<i>Background</i>: Telemedicine health service has revolutionized delivery of health care to the doorsteps of needy and deserving cases especially since the onset of the recent pandemic; COVID-19. Beta thalassemia major children require lifelong medical care, despite any untoward circumstances. Aim of this study is to identify role of telemedicine in diagnosing, treating and following-up of thalassemia cases and assess patient satisfaction. <i>Objective</i>: To assess implementation, patient satisfaction and usability of telemedicine health service for pediatric thalassemia cases in Northern Balochistan. <i>Study place and duration</i>: Study was carried out at District Headquarter Hospital Zhob from April 2021 to September 2021. <i>Material & methods</i>: Telemedicine service was established at Pediatric Department DHQ Hospital Zhob during COVID-19 pandemic. The data of all thalassemia cases that were managed/provided health care via telemedicine was collected and recorded. Patient satisfaction and usability of telemedicine service provided was assessed using a questionnaire prepared after thorough study of literature. The parents and care-givers of thalassemia cases treated via telemedicine were interviewed on mobile cell phones/telephone and the questionnaires were filled. Overall satisfaction was also gauged according to a scale from 1 to 5, where 1 depicted extremely dissatisfaction and 5 showed extreme satisfaction. The data was analyzed with Statistical package for social science (SPSS 21). <i>Results</i>: Total 347 patients of thalassemia received telemedicine health service during the study period. There were 182 (52.4%) males and 165 (47.6%) females with age range of 1 – 15 years (mean age 8.09 + 3.47 years). Total 289 (83.3%) telemedicine service seekers were residing in rural areas and rest 58 (16.7%) resided in urban areas. Ninety seven families (28%) belonged to poor class, 192 (55.35) belonged to lower middle class, 55 (15.9%) belonged to upper middle class and 3 (0.9%) belonged to rich class. Majority of participants (97.4%) were significantly satisfied with telemedicine service (p value 0.001). <i>Conclusion</i>: During current COVID-19 pandemic telemedicine has emerged as an exceptional alternate mode of healthcare delivery. It saves cost and time of travelling for thalassemia cases who require life-long hospital visits and follow-up. People have shown high satisfaction and positive attitude towards telemedicine health service provision.