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CASE REPORT
Year : 2023 | Volume
: 22
| Issue : 2 | Page : 235-238
Keloid masking a parotid tumor: A rare case presentation
Muhammad Lawal Abubakar1, Ibrahim Babatunde Mohammed2, Murtala Abubakar3, Wasiu Olushola Adebayo1, Mohammed Abdullatif Kabir1, Oluwatobi Adebayo Abass1, Abdulrasheed Ibrahim1
1 Department of Surgery, Division of Plastic and Reconstructive Surgery, Ahmadu Bello University Teaching Hospital, Zaria, Nigeria
2 Department of Surgery, Division Otorhinolaryngology, Ahmadu Bello University Teaching Hospital, Zaria, Nigeria
3 Department of Pathology, Ahmadu Bello University Teaching Hospital, Zaria, Nigeria
Correspondence Address:
Muhammad Lawal Abubakar
Department of Surgery, Division of Plastic and Reconstructive Surgery, Ahmadu Bello University Teaching Hospital Shika, Zip Code 810105, Zaria
Nigeria
Source of Support: None, Conflict of Interest: None
CheckDOI: 10.4103/aam.aam_143_21
Abstract
A 37-year-old Nigerian woman presented with an itchy and occasionally painful scar that appeared after the healing of an acne lesion over the left parotid region, 13 years ago. She noticed a progressive increase; however, there was no history of facial weakness. Examination revealed a keloid lesion overlying a firm nontender mass. Ultrasound scan and fine-needle aspiration cytology indicated a benign tumor of the left parotid. Superficial parotidectomy was performed, and the histology revealed a keloid overlying a pleomorphic adenoma. This is an unusual presentation of keloid which developed over a pleomorphic adenoma. Hence, we report the above case for its rarity.
Abstract in French Résumé
Une femme Nigériane de 37 ans s'est présentée avec une cicatrice qui démangeait et parfois était douloureuse, apparue après la cicatrisation d'une lésion acnéique sur le région parotide gauche, il y a 13 ans. Elle a remarqué une augmentation progressive; cependant, il n'y avait aucun antécédent de faiblesse faciale. Examen révélé une lésion chéloïde recouvrant une masse ferme non sensible. L'échographie et la cytoponction à l'aiguille fine ont révélé une tumeur bénigne du côté gauche parotide. Une parotidectomie superficielle a été réalisée et l'histologie a révélé une chéloïde recouvrant un adénome pléomorphe. C'est un peu inhabituel présentation d'une chéloïde qui s'est développée sur un adénome pléomorphe. Par conséquent, nous signalons le cas ci-dessus pour sa rareté.
Mots-clés: Cicatrice chéloïde, adénome pléomorphe, rare
Keywords: Keloid scar, pleomorphic adenoma, rare
Introduction 
Keloids are pathological scars resulting from the excessive growth of dermal collagen due to an abnormal healing response to injury or inflammation in genetically predisposed individuals.[1],[2] Common sites for keloids include the chest, shoulders, earlobes, nape of the neck, upper arms, and cheeks. The common causes are ear piercing, inflammation, folliculitis, trauma, tattooing, burns, and acne.[3] Patients with keloids typically have pruritus, pain, tenderness, secondary infection, and ulceration. However, the most disturbing feature for patients with facial keloids is cosmetic disfigurement.[4]
Pleomorphic adenoma is a common parotid benign mixed tumor of dual origin. It arises from epithelial and myoepithelial elements of the gland.[5] Commonly, it is seen in the third to sixth decades of life with a slight female preponderance and the right parotid gland is more frequently affected.[5] The clinical presentation of parotid tumors is varied and associated clinical latency explains the long interval between onset and consultation found in most developing countries.[6] This delay is usually compounded by a lack of financial means, the fear of surgical intervention, neglect, and primary consultation in unorthodox medicine.[6]
This presentation highlights the peculiar clinical presentation and management of a rare case of keloid which developed over a parotid tumor.
Case Report W. T is a 37-year-old Nigerian woman who presented to our clinic with a scar which was gradually increasing in size over the preceding 13 years. The scar appeared after the healing of an acne lesion over the left parotid region. There was associated itching and it was occasionally painful. There was no history of similar lesions on the other parotid area or any part of the body. There was no facial weakness or systemic symptoms. There was no known family history of keloids. She used local herbs with no improvement in symptoms or regression of scar. Cosmetic concerns necessitated her presentation.
Examination revealed a hyperpigmented, firm nontender scar 8 cm × 6 cm in its widest dimensions with irregular edges [Figure 1]a overlying a firm nontender mass 5 cm × 4 cm in the left parotid region. Multiple acne lesions were also noticed on the face. No left-side facial palsy, submandibular, or cervical lymphadenopathy was noted. Other systemic examinations were essentially normal. A diagnosis of keloid overlying a left parotid tumor was made.
Figure 1: (a) Preoperative lesion. (b) Intraoperative lesion showing parotid tumor (yellow arrow) and keloid (black arrow)Ultrasound-guided fine-needle aspiration cytology (FNAC) indicated a benign tumor of the left parotid gland lobulated measuring 3.3 cm × 1.55 cm with mixed echogenicity. It showed normal color Doppler interrogation. Cytology showed an epithelial cluster of monomorphic cells with scanty cytoplasm and stromal elements composed of myxoid structures.
The patient was informed of the diagnosis and counseled for superficial parotidectomy and extramarginal excision of the keloid. The procedure was performed utilizing general anesthesia.
Operative technique
Preliminary skin markings were made for a modified lazy-S incision from the anterior border of the tragus down to the lobule and then to the angle of the mandible. A local anesthetic (0.5% lidocaine with 1:200,000 epinephrine) was injected into the skin. The flaps were elevated at the level of subcutaneous tissue by sharp dissection and made down to the tumor. The keloid (black arrow) and parotid gland (yellow arrow) were identified [Figure 1]b. The superficial lobe of the parotid gland was removed with gentle separation of the gland from the facial nerve, and an extramarginal excision of the keloid was done. Hemostasis was ensured and the wound closed in two layers: vicryl 3-0 and prolene 4-0 to the skin with a penrose drain.
The patient did well postoperatively, and the drain was removed postoperative day 3. Sutures were removed after 1 week, and the patient was discharged home. Postoperative outcome was satisfactory.
She was lost to follow-up initially. She, however, commenced injection of 40-mg triamcinolone into the edge of the scar. This was repeated at forthrightly intervals for a maximum of six doses [Figure 2].
The histology revealed pigmented epidermis overlying fibrocollagenous dermis containing dense broad bands of eosinophilic collagen (features in keeping with keloid). Lying beneath is an encapsulated biphasic tumor having a lobulated pattern composed of nests, cords, tubules, and strands of epithelial cells within abundant fibromyxoid stroma. The epithelial cells have rounded to oval to elongated vesicular nuclei, small nucleoli, and moderate cytoplasm. Sprinkles of mononuclear inflammatory infiltrates and residual salivary glands were seen (features in keeping with pleomorphic adenoma) [Figure 3].
Figure 3: (a) Shows specimen (keloid above and parotid tumor below). (b) Shows histologic features of keloid. (c) Shows histologic features of pleomorphic adenoma Discussion Keloids are common among darker-skinned races and in populations of Asian origin.[3] An incidence of 4.5% to 16% has been reported in African-Americans and Hispanic populations, and a geographical variation among regions was estimated to range from 0.09% to 16% in the African population.[3] Keloids may develop up to several years after minor injuries and persist for a long period of time, and do not regress spontaneously as seen in our patient. Although our patient presented with occasional itch, pain, and cosmetic concerns, sleep disturbance and depression have been reported.[7]
The face is central to many aspects of social interaction, and the visual perception of the face is influenced by a complex combination of various factors such as appearance and symmetry.[4] Asymmetry of the parotid region affected the patient's overall facial appearance and was associated with significant emotional distress. This is hardly surprising because any deformity of the face has always been considered one of the least desirable handicaps.[4]
Salivary gland tumors are rare.[7] The prevalence of salivary gland tumors in some African countries ranges from 2.8% to 10% of all head-and-neck tumors, and the parotid gland is the most common site in 46.5% to 60% of the cases.[7] The tumors of salivary glands are mostly benign with pleomorphic adenoma as the most common variant. Studies in Nigeria showed an incidence of 49.1% to 61% for parotid tumors, while similar a study in Kumasi Ghana, revealed an incidence of 52%.[8],[9],[10]
Despite the common occurrence of facial keloids and parotid tumors in Africans, to our knowledge, the association of a parotid pleomorphic adenoma with a keloid scar has not been previously reported. However, some authors have reported keloids in rare situations such as malignant melanoma with a keloid scar, tuberculosis verrucosa cutis developing over a keloid, carcinoma en cuirasse presenting as unusual keloid-like nodules, and multiple asymptomatic cutaneous pilar leiomyoma mimicking spontaneous eruptive keloids.[11],[12],[13],[14]
Detecting pleomorphic adenoma beneath a keloid lesion such as the one presented may be challenging, due to its relative rarity. It is possible that the keloid scar initially formed following acne, while the adenoma developed later, and the diagnosis may well have been missed at the time.
Definitive diagnosis of parotid tumor requires parotidectomy. Preoperative diagnosis is founded on fine-needle aspiration cytology and medical imaging. Magnetic resonance imaging is the radiologic examination of choice in case of suspected malignancy.[15],[16]
FNAC can be used to diagnose parotid tumors preoperatively with a high degree of sensitivity and specificity.[15],[17] With small, mobile tumors preoperative histologic diagnosis may not be required, however, in this patient, a histologic diagnosis contributed to patient counseling and informed consent.
Conclusion Presenting this case is relevant for clinical decision-making, in the context of integrating all tools available: Clinical, radiological, and histological examinations, for better case management due to the paucity of related literature. The ability to identify rare presentations of keloid scar coexisting with other pathologies is critical to avoid delay or inappropriate treatment.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
References 
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