1
|
Kazacheuskaya L, Arora K. Esophageal melanosis: Two case reports and review of literature. World J Gastroenterol 2024; 30:4557-4565. [PMID: 39563752 PMCID: PMC11572630 DOI: 10.3748/wjg.v30.i42.4557] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/26/2024] [Revised: 09/29/2024] [Accepted: 10/14/2024] [Indexed: 10/31/2024] Open
Abstract
BACKGROUND Esophageal melanosis (EM) is a rare condition characterized by melanin pigmentation in the esophageal mucosa. It is not well understood and has been documented in less than 100 cases worldwide. CASE SUMMARY We report two cases of African American patients who complained of significant weight loss (over 20 pounds in approximately six months) and abdominal pain during their first visit. The first case involves a 54-year female with a history of hepatic steatosis and polysubstance abuse, who also experiences nausea and vomiting. The second case is a 59-year-old male with hypertension and gastroesophageal reflux disease (GERD), who was diagnosed with esophageal squamous cell carcinoma. Both cases show benign melanocytes in the basal layer on the esophagus biopsy and are diagnosed as EM. CONCLUSION It is important to note that EM has been associated with malignancies such as carcinoma and melanoma. Therefore, accurate diagnosis and appropriate management are crucial. Patients with EM, especially those with concurrent risk factors (e.g., GERD, smoking), should be carefully monitored for any signs of malignancy.
Collapse
Affiliation(s)
- Liubou Kazacheuskaya
- Department of Pathology, Louisiana State University Health, Shreveport, LA 71103, United States
| | - Kshitij Arora
- Department of Pathology, Louisiana State University Health, Shreveport, LA 71103, United States
| |
Collapse
|
2
|
Toedtling V, Crawford FC. Clinical and histopathological differential diagnosis of Laugier-Hunziker syndrome: An extremely rare case with unusual extensive oral hyperpigmentation. Clin Case Rep 2021; 9:309-313. [PMID: 33489178 PMCID: PMC7813034 DOI: 10.1002/ccr3.3522] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/14/2020] [Revised: 10/16/2020] [Accepted: 10/17/2020] [Indexed: 11/10/2022] Open
Abstract
Laugier-Hunziker syndrome is a rare and benign disorder characterized by hyperpigmentation of the lips and buccal mucosae with associated longitudinal melanonychia of nails. Clinical correlation is needed to rule out other pigmentary disorders.
Collapse
Affiliation(s)
- Verena Toedtling
- Division of DentistrySchool of Medical SciencesFaculty of Biology, Medicine and Health, Oral and Maxillofacial SurgeryThe University of ManchesterManchesterUK
| | | |
Collapse
|
3
|
Sputa‐Grzegrzolka P, Wozniak Z, Akutko K, Pytrus T, Baran W, Calik J, Glatzel‐Plucinska N, Domagala Z, Podhorska‐Okolow M, Stawarski A, Dziegiel P. Laugier‐Hunziker syndrome: a case report of the pediatric patient and review of the literature. Int J Dermatol 2020; 59:1513-1519. [DOI: 10.1111/ijd.15262] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/03/2020] [Revised: 08/12/2020] [Accepted: 09/23/2020] [Indexed: 02/06/2023]
Affiliation(s)
| | | | - Katarzyna Akutko
- 2nd Department and Clinic of Pediatrics, Gastroenterology and Nutrition Wroclaw Medical University Poland
| | - Tomasz Pytrus
- 2nd Department and Clinic of Pediatrics, Gastroenterology and Nutrition Wroclaw Medical University Poland
| | - Wojciech Baran
- Department of Dermatology, Venereology and Allergology Wroclaw Medical University Poland
| | - Jacek Calik
- Department of ChemotherapyLower Silesian Oncology Center Wroclaw Poland
| | - Natalia Glatzel‐Plucinska
- Division of Histology and Embryology Department of Human Morphology and Embryology Wroclaw Medical University Poland
| | - Zygmunt Domagala
- Division of Anatomy Department of Human Morphology and Embryology Wroclaw Medical University Poland
| | | | - Andrzej Stawarski
- 2nd Department and Clinic of Pediatrics, Gastroenterology and Nutrition Wroclaw Medical University Poland
| | - Piotr Dziegiel
- Division of Histology and Embryology Department of Human Morphology and Embryology Wroclaw Medical University Poland
| |
Collapse
|
4
|
Duan N, Zhang YH, Wang WM, Wang X. Mystery behind labial and oral melanotic macules: Clinical, dermoscopic and pathological aspects of Laugier-Hunziker syndrome. World J Clin Cases 2018; 6:322-334. [PMID: 30283795 PMCID: PMC6163135 DOI: 10.12998/wjcc.v6.i10.322] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/21/2018] [Revised: 06/28/2018] [Accepted: 07/24/2018] [Indexed: 02/05/2023] Open
Abstract
Labial and oral melanotic macules are commonly encountered in a broad range of conditions ranging from physiologic pigmentation to a sign of an underlying life-threatening disease. Although Laugier-Hunziker syndrome (LHS) shares some features of labial and oral pigmentation with a variety of conditions, it is a benign and acquired condition, frequently associated with longitudinal melanonychia. Herein, the demographic, clinical, dermoscopic, and pathological aspects of LHS were reviewed comprehensively. The important differential diagnoses of mucocutaneous and nail pigmentation are provided. An accurate diagnosis is crucial to design a reasonable medical strategy, including management options, malignant transformation surveillance, and psychological support. It is important that clinicians conduct long-term follow-up and surveillance due to the potential risks of malignant transformation and local severe complications in some conditions.
Collapse
Affiliation(s)
- Ning Duan
- Department of Oral Medicine, Nanjing Stomatological Hospital, Medical School of Nanjing University, Nanjing 210008, Jiangsu Province, China
| | - Yang-Heng Zhang
- Department of Periodontology, Nanjing Stomatological Hospital, Medical School of Nanjing University, Nanjing 210008, Jiangsu Province, China
| | - Wen-Mei Wang
- Department of Oral Medicine, Nanjing Stomatological Hospital, Medical School of Nanjing University, Nanjing 210008, Jiangsu Province, China
| | - Xiang Wang
- Department of Oral Medicine, Nanjing Stomatological Hospital, Medical School of Nanjing University, Nanjing 210008, Jiangsu Province, China
| |
Collapse
|
5
|
Destek S, Gul VO, Ahioglu S, Erbil Y. A Rare Disease of the Digestive Tract: Esophageal Melanosis. Gastroenterology Res 2016; 9:56-60. [PMID: 27785326 PMCID: PMC5040545 DOI: 10.14740/gr670w] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 07/15/2015] [Indexed: 11/12/2022] Open
Abstract
Esophageal melanosis which is characterized by melanocytic proliferation in the squamous epithelium of the esophagus and melanin accumulatin of esophageal mucosa (EM) is a rare disease of the digestive system. Although esophageal melanosis is considered to be a benign disease, its etiology is not cleared and has been reported to be the precursor lesion of esophageal primary melanomas. In this report, we aimed to note esophageal melanosis in a 55-year-old female case who applied to our clinic with difficulty in swallowing, burning behind the breastbone in the stomach, heartburn, indigestion, and pain in the upper abdomen after endoscopic and pathologic evaluation. Complaints dropped with anti-acid therapy and case was followed by intermittent endoscopic procedures because of precursor melanocytic lesions.
Collapse
Affiliation(s)
| | - Vahit Onur Gul
- General Surgery Department, Edremit Military Hospital, Edremit, Balikesir, Turkey
| | - Serkan Ahioglu
- Biochemistry Department, Edremit Military Hospital, Edremit, Balikesir, Turkey
| | - Yesim Erbil
- General Surgery Department, Medical Faculty, Istanbul University, Capa, Istanbul, Turkey
| |
Collapse
|
6
|
Nikitakis NG, Koumaki D. Laugier-Hunziker syndrome: case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol 2013; 116:e52-8. [PMID: 23562360 DOI: 10.1016/j.oooo.2012.12.012] [Citation(s) in RCA: 21] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/09/2012] [Revised: 12/14/2012] [Accepted: 12/21/2012] [Indexed: 12/22/2022]
Abstract
Laugier-Hunziker syndrome is a rare acquired disorder characterized by diffuse pigmented macules of the oral mucosa. Other mucosal and cutaneous surfaces may be affected, and nail involvement is frequent. A case of Laugier-Hunziker syndrome in a Greek female with multiple buccal and labial mucosal lesions and unusual conjunctival involvement is presented here. In addition, a thorough review of the English language literature of this rather under-recognized syndrome is included and the main differential diagnostic possibilities are discussed.
Collapse
Affiliation(s)
- Nikolaos G Nikitakis
- Department of Oral Pathology and Medicine, Dental School, National and Kapodistrian University of Athens, Athens, Greece.
| | | |
Collapse
|
7
|
Urbina F, Sudy E. Pigmented Fungiform Papillae of the Tongue in Laugier Disease (or Laugier-Hunziker Syndrome). ACTAS DERMO-SIFILIOGRAFICAS 2013. [DOI: 10.1016/j.adengl.2012.10.004] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022] Open
|
8
|
Urbina F, Sudy E. Pigmented fungiform papillae of the tongue in Laugier disease (or Laugier-Hunziker syndrome). ACTAS DERMO-SIFILIOGRAFICAS 2012; 104:173-4. [PMID: 23266057 DOI: 10.1016/j.ad.2012.10.009] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/04/2012] [Revised: 10/04/2012] [Accepted: 10/14/2012] [Indexed: 12/19/2022] Open
|
9
|
Abstract
Laugier–Hunziker syndrome (LHS) is an acquired pigmentary condition affecting lips, oral mucosa and acral area, frequently associated with longitudinal melanonychia. There is neither malignant predisposition nor underlying systemic abnormality associated with LHS. Herein, we present three uncommon cases of LHS with possibly new feature of nail pigmentation, which were diagnosed during the past 2 years. We also review the clinical and histological findings, differential diagnosis, and treatment of the syndrome in published literature.
Collapse
|
10
|
Abstract
Laugier-Hunziker syndrome is a rare acquired disorder characterized by diffuse hyperpigmentation of the oral mucosa and longitudinal melanonychia in adults. They appear as macular lesions less than 5 mm in diameter. Laugier-Hunziker syndrome is considered to be a benign disease with no systemic manifestation or malignant potential. Therefore, it is important to rule out other mucocutaneous pigmentary disorders that do require medical management. Prompt clinical recognition also averts the need for excessive and invasive procedures and treatments. In India, the reported cases of this syndrome are very few. We provide a review of literature on Laugier-Hunziker syndrome with its differential diagnosis.
Collapse
Affiliation(s)
- Ramakant S Nayak
- Department of Oral Pathology and Microbiology, Maratha Mandal's NG Halgekar Institute of Dental Sciences and Research Centre, Belgaum, Karnataka, India
| | | | | |
Collapse
|
11
|
|
12
|
Aliagaoglu C, Yanik ME, Albayrak H, Güvenç SC, Yildirim U. Laugier-Hunziker syndrome: diffuse large hyperpigmentation on atypical localization. J Dermatol 2009; 35:806-7. [PMID: 19239567 DOI: 10.1111/j.1346-8138.2008.00577.x] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/27/2022]
|
13
|
Abstract
Volar melanotic macules are asymptomatic, tan-brown to brownish-black macules found on the palms, fingers, soles, and/or toes of dark-skinned individuals and are infrequently reported in light-skinned individuals. Herein, we report the acquisition of multiple, brownish-black volar digital macules in a 63-year-old white woman and avid gardener that were noted after the onset of pruritic, chronic hand dermatitis over a 10-month period. She was healthy without any medical conditions or medications. No family history of intestinal polyposis or mucosal melanosis existed. At presentation, she had less than 20 brownish-black, 2- to 10-mm irregular macules over the volar aspect of the thumbs and index fingers, left greater than right hand, associated with erythema, scale, and a negative potassium hydroxide examination. In addition, she had melanotic macules of her lips, buccal mucosa, and conjuctiva. No nail or anogenital macules were found. Biopsy demonstrated a region of epidermal hyperplasia, hyperkeratosis, and interface dermatitis associated with melanophages and sparse lymphocytic infiltrate. Melanin staining revealed diffuse pan-epidermal hypermelanization, dermal melanin, and dendritic melanocytes, which expressed HMB-45. After 1-year follow-up and treatment with topical corticosteroid, the volar macules have faded to tan brown. Based on the location, avocation, and coexisting mucosal melanotic macules, an irritant phytodermatitis with postinflammatory hyperpigmentation was diagnosed in the setting of Laugier-Hunziker syndrome-a disorder denoted by macular hyperpigmentation of nail, volar, and/or mucosal surfaces in healthy adults. Volar melanotic macules can also complicate 5-flourouracil therapy, tinea pedis, and other inflammatory disorders; be associated with acral melanomas; or represent a cutaneous manifestation of systemic disease such as Addison disease, Peutz-Jegher syndrome, Leopard syndrome, Carney syndrome, AIDS, and neurofibromatosis.
Collapse
|
14
|
Chang F, Deere H. Esophageal melanocytosis morphologic features and review of the literature. Arch Pathol Lab Med 2006; 130:552-7. [PMID: 16594751 DOI: 10.5858/2006-130-552-emmfar] [Citation(s) in RCA: 51] [Impact Index Per Article: 2.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/06/2022]
Abstract
Endoscopic or macroscopic esophageal melanocytosis is a benign clinicopathologic entity characterized by melanocytic proliferation in esophageal squamous epithelium and melanin deposition in the mucosa. Little is known about the etiology and natural course of this condition, although it has been suggested to be a precursor of primary esophageal melanoma by some authors. Following a search of the bibliographic databases (PubMed and Medline) regarding esophageal melanocytosis and melanosis, thirty-four cases of isolated esophageal melanocytosis (including one unpublished case from us) were found. The histopathologic features of esophageal melanocytosis are reviewed and its differential diagnosis with other pigmented esophageal lesions is discussed.
Collapse
Affiliation(s)
- Fuju Chang
- Department of Histopathology, St Thomas' Hospital, Guy's & St Thomas' NHS Foundation Trust, London, United Kingdom.
| | | |
Collapse
|
15
|
|
16
|
Jones BH, Fleischer DE, De Petris G, Heigh RI, Shiff AD. Esophageal melanocytosis in the setting of Addison's disease. Gastrointest Endosc 2005; 61:485-7. [PMID: 15758932 DOI: 10.1016/s0016-5107(04)02845-7] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/22/2022]
Affiliation(s)
- Bradford H Jones
- Division of Gastroenterology and Hepatology, Mayo Clinic Scottsdale, 13400 E. Shea Boulevard, Scottsdale, AZ 85259, USA
| | | | | | | | | |
Collapse
|
17
|
Ayoub N, Barete S, Bouaziz JD, Le Pelletier F, Frances C. Additional conjunctival and penile pigmentation in Laugier-Hunziker syndrome: a report of two cases. Int J Dermatol 2004; 43:571-4. [PMID: 15304179 DOI: 10.1111/j.1365-4632.2004.02173.x] [Citation(s) in RCA: 19] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/12/2022]
Abstract
BACKGROUND Laugier-Hunziker syndrome is a rare acquired hyperpigmentation of the oral mucosa and lips which is often associated with longitudinal melanonychia. We report two patients exhibiting the classical features of Laugier-Hunziker syndrome with additional and previously unreported conjunctival and penile pigmentation. PATIENTS AND METHODS Two patients presented with a history of progressive acquired macular pigmentation of the oral mucosa, the lips, the ocular conjunctiva and the penis. Longitudinal melanonychia was found in one patient. Microscopic features (basal epithelial melanosis, moderate acanthosis and superficial pigmentary incontinence) and ultrastructural details (increased number of normal-appearing melanosomes inside basal keratinocytes and dermal melanophages) were characteristic of Laugier-Hunziker syndrome. CONCLUSIONS These two observations illustrate the topographical variations of Laugier-Hunziker syndrome and highlight the need to expand the originally described features to include more widespread areas of hyperpigmentation.
Collapse
Affiliation(s)
- Nakhlé Ayoub
- Dermatology Department, Groupe Hospitalier Pitié-Salpêtrière, Paris, France
| | | | | | | | | |
Collapse
|
18
|
|
19
|
Moore RT, Chae KAM, Rhodes AR. Laugier and Hunziker pigmentation: a lentiginous proliferation of melanocytes. J Am Acad Dermatol 2004; 50:S70-4. [PMID: 15097932 DOI: 10.1016/j.jaad.2003.09.016] [Citation(s) in RCA: 25] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/25/2022]
Abstract
Macular pigmentation of Laugier and Hunziker is an acquired disorder involving lips, oral mucosa, acral surfaces, nail apparatus, or a combination of these in the absence of systemic disease, and is reported to show intraepidermal melanosis without melanocytosis. We present a 69-year-old white man with typical features of this disorder, involving lips and oral mucosa, first appearing 4 years before presentation. A pigmented macule on sun-exposed lip vermilion revealed increased numbers of slightly atypical melanocytes in a lentiginous epidermal pattern. A buccal mucosa pigmented macule and adjacent nonpigmented mucosa were studied using hematoxylin and eosin staining, and S-100 and L-3,4 dihydroxyphenylalanine histochemistry. The buccal mucosa macule revealed markedly increased numbers of dendritic, L-3,4 dihydroxyphenylalanine-reactive intraepithelial melanocytes. Further studies are warranted to determine if this pigmentation disorder represents a spectrum of histopathologic change or, in fact, should be renamed mucocutaneous lentiginosis of Laugier and Hunziker.
Collapse
Affiliation(s)
- Roger T Moore
- Department of Dermatology, Rush Medical College, Rush-Presbyterian-St Luke's Medical Center, 1653 W Congress Parkway, 507 Kidston, Chicago, IL 60612, USA
| | | | | |
Collapse
|
20
|
Abstract
We report the unusual occurrence of 3 cases of Laugier-Hunziker syndrome in the same family.
Collapse
Affiliation(s)
- Edouard N Makhoul
- Department of Dermatology, Hôtel-Dieu de France, Achrafieh, PO Box 16-1830, Beirut, Lebanon
| | | | | | | |
Collapse
|
21
|
Abstract
Laugier and Hunziker described a syndrome consisting of asymptomatic benign areas of hyperpigmentation affecting the lips, buccal mucosa and, in 50%, the fingernails. We report a 67-year-old woman with the clinical features of Laugier-Hunziker syndrome in association with vulval pigmentation. Histology, immunohistochemistry and electron microscopy from the various areas of pigmentation on the body confirmed the benign nature of the pigmentation. We review potential causes of oral and genital pigmentation, and suggest an expansion of the original syndrome described by Laugier and Hunziker to include more widespread areas of benign hyperpigmentation, which may associated.
Collapse
Affiliation(s)
- P Lenane
- Regional Centre of Dermatology, Mater Misericordiae Hospital, Dublin, Ireland
| | | | | | | |
Collapse
|
22
|
Abstract
Oral pigmentation may be physiological or pathological in nature. It may represent a localized anomaly of limited significance or the presentation of potentially life-threatening multisystem disease. Evaluation of a patient with oral pigmentation requires a systematic approach with resource to appropriate investigations in certain circumstances. A full history of evolution of the pigmentary changes, as well as inquiring into family history, drug ingestion and systemic symptoms of concurrent disease are clearly important in the assessment. The duration, pattern, hue and distribution of colour changes can provide useful diagnostic clues. Special attention is given to newly appearing lesions, or those that have changed significantly in appearance, and biopsy may be needed to validate the clinical impression. This review should enable the reader to increase their familiarity with the assessment of oral pigmentation, the common causes of oral pigmentary change and the rarer disorders of pigmentation seen in this area. The systemic diseases that may give rise to oral pigmentation are detailed and the early signs of oral melanoma are highlighted, as well as the drugs which may cause pigmentary changes in this area and the different pattern of pigmentation they may induce.
Collapse
Affiliation(s)
- P Lenane
- Regional Centre of Dermatology Mater Misericordiae Hospital, Dublin, Ireland
| | | |
Collapse
|