Introduction: Langerhans cell histiocytosis (LCH) is a rare atypical cellular disorder characterized by clonal proliferation of Langerhans cells leading to myriad clinical presentations and variable outcomes. It usually occurs in children and young adults. It can be present with local and systemic manifestation involving skin, bone, mucosal tissues and internal organs.
Aims and objectives: The stomatologist plays an important role in management of the disease by keeping in mind the various oral manifestations of the disease.
Case report: Of a child with disseminated LCH with multiorgan involvement who presented with failure to thrive, osteolytic bony lesions and extensive cutaneous eruptions.
Conclusion: Early diagnosis and awareness is necessary to treat the patients.
Mehta B, Amladi S. Langerhans cell histiocytosis presenting as hypopigmented papule. Pediatr Dermatol 2010;27(2):215-217.
Histiocytosis syndromes in children. Writing Group of the Histiocyte Society. Lancent 1987;1(8516):208-209.
Lahey E. Histiocytosis X: An analysis of prognostic factors. J Pediatr 1975;87(2):184-189.
A multicenter retrospective survey of Langerhans’ cell histiocytosis: 348 cases observed between 1983 and 1993. The French Langerhans’ Cell Study Group. Arch Dis Child 1996; 75(1):17-24.
Isaacs H Jr. Fetal and neonatal histiocytoses. Pediatr Blood Cancer 2006;47(2):123-129.
Longaker MA, Frieden IJ, LeBoit PE, Sherertz EF. Congenital ‘self-healing’ Langerhans cell histiocytosis: The need for long-term follow-up. J Am Acad Dermatol 1994;31(5):910-916.
Satter EK, Whitney. Langerhans cell histiocytosis: A case report and summary of the current recommendations of the Histiocyte Society. Dermatol Online J 2008;14(3):3.
Lau L, Krafchik B, Trebo MM, Weitzman S. Cutaneous Langerhans cell histiocytosis in children under 1 year. Pediatr Blood Cancer 2006;46(1):66-71.
Gadner H, Grois N, Arico M, Broadbent V, Ceci A, Jakobson A, Komp D, Michaelis J, Nicholson S, Pötschger U, et al. A randomized trial of treatment for multisystem Langerhans’ cell histiocytosis. J Pediatr 2001;138(5):728-734.
Shafer WG, Hine MK, Levy BM, editors. A Text Book of Oral Pathology. 4th edition. Philadelphia: WB Saunders; 1993. 308-312 p.
Kramer IRH, Pindborg JJ, Shear M. WHO International histological classification of tumours. Berlin: Springer; 1992.16-21 p.
Hitchin AD. The etiology of the calcified composite odontomes. Br Dent J 1971;130(11):475-482.
Piattelli A, Perfetti G, Carrano A. Complex odontome as a periapical and interradicular radiopacity in a primary molar. J Endod 1996;22(10):561-563.
Torreti EF, Carrel R. Compound odontoma in a 12-year-old girl. ASDC J Dent Child 1983;50:376-378.
Kokich VG, Mathews DP. Surgical and orthodontic management of impacted teeth. Dent Clin North Am 1993;37(2):181-204.
Veis A, Tziafas D, Lambrianidis T. A case report of a compound odontoma causing delayed eruption of a central maxillary incisor: Clinical and microscopic evaluation. J Endod 2000; 26(8):477-479.
Hisatomi M, Asaumi JI, Konouchi H, Honda Y, Wakasa T, Kishi K. A case of complex odontoma associated with an impacted lower deciduous second molar and analysis of the 107 odontomas. Oral Dis 2002;8(2):100-105.
Haavikko K. Hypodontia of permanent teeth: An orthopantomographic study. Suom Hammaslaak Toim 1971;67(4):219-225.
Kupietzky A, Houpt M. Hypohidrotic ectodermal dysplasia: Characteristics and treatment. Quintessence Int 1995;26(4): 285-291.
Nieminen P, Kotilainen J, Aalto Y, Knuutila S, Pirinen S, Thesleff I. MSX 1 gene is deleted in Wolf-Hirschhorn Syndrome patients with oligodontia. J Dent Res 2003;82(12):1013-1017.
Jorgenson RJ. Clinician's view of Hypodontia. J Am Dent Assoc 1980;101(2):283-286.
Ranta R. Herediatry agenesis of ten maxillary posterior teeth: A family history. ASDC J Dent Child 1985;52(2):125-127.
Mallineni SK, Jayaraman J, Yiu CK, King NM. Concomitant occurrence of hypohyperdontia in a patient with Marfan syndrome: A review of the literature and report of a case. J Investig Clin Dent 2012;3(4):253-257.
Kuppuswamy B. Manual of Socioeconomic Status (urban), Manasayan, Delhi 1981.
Parikh U, Trivedi G. Manual of socioeconomic status scale (Rural), Manasayan, Delhi, 1964.
Prasad BG. Social classification of Indian families. J Indian Med Assoc 1961;37:250-251.
Prasad BG. Social classification of Indian families. J Indian Med Assoc 1968;51:365-56.
Prasad BG. Changes proposed in Social classification of Indian families. J Indian Med Assoc 1970;55:198-199.
Mahmoodian J, Afshar H, Hadjhashem M. Determination of primate space on 4 to 5 Years Old Children of Tehran's Kindergarten in 2000. J Dent (Tehran) 2004;1(1):21-26.
Shivakumar KM, Chandu GN, Subba Reddy VV, Shafiulla MD. Prevalence of malocclusion and orthodontic treatment needs among middle and high school children of Davangere city, India by using Dental Aesthetic Index. J Indian Soc Pedod Prev Dent 2009;27(4):211-218.
Ferreira MC, Pastore C, Imada R, Guaré R, Leite M, Poyares D, Santos MT. Autonomic nervous system in individuals with cerebral palsy: a controlled study. J Oral Pathol Med 2011 Aug;40(7):576-581.
Sari ME, Ozmen B, Koyuturk AE, Tokay U. A retrospective comparison of dental treatment under general anesthesia on children with and without mental disabilities. Niger J Clin Pract 2014 May-Jun;17(3):361-365.
Gáspár J, Tóth Z, Fejérdy L, Kaán B, Fábián TK. Some background data about the high dental anxiety of the Hungarian population. Fogorv Sz 2004 Apr;97(2):85-89.
Shin WJ, Kang SJ, Kim YK, Seong SH, Han SM, Hwang GS. Link between heart rate and blood pressure Mayer wave during general anesthesia. Clin Auton Res 2011 Oct;21(5):309-317.
Li YX, Tang HT, Zhou WF, Hu XY, Xiao SC, Niu XH, Li YC, Wu YS, Yao M, Wang HX, et al. Analgesic and sedative effects of inhaling a mixture of nitrous oxide and oxygen on burn patient during and after dressing change. Zhonghua Shao Shang Za Zhi 2013 Dec;29(6):537-540.
Wongprasartsuk P, Stevens J. Cerebral palsy and anaesthesia. Paediatr Anaesth 2002 May;12(4):296-303.
Verschuren O, Wiart L, Ketelaar M. Stages of change in physical activity behavior in children and adolescents with cerebral palsy. Disabil Rehabil 2013 Jan;35(19):1630-1635.
Surén P, Bakken IJ, Aase H, Chin R, Gunnes N, Lie KK, Magnus P, Reichborn-Kjennerud T, Schj⊘lberg S, Øyen AS, et al. Autism spectrum disorder, ADHD, epilepsy, and cerebral palsy in norwegian children. Pediatrics 2012 Jul;130(1):e152-e158.
Pollare T, Lithell H, Selinus I, Berne C. Application of prazosin is associated with an increase of insulin sensitivity in obese patients with hypertension. Diabetologia 1988 Jul;31(7):415-420.
Berne C, Pollare T, Lithell H. Effects of antihypertensive treatment on insulin sensitivity with special reference to ACE inhibitors. Diabetes Care 1991 Nov;14(Suppl 4):39-47.
Cohn JN. Relationship of plasma volume changes to resistance and capacitance vessel effects of sympathomimetic amines and angiotensin in man. Clin Sci 1966 Apr;30(2):267-278.
Julius S, Pascual AV, Reilly K, London R. Abnormalities of plasma volume in borderline hypertension. Arch Intern Med 1971 Jan;127(1):116-119.
Greenwood JP, Scott EM, Stoker JB, Mary DA. Hypertensive left ventricular hypertrophy: relation to peripheral sympathetic drive. J Am Coll Cardiol 2001 Nov;38(6):1711-1717.
Spirito P, Bellone P, Harris KM, Bernabo P, Bruzzi P, Maron BJ. Magnitude of left ventricular hypertrophy and risk of sudden death in hypertrophic cardiomyopathy. N Engl J Med 2000 Jun;342:1778-1785.
Haider AW, Larson MG, Benjamin EJ, Levy D. Increased left ventricular mass and hypertrophy are associated with increased risk for sudden death. J Am Coll Cadiol 1998 Nov;32(5):1454-1459.
Jouven X, Zureik M, Desnos M, Guérot C, Ducimetière P. Resting heart rate as a predictive factor for sudden death in middle-aged men. Cardiovasc Res 2001 May;50(2):373-378.
Gillman MW, Kannel WB, Belanger A, D'Agostino RB. Influence of heart rate on mortality among persons with hypertension: the Framingham Study. Am Heart J 1993 Apr;125(4):1148-1154.
Julius S, Palatini P, Kjeldsen SE, Zanchetti A, Weber MA, McInnes GT, Brunner HR, Mancia G, Schork MA, Hua TA, et al. Usefulness of heart rate to predict future cardiac events in treated patients with high-risk systemic hypertension. Am J Cardiol 2012 Mar;109(5):685-692.
Palatini P, Roawi W, Casiglia E, Chalmers J, Ferrari R, Grassi G, Inoui T, Jelakovic B, Jensen MT, Julius S, et al. Management of the hypertensive patient with elevated heart rate: Statement of the Second Consensus Conference endorsed by the European Society of Hypertension. J Hypertens 2016 May;34(5):813-821.
Benedict CR, Shelton B, Johnstone DE, Francis G, Greenberg B, Konstam M, Probstfield JL, Yusuf S. Prognostic significance of plasma norepinephrine in patients with asymptomatic left ventricular dysfunction. Circulation 1996 Aug;94(4):690-697.
Grassi G, Seravalle G, Mancia G. Sympathetic activation in cardiovascular disease: evidence, clinical impact and therapeutic implications. Eur J Clin Invest 2015 Dec;45(12):1367-1375.
Cullington D, Goode KM, Zhang J, Cleland JG, Clark AL. Is heart rate important for patients with heart failure in atrial fibrillation? JACC Heart Fail 2014 Jun;2(3):213-220.
Wikstrand J, HJalmarson A, Waagstein F, Fagerberg J, Goldstein S, Kjekshus J, Wedel H; MERIT-HF Study Group. Dose of metoprolol CR/XL and clinical outcomes in patients with heart failure: analysis of the experience in metoprolol CR/XL randomized intervention trial in chronic heart failure (MERIT-HF). J Am Coll Cardiol 2002 Aug;40(3):491-498.
Yancy CW, Jessup M, Bozkurt B, Bulter J, Casey DE Jr, Colvin MM, Drazner MH, Filippatos GS, Fonarow GC, Givertz MM, et al. 2017 ACC/AHA/HFSA focused update of the 2013 ACCF/AHA guideline for the management of heart failure: A Report of the American College of Cardiology/American Heart Association Task Force on Clinical Practice Guidelines and the Heart Failure Society of America. J Am Coll Cardiol 2017 Aug;70(6):776-803.
Luetic AT. First experience in clinical application of KANET. Donald School J Ultrasound Obstet Gynecol 2015;9(1):96-99.
Vasilj O. Is in utero fetal neurological assessment comparible to postnatal neurological assessment? Donald School J Ultrasound Obstet Gynecol 2015;9(1):91-95.
Tomasovic S, Predojevic M. 4D ultrasound medical devices for recent advances on the etiology of cerebral palsy. Acta Inform Med 2011 Dec;19(4):228-234.
Vladareanu R, Lebit D, Constantinescu S. Ultrasound assessment of fetal neurobehavior in high-risk pregnancies. Donald School J Ultrasound Obstet Gynecol 2012 Apr-Jun;6(2):132-147.
Honemeyer U, Talic A, Therwat A, Paulose I, Patidar R. The clinical value of KANET test in studying fetal neurobehavior in normal and at-risk pregnancies. J Perinat Med 2013 Mar;41(2):187-197.
Kadic AS. Fetal neurology: the role of fetal stress. Donald School J Ultrasound Obstet Gynecol 2015;9(1):30-39.
Hata T, Kanenishi J, Hanoka U, Uematsu R, Marumo G, Tanaka H. HDlive study of fetal development and behavior. Donald School J Ultrasound Obstet Gynecol 2014;8(3):250-265.
Reynoso C, Crespo-Eguílaz N, Alcázar JL, Narbona J. Motor behavior of human fetuses during the second trimester of gestation: a longitudinal ultrasound study. An Pediatr (Barc) 2015 Mar;82(3):183-191.
Predojević M, Talić A, Stanojević M, Kurjak A, Salihagić Kadić A. Assessment of motoric and hemodynamic parameters in growth restricted fetuses-case study. J Matern Fetal Neonatal Med 2014 Feb;27(3):247-251.
Kurjak A, Talic A, Stanojevic M, Honemeyer U, Serra B, Prats P, Di Renzo GC. The study of fetal neurobehavior in twins in all three trimesters of pregnancy. J Matern Fetal Neonatal Med 2013 Aug;26(12):1186-1195.
Athanasiadis AP, Mikos T, Tambakoudis GP, Theodoridis TD, Papastergiou M, Assimakopoulos E, Tarlatzis BC. Neuro-developmental fetal assessment using Kanet scoring system in low and high-risk pregnancies. J Matern Fetal Neonatal Med 2013;26(4):363-368.
Kurjak A, Stanojević M, Predojević M, Laušin I, Salihagić-Kadić A. Neurobehavior in fetal life. Semin Fetal Neonatal Med 2012 Dec;17(6):319-323.
Hepper PG, Dornan JC, Lynch C, Maguire JF. Alcohol delays the emergence of the fetal elicited startle response, but only transiently. Physiol Behav 2012 Aug 20;107(1):76-81.
Talic A, Kurjak A, Stanojevic M, Honemeyer U, Badreldeen A, Direnzo GC. The assessment of fetal brain function in fetuses with ventriculomegaly: the role of the Kanet test. J Matern Fetal Neonatal Med 2012 Aug;25(8):1267-1272.
Kurjak A, Honemeyer U, Stanojevic M, Zalud I. Comparison between antenatal neurodevelopmental test and fetal Doppler in the assessment of fetal well-being. J Perinat Med 2013 Jan;41(1):107-114.
Neto MR. KANET in Brazil: first experience. Donald School J Ultrasound Obstet Gynecol 2015;9(1):1-5.
Morokuma S, Fukushima K, Yumoto Y, Uchimura M, Fujiwara A, Matsumoto M, Satoh S, Nakano H. Simplified ultrasound screening for fetal brain function based on behavioral pattern. Early Hum Dev 2007 Mar;83(3):177-181.
Kurjak A, Tikvica Luetic A, Stanojevic M, Talic A, Zalud I, Al-Noobi M, Perva S, Abushama M, Tomasovic S, Zaputovic S, et al. Further experience in the clinical assessment of fetal neurobehavior. Donald School J Ultrasound Obstet Gynecol 2010 Jan-Mar;4(1):59-71.
Kurjak A, Abo-Yaqoub S, Stanojevic M, Yigiter AB, Vasilj O, Lebit D, Shaddad AN, Ahmed B, Kavak ZN, Miskovic B, et al. The potential of 4D sonography in the assessment of fetal neurobehavior: multicentric study in high-risk pregnancies. J Perinat Med 2010;38(1):77-82.
Miskovic B, Vasilj O, Stanojevic M, Ivankovicc D, Kerner M, Tikvica A. The comparison of fetal behavior in high-risk and normal pregnancies assessed by four-dimensional ultrasound. J Matern Fetal Neonatal Med 2010 Dec;23(12):1461-1467.
Kurjak A, Ahmed B, Abo-Yaguab S, Younis M, Saleh H, Shaddad AN, Vasilj O, Al Bahar AJ, Miskovic B, Khenyab N. An attempt to introduce neurological test for fetus based on 3D and 4D sonography. Donald School J Ultrasound Obstet Gynecol 2008;2:29-44.
Livingston JH, Stivaros S, Warren D, Crow YJ. Intracranial calcification in childhood: a review of aetiologies and recognizable phenotypes. Dev Med Child Neurol 2014 Jul;56(7):612-626.
Couvreur J, Desmonts G, Tournier G, Szusterkac M. A homogeneous series of 210 cases of congenital toxoplasmosis in 0 to 11-month-old infants detected prospectively. Ann Pediatr (Paris) 1984 Nov;31(10):815-819. (French).
Valentini P, Annunziata ML, Angelone DF, Masini L, De Santis M, Testa A, Grillo RL, Speziale D, Ranno O. Role of spiramycin/cotrimoxazole association in the mother-to-child transmission of toxoplasmosis infection in pregnancy. Eur J Clin Microbiol Infect Dis 2009 Mar;28(3):297-300.
Valentini P, Buonsenso D, Barone G, Serranti D, Calzedda R, Ceccarelli M, Speziale D, Ricci R, Masini L. Spiramycin/cotrimoxazole versus pyrimethamine/sulfonamide and spiramycin alone for the treatment of toxoplasmosis in pregnancy. J Perinatol 2015 Feb;35(2):90-94.
Masini L, De Santis M, Apicella M, Noia G, Santangelo R, Speziale D, Lanzone A, Valentini P. Prenatal diagnosis by ultrasound and amniocentesis in a single center cohort of 1065 pregnancies complicated by maternal Toxoplasma infection. ISUOG 2017—27th World Congress—16-19 September, Vienna, Austria OC05.05. Ultrasound Obstet Gynecol 2017;50(Suppl 1):10.
Vynnycky E, Adams EJ, Cutts FT, Reef SE, Navar AM, Simons E, Yoshida LM, Brown DW, Jackson C, Strebel PM, et al. Using seroprevalence and immunisation coverage data to estimate the global burden of congenital rubella syndrome, 1996-2010: a systematic review. PLoS One 2016 Mar 10;11(3):e0149160.
Hutton J. Does rubella Cause autism: a 2015 reappraisal? Front Hum Neurosci 2016 Feb 1;10:25.
Thompson KM, Simons EA, Badizadegan K, Reef SE, Cooper LZ. Characterization of the risks of adverse outcomes following rubella infection in pregnancy. Risk Anal 2016 Jul;36(7):1315-1331.
De Santis M, Cavaliere AF, Straface G, Caruso A. Rubella infection in pregnancy. Reprod Toxicol 2006 May;21(4):390-298.
Harger JH, Ernest JM, Thurnau GR, Moawad A, Thom E, Landon MB, Paul R, Miodovnik M, Dombrowski M, Sibai B, et al. Frequency of congenital varicella syndrome in a prospective cohort of 347 pregnant women. Obstet Gynecol 2002 Aug;100(2):260-265.
Enders G, Miller E, Cradock-Watson J, Bolley I, Ridehalgh M. Consequences of varicella and herpes zoster in pregnancy: prospective study of 1739 cases. Lancet 1994 Jun 18;343(8912):1548-1551.
Tan MP, Koren G. Chickenpox in pregnancy: revisited. Reprod Toxicol 2006 May;21(4):410-420.
Pastuszak AL, Levy M, Schick B, Zuber C, Feldkamp M, Gladstone J, Bar-Levy F, Jackson E, Donnenfeld A, Meschino W, et al. Outcome after maternal varicella infection in the first 20 weeks of pregnancy. N Engl J Med 1994 Mar 31;330(13):901-905.
Pretorius DH, Hayward I, Jones KL, Stamm E. Sonographic evaluation of pregnancies with maternal varicella infection. J Ultrasound Med 1992 Sep;11(9):459-463.
Verstraelen H, Vanzieleghem B, Defoort P, Vanhaesebrouck P, Temmerman M. Prenatal ultrasound and magnetic resonance imaging in fetal varicella syndrome: correlation with pathology findings. Prenat Diagn 2003 Sep;23(9):705-709.
Paryani SG, Arvin AM. Intrauterine infection with varicella-zoster virus after maternal varicella. N Engl J Med 1986 Jun 12;314(24):1542-1546.
Meyberg-Solomayer GC, Fehm T, Muller-Hansen I, Enders G, Poets C, Wallwiener D, Solomayer EF. Prenatal ultrasound diagnosis, follow-up, and outcome of congenital varicella syndrome. Fetal Diagn Ther 2006;21(3):296-301.
Pichler M, Staffler A, Bonometti N, Messner H, Deluca J, Thuile T, Kluge R, Schmuth M, Eisendle K. Premature newborns with fatal intrauterine herpes simplex virus-1 infection: first report of twins and review of the literature. J Eur Acad Dermatol Venereol 2015 Jun;29(6):1216-1220.
Plourde AR, Bloch EM. A literature review of Zika virus. Emerg Infect Dis 2016 Jul;22(7):1185-1192.
Paixão ES, Barreto F, Teixeira Mda G, Costa Mda C, Rodrigues LC. History, epidemiology, and clinical manifestations of Zika: a systematic review. Am J Public Health 2016 Apr;106(4):606-612.
Rasmussen SA, Jamieson DJ, Honein MA, Petersen LR. Zika virus and birth defects—reviewing the evidence for causality. N Engl J Med 2016 May 19;374(20):1981-1987.
Honein MA, Dawson AL, Petersen EE, Jones AM, Lee EH, Yazdy MM, Ahmad N, Macdonald J, Evert N, Bingham A, et al. Birth defects among fetuses and infants of US women with evidence of possible Zika virus infection during pregnancy. JAMA 2017 Jan 3;317(1):59-68.
Chervenak FA, Jeanty P, Cantraine F, Chitkara U, Venus I, Berkowitz RL, Hobbins JC. The diagnosis of fetal microcephaly. Am J Obstet Gynecol 1984 Jul 1;149(5):512-517.
Papageorghiou AT, Thilaganathan B, Bilardo CM, Ngu A, Malinger G, Herrera M, Salomon LJ, Riley LE, Copel JA. ISUOG Interim Guidance on ultrasound for Zika virus infection in pregnancy: information for healthcare professionals. Ultrasound Obstet Gynecol 2016 Apr;47(4):530-532.
Chibueze EC, Parsons AJ, Lopes KD, Yo T, Swa T, Nagata C, Horita N, Morisaki N, Balogun OO, Dagvadorj A, et al. Diagnostic accuracy of ultrasound scanning for prenatal microcephaly in the context of Zika virus infection: a systematic review and meta-analysis. Sci Rep 2017 May 23;7(1):2310.
Cordeiro MT. Zika virus: laboratory diagnosis. In: Zika in focus. Springer International Publishing; 2017. pp. 59-62.
Mlakar J, Korva M, Tul N, Popović M, Poljšak-Prijatelj M, Mraz J, Kolenc M, Resman Rus K, Vesnaver Vipotnik T, Fabjan Vodušek V, et al. Zika virus associated with microcephaly. N Engl J Med 2016 Mar 10;374(10):951-958.
Zare Mehrjardi M, Keshavarz E, Poretti A, Hazin AN. Neuroimaging findings of Zika virus infection: a review article. Jpn J Radiol 2016 Dec;34(12):765-770.
Oliveira Melo AS, Malinger G, Ximenes R, Szejnfeld PO, Alves Sampaio S, Bispo de Filippis AM. Zika virus intrauterine infection causes fetal brain abnormality and microcephaly: tip of the iceberg? Ultrasound Obstet Gynecol 2016 Jan;47(1):6-7.
Brasil P, Pereira JP Jr, Moreira ME, Ribeiro Nogueira RM, Damasceno L, Wakimoto M, Rabello RS, Valderramos SG, Halai UA, Salles TS, et al. Zika virus infection in pregnant women in Rio de Janeiro. N Engl J Med 2016 Dec 15;375(24):2321-2334.