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Case Reports
. 2016 Jan 20:5:Doc04.
doi: 10.3205/iprs000083. eCollection 2016.

Cheilitis glandularis: Case report with respect to immunohistochemical findings

Reinhard E Friedrich  1 , Thomas Löning  2
Affiliations
Case Reports

Cheilitis glandularis: Case report with respect to immunohistochemical findings

Reinhard E Friedrich et al. GMS Interdiscip Plast Reconstr Surg DGPW. .

Abstract
in English, German

Cheilitis glandularis (CG) is a rare benign affection of the lip mucosa. The etiology and pathogenesis of CG are unknown. Surgical measures are the leading therapeutic options to treat CG. This case report on a 55-year-old female illustrates the recurrent affection of the lips over several years and local therapy. Furthermore, the study of the resected glands intends to differentiate the lesions with respect to the recently introduced concept of immunoglobulin G4-related diseases.

Die Cheilitis glandularis ist eine seltene, gutartige Affektion der Lippenschleimhaut. Die Ätiopathogenese ist unbekannt. Chirurgische Maßnahmen sind in der Behandlung führend. Der Fallbericht einer 55-jährigen Frau beschreibt das wiederholte Auftreten von typischen Befunden über mehrere Jahre und die lokale Therapie. Darüber hinaus unternimmt die Untersuchung der Exzidate den Versuch, die Läsionen in Zusammenhang mit den neuerlich eingeführten IgG4-assoziierten Erkrankungen zu differenzieren.

Keywords: IgG4-related disease; cheilitis glandularis; lip surgery; minor salivary gland.

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Figures

Figure 1
Figure 1. (A) Detail of patient’s photograph en face showing multiple pigmentary disorders of her light skin. The lips also show pigmentary disorders but the lip contour is not affected. The disease cannot be estimated from the external evaluation. (B) Oral inspection reveals clear mucous drooling after light compression of the upper lip (C) Close-up view of the oral aspect shown in (B) reveals the extended orifice of the labial minor salivary gland. (D) Expulsion of clear saliva after slight pressure on the lower lip from minor salivary glands of the lower lip vermilion during oral investigation for recurrent cheilitis glandularis. (E) After incision of the lower lip, berry-like enlarged minor salivary glands come to light.
Figure 2
Figure 2. (A) Minor salivary gland with interstitial lymphoid infiltrates penetrating atrophic acinar residues; excretory ducts (left) showing moderate ectasia and mucous retention. (B) Closer look at the duct system showing epithelial hyperplasia and apocrine metaplasia (A and B: haematoxylin-eosin staining). (C) Low magnification of the ectatic ductal tree and the irregular atrophia of acinar tissues with some fibrosis and lymphoid infiltrates in between. (D) Closer look at the lymphoid infiltrates penetrating the hyperplastic ductal epithelium (C and D: periodic acid-Schiff reaction).
Figure 3
Figure 3. Immunohistochemical decoration of the interstitial lymphoid infiltrate (A: CD45, B: CD138). IgG4 staining showing a scattered pattern in (C), and very focal clustering in (D).
See this image and copyright information in PMC

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