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Journal of Prosthetic Dentistry

Implant and prosthetic rehabilitation of a patient with mucous membrane pemphigoid

  • Thomas Fuschetto
    Correspondence
    Corresponding author: Dr Thomas Fuschetto, Department of Prosthodontics and Digital Technology, School of Dental Medicine, Stony Brook University, 1105 Westchester Hall, Stony Brook, NY 11794-8712
    Affiliations
    Prosthodontic Resident, Department of Prosthodontics and Digital Technology, School of Dental Medicine, Stony Brook University, Stony Brook, NY

    Private practice, Westchester County, NY
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  • Kenneth S. Kurtz
    Affiliations
    Clinical Professor of Prosthodontics and Director, Division of Maxillofacial Prosthetics, Department of Prosthodontics and Digital Technology, School of Dental Medicine, Stony Brook University, Stony Brook, NY
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  • Rafael A. Delgado-Ruiz
    Affiliations
    Associate Professor, Department of Prosthodontics and Digital Technology, School of Dental Medicine, Stony Brook University, Stony Brook, NY
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Published:December 31, 2020DOI:https://doi.org/10.1016/j.prosdent.2020.09.020

      Abstract

      Mucous membrane pemphigoid (MMP) is an autoimmune condition characterized by subepithelial separation and deposition of autoantibodies and complement along the basement membrane zone. The disease results in the development of vesiculobullous lesions of the mucous membranes and skin. This report discusses the surgical treatment and management and the prosthetic implant rehabilitation of a patient with mucous membrane pemphigoid. The rationale for this treatment was to fabricate a prosthesis that was stable and did not rub against the gingival tissues and that was easily cleaned. The overdenture attachment system used provides more stability than other attachment systems while allowing the prosthesis and abutments to be easily cleaned.
      Mucous membrane pemphigoid (MMP) is a chronic, autoimmune, vesiculobullous disease characterized by erosive lesions in the mucous membranes and occasionally the skin.
      • Xu H.-H.
      • Werth V.P.
      • Parisi E.
      • Sollecito T.P.
      Mucous membrane pemphigoid.
      • Megarbane J.-M.
      • Freiha C.
      • Mokbel N.
      Oral rehabilitation of a severe periodontally involved patient with mucous membrane pemphigoid: a 15-year follow-up case report.
      • Arduino P.G.
      • Lopetuso E.
      • Carcieri P.
      • Giacometti S.
      • Carbone M.
      • Tanteri C.
      • et al.
      Professional oral hygiene treatment and detailed oral hygiene instructions in patients affected by mucous membrane pemphigoid with specific gingival localization: a pilot study in 12 patients.
      • Chan M.H.
      • Wolf J.C.
      Biopsy techniques and diagnoses & treatment of mucocutaneous lesions.
      It is caused by deposits of autoantibodies (IgG, IgA) and complement (C3) along the basement membrane zone.
      • Xu H.-H.
      • Werth V.P.
      • Parisi E.
      • Sollecito T.P.
      Mucous membrane pemphigoid.
      ,
      • Hayes E.
      • Challacombe S.J.
      An overview of vesicobullous conditions affecting the oral mucosa.
      ,
      • Tricamo M.B.
      • Rees T.D.
      • Hallmon W.W.
      • Wright J.M.
      • Cueva M.A.
      • Plemons J.M.
      Periodontal status in patients with gingival mucous membrane pemphigoid.
      The severity of MMP varies from mild expression involving only the oral mucosa to severe instances involving esophageal, genital, and ocular mucosas.
      • Xu H.-H.
      • Werth V.P.
      • Parisi E.
      • Sollecito T.P.
      Mucous membrane pemphigoid.
      ,
      • Chan M.H.
      • Wolf J.C.
      Biopsy techniques and diagnoses & treatment of mucocutaneous lesions.
      MMP is predominant in women and is commonly observed in the fifth decade of life.
      • Xu H.-H.
      • Werth V.P.
      • Parisi E.
      • Sollecito T.P.
      Mucous membrane pemphigoid.
      ,
      • Chan M.H.
      • Wolf J.C.
      Biopsy techniques and diagnoses & treatment of mucocutaneous lesions.
      ,
      • Hayes E.
      • Challacombe S.J.
      An overview of vesicobullous conditions affecting the oral mucosa.
      The reported prevalence is approximately 1:40 000, with the incidence being 1.16 to 2.00 per million.
      • Ong H.S.
      • Setterfield J.F.
      • Minassian D.C.
      • Dart J.K.
      Mucous membrane pemphigoid with ocular involvement: the clinical phenotype and its relationship to direct immunofluorescence findings.
      Diagnosis is based on clinical, histologic, and direct immunofluorescence methods.
      • Xu H.-H.
      • Werth V.P.
      • Parisi E.
      • Sollecito T.P.
      Mucous membrane pemphigoid.
      ,
      • Megarbane J.-M.
      • Freiha C.
      • Mokbel N.
      Oral rehabilitation of a severe periodontally involved patient with mucous membrane pemphigoid: a 15-year follow-up case report.
      ,
      • Chan M.H.
      • Wolf J.C.
      Biopsy techniques and diagnoses & treatment of mucocutaneous lesions.
      Treatments for MMP include topical and systemic corticosteroids, immunosuppressants, tetracyclines plus niacinamide, and laser light therapy in combination with local corticosteroids.
      • Oliveira P.C.
      • Reis J.A.
      • Lacerda J.A.
      • Silveira N.T.
      • Santos J.M.
      • Vitale M.C.
      • et al.
      Laser light may improve the symptoms of oral lesions of cicatricial pemphigoid: a case report.
      • Yilmaz H.G.
      • Kusakci-Seker B.
      • Bayindir H.
      • Tözüm T.F.
      Low-level laser therapy in the treatment of mucous membrane pemphigoid: a promising procedure.
      • Maglie R.
      • Hertl M.
      Pharmacological advances in pemphigoid.
      • Schellinck A.E.
      • Rees T.D.
      • Plemons J.M.
      • Kessler H.P.
      • Rivera-Hidalgo F.
      • Solomon E.S.
      A comparison of the periodontal status in patients with mucous membrane pemphigoid: a 5-year follow-up.
      • Radulescu M.
      The pharmacologic management of common lesions of the oral cavity.
      Doxycycline has been prescribed combined with or instead of corticosteroids in patients with minimal disease activity or where comorbidities limit the use of corticosteroids because of its antimicrobial and anti-inflammatory properties.
      • Schellinck A.E.
      • Rees T.D.
      • Plemons J.M.
      • Kessler H.P.
      • Rivera-Hidalgo F.
      • Solomon E.S.
      A comparison of the periodontal status in patients with mucous membrane pemphigoid: a 5-year follow-up.
      Few reports have described the use of dental implants or complete-mouth rehabilitation in patients with MMP. Megarbane et al
      • Megarbane J.-M.
      • Freiha C.
      • Mokbel N.
      Oral rehabilitation of a severe periodontally involved patient with mucous membrane pemphigoid: a 15-year follow-up case report.
      described the 15-year follow-up of a patient with MMP and concluded that fixed implant rehabilitation is appropriate for patients with MMP. A recent systematic review
      • Strietzel F.
      • Schmidt-Westhausen A.
      • Neumann K.
      • Reichart P.
      • Jackowski J.
      Implants in patients with oral manifestations of autoimmune or muco-cutaneous diseases - a systematic review.
      concluded that guidelines regarding implant-supported or implant-retained restorations for patients with oral mucocutaneous disorders are lacking, that implant survival rates in affected patients are comparable with those of healthy patients, that these patients seem to benefit from implant-retained or implant-supported fixed prostheses, and that implant-prosthetic treatment guidelines regarding healthy patients should be followed.
      The prosthetic dental literature is sparse regarding the clinical management of patients with MMP, particularly those with edentulism. This clinical report describes the treatment and clinical rationale of a patient with MMP that included tooth extractions, an immediate complete denture, dental implant placement, and an implant-retained mandibular overdenture.

      Clinical report

      A 68-year-old woman presented to the Stony Brook University School of Dental Medicine Postgraduate Prosthodontics clinic with the chief complaint that she wanted to smile comfortably and without pain. An oral pathologist had diagnosed MMP of the mandibular mucosa, right eye, and genitals. Her oral ulcerations had been managed with doxycycline 20 mg 2 times a day. She was edentulous in the maxilla and partially edentulous in the mandible with nonrestorable mandibular anterior teeth and some premolar roots present. Gingivitis and multiple mandibular ulcerations on the alveolar ridge were observed (Fig. 1). She had an ill-fitting maxillary complete denture and a mandibular interim removable partial denture. She demonstrated poor oral hygiene and inflamed gingiva and reported difficulty in brushing her teeth because of the pain, which is commonly seen in patients with MMP.
      • Tricamo M.B.
      • Rees T.D.
      • Hallmon W.W.
      • Wright J.M.
      • Cueva M.A.
      • Plemons J.M.
      Periodontal status in patients with gingival mucous membrane pemphigoid.
      ,
      • Schellinck A.E.
      • Rees T.D.
      • Plemons J.M.
      • Kessler H.P.
      • Rivera-Hidalgo F.
      • Solomon E.S.
      A comparison of the periodontal status in patients with mucous membrane pemphigoid: a 5-year follow-up.
      ,
      • Arduino P.
      • Farci V.
      • D’Aiuto F.
      • Carcieri P.
      • Carbone M.
      • Tanteri C.
      • et al.
      Periodontal status in oral mucous membrane pemphigoid: initial results of a case-control study.
      Her treatment goals aligned with removable prostheses because of hygiene concerns. The oral pathologist recommended delaying surgery until the oral ulcerations were at a minimum and to exercise caution when manipulating the mandibular gingival tissues. Oral or systemic corticosteroids were not used before the surgical procedures because they had not been effective during the initial treatment of her condition.
      Figure thumbnail gr1
      Figure 1Initial photograph depicting extent of mucous membrane pemphigoid ulcerations and unfavorable prognosis of mandibular teeth.
      Under local anesthesia, with slow administration to avoid bullae formation, a full-thickness mucoperiosteal flap was reflected. The teeth were removed conventionally, as bone is not affected by MMP, and alveoloplasty of the interproximal bone was completed to create smooth bony contours. It was decided not to graft because of the presence of intact sockets. The flap was closed by using polyglactin 910 sutures (Coated VICRYL; Ethicon Inc), and care was taken not to tear the tissues. However, desquamation did occur during surgical manipulation (Fig. 2A). A mandibular immediate complete denture was delivered. Postoperatively, she was prescribed amoxicillin 500 mg, 3 times a day for 7 days, to prevent infection in the broad area of desquamation. She was evaluated at 24 hours and 2 weeks (Fig. 2B, 2C) for healing, discomfort management, suture removal, and completion of a soft reline (COE-SOFT; GC America Inc) of the immediate denture.
      Figure thumbnail gr2
      Figure 2A, Day of extractions. Friability of gingival tissues seen from subsequent desquamation of epithelium. B, Twenty-four hours after extractions. Large fibrin clot observed covering surgical site. Pain prevented wear of immediate complete denture at this stage. C, Two weeks after extractions and after removal of sutures. Healing appeared normal given extensive desquamation. D, Six months after extractions. Healed gingival tissues without evidence of ulcerations before implant placement.
      After 3 months, a cone beam computed tomography (CBCT) scan was made by using the dual scan technique,
      • Vercruyssen M.
      • Fortin T.
      • Widmann G.
      • Jacobs R.
      • Quiryen M.
      Different techniques of static/dynamic guided implant surgery: modalities and indications.
      with the goal of executing flapless guided surgery to minimize damage to the desquamation that occurred at the time of the extractions. Implant planning was completed, and a surgical template was designed and fabricated (Implant Studio; 3Shape A/S). At the preoperative visit, no ulcerations were present on the mandibular mucosa, and she reported not having had ulcerations for the past month (Fig. 2D). The oral pathologist advised that she might be in a state of remission and to continue to proceed cautiously. She was prescribed 0.12% chlorhexidine rinse, which was started 3 days before surgery, and a 7-day regimen of amoxicillin 500 mg, which was started 1 day before surgery. Under local anesthesia, 4 implants (Tapered Internal Plus; Biohorizons Implant Systems Inc) were placed fully guided by using a flapless approach (Fig. 3). Cover screws were placed, and the osteotomy sites were sutured closed using polyglactin 910 sutures. She reported minimal postoperative discomfort. Stage II surgery was completed after 3 months (Fig. 4). Because of the growth of bone over the cover screws, small full-thickness mucoperiosteal flaps were elevated, bone was removed and contoured, healing abutments (Healing Abutment; Biohorizons Implant Systems Inc) were placed, and the flaps were sutured with polyglactin 910 sutures. Desquamation was not a concern because her MMP was in remission.
      Figure thumbnail gr3
      Figure 3Implants placed flapless, uneventfully, via guided surgery. Ecchymosis evident where anesthetic delivered.
      Figure thumbnail gr4
      Figure 4Two mucoperiosteal flaps elevated for stage II surgery and healing abutments seated uneventfully.
      After 1 month of healing, the definitive impressions were obtained with border molded custom trays at the implant level for the mandible with polyvinyl siloxane impression material (Aquasil Ultra; Dentsply Sirona). A stone verification device was fabricated for the mandibular definitive cast by using Type IV dental stone (Prima Rock; Whip Mix Corp) and the appropriate nonengaging interim abutments (Titanium Temporary Abutment, Nonhexed Abutment; Biohorizons Implant Systems Inc).
      • Alhashim A.
      • Flinton R.J.
      Dental gypsum verification jig to verify implant positions: a clinical report.
      Maxillomandibular relations were recorded, and the mandibular definitive cast was verified after the stone device passed the 1-screw test
      • Alhashim A.
      • Flinton R.J.
      Dental gypsum verification jig to verify implant positions: a clinical report.
      and did not fracture after the screws were tightened to 30 Ncm. Artificial tooth arrangements were completed with a balanced lingualized occlusion. The tooth arrangements and casts were scanned and digitized, and an integrated bar was fabricated with computer-aided design and computer-aided manufacturing (CAD-CAM).
      The proprietary components were evaluated, and the housings were attached intraorally to the bar by using resin cement (RelyX Unicem II Cement; 3M) (Fig. 5). The bar was picked up by using an extended custom tray and polyvinyl siloxane impression material to capture the supporting soft tissue accurately. A new definitive cast was fabricated with the appropriate analog components, and the artificial tooth arrangement was transferred to the bar. After the definitive clinical evaluation, the prostheses were processed (Fig. 6). The bar components were tightened to their manufacturer-recommended torque values, and the prostheses were delivered with the least retentive components in place.
      Figure thumbnail gr5
      Figure 5A, Custom and stock integrated bar components seated. B, Bar cemented to proprietary housings and path of insertion verified.
      Figure thumbnail gr6
      Figure 6Intaglio of prosthesis showing nylon retentive components and bar encased in acrylic resin.
      The patient was seen for follow-ups periodically after delivery of the definitive prostheses. At the follow-up appointments, she reported no significant ulcerations related to her MMP. The gingival tissues around the abutments were within normal limits and without signs of inflammation. She was happy with the prosthetic result (Fig. 7), demonstrated good oral hygiene, and continued doxycycline 20 mg 2×/d as prescribed by her oral pathologist. At the 8-month follow-up, she reported being able to eat without pain or discomfort, the gingiva had adapted well to the abutments, and radiographic evaluation showed stable bone levels.
      Figure thumbnail gr7
      Figure 7Definitive prosthesis delivered and esthetic outcome.

      Discussion

      This clinical report describes the prosthetic rehabilitation of a patient with MMP. Interestingly, her MMP went into remission approximately 6 months after extraction of her teeth. Although plaque removal alone cannot induce MMP regression,
      • Arduino P.G.
      • Lopetuso E.
      • Carcieri P.
      • Giacometti S.
      • Carbone M.
      • Tanteri C.
      • et al.
      Professional oral hygiene treatment and detailed oral hygiene instructions in patients affected by mucous membrane pemphigoid with specific gingival localization: a pilot study in 12 patients.
      the doxycycline treatment, extracting the teeth in poor condition, thus removing the plaque irritant, may have precipitated the MMP remission.
      Other treatment options included a fixed implant-supported prosthesis and an implant-supported bar overdenture (based on overall rigidity, full implant support, and minimal tissue trauma). The disadvantages of these prostheses include difficulty with hygiene, which may have led to the increased plaque retention and gingival inflammation often experienced by patients with MMP.
      • Payne A.G.
      • Solomons Y.F.
      Mandibular implant-supported overdentures: a prospective evaluation of the burden of prosthodontic maintenance with 3 different attachment systems.
      The prosthesis type chosen for the mandible was the integrated bar, an implant-and tissue-supported bar with increased rigidity that is processed into the definitive prosthesis and supported by a combination of stock and custom implant components, and nylon inserts. Based on the prosthodontic team's experience, the benefits of this prosthetic solution include a more rigid prosthesis than resilient component options, the ability to parallel all of the retentive components with the custom angulated abutments, and ease of oral hygiene as experienced by this patient with minimal plaque accumulation. A disadvantage of this type of prosthesis is that it is implant- and tissue-supported. However, because of its increased rigidity, tissue trauma should be low. Other prosthetic designs that may be suitable for these patients include telescopic copings such as the ATLANTIS Conus concept (Dentsply Sirona); however, limitations include implant compatibility.
      Surgical management and treatment planning was essential for patient comfort. Although it was not specifically examined, Toscano et al
      • Toscano N.J.
      • Holtzclaw D.J.
      • Shumaker N.D.
      • Stokes S.M.
      • Meehan S.C.
      • Rees T.D.
      Surgical considerations and management of patients with mucocutaneous disorders.
      noted that when treating patients with vesiculobullous diseases, the medication dosage and frequency should be increased before professional oral hygiene or surgical intervention. They also recommended the prescription of a topical corticosteroid for several days before the planned surgical procedure or manipulation of the gingiva. Megarbane et al
      • Megarbane J.-M.
      • Freiha C.
      • Mokbel N.
      Oral rehabilitation of a severe periodontally involved patient with mucous membrane pemphigoid: a 15-year follow-up case report.
      managed their patient with MMP with systemic corticosteroids 3 days before and 2 days after surgical intervention and administered a course of antibiotics, analgesics, and 0.12% chlorhexidine rinse for 1 week postoperatively.
      To date, this treatment has been successful, although further follow-up will be needed to evaluate biologic or prosthetic complications. The literature lacks guidelines on the dental rehabilitation of patients with MMP. Further research and reports are needed.

      Summary

      This clinical report describes the prosthetic rehabilitation of a patient with mucous membrane pemphigoid with tooth extractions, an immediate mandibular complete denture, flapless guided implant surgery, a maxillary complete denture, and an implant-retained mandibular overdenture with an integrated bar. The selection of this definitive prosthetic design resulted in a stable implant- and tissue-supported mandibular prosthesis that is comfortable and easily cleaned by the patient. The treatment outcome demonstrates that this prosthesis design may be an option for patients with MMP.

      Acknowledgments

      The authors thank Dr John Fantasia (Oral Pathologist–Long Island Jewish Medical Center/Northwell Health, New Hyde Park, NY) for his expertise and recommendations during the treatment of this patient.

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