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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 4  |  Issue : 2  |  Page : 168-169

Tongue harmatoma in association with cleft palate: Case report


1 Department of Surgery, ESUT Teaching Hospital Parklane; Department of Plastic Surgery, National Orthopaedic Hospital, Enugu, Nigeria
2 Department of Plastic Surgery, National Orthopaedic Hospital, Enugu, Nigeria

Date of Web Publication11-Aug-2017

Correspondence Address:
Uwakwe Cosmas Mba
Department of Surgery, ESUT Teaching Hospital Parklane, P.M.B. 1030, Enugu
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jclpca.jclpca_34_17

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  Abstract 


Tongue hamartoma is not a common congenital disorder. Common associations include cardiac defects and limb anomalies such as polydactyly and syndactyly. Its association with cleft palate is unusual and as such only very few cases have been reported. A 15-month-old girl who presented to us with a mass on the tongue in association with cleft of the soft palate is presented. She had cleft palate repair and incisional biopsy of the mass. Initial histology was lipoma. The mass was excised a year later and histology confirmed lipomatous harmatoma.

Keywords: Case report, cleft palate, tongue harmatoma


How to cite this article:
Mba UC, Onah II. Tongue harmatoma in association with cleft palate: Case report. J Cleft Lip Palate Craniofac Anomal 2017;4:168-9

How to cite this URL:
Mba UC, Onah II. Tongue harmatoma in association with cleft palate: Case report. J Cleft Lip Palate Craniofac Anomal [serial online] 2017 [cited 2019 Oct 18];4:168-9. Available from: http://www.jclpca.org/text.asp?2017/4/2/168/212837




  Introduction Top


Hamartoma was first defined by Alberecht in 1904 as a benign, tumor-like malformation composed of a disordered mixture of mature tissues that normally occur in the affected part, but with a predominance of one particular tissue. Commonly internal organs are affected.[1] When it occurs in the tongue, common associations are cardiac defects or limb anomalies and very rarely cleft palate. These occur individually or as part of oro-facial-digital syndrome.[2] We present a 15-month-old girl with tongue hamartoma and cleft of the soft palate.


  Case Report Top


A 15-month-old girl was referred to us with a hole in the roof of the mouth and a mass on the tongue. No other defect was noticed. The antenatal period was uneventful. The child was the second of a set of twins. The other sibling had no similar anomaly.

Examination showed a soft fleshy mass on the posterior ½ of the dorsum of the tongue, and a cleft of the soft palate [Figure 1] and [Figure 2]. Ultrasound revealed normal thyroid gland.
Figure 1: Cleft palate immediate preoperative

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Figure 2: Tongue mass immediate preoperative

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She had cleft palate repair with intravelar veloplasty and incisional biopsy of the mass. Histology revealed lipoma. About 1 year later, the mass was completely excised with primary closure of the resulting defect. Histology confirmed lipomatous hamartoma.


  Discussion Top


Tongue mass with cleft palate is an unusual association and can be considered a developmental sequence. Tongue development is one of the earliest events in fetal life (4th–5th week) and affects the development of other maxillofacial structures.[3] The palate develops between 6th and 7th week of fetal life. Tongue malposition can result in palatal defect as in Pierre-Robbin sequence.[4],[5] A mass on the dorsum of the tongue can, therefore, cause palatal cleft if present during palatal shelf development.

Tongue hamartomas are usually composed of indigenous tissues with a predominance of one. Smooth muscle was predominant tissue in most cases reported,[1],[6],[7] but in our patient, fat was predominant.

Irrespective of predominant tissue component, complete excision is the treatment of choice as it is curative with only one report of recurrence following incomplete excision.[1],[7],[8] If associated with cleft palate, it seems rational to excise the mass before or at the same time the cleft palate repair is done. This avoids the mass interfering with the repair. In our patient, the cleft palate repair with incisional biopsy was done before excision of the hamartoma without any effect on the repair. We needed to determine the histology of the mass and because our patient presented at 15 months of age, the cleft palate needed to be repaired to ensure good speech development. There is general agreement that cleft palate is repaired between 6 and 12 months of age, and hence that function of the velum and palate is optimized before speech development.[9]


  Conclusion Top


Tongue hamartoma is very rarely associated with cleft palate and may result from the hamartoma obstructing palate development. In isolated nonsyndromic cases, this association should be considered a developmental sequence.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Wang HL, Chiang FY, Tai CF, Tsai KB, Wang LF. Lingual leiomyomatous hamartoma with bifid tip and ankyloglossia in a patient without oral-facial-digital syndrome: A case report and literature review. World J Surg Oncol 2013;11:230.  Back to cited text no. 1
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2.
Poretti A, Vitiello G, Hennekam RC, Arrigoni F, Bertini E, Borgatti R, et al. Delineation and diagnostic criteria of Oral-Facial-Digital Syndrome type VI. Orphanet J Rare Dis 2012;7:4.  Back to cited text no. 2
[PUBMED]    
3.
Hong SJ, Cha BG, Kim YS, Lee SK, Chi JG. Tongue growth during prenatal development in Korean Fetuses and Embryos. J Pathol Transl Med 2015;49:497-510.  Back to cited text no. 3
[PUBMED]    
4.
Bjork BC, Turbe-Doan A, Prysak M, Herron BJ, Beier DR. Prdm16 is required for normal palatogenesis in mice. Hum Mol Genet 2010;19:774-89.  Back to cited text no. 4
[PUBMED]    
5.
Parada C, Han D, Grimaldi A, Sarrión P, Park SS, Pelikan R, et al. Disruption of the ERK/MAPK pathway in neural crest cells as a potential cause of Pierre Robin sequence. Development 2015;142:3734-45.  Back to cited text no. 5
    
6.
Arjuna B, Miran N, Qurashi H. Case report of congenital leiomyomatous hamartoma: New epidemiological findings and review of literature. Int J Otorhinolaryngol2012;76:1528-30.  Back to cited text no. 6
    
7.
Nakanishi K, Nomura J, Matsumura Y, Yanase S, Kato H, Tagawa T. Leiomyomatous hamartoma of the tongue in an infant: A case report. J Dent Child (Chic) 2012;79:111-4.  Back to cited text no. 7
[PUBMED]    
8.
Demuth RJ, Johns DF. Recurrent aspiration pneumonitis in a cleft palate child with hamartoma of the tongue. Cleft Palate J 1981;18:299-303.  Back to cited text no. 8
[PUBMED]    
9.
Nahai FR, Williams JK, Burstein FD, Martin J, Thomas J. The management of cleft lip and palate: Pathways for treatment and longitudinal assessment. Sem Plast Surg 2005;19:275-85.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2]



 

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