Identifying Pain Subtypes in Patients With Craniofacial Lesions of Fibrous Dysplasia/McCune-Albright Syndrome.
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| Názov: | Identifying Pain Subtypes in Patients With Craniofacial Lesions of Fibrous Dysplasia/McCune-Albright Syndrome. |
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| Autori: | Berry C; PhD Candidate, Department of Anesthesiology, Critical Care and Pain Medicine, Boston Children's Hospital, Harvard Medical School, Boston, MA., Boyce AM; Principal Investigator, Metabolic Bone Disorders Unit, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD., Kaban LB; Chief, Emeritus, Department of Oral & Maxillofacial Surgery, Harvard School of Dental Medicine, Boston, MA; Chief, Emeritus, Division of Oral and Maxillofacial Surgery, Massachusetts General Hospital, Boston, MA., Peacock ZS; Chief, Department of Oral & Maxillofacial Surgery, Harvard School of Dental Medicine, Boston, MA; Chief, Division of Oral and Maxillofacial Surgery, Massachusetts General Hospital, Boston, MA., Mannstadt M; Chief, Endocrine Unit, Massachusetts General Hospital, Harvard Medical School, Boston, MA., Upadhyay J; Assistant Professor, Department of Anesthesiology, Critical Care and Pain Medicine, Boston Children's Hospital, Harvard Medical School, Boston, MA; Assistant Professor, Department of Psychiatry, McLean Hospital, Harvard Medical School, Belmont, MA. Electronic address: jaymin.upadhyay@childrens.harvard.edu. |
| Korporácia: | Craniofacial Fibrous Dysplasia Research Team |
| Zdroj: | Journal of oral and maxillofacial surgery : official journal of the American Association of Oral and Maxillofacial Surgeons [J Oral Maxillofac Surg] 2025 Mar; Vol. 83 (3), pp. 366-373. Date of Electronic Publication: 2024 Dec 04. |
| Spôsob vydávania: | Journal Article |
| Jazyk: | English |
| Informácie o časopise: | Publisher: W.B. Saunders Co Country of Publication: United States NLM ID: 8206428 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1531-5053 (Electronic) Linking ISSN: 02782391 NLM ISO Abbreviation: J Oral Maxillofac Surg Subsets: MEDLINE |
| Imprint Name(s): | Original Publication: [Philadelphia, PA : W.B. Saunders Co., c1982- |
| Výrazy zo slovníka MeSH: | Fibrous Dysplasia, Polyostotic*/complications , Fibrous Dysplasia, Polyostotic*/psychology , Facial Pain*/etiology , Facial Pain*/classification, Humans ; Male ; Female ; Cross-Sectional Studies ; Prospective Studies ; Adolescent ; Pain Measurement ; Child ; Adult ; Anxiety/etiology ; Depression/etiology ; Surveys and Questionnaires ; Young Adult ; Photophobia/etiology |
| Abstrakt: | Background: Fibrous dysplasia/McCune-Albright syndrome (FD/MAS) is a genetic disorder, marked by bone lesions, often affecting the craniofacial skeleton. Pain is a prevalent yet heterogeneous symptom reported by patients with craniofacial FD. Effective treatments are currently lacking, posing a significant clinical challenge to patient care. Purpose: This preliminary study examined pain profiles in craniofacial FD and aimed to identify subtypes of patients based on pain phenotypes and emotional health. Study Design, Setting, Sample: A prospective, cross-sectional study involving 15 patients with FD/MAS, conducted at Boston Children's Hospital and Massachusetts General Brigham's Hospitals. Predictor/exposure/independent Variable: Headache frequency, craniofacial pain severity, neuropathic pain quality, pain interference, allodynia, photophobia, depression, and anxiety were assessed using clinical questionnaires. Main Outcome Variable(s): The primary outcome variable was the symptom profile derived from standardized clinical questionnaires and analyzed using principal component analysis and K-means clustering. Covariates: Covariates included demographic data, diagnosis, and lesion location(s). Analyses: Principal component analysis and K-means clustering of patient-reported measures of pain and emotional health were performed. Analysis of variance was conducted to determine significant differences among patient subtypes. Statistical significance was set at (P < .05). Results: The study included 15 subjects with FD/MAS, with a mean age of 36.2 (13.9) years, including 1 male. Clustering analysis identified 3 subtypes of patients with distinct symptom profiles. Cluster 1 (n = 2) averaged 70 (28.3) headache days in a 90-day period, pain level of 7.5 (0.7) on a 0-10 scale, and severe anxiety, depression, allodynia, photophobia, and pain interference. Cluster 2 (n = 7) patients reported an average of 5.4 (7.5) headache days, an average pain level of 2.7 (2.6), mild or no anxiety, depression, allodynia, photophobia, and pain interference. Cluster 3 (n = 6) patients displayed a mixed symptom profile with an average of 47.3 (36.4) headache days and a pain level of 5.25 (1.4). Notably, patients with temporal and skull base lesions were predominantly found in Clusters 1 and 3, which exhibited the most severe symptomatology. Conclusions and Relevance: This study establishes a basis for future longitudinal research aimed at understanding underlying pain mechanisms and evaluating the response to personalized pain management strategies in subtypes of patients with craniofacial FD. (Copyright © 2025 American Association of Oral and Maxillofacial Surgeons. All rights reserved.) |
| Contributed Indexing: | Investigator: H van der Heijden; E Golden; A Westbrook; N Shah; LA Drubach; S Voss; N Kwatra; L Romo; D Ebb; M Cay |
| Entry Date(s): | Date Created: 20241222 Date Completed: 20250429 Latest Revision: 20250429 |
| Update Code: | 20250429 |
| DOI: | 10.1016/j.joms.2024.12.001 |
| PMID: | 39710366 |
| Databáza: | MEDLINE |
Nájsť tento článok vo Web of Science | Abstrakt: | Background: Fibrous dysplasia/McCune-Albright syndrome (FD/MAS) is a genetic disorder, marked by bone lesions, often affecting the craniofacial skeleton. Pain is a prevalent yet heterogeneous symptom reported by patients with craniofacial FD. Effective treatments are currently lacking, posing a significant clinical challenge to patient care.<br />Purpose: This preliminary study examined pain profiles in craniofacial FD and aimed to identify subtypes of patients based on pain phenotypes and emotional health.<br />Study Design, Setting, Sample: A prospective, cross-sectional study involving 15 patients with FD/MAS, conducted at Boston Children's Hospital and Massachusetts General Brigham's Hospitals.<br />Predictor/exposure/independent Variable: Headache frequency, craniofacial pain severity, neuropathic pain quality, pain interference, allodynia, photophobia, depression, and anxiety were assessed using clinical questionnaires.<br />Main Outcome Variable(s): The primary outcome variable was the symptom profile derived from standardized clinical questionnaires and analyzed using principal component analysis and K-means clustering.<br />Covariates: Covariates included demographic data, diagnosis, and lesion location(s).<br />Analyses: Principal component analysis and K-means clustering of patient-reported measures of pain and emotional health were performed. Analysis of variance was conducted to determine significant differences among patient subtypes. Statistical significance was set at (P < .05).<br />Results: The study included 15 subjects with FD/MAS, with a mean age of 36.2 (13.9) years, including 1 male. Clustering analysis identified 3 subtypes of patients with distinct symptom profiles. Cluster 1 (n = 2) averaged 70 (28.3) headache days in a 90-day period, pain level of 7.5 (0.7) on a 0-10 scale, and severe anxiety, depression, allodynia, photophobia, and pain interference. Cluster 2 (n = 7) patients reported an average of 5.4 (7.5) headache days, an average pain level of 2.7 (2.6), mild or no anxiety, depression, allodynia, photophobia, and pain interference. Cluster 3 (n = 6) patients displayed a mixed symptom profile with an average of 47.3 (36.4) headache days and a pain level of 5.25 (1.4). Notably, patients with temporal and skull base lesions were predominantly found in Clusters 1 and 3, which exhibited the most severe symptomatology.<br />Conclusions and Relevance: This study establishes a basis for future longitudinal research aimed at understanding underlying pain mechanisms and evaluating the response to personalized pain management strategies in subtypes of patients with craniofacial FD.<br /> (Copyright © 2025 American Association of Oral and Maxillofacial Surgeons. All rights reserved.) |
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| ISSN: | 1531-5053 |
| DOI: | 10.1016/j.joms.2024.12.001 |