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Case report

Oxford, UK
International
IJD
Blackwell
1365-4632
45 Publishing,
Publishing
Journal Ltd,
of
Ltd.
Dermatology
2004

Lingual traumatic ulceration (Riga–Fede disease)


Lingualettraumatic
Baroni
Cameo al. ulceration

Adone Baroni, MD, Carlo Capristo, MD, Luigi Rossiello, MD, Franco Faccenda, MD, and
Rocco Alfredo Satriano, MD

From the Departments of Dermatology and An 11-month-old male infant was referred to our clinic because of a painful ulcer of
Pediatrics, 2nd University of Naples, Naples, approximately 5 months’ duration on the ventral surface of the tongue (Fig. 1). On physical
Italy
examination, the lesion was circular (3 cm × 2 cm) with erythematous, raised, and indurated
borders.
Correspondence
Rocco Alfredo Satriano, MD No pathologic findings emerged from the laboratory data, neurologic examination, or
2nd University of Naples clinical history. The family history was also negative for developmental disorders and congenital
via Sergio Pansini 5 syndromes. No biopsy was performed in view of the age of the infant, the particular site of the
80131 Napoli lesion, and the clinical evidence of diagnosis. The treatment included odontologic cream
Italy
(methylvinylether/maleic acid) as a protective shield, a collutorium (chlorhexidine 0.2%), and
E-mail: vincruoc@tin.it
the use of a teething ring. Complete healing of the lesion (Fig. 2) occurred within 3 weeks.

A broad variety of terms have been used to describe RFD,


Discussion
such as eosinophilic ulcer of the oral mucosa, sublingual
Riga–Fede disease (RFD) is extremely rare and is character- fibrogranuloma, sublingual growth in infants, lingual traumatic
ized by an ulceration of the tongue, often caused by repetitive ulceration, traumatic atrophic glossitis, traumatic granuloma
traumatic injuries from back and forward movement of the of the tongue, and traumatic ulcerative granuloma with stro-
tongue over the mandibular anterior incisors. The lesion was mal eosinophilia.1–3 RFD may be considered as a benign con-
first identified by Antonio Riga, an Italian physician, in 1881. dition in healthy infants, but it may be associated with other
Further cases of the disease were described by Fede, who also serious neurologic disorders.2,6 Early detection of RFD is
performed histologic studies of the lesion.1–3 Microscopic recommended to treat these severe diseases, but also for other-
findings mainly show an inflammatory infiltrate composed of wise healthy infants. Indeed, a failure to diagnose or treat
numerous eosinophils with lymphocytes, macrophages, plasma these lesions properly may induce deformity or mutilation of
cells, and mast cells.4,5 the tongue and dehydration or inadequate nutrient intake for

1096 Figure 1 Ulcer on the tongue

International Journal of Dermatology 2006, 45, 1096–1097 © 2004 The International Society of Dermatology
Baroni et al. Lingual traumatic ulceration Case report 1097

Figure 2 Healing of the ulcer after treatment

the infant.1,2,6 Nevertheless, traumatic ulceration related to


References
teething is highly unusual in infants, so that the lack of experi-
ence may result in a failure to diagnose. 1 Ahmet T, Ferruh B, Gurcan A. Lingual traumatic ulceration
Ulceration of the tongue may also be due to other causes, (Riga–Fede disease). Br J Oral Maxillofac Surg 2003;
including bacterial or mycotic infections, allergy and immuno- 41: 201.
logic diseases, tumors, or genetic disorders.2,3,6 The clinical 2 Baghdadi ZD. Riga–Fede disease: association with
presentation, laboratory data, and positive results obtained microcephaly. Int J Paediatr Dent 2002; 12: 442– 445.
after treatment rule out these diagnostic hypotheses in this 3 Elzay RP. Traumatic ulcerative granuloma with stromal
case, so that a biopsy was not performed. With regard to the eosinophilia (Riga–Fede disease) and traumatic eosinophilic
treatment of RFD, several management options have been granuloma. Oral Surg 1983; 55: 497 –506.
4 Zaenglein AL, Chang MW, Meehan SA, et al. Extensive
reported that may be used alone or in combination: (i) cellu-
Riga–Fede disease of the lip and tongue. J Am Acad
lose film or other protective dental appliance; (ii) oral dis-
Dermatol 2002; 47: 445– 447.
infectant; (iii) corticosteroids; (iv) teething ring; and (v) filing of
5 Terzioglu A, Bingul F, Aslan G. Lingual traumatic ulceration
the teeth to smooth sharp cutting edges.1,3,6 The extraction of (Riga–Fede disease). J Oral Maxillofac Surg 2002;
the lower incisors may also be indicated, but must be regarded 60: 478.
as a last resort.2,6 In the present case, we used conservative 6 Slayton RL. Treatment alternatives for sublingual traumatic
treatment, consisting of a protective composite over the sharp ulceration (Riga–Fede disease). Pediatr Dent 2000; 22:
edges, oral disinfectant, and a teething ring. 413 –414.

© 2004 The International Society of Dermatology International Journal of Dermatology 2006, 45, 1096–1097

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