Congenital infiltrating lipomatosis of face: Two cases reports and review

doi:10.3877/cma.j.issn.1674-1366.2024.01.008

Abstract

Abstract:

Congenital infiltrating lipomatosis of face (CILF) is a rare disease that is congenital, non-hereditary, and increased infiltrating adipose tissue. CILF is mostly found at birth, with the main clinical manifestations of obvious facial soft tissue swelling, jaw bone swelling, early eruption of permanent teeth, macrodontism, etc. It is often occurring to the lower 2/3 parts of the face, and the left side. Imaging examination shows fatty infiltration of soft tissues in the affected face. This article reported two cases of CILF, and reviewed its etiology, clinical manifestations, diagnosis, and treatment progress.

Key words: Congenital infiltrating lipomatosis of face, Facial hyperplasia, Early eruption of permanent tooth, Macrodontism

Yingqian Wu, Jimei Su. Congenital infiltrating lipomatosis of face: Two cases reports and review[J]. Chinese Journal of Stomatological Research(Electronic Edition), 2024, 18(01): 48-53.

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References 26

[1]	Li Y，Chang G，Si L，et al. Congenital infiltrating lipomatosis of the face：Case report and literature review[J]. Ann Plast Surg，2018，80（1）：83-89. DOI：10.1097/SAP.0000000000001213.
[2]	Kamal D，Breton P，Bouletreau P. Congenital infiltrating lipomatosis of the face：Report of three cases and review of the literature[J]. J Craniomaxillofac Surg，2010，38（8）：610-614. DOI：10.1016/j.jcms.2010.02.014.
[3]	Gupta R，Mukul SK，Kumar P，et al. Congenital infiltrating lipomatosis of the face with temporomandibular joint ankylosis[J]. Natl J Maxillofac Surg，2020，11（1）：117-120. DOI：10.4103/njms.NJMS_59_17.
[4]	樊丽娜，傅升，姚丽青.罕见巨大舌先天性浸润型脂肪增殖症1例[J].实用口腔医学杂志，2006，22（5）：634. DOI：10.3969/j.issn.1001-3733.2006.05.049.
[5]	Slavin SA，Baker DC，McCarthy JG，et al. Congenital infiltrating lipomatosis of the face：Clinicopathologic evaluation and treatment[J]. Plast Reconstr Surg，1983，72（2）：158-164. DOI：10.1097/00006534-198308000-00006.
[6]	Donati L，Candiani P，Grappolini S，et al. Congenital infiltrating lipomatosis of the face related to cytomegalovirus infection[J]. Br J Plast Surg，1990，43（1）：124-126. DOI：10.1016/0007-1226(90)90058-8.
[7]	Couto RA，Mulliken JB，Padwa BL，et al. Facial infiltrating lipomatosis：Expression of angiogenic and vasculogenic factors[J]. J Craniofac Surg，2011，22（6）：2405-2408. DOI：10.1097/SCS.0b013e318231fe6c.
[8]	Capra V，Severino M，Rossi A，et al. Pituitary deficiency and congenital infiltrating lipomatosis of the face in a girl with deletion of chromosome 1q24.3q31.1[J]. Am J Med Genet A，2014，164A（2）：495-499. DOI：10.1002/ajmg.a.36283.
[9]	Maclellan RA，Luks VL，Vivero MP，et al. PIK3CA activating mutations in facial infiltrating lipomatosis[J]. Plast Reconstr Surg，2014，133（1）：12e-19e. DOI：10.1097/01.prs.0000436822.26709.7c.
[10]	Couto JA，Konczyk DJ，Vivero MP，et al. Somatic PIK3CA mutations are present in multiple tissues of facial infiltrating lipomatosis[J]. Pediatr Res，2017，82（5）：850-854. DOI：10.1038/pr.2017.155.
[11]	Briand C，Galmiche-Rolland L，Vabres P，et al. Congenital infiltrating lipomatosis of the face with lingual mucosal neuromas associated with a PIK3CA mutation[J]. Pediatr Dermatol，2020，37（6）：1128-1130. DOI：10.1111/pde.14302.
[12]	刘珊，杨利洒，丁张帆，等.面部先天性浸润性脂肪增殖症伴发癫痫1例[J].华西口腔医学杂志，2019，37（1）：109-112. DOI：10.7518/hxkq.2019.01.022.
[13]	Sun R，Sun L，Li G，et al. Congenital infiltrating lipomatosis of the face：A subtype of hemifacial hyperplasia[J]. Int J Pediatr Otorhinolaryngol，2019，125：107-112. DOI：10.1016/j.ijporl.2019.06.032.
[14]	Serpa MS，Scully C，Molina Vivas AP，et al. Infiltrating lipomatosis of the face：Case series and literature review[J]. Oral Surg Oral Med Oral Pathol Oral Radiol，2017，123（3）：e99-e105. DOI：10.1016/j.oooo.2016.10.009.
[15]	Sahai S，Rajan S，Singh N，et al. Congenital infiltrating lipomatosis of the face with exophytic temporomandibular joint ankylosis：A case report and review of the literature[J]. Dentomaxillofac Radiol，2013，42（3）：16128745. DOI：10.1259/dmfr/16128745.
[16]	Shenoy AR，Nair KK，Lingappa A，et al. Congenital infiltrating lipomatosis of face：Case report and review of literature[J]. J Indian Soc Pedod Prev Dent，2015，33（2）：156-160. DOI：10.4103/0970-4388.155134.
[17]	Sant′Anna GD，Saffer M，Mauri M，et al. Encephalocraniocutaneous lipomatosis with otolaryngologic manifestations：A rare neurocutaneous syndrome[J]. Int J Pediatr Otorhinolaryngol，1999，49（3）：231-235. DOI：10.1016/s0165-5876(99)00199-8.
[18]	Islam MN，Bhattacharyya I，Ojha J，et al. Comparison between true and partial hemifacial hypertrophy[J]. Oral Surg Oral Med Oral Pathol Oral Radiol Endod，2007，104（4）：501-509. DOI：10.1016/j.tripleo.2006.11.053.
[19]	Bou-Haidar P，Taub P，Som P. Hemifacial lipomatosis，a possible subtype of partial hemifacial hyperplasia：CT and MR imaging findings[J]. AJNR Am J Neuroradiol，2010，31（5）：891-893. DOI：10.3174/ajnr.A1857.
[20]	Kim JE，Gottschall JA，Bachman RP，et al. Facial infiltrating lipomatosis：Physical，radiological，and histopathological findings[J]. Arch Otolaryngol Head Neck Surg，2010，136（3）：301-303. DOI：10.1001/archoto.2010.21.
[21]	Bashizadehfakhar H，Panjnoush M，Kheirandish Y，et al. Radiographic findings of congenital facial infiltrating lipomatosis：A case report[J]. Front Dent，2022，19：27. DOI：10.18502/fid.v19i27.10598.
[22]	van Wingerden JJ，Erlank JD，Becker JH. Liposuction for congenital infiltrating lipomatosis of the face[J]. Plast Reconstr Surg，1988，81（6）：989. DOI：10.1097/00006534-198806000-00047.
[23]	Padwa BL，Mulliken JB. Facial infiltrating lipomatosis[J]. Plast Reconstr Surg，2001，108（6）：1544-1554. DOI：10.1097/00006534-200111000-00017.
[24]	Kalantary S，van de Casteele E，Nadjmi N. Congenital infiltrating lipomatosis of the face：Case report with presentation of a new multistep surgical approach[J]. J Oral Maxillofac Surg，2018，76（6）：1334-1343. DOI：10.1016/j.joms.2017.10.015.
[25]	Tracy JC，Klement GL，Scott AR. Interdisciplinary management of congenital infiltrating lipomatosis[J]. Int J Pediatr Otorhinolaryngol，2013，77（12）：2071-2074. DOI：10.1016/j.ijporl.2013.08.008.
[26]	Oh KS，Bahmad HF，Stoyanov KV，et al. Recurrent PIK3CA H1047R-mutated congenital infiltrative facial lipomatosis：A case report and review of literature[J]. Curr Issues Mol Biol，2023，45（2）：1712-1719. DOI：10.3390/cimb45020110.

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