Dental Implants in a Young Patient with Papillon-Lefevre Syndrome: A Case Report
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Dental Implants in a Young Patient with
Papillon-Lefevre Syndrome: A Case Report
Ian Woo, MSc, DDS,* Daniel P. Brunner, DDS, MD,** Dennis-Duke R. Yamashita, DDS,*** Bach T. Le, DDS, MD†
apillon-Lefevre Syndrome is A case is reported of dental im- cessful osseointegration and preser-
P characterized by generalized
rapid destruction of the dental
alveolar supporting bone and diffused
plant placement in a 13-year-old pa-
tient diagnosed with Papillon-
Lefevre Syndrome. Two titanium
vation of alveolar bone 1 year after
implant placement and the continual
wearing of a functional dental
palmoplantar hyperkeratosis. The dis- dental implants were placed in the prosthesis. (Implant Dent 2003;12:
order manifests itself as an autosomal
mandible for an implant-retained 140 –144)
recessive disease with an occurrence
of about one to four cases per million.1 denture after the patient complained Key Words: alveolar bone preserva-
It affects both the primary and second- of having an unstable prosthesis. tion, implant-retained overdenture,
ary dentition. The periodontal changes Follow-up radiographs showed suc- early edentulism
usually appear before the age of 4
years. Inflammatory response in the
vere loss of alveolar bone is often the Fentanyl (Abbott Labs, Chicago, IL)
periodontium leads to rapid bone loss
result.2,5,6 Early extractions of all per- were used. A total of 11 teeth were
and exfoliation of teeth. Because both
manent teeth has been considered as extracted. Tooth 1 and 16 were spared
sets of dentitions are affected, these
the treatment of choice to preserve the because they were unerupted and
patients are usually edentulous and
remaining supporting bone.7 would not interfere with the dental
wearing complete dentures by their
teen years. prosthesis. The patient was given an-
The exact immunologic abnor- CASE REPORT tibiotics and analgesics postopera-
mality of Papillon-Lefevre Syndrome A 13-year-old male diagnosed tively. The follow-up took place with
is unknown. It has been reported that with Papillon-Lefevre syndrome was the general dentist at the dental clinic.
the disease may be associated with presented at the Los Angeles County/ Complete upper and lower dentures
diminished neutrophil activity.2 Mi- University of Southern California were fabricated for the patient. One
croscopic changes include marked (LAC/USC) Medical Center outpa- year later, the patient complained
chronic inflammation with predomi- tient dental clinic in April 1999. The about the instability of the lower
nant plasma-cell infiltration, oste- patient was also seen by the dermatol- denture.
oclastic activity, and lack of osteoblas- ogy and ophthalmology departments. He was consulted for dental im-
tic activity.3 The bacterial flora in this The patient displayed the classic signs plant placement at the oral and maxil-
disease a similar to those found in of diffused palmoplantar hyperkerato- lofacial department of the LAC/USC
adult periodontitis with a prevalence sis (Fig. 1). Intraoral examination re- Medical Center. The treatment plan
of gram negative cocci, rods, and vealed class III hypermobility in all was to place two dental implants and
spirochetes.4 remaining dentition with severe gingi- fabricate an implant-retained overden-
Because conventional periodontal val inflammation. A panoramic radio- ture for his mandible. A preoperative
treatment usually fails to arrest the graph showed generalized advanced Panorex was taken before implant
rapid progression of periodontitis, se- bone loss with both an atrophic max- placement (Fig. 3). Two 4.0 ⫻ 13 mm
illa and mandible (Fig. 2). It was de- Branemark system titanium implants
*Resident, Department of Oral and Maxillofacial Surgery, Los
termined that all erupted teeth were (Nobel Biocare, Goteborg, Sweden)
Angeles County/University of Southern California Medical Cen-
ter, Los Angeles, CA.
nonrestorable and required were placed in the left and right canine
**Chief Resident, Department of Oral and Maxillofacial Surgery, extractions. areas of the mandible. The implant
Los Angeles County/University of Southern California Medical
Center, Los Angeles, CA. Upon physical examination, the placement surgery was done in the
***Chairman, Department of Oral and Maxillofacial Surgery, Los
Angeles County/University of Southern California Medical Cen- patient showed no other abnormalities. operating room under general anesthe-
ter, Los Angeles, CA.
†Clinical Assistant Professor, Department of Oral and Maxillofa- Intravenous sedation was scheduled sia. An immediate postoperative Pan-
cial Surgery, Los Angeles County/University of Southern Califor-
nia Medical Center, Los Angeles, CA. because the patient was apprehensive orex was taken (Fig. 4). The patient
to having dental extractions. The com- was placed on an antibiotic regimen
ISSN 1056-6163/03/01202-140$3.00
Implant Dentistry bination of Brevital (Eli Lily and Co., (tetracycline 500 mg) and Peridex (Al-
Volume 12 • Number 2
Copyright © 2003 by Lippincott Williams & Wilkins, Inc. Indianapolis, IN), Midazolam (Ben pharma USPD Inc., Baltimore, MD)
DOI: 10.1097/01.ID.0000041223.08656.A7 Venue Labs Inc., Bedford, OH), and rinse for 2 weeks. He was followed-up
140 DENTAL IMPLANTS AND PAPILLON-LEFEVRE SYNDROME
Center outpatient dental clinic for den- like ankylosed teeth.9 It has been
tal extractions because of his oral con- stated that because of this characteris-
dition of Papillon-Lefevre Syndrome. tic they are contraindicated in growing
The fabrication of upper and lower individuals because they may result in
complete dentures restored form and the infrapositioning of implants.10 It is
function for this patient. However, be- speculated that as bone growth occurs,
cause of the inadequate and continual the implant fixture would remain at its
loss of bone support in the mandible, original position, resulting in a new
the stability of the lower denture was but inferior position of the implant
compromised. The treatment option of relative to the alveolar crest, thus
Fig. 1. Hyperkeratosis in both palms. an implant-retained overdenture was termed infrapositioning. According to
then deemed appropriate. This study
Behrendts,11 the apposition of alveolar
has shown that the successful outcome
bone and the increase of alveolar
weekly for the first month and then at of implant treatment in patients with
Papillon-Lefevre Syndrome is achiev- height are completed during the early
regular intervals by the oral surgery teen years. Whether infrapositioning
department. able. Not only did the two titanium
implants successfully osseointegrate, will be of significance as the patient
The postsurgical recovery period ages remains to be seen. Our success-
was unremarkable with no chief com- but the supporting alveolar bone was
also preserved. These implants helped ful implant placement in this 13-year-
plaint or complications. The lower
increase the retention and stability of old patient certainly allows us to fol-
complete denture was first relined pe-
the mandibular denture through their low his growth, the prognosis, and the
riodically with Viscogel (Dentsply,
Munich, Germany) to avoid immedi- attachments and by preserving the un- positions of these integrated implants
ate loading of the implant fixtures dur- derlying bone structure. These results in the near future. Nonetheless, the
ing osseointegration. The implants concurred with the findings of Ullbro treatment approach in this case has
were subsequently uncovered 4 et al.8 shown initial success and has en-
months after their placements into the Dental implants function much hanced the therapeutic options in pa-
mandible. They were clinically and ra-
diographically determined to be os-
seointegrated successfully. The crite-
ria for success were the absence of
mobility, the absence of radiographic
gap in the bone-implant interface, and
the absence of pain or infection at the
periimplant area. The lower overden-
ture was then modified with the
O–ring-type of attachments and stabi-
lized through these implants. A
follow-up panoramic radiograph was
taken at week 23 showing preservation
of supporting bone (Figs. 5). One year
follow-up appointment showed con-
tinued success of the treatment with no
further bone loss. Periapical radio-
graphs and clinical photographs of the
implants were taken (Figs. 6 and 7).
DISCUSSION
Papillon-Lefevre Syndrome is a
devastating disease process character-
ized by rapid destruction of the dental
alveolar complex. It starts affecting
the individual during childhood and
poses both physical and psychological
challenges to these patients. Rapid
bone loss and exfoliation of teeth often
lead to early edentulism and the need
to wear removable dental prostheses. Fig. 2. Generalized advanced bone loss with hopeless dentition at the initial visit.
In this case study, the patient was Fig. 3. Panorex of oral condition before implant placement.
referred to the LAC/USC Medical
IMPLANT DENTISTRY / VOLUME 12, NUMBER 2 2003 141Fig. 6. Periapical radiograph of the dental
implants at 1-year follow-up.
Fig. 7. Clinical picture of osseointegrated im-
plants at 1-year follow-up.
without extraction. J Periodontol. 1989;60:
512–515.
Fig. 4. Immediate postimplant placement Panorex. 8. Ullbro C, Crossner CG, Lundgren T,
Fig. 5. Panorex at week 23 showing preservation of the alveolar bone. et al. Osseointegrated implants in a patient
with Papillon-Lefèvre syndrome: A 4 1/2-
year follow-up. J Clin Periodontol. 2000;
tients with Papillon-Lefevre perkeratosis and premature periodontal
27:951–954.
Syndrome. destruction of the teeth. J Pediatr. 1964;
9. Oesterle LJ, Cronin RJ, Ranly DM.
65:895–898. Maxillary implants and the growing patient.
CONCLUSION 2. Van Dyke TE, Taubman MA, Eber- Int J Oral Maxillofacial Implants. 1993;8:
sole JL, et al. The Papillon-Lefevre 377–387.
This report has shown successful Syndrome: Neutrophil dysfunction with se- 10. Ödman J, Gröndahl K, Lekholm U,
1-year follow-up of implant osseointe- vere periodontal disease. Clin Immunol Im- et al. The effect of osseointegrated im-
gration and alveolar bone preservation munopathol. 1984;31:419–429. plants on the dento-alveolar development.
in a Papillon-Lefevre Syndrome pa- 3. Martinez Lalis RR, Lopez Otero R, A clinical and radiographic study in grow-
tient wearing a functional implant- Carranza FA Jr. A case of Papillon-Lefevre ing pigs. Eur J Orthod. 1991;13:279–286.
Syndrome. Periodontics. 1965;3:292– 11. Behrendts RG. Growth in the age-
retained overdenture. This result pro- 295.
vides a viable option for restoring ing craniofacial skeleton. In: Craniofacial
4. Newman MG, Angel I, Karge H, et al. Growth Series, Monograph No. 17. Ann
edentulism in young individuals suf- Bacterial studies of the Papillon-Lefevre Arbor, Michigan: Center for Human
fering from this disorder. Syndrome. J Dent Res. 1977;56:545–547. Growth and Development, University of
5. Rateitschak-Pluss EM, Schroeder Michigan; 1985.
Disclosure HE. History of periodontitis in a child with
The authors claim to have no fi- Papillon-Lefevre syndrome. A case report.
nancial interest in any company or any J Periodontol. 1984;55:35–46. Reprint requests and correspondence to:
of the products mentioned in this 6. Shapira J, Eidelman E, Fuks A, et al. Ian Woo, MSc, DDS
Treatment of Papillon-Lefevre syndrome Department of Dentistry
article.
with chemotherapy. Report of cases. Spec Los Angeles County/USCMS
REFERENCES Care Dentist. 1985;5:71–74. 1175 Cummings Street, OPD 1P51
7. Machtei EE, Zubrey Y, Ben Yehuda Los Angeles, CA 90033
1. Gorlin RJ, Sedano HD, Anderson A, et al. Proximal bone loss adjacent to Fax: (323) 226 –5241
VE. The syndrome of palmar-plantar hy- periodontally “hopeless” teeth with and E-mail: ianwoo@doctor.com
142 DENTAL IMPLANTS AND PAPILLON-LEFEVRE SYNDROMEAbstract Translations [German, Spanish, Portuguese, Japanese]
AUTOR(EN): Ian Woo, MSc, DDS*, Daniel P. ZUSSAMENFASSUNG: Innerhalb des vorliegenden Artikels wird der Fall eines 13 Jahre
Brunner, DDS, MD**, Dennis-Duke R. Ya- alten, am Papillon-Lefèvre-Syndrom erkrankten Patienten geschildert, der einer Implan-
mashita, DDS***, Bach T. Le, DDS, MD****. tierungsbehandlung unterzogen wurde. Nachdem der Patient über den mangelnden Sitz
*Assistenzarzt, Abteilung für Gesichts- und Kief- seines bisherigen Zahnersatzes geklagt hatte, wurden im Unterkiefer zwei Titanimplantate
erchirurgie, Bezirk Los Angeles / Universität des zur Befestigung einer implantatfixierten Prothese eingepflanzt. Bei der Nachuntersu-
Medizinischen Fachzentrums von Südkali- chung, die ein Jahr nach erfolgter Implantierung und dem ständigen Tragen der funktion-
fornien, Los Angeles, Kalifornien. **Leitender alen Zahnprothese vorgenommen wurde, wurden Röntgenaufnahmen gemacht. Diese
Assistenzarzt, Abteilung für Gesichts- und Kief- erwiesen eine erfolgreiche Integration der Implantate in das umliegende Knochengewebe
erchirurgie, Bezirk Los Angeles / Universität des sowie die vollständige Erhaltung des vorhandenen Alveolarknochens.
Medizinischen Fachzentrums von Südkali-
fornien, Los Angeles, Kalifornien. ***Vorsit- SCHLÜSSELWÖRTER: Erhaltung des Alveolarknochens, implantatfixierte Deckproth-
zender, Abteilung für Gesichts- und Kieferchiru- ese, frühzeitiger Zahnverlust
rgie, Bezirk Los Angeles / Universität des
Medizinischen Fachzentrums von Südkali-
fornien, Los Angeles, Kalifornien. ****Stellver-
tretender Professor für klinische Medizin, Abtei-
lung für Gesichts- und Kieferchirurgie, Bezirk
Los Angeles / Universität des Medizinischen
Fachzentrums von Südkalifornien, Los Angeles,
Kalifornien. Schriftverkehr: Dr. Ian Woo, Abtei-
lung für Zahnheilkunde (Department of Dentist-
ry), Bezirk Los Angeles (Los Angeles County) /
USCMS, 1175 Cummings Street, OPD 1P51,
Los Angeles, California 90033. Fax: (323) 226 –
5241; eMail: Ianwoo@doctor.com
AUTORES: Ian Woo, MSc, DDS,* Daniel P. ABSTRACTO: Se informa el caso de un informe dental colocado en un paciente de 13
Brunner, DDS, MD,** Dennis-Duke R. Ya- años diagnosticado con el síndrome de Papillon-Lefevre. Se colocaron dos implantes
mashita, DDS,*** Bach T. Lee, DDS, dentales de titanio en la mandíbula de una dentadura retenida por implantes después de
MD****. *Residente, Departamento de Cir- que el paciente se quejó de tener una prótesis inestable. Las radiografías de seguimiento
ugía Oral y Maxilofacial, Condado de Los demuestran una exitosa oseointegración y preservación del hueso alveolar un año después
Angeles/Centro Médico de la Universidad del de la colocación del implante y el uso continuo de una prótesis dental funcional.
Sur de California, Los Angeles, CA. **Jefe de
Residentes, Departamento de Cirugía Oral y PALABRAS CLAVES: preservación del hueso alveolar, sobredentadura retenida con
Maxilofacial, Condado de Los Angeles/Centro implantes, edentulismo temprano
Médico de la Universidad del Sur de Califor-
nia, Los Angeles, CA. ***Jefe, Departamento
de Cirugía Oral y Maxilofacial, Condado de
Los Angeles/Centro Médico de la Universidad
del Sur de California, Los Angeles, CA.
****Profesor Asistente Clínico, Departa-
mento de Cirugía Oral y Maxilofacial, Con-
dado de Los Angeles/Centro Médico de la
Universidad del Sur de California, Los Ange-
les, CA. Correspondencia a: Dr. Ian Woo,
Department of Dentistry, Los Angeles County/
USCMS, 1175 Cummings Street, OPD 1P51,
Los Angeles, CA 90033. Fax: (323) 226-5241;
Correo electrónico: ianwoo@doctor.com
IMPLANT DENTISTRY / VOLUME 12, NUMBER 2 2003 143AUTOR(ES): Ian Woo, MSc DDS*, Daniel P. SINOPSE: registrou-se um relatório de caso de uma colocação de um implante odon- Brunner, DDS, MD**, Dennis-Duke R. Ya- tológico em um paciente de 13 anos de idade diagnosticado com a Síndrome de Papillon- mashita, DDS***, Bach T. Le, DDS, Lefevre. Dois implantes odontológicos de titânio foram colocados na mandíbula para uma MD****. *Residente, Departamento de Ciru- dentadura fixada por implante após reclamação do paciente a respeito de instabilidade da rgia Oral e Maxilofacial, Comarca de Los prótese. Um ano após a colocação do implante e o desgaste contínuo de uma prótese Angeles/Centro Médico da Universidade do odontológica funcional, as radiografias de acompanhamento exibiram osseointegração e Sul da Califórnia, Los Angeles, CA . preservação do osso alveolar bem sucedidas. **Residente-chefe, Departamento de Cirurgia Oral e Maxilofacial, Comarca de Los Angeles/ PALAVRAS-CHAVES: preservação óssea alveolar, sobredentadura fixada por implante, Centro Médico da Universidade do Sul da edentulismo prematuro Califórnia, Los Angeles, CA. ***Presidente, Departamento de Cirurgia Oral e Maxilofa- cial, Comarca de Los Angeles/Centro Médico da Universidade do Sul da Califórnia, Los Angeles, CA. ****Professor Clínico Adjunto, Departamento de Cirurgia Oral e Maxilofa- cial, Comarca de Los Angeles/Centro Médico da Universidade do Sul da Califórnia, Los Angeles, CA. Correspondências devem ser en- viadas a: Dr. Ian Woo, Departamento de Od- ontologia, Comarca de Los Angeles/USCMS, 1175 Cummings Street, OPD 1P51, Los Ange- les, Califórnia 90033.Fax: (323) 226-5241; E-mail: ianwoo@doctor.com 144 DENTAL IMPLANTS AND PAPILLON-LEFEVRE SYNDROME
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