International Pediatric Multidisciplinary Management Using Telemedicine to Promote Equitable Care

Introduction

For decades, inaccurate or delayed diagnoses that persist across all treatment areas have compromised health care and put an unacceptable number of people at risk. Diagnostic inaccuracies can adversely affect health, cause psychological distress, and increase financial expenses. A patient may receive incorrect or unnecessary therapy if a diagnostic error occurs, or essential, potentially life-saving treatment may be withheld or delayed. ¹

According to Human Resources for Health, imbalances in the shortage, composition, and distribution of the health care system have shaped the Latin American and Caribbean region. ² Within countries in the vicinity, there is wide variation in the regional distribution of health professionals and standards of care. For example, while Grenada has six nurses for every physician, the Dominican Republic (DR) has only 0.2. ²

The health care system in the DR faces several challenges of socioeconomic constraints, shortage of health care professionals, and availability of adequate, cost-effective, and quality health care facilities. Many patients are undiagnosed or misdiagnosed for diverse reasons, including a lack of knowledge of these diseases among physicians and inadequate screening. In addition, primary care providers often overlook the need for referral. An even greater disadvantage is that patients cannot afford the cost of consultation in a hospital. ³

Hence, telemedicine has emerged as a powerful platform to deliver health services through Information and Communication Technologies, reduce health inequalities, enhance health outcomes, and guide health care providers. ^4,5 It also provides remote access to health services, particularly to isolated or underserved communities. ^6,7

In 2020, a leading telegenetics study was conducted in the city of Santo Domingo, DR, aiming to determine the feasibility of telemedicine as an appraisal tool for the clinical and morphological assessment of children with undiagnosed genetic diseases. ³ However, this research explores the application of telemedicine as a collaboration tool looking to address many challenges existing in the country when it comes to consultations that require referral. This study also reports a collaborative telehealth service model with a pediatrician to improve access to pediatric subspecialty care in an underserved region. It is expected that communication and collaboration within multidisciplinary teams lead to more precise and higher quality care.

Methods

Results

The medical team evaluated a total of 65 patients during this 6-year study. The patients' age ranged from 2 months to 16 years of age (mean 8 years old). The most common reasons for referral were masses or malignancies, vascular malformations, urogenital anomalies, stuttering, and cochlear implant (CI) programming. The most frequent subspecialties consulted were orthopedic surgery (15%), neurology (12%), gastroenterology (8%), hematology (8%), oncology (8%), rheumatology (8%), and urology (8%) (see Table 2 for classification of patients by subspecialty).

Subspecialty consultation frequently influenced diagnosis and management (Fig. 1). A total of 41 of 65 cases (63%) had been diagnosed already. A change in medical management occurred in 92% of cases (n = 60). Out of 60 cases, subspecialty consultation presented a change of diagnosis and treatment plan of 55% (n = 33), whereas, in the 60 cases involving management change, 43.3% (n = 26) changed only treatment and 2% (n = 1) diagnosis alone. In 43% (n = 28), changes improved symptoms and/or overall health. Subspecialty consultation led to surgical intervention in 12.3% (8/65) and diagnostic imaging in 32.3% (21/65).

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Fig. 1.

Effect of subspecialty consultation on change of patient diagnosis and/or management.

In this study, we describe four patients from our cohort (Table 3 details diagnostic evaluation and intervention of all participants): an 11-year-old child presented persistent leg pain and limping. Interventional radiology biopsied a leg mass identified by a leg magnetic resonance imaging (MRI) scan. Histopathology was consistent with CRMO (chronic recurrent multifocal osteomyelitis), a rare condition with a prevalence of 0.5 per 10⁶. Technetium bone scan identified lumbar vertebral column involvement (Fig. 2A), a high-risk lesion for compression fracture. ⁹ She received bisphosphonate therapy in Santo Domingo; subsequent MRIs and bone scans displayed marked improvement of the lesions (Fig. 2B).

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Fig. 2.

Magnetic resonance imaging scan presenting lesion (A): before child was treated and (B): showing a marked improvement of lesions after bisphosphonate therapy. The arrow represents the contrast enhanced image showing a leg mass proximal to the femur.

A full-term female neonate 14 days old was diagnosed with an amniotic band on her right leg by a sonogram 2 weeks before being born. Clinical examination revealed one skin fissure at the level of the right leg; the wound's edges were red and inflamed, a class 2 on the diagnostic criteria by Patterson ¹⁰ (Fig. 3A). Constriction band showed a marked lymphedema and there was an important improvement after surgical release (Fig. 3B). Graded compression stocking and lymphatic massages after the surgery were recommended by a CCHMC team composed of a pediatric surgeon, an interventional radiologist, and a hematologist.

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Fig. 3.

Child's leg showing constriction band and a marked lymphedema before surgery (A: November 2019). After surgery, a significant improvement of the leg can be seen (B: August 2021).

A 3-year-old boy who experienced a diffusely violaceous, macular rash throughout his body had increased inflammatory biomarkers (Fig. 4A). Local rheumatologists treated him with systemic glucocorticoids and methotrexate for presumed systemic onset juvenile idiopathic arthritis. However, neither his clinical examination nor laboratory results improved. Multiple local dermatologists consulted, yielding a skin biopsy without a definitive diagnosis. Telemedicine consultation followed with University of Cincinnati dermatopathology determined that it was lupus erythematosus panniculitis, a rare type of Antinuclear antibody (ANA) negative pediatric cutaneous lupus erythematosus. ¹¹ CCHMC consultants in dermatology and rheumatology prescribed hydroxychloroquine, allowing him to discontinue systemic steroids successfully. He has gradually improved per serial follow-up with his pediatrician and every 3-month digital image review by his CCHMC consultants using store and forward technology (Fig. 4B).

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Fig. 4.

Child before (A: August 2020) and during treatment with hydroxychloroquine (B: July 2021).

A 3-year-old child showed worsening ataxia and incremental balance problems for many months. She had visited multiple specialists, including a physical visit to a tertiary freestanding children's U.S.-based hospital. After a detailed examination, she was diagnosed with opsoclonus myoclonus, most likely due to a Mycoplasma pneumonia infection. Meta-iodobenzylguanidine scan excluded neuroblastoma. Following treatment with a single rituximab infusion in Santo Domingo, she experienced a durable recovery sustained for the past 4 years with a resolution of her presenting symptoms. She avidly participates in sports (video in Supplementary Video S1 digital content).

Discussion

This article illustrates the importance of telemedicine as a successful tool to solve challenging cases between countries of different cultures by fostering an established international collaboration between CCHMC and pediatric patients at Centro de Obstetricia y Ginecologia. We previously reported a partnership between genetic services in the DR and CCHMC, showing that telemedicine programs can facilitate access to genetic services for patients living in remote locations with a very high diagnostic yield and patient satisfaction. ³

In 2019, 11% of patients in the United States used some form of telehealth, whereas by May 2020, that figure increased to 50%. All health care systems quickly adapted. Providers across all specialties now see between 50 to 175 times the number of patients by telehealth compared with pre-pandemic levels. ⁴

Currently, cost-effectiveness plays a pivotal role in decision management. Our cohort includes patients with the same diagnoses and different care approaches. One patient traveled to CCHMC to obtain expert oncology consultation for an anaplastic brain ependymoma, while another patient received care through telemedicine in the DR. Neuroradiology reviewed the MRI scan from the latter patient; both patients received similar chemotherapy regimen modifications per oncology recommendations.

Similarly, a patient with portal hypertension due to cavernous portal vein thrombosis received a Rex shunt at CCHMC. He obtained serial monitoring with serial abdominal ultrasounds and platelet counts in the DR, supervised using telemedicine by his CCHMC surgeon. Another patient who underwent neurosurgery at CCHMC for brain cavernoma obtained subsequent MRI scans in the DR that the CCHMC neurosurgeon reviewed during monitoring visits using telemedicine, avoiding expensive travel for this patient's ongoing care. The incremental difference in travel costs favors telemedicine.

Telemedicine offers to revolutionize health equity by addressing existing health care disparities in the developing world. Patients obtain access to care without the burden of travel costs or geographic barriers. The remarkable alliance between the DR hub and CCHMC distance sites allows patients to attain efficient ongoing management that improves their quality of life. For example, bilateral CI used to treat congenital sensorineural hearing loss requires precise adjustments for new hearing needs that can be performed by serial, sophisticated software programming by telemedicine. One young man residing in the DR who obtained adjustments provided immediate gratitude to his Cincinnati-based audiologist during a telemedicine follow-up visit.

The lack of access to CI services in rural locations creates a significant health care disparity. Moreover, rural residence is a cause for delayed implantation in eligible adult and pediatric CI candidates. Our two CI cases support a previous study, ⁹ which evaluated the effectiveness of remote CI programming through telehealth. Smaller hospitals and hospital systems can collaborate with larger academic institutions to build systems that enable emergent telehealth services. ¹²

Telemedicine's positive impact on the appropriateness of referrals to medical/surgical subspecialists and redundancy reduction in evaluative and diagnostic studies has been established. ^13,14 However, studies also show that improved communication between the pediatric health care team and the patient has the potential to enhance disease management. ¹⁵

One growing subspecialty of rising importance is Pediatric Palliative Care (PPC). PPC targets the best possible quality of life for children with life-threatening illnesses along with their families, ¹⁶ for example, by enhancing the care of patients with serious malignancies. The American Academy of Pediatrics issued recommendations in 2000 for an integrated palliative care model for children with life-threatening illnesses, which begins at the diagnosis and stresses the provision of curative therapies. Furthermore, they suggested comfort measures to improve the quality of life throughout their disease. ¹⁴

While local expertise had previously recommended leg amputation in a case of alveolar rhabdomyosarcoma, the CCHMC counterpart understood that the outcome would be the same, independent of any further action. Parents were advised to avoid amputation and focus on the quality of life. Even though the patient passed away a couple of months later, they did not have to go through the agony of an amputation.

The following three cases highlight the significance of pathology for a proper diagnosis. Case 1 was initially embryonal rhabdomyosarcoma. After a CCHMC pathologist reviewed the slides, there were suggestions that it could be an alveolar rhabdomyosarcoma, which is more aggressive and justified the patient's course of disease. Case 2 initially was thought to be a fibrous histiocytoma; however, University of Cincinnati's pathologists determined a desmoid tumor. An oncologist recommended a change in the chemotherapy treatment; 5 years later, the patient is well. Case 3 (Fig. 4) had a diagnosis, by the DR pathologist, as vasculitis with lobular panniculitis. However, the dermatopathologist from the University of Cincinnati diagnosed it as lupus panniculitis, leading to treatment with hydroxychloroquine and improving the clinical course for the child.

A limitation of this study is that, occasionally therapeutic options offered to some patients and specific subspecialists were unavailable in the country. Some patients did not have the possibility of traveling to the United States, presenting a barrier to accessing specific subspecialty care. Another limitation is that as the sole provider in the DR, there is a potential to introduce referral and participation bias. Many patients do not seek care, assuming that they cannot obtain a diagnosis in the country, and those who do are because they are patients who often seek consultation in the clinic. Also, this study is limited from an epidemiologic perspective due to its retrospective, descriptive, and observational nature.

Conclusion

This study assessed 65 patients referred for subspecialty consultation, using a collaborative telemedicine protocol involving a local pediatrician and 17 subspecialties at a leading academic pediatric hospital. This approach showed high patient satisfaction with the technology platform, direct patient savings from transportation costs, access to subspecialty care, and the quality of care derived. It confirmed that telemedicine can be used safely and efficiently and proved the importance of accurate diagnosis to offer optimal management.

Telemedicine offers to revolutionize health equity by addressing existing health care disparities in the developing world through its capacity for global outreach. We demonstrate how a collaborative, international partnership between institutions can provide high-quality care from subspecialists at a distance pediatric academic center facilitated by a hub specialist in-country to patients in underserved regions. This approach is a cost-effective, personalized, and targeted solution that assists ongoing care for patients with chronic conditions. Telemedicine implementation needs further examination to improve access to subspecialty care.