Prevalence and clinical characteristics of children with medical complexity in Tottori Prefecture, Japan: A population-based longitudinal study



      To investigate the prevalence and background of children with medical complexity (CMC) and its secular trend in Japan.


      CMC were defined as patients under the age of 20 years requiring medical care and devices. The patients were enrolled using the national health insurance claims data of three hospitals and two rehabilitation centers in Tottori Prefecture. The study period was divided into three periods: Period 1, 2007–2010; Period 2, 2011–2014; and Period 3, 2015–2018.


      A total of 378 CMC were enrolled. The prevalence of CMC was 1.88 per 1000 population among subjects aged <20 years in 2018, and it increased by approximately 1.9 times during the study period. The number of CMC who presented with severe motor and intellectual disabilities did not change from Period 1 to Period 3. Meanwhile, the number of CMC who had relatively preserved motor and intellectual abilities increased from 58 to 98. The proportion of CMC who required respiratory management and oxygen therapy increased by 1.3 and 1.8 times, respectively. By contrast, the proportion of CMC who need tube feeding decreased significantly between periods 1 and 3 (P < 0.05).


      The prevalence of CMC increased almost twice during the 12-year study period; however, the increase in the number of patients with relatively preserved motor and intellectual abilities was pronounced. This study showed that the need for medical care and devices differed based on the underlying disorders and severity of CMC; therefore, individualized medical, welfare, and administrative services and education about the various types of CMC must be provided.


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        • Ohshima K.
        Basic problem of severely, mentally and physically disabled children (in Japanese).
        Koshu Eisei (Tokyo). 1971; 35: 648-655
        • Suzuki Y.
        • Takai S.
        • Takechi N.
        • Yamada M.
        • Morooka M.
        • Hiramoto A.
        • et al.
        New scoring system for patients with severe motor and intellectual disabilities, medical care dependent group (in Japanese).
        Nihon Jushou Shinshin Shougai Gakkaishi (Tokyo). 2008; 33: 303-309
        • Cohen E.
        • Kuo D.Z.
        • Agrawal R.
        • Berry J.G.
        • Bhagat S.K.
        • Simon T.D.
        • et al.
        Children with medical complexity: an emerging population for clinical and research initiatives.
        Pediatrics. 2011; 127: 529-538
        • Burns K.H.
        • Casey P.H.
        • Lyle R.E.
        • Bird T.M.
        • Fussell J.J.
        • Robbins J.M.
        Increasing prevalence of medically complex children in US hospitals.
        Pediatrics. 2010; 126: 638-646
        • Cohen E.
        • Berry J.G.
        • Camacho X.
        • Anderson G.
        • Wodchis W.
        • Guttmann A.
        Patterns and costs of health care use of children with medical complexity.
        Pediatrics. 2012; 130: e1463-e1470
        • Srivastava R.
        • Downie J.
        • Hall J.
        • Reynolds G.
        Costs of children with medical complexity in Australian public hospitals.
        J Paediatr Child Health. 2016; 52: 566-571
        • Takinami Y.
        • Ujiie Y.
        • Takeshita H.
        • Fujihara J.
        Current status and issues of medical care in schools for special need education (in Japanese).
        Rinsho Shinrigaku Kenkyu (Tokyo). 2016; 53: 80-86
        • Kumazaki K.
        • Yoshioka T.
        • Tamasaki A.
        • Maegaki Y.
        A study of the utilization of welfare systems by patients with severe motor and intellectual disabilities and their families (in Japanese).
        Yonago Igaku Zasshi (Tottori). 2015; 66: 81-89
        • Nishigaki K.
        • Yoneyama A.
        • Ishii M.
        • Kamibeppu K.
        An investigation of factors related to the use of respite care services for children with severe motor and intellectual disabilities (SMID) living at home in Japan.
        Health Soc Care Community. 2017; 25: 678-689
        • Caicedo C.
        Children with special health care needs: child health and functioning outcomes and health care service use.
        J Pediatr Health Care. 2016; 30: 590-598
        • Palisano R.
        • Rosenbaum P.
        • Walter S.
        • Russell D.
        • Wood E.
        • Galuppi B.
        Development and reliability of a system to classify gross motor function in children with cerebral palsy.
        Dev Med Child Neurol. 1997; 39: 214-223
        • Kumode M.
        • Hoshino R.
        • Sato S.
        • Matsubasa T.
        • Nagae A.
        • Fujita Y.
        Survey of severe children with medical complexity (in Japanese).
        Nihon Shounika Gakkai Zasshi (Tokyo). 2018; 122: 1519-1526
      1. Nagura M, Tamura M [Internet]. 3. Survey of the number of children with medical complexities and resources. Intermediate report of “Survey on medical care children and research on collaboration between medical care, welfare, health care and education” (in Japanese) [cited 2020 Feb 14]. Available from:

        • Glasson E.J.
        • Jacques A.
        • Wong K.
        • Bourke J.
        • Leonard H.
        Improved survival in Down syndrome over the last 60 years and the impact of perinatal factors in recent decades.
        J Pediatr. 2016; 169e1
        • Imataka G.
        • Suzumura H.
        • Arisaka O.
        Clinical features and survival in individuals with trisomy 18: A retrospective one-center study of 44 patients who received intensive care treatments.
        Mol Med Rep. 2016; 13: 2457-2466
        • Ishitsuka K.
        • Matsui H.
        • Michihata N.
        • Fushimi K.
        • Nakamura T.
        • Yasunaga H.
        Medical procedures and outcomes of Japanese patients with trisomy 18 or trisomy 13: Analysis of a nationwide administrative database of hospitalized patients.
        Am J Med Genet A. 2015; 167A: 1816-1821
        • Reid S.M.
        • Carlin J.B.
        • Reddihough D.S.
        Rates of cerebral palsy in Victoria, Australia, 1970 to 2004: has there been a change?.
        Dev Med Child Neurol. 2011; 53: 907-912
        • Sellier E.
        • Platt M.J.
        • Andersen G.L.
        • Krageloh-Mann I.
        • De La Cruz J.
        • Cans C.
        • et al.
        Decreasing prevalence in cerebral palsy: a multi-site European population-based study, 1980 to 2003.
        Dev Med Child Neurol. 2016; 58: 85-92
        • Touyama M.
        • Touyama J.
        • Toyokawa S.
        • Kobayashi Y.
        Trends in the prevalence of cerebral palsy in children born between 1988 and 2007 in Okinawa, Japan.
        Brain Dev. 2016; 38: 792-799
        • Brooks J.C.
        • Strauss D.J.
        • Shavelle R.M.
        • Tran L.M.
        • Rosenbloom L.
        • Wu Y.W.
        Recent trends in cerebral palsy survival. Part I: period and cohort effects.
        Dev Med Child Neurol. 2014; 56: 1059-1064
        • Liu Y.
        • Chen S.
        • Zuhlke L.
        • Black G.C.
        • Choy M.K.
        • Li N.
        • et al.
        Global birth prevalence of congenital heart defects 1970–2017: updated systematic review and meta-analysis of 260 studies.
        Int J Epidemiol. 2019;
        • Hoashi T.
        • Miyata H.
        • Murakami A.
        • Hirata Y.
        • Hirose K.
        • Matsumura G.
        • et al.
        The current trends of mortality following congenital heart surgery: the Japan Congenital Cardiovascular Surgery Database.
        Interact Cardiovasc Thorac Surg. 2015; 21: 151-156
        • Yamaoka Y.
        • Tamiya N.
        • Watanabe A.
        • Miyazono Y.
        • Tanaka R.
        • Matsuzawa A.
        • et al.
        Hospital-based care utilization of children with medical complexity in Japan.
        Pediatr Int. 2018; 60: 626-633
        • Sasaki M.
        • Miyanomae T.
        • Yamamoto S.
        • Imai M.
        The surveillance of serious patients with severe motor and intellectual disabilities through SMID data base (in Japanese).
        Iryo (Tokyo). 2009; : 708-713
        • Chan T.
        • Rodean J.
        • Richardson T.
        • Farris R.W.D.
        • Bratton S.L.
        • Di Gennaro J.L.
        • et al.
        Pediatric critical care resource use by children with medical complexity.
        J Pediatr. 2016; 177: 197-203
        • Hanaoka T.
        • Mita K.
        • Hiramoto A.
        • Suzuki Y.
        • Maruyama S.
        • Nakadate T.
        • et al.
        Survival prognosis of Japanese with severe motor and intellectual disabilities living in public and private institutions between 1961 and 2003.
        J Epidemiol. 2010; 20: 77-81
        • Rose L.
        • McKim D.A.
        • Katz S.L.
        • Leasa D.
        • Nonoyama M.
        • Pedersen C.
        • et al.
        Home mechanical ventilation in Canada: a national survey.
        Respir Care. 2015; 60: 695-704
        • Valko L.
        • Baglyas S.
        • Gal J.
        • Lorx A.
        National survey: current prevalence and characteristics of home mechanical ventilation in Hungary.
        BMC Pulm Med. 2018; 18: 190
        • Racca F.
        • Berta G.
        • Sequi M.
        • Bignamini E.
        • Capello E.
        • Cutrera R.
        • et al.
        Long-term home ventilation of children in Italy: a national survey.
        Pediatr Pulmonol. 2011; 46: 566-572
        • Nasilowski J.
        • Wachulski M.
        • Trznadel W.
        • Andrzejewski W.
        • Migdal M.
        • Drozd W.
        • et al.
        The evolution of home mechanical ventilation in Poland between 2000 and 2010.
        Respir Care. 2015; 60: 577-585
        • Mundi M.S.
        • Pattinson A.
        • McMahon M.T.
        • Davidson J.
        • Hurt R.T.
        Prevalence of home parenteral and enteral nutrition in the United States.
        Nutr Clin Pract. 2017; 32: 799-805
        • Lezo A.
        • Capriati T.
        • Spagnuolo M.I.
        • Lacitignola L.
        • Goreva I.
        • Di Leo G.
        • et al.
        Paediatric home artificial nutrition in Italy: Report from 2016 survey on behalf of artificial nutrition network of Italian Society for Gastroenterology, Hepatology and Nutrition (SIGENP).
        Nutrients. 2016; : 10
        • Sako A.
        • Yasunaga H.
        • Horiguchi H.
        • Fushimi K.
        • Yanai H.
        • Uemura N.
        Prevalence and in-hospital mortality of gastrostomy and jejunostomy in Japan: a retrospective study with a national administrative database.
        Gastrointest Endosc. 2014; 80: 88-96