series or anectodal experiences. Here, we describe three very refractory pediatric ES cases treated on bortezomib without adverse effects. Two of the three patients had dramatical and long-lasting recovery that started following the first doses of the drug. Clinical trials to assess bortezomib role in ES treatment are warrented and results may lead to inclusion of this drug as an option, even as a first-line therapy.

The challenge of multiple paediatric febrile neutropenia clinical decision rules has been well described, and the difficulties with ever-expanding variants based on individual datasets discussed extensively.1 The re-calibration of the SPOG rule undertaken by Haeusler and colleagues2 (creating the ‘AUS’ rule) for a practical, bedside tool which would be able to risk stratify episodes of FN. This letter reports a further validation in a meta-analytic dataset from the ‘Predicting Infectious Complications in Children with Cancer’ (PICNICC) collaboration.Briefly, the PICNICC collaboration was formed to develop a robust prediction rule for febrile neutropenia, with methods and materials detailed in previous publications.1,3 Over 8000 episodes of 33 different candidate predictor variables and seven clinically relevant outcomes have been collated from 26 study groups, with varied completeness. The ‘AUS’ rule uses three equally weighted factors to scale the risk of complications in an episode of FN, summed between 0 and 3.These factors are: preceding chemotherapy more intensive than acute lymphoblastic leukaemia maintenance, platelet count less than 50 g/L, total white cell count lower than 300 cells/mm3. The clinical response can be scaled according to this score, with values of 0 and 1 considered as ‘lower risk’.We evaluated the association between the AUS rule and bacterial infection using these data, reporting the discrimination (Area Under the receiver-operator Curve, AUC) and proportion of episodes classed as lower risk (scoring 0 or 1). Analyses were undertaken using R (v3.2.0).1520 episodes contributed to the analysis, with 301 episodes of documented bacterial infection. The discriminatory ability appeared very similar to Haeusler’s values; AUC 0.64 (95% CI 0.61 to 0.68) compared with the original AUC 0.67 (95% CI 0.63 to 0.71). Using the AUS rule on the PICNICC dataset would have identified 44% (668/1520) of the population as a lower risk group (score 1 or 0).This reassuring data has led to the introduction of an AUS rule based system to shorten the duration of antibiotic therapy, in concordance with UK National and International guidelines[3,4]. This now-validated rule will reduce hospitalisation for febrile neutropenic episodes in the UK, which was of particular importance during the 2020 SARS-CoV-2 coronavirus pandemic.

Objectives: Clinical studies have shown low toxicity and a favorable safety profile for sirolimus in vascular anomalies. Here, we describe severe adverse events (SAEs) observed during “off-label use”. Methods: We performed a retrospective, multicenter chart review for SAEs during “off-label” sirolimus therapy for vascular anomalies and analyzed these cases by a pre-designed workflow. Results: We identified 17 SAEs in 14 patients diagnosed with generalized lymphatic anomaly (n=4), Gorham-Stout disease (n=2), central conducting lymphatic anomaly (n=1), lymphatic malformation (n=4), tufted angioma (n=1), kaposiform hemangioendothelioma (n=1), and venous malformation in a CLOVES syndrome (n=1). Three patients presented two SAEs. The age at initiation of sirolimus therapy was under the age of 2 years (5x), 2 to 6 years (5x) and older than 12 years (4x). SAEs occurred during the first 3 months of sirolimus therapy (7x), between 3 and 12 months (7x) and after one year of therapy (3x). The most frequent SAE was viral pneumonia (8x) resulting in death due to a metapneumovirus infection in a 3 months old and generalized adenovirus infection in a 28 months old child. Sirolimus blood level at the time of SAEs ranged between 2.7 and 21 ng/L. Five patients were on antibiotic prophylaxis during sirolimus therapy. Conclusions: Most SAEs are observed in the first year of sirolimus therapy; however, SAEs can also occur after a longer treatment period. SAEs are potentially life threatening, especially in early infancy. Presence risk factors, i.e. underlying vascular anomaly or immune status, may contribute to the risk of SAEs.

Sickle cell disease (SCD) is a hematologic disorder defined by presence of sickle-shaped red blood cells that can occlude blood vessels and cause tissue ischemia and pain. Treating SCD pain adequately and safely is difficult given today’s opioid climate. Buprenorphine-naloxone has been described as an alternative option to treat chronic pain in the adult literature, however, it historically has required discontinuation of full-agonist opioids before initiation, resulting in opioid withdrawal symptoms. Here we present two adolescents with SCD who successfully weaned off large doses of full-agonist opioids by using a micro-dose induction of buprenorphine-naloxone in clinic without patients experiencing withdrawal symptoms. Ambulatory micro-dose induction may remove hurdles that otherwise would discourage patients from trying this regimen while still controlling pain safely.

Bright IDEAS Problem-Solving Skills Training (BI) is an evidence-based behavioral intervention that has been utilized extensively with caregivers of children recently diagnosed with cancer. Considerable evidence has shown that BI is acceptable to caregivers and improvements in problem-solving skills mediate reduced symptoms of distress; and it is most effective with single, minority caregivers. A slightly modified version of BI was offered to caregivers of children with sickle cell disease (SCD) in a two-site pilot feasibility trial. BI was modified to reduce barriers to care, logistical challenges, and stigma associated with receiving behavioral health services. Our goal was to establish high rates of recruitment and retention amongst caregivers of children with SCD. Recruitment was acceptable (94%; N = 72) and retention reasonable (48.6%) across both sites with 35 caregivers successfully completing the BI program. Results showed that caregivers of children with SCD, who successfully completed the BI program reported, significant improvements in problem-solving skills immediately and three months post-intervention completion. Interestingly, initial levels of distress were low with few caregivers reporting clinically significant levels of distress; distress remained low over time. Findings are discussed in the context of psychosocial screening and assumptions regarding caregivers of children with SCD.