Petrified Ear in Adrenal Insufficiency: Systematic Literature Review
Abstract
1. Introduction
2. Materials and Methods
2.1. Literature Search Strategy
2.2. Selection Criteria—Data Extraction
2.3. Reporting Thoroughness—Statistics
3. Results
3.1. Search Results
3.2. Findings
4. Discussion
5. Conclusions
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Conflicts of Interest
References
- Bochdalek, V.A. Verknöcherung der Aurecula. Prag. Vierteljahrschr. 1866, 89, 33–46. [Google Scholar]
- Wassmund. Verknöcherung der Ohrmuschel und Röntgographie. Dtsch. Med. Wochenschr. 1899, 25, 439–440. [Google Scholar] [CrossRef]
- High, W.A.; Larson, M.J.; Hoang, M.P. Idiopathic bilateral auricular ossificans: A case report and review of the literature. Arch. Pathol. Lab. Med. 2004, 128, 1432–1434. [Google Scholar] [CrossRef] [PubMed]
- Karrs, J.X.; Bass, J.; Karrs, T. Inflexible ears. JAMA Dermatol. 2016, 152, 335–336. [Google Scholar] [CrossRef]
- Valea, A.; Nistor, C.; Ciobica, M.L.; Sima, O.C.; Carsote, M. Endocrine petrified ear: Associated endocrine conditions in auricular calcification/ossification (a sample-focused analysis). Diagnostics 2024, 14, 1303. [Google Scholar] [CrossRef] [PubMed]
- Jarvis, J.L.; Jenkins, D.; Sosman, M.C.; Thorn, G.W. Röntgenologic observations in Addison’s disease; a review of 120 cases. Radiology 1954, 62, 16–29. [Google Scholar] [CrossRef] [PubMed]
- Nieman, L.K.; Chanco Turner, M.L. Addison’s disease. Clin. Dermatol. 2006, 24, 276–280. [Google Scholar] [CrossRef]
- Hahner, S.; Ross, R.J.; Arlt, W.; Bancos, I.; Burger-Stritt, S.; Torpy, D.J.; Husebye, E.S.; Quinkler, M. Adrenal insufficiency. Nat. Rev. Dis. Primers 2021, 7, 19. [Google Scholar] [CrossRef]
- Kumar, R.; Wassif, W.S. Adrenal insufficiency. J. Clin. Pathol. 2022, 75, 435–442. [Google Scholar] [CrossRef]
- Montoli, A.; Colussi, G.; Minetti, L. Hypercalcaemia in Addison’s disease: Calciotropic hormone profile and bone histology. J. Intern. Med. 1992, 232, 535–540. [Google Scholar] [CrossRef]
- Kottanattu, L.; Lava, S.A.G.; Helbling, R.; Simonetti, G.D.; Bianchetti, M.G.; Milani, G.P. Pancreatitis and cholecystitis in primary acute symptomatic Epstein-Barr virus infection—Systematic review of the literature. J. Clin. Virol. 2016, 82, 51–55. [Google Scholar] [CrossRef]
- Betti, C.; Lavagno, C.; Bianchetti, M.G.; Kottanattu, L.; Lava, S.A.G.; Schera, F.; Lacalamita, M.C.; Milani, G.P. Transient secondary pseudo-hypoaldosteronism in infants with urinary tract infections: Systematic literature review. Eur. J. Pediatr. 2024, 183, 4205–4214. [Google Scholar] [CrossRef]
- Papageorgiou, G.; Grant, S.W.; Takkenberg, J.J.M.; Mokhles, M.M. Statistical primer: How to deal with missing data in scientific research? Interact. Cardiovasc. Thorac. Surg. 2018, 27, 153–158. [Google Scholar] [CrossRef] [PubMed]
- Gaddis, G.M.; Gaddis, M.L. Introduction to biostatistics: Part 5, Statistical inference techniques for hypothesis testing with nonparametric data. Ann. Emerg. Med. 1990, 19, 1054–1059, Erratum in Ann. Emerg. Med. 1991, 20, 573. [Google Scholar] [CrossRef]
- Anschütz, F.; Dontenwill, W. Über Verkalkung des Ohrknorpels bei der Addisonschen Erkrankung [Calcification of the ear cartilage in Addison’s disease]. Ärztl Wochensch. 1955, 10, 136–137. [Google Scholar] [PubMed]
- Novick, W.H. Calcification of auricular cartilage in Addison’s disease. Arch. Otolaryngol. 1960, 72, 448–449. [Google Scholar] [CrossRef]
- McKusick, V.A.; Goodman, R.M. Pinnal calcification: Observations in systemic diseases not associated with disordered calcium metabolism. JAMA 1962, 179, 230–232. [Google Scholar] [CrossRef]
- Randall, R.E., Jr.; Spong, F.W. Calcification of the auricular cartilage in a patient with hypopituitarism. N. Engl. J. Med. 1963, 269, 1135–1137. [Google Scholar] [CrossRef]
- Gordon, D.L. Calcification of auricular cartilage. Arch. Intern. Med. 1964, 113, 23–27. [Google Scholar] [CrossRef]
- Bedsole, A. Calcified auricular cartilages in Addison’s disease. South. Med. J. 1966, 59, 1268–1270. [Google Scholar] [CrossRef]
- Siebenmann, R.E. Die Ohrknorpelverknöcherung beim Morbus Addison [The ossification of the ear cartilage in Addison’s disease]. Schweiz. Med. Wochenschr. 1977, 107, 468–474. [Google Scholar]
- Chadwick, J.M.; Downham, T.F., 2nd. Auricular calcification. Int. J. Dermatol. 1978, 17, 799–801. [Google Scholar] [CrossRef]
- Zillessen, E.; Gless, K.H.; Baldauf, G. Verknöcherung der Ohrmuschel bei partieller Hypophyseninsuffizienz mit selektivem ACTH- und STH-Mangel [Ossification of the auricle in partial pituitary failure with selective ACTH and STH deficiency (author’s transl)]. Dtsch. Med. Wochenschr. 1978, 103, 698–701. [Google Scholar] [CrossRef]
- Barkan, A.; Glantz, I. Calcification of auricular cartilages in patients with hypopituitarism. J. Clin. Endocrinol. Metab. 1982, 55, 354–357. [Google Scholar] [CrossRef]
- Gómez Huelgas, R.; Obeso Fernández, G.; Lacambra Calve, C.; Pereda García, J.M. Calcificación de los cartílagos auriculares en dos pacientes con enfermedad de Addison [Calcification of auricular cartilages in two patients with Addison’s disease]. Rev. Clin. Esp. 1988, 182, 118. [Google Scholar]
- Cohen, A.M.; Talmi, Y.P.; Floru, S.; Bar Ziv, J.; Zohar, Y.; Djaldetti, M. Ossification of the auricle in Addison’s disease. J. Laryngol. Otol. 1989, 103, 885–886. [Google Scholar] [CrossRef]
- Talmi, Y.P.; Cohen, A.M.; Bar-Ziv, J.; Finkelstein, Y.; Floru, S.; Zohar, Y. Ossified auricle in Addison’s disease. Ann. Otol. Rhinol. Laryngol. 1990, 99 Pt 1, 499–500. [Google Scholar] [CrossRef] [PubMed]
- Cohen, A.M.; Talmi, Y.P.; Floru, S.; Tsigelman, R.; Kalmanovitz, M.; Zohar, Y.; Djaldetti, M. X-ray microanalysis of ossified auricles in Addison’s disease. Calcif. Tissue Int. 1991, 48, 88–92. [Google Scholar] [CrossRef]
- Calvo Catalá, J.; Hortelano Martínez, E.; González-Cruz Cervellera, M.I.; Climent Díaz, B.; Ortega Gonzålez, E.; Valero Prieto, I. Calcificación de los cartílagos auriculares en un paciente con insuficiencia suprarrenal: Aportación de un caso y revisión de la literatura [Calcification of auricular cartilages in a patient with adrenal insufficiency: Presentation of a case and review of the literature]. Ann. Med. Interna. 1994, 11, 496–498. [Google Scholar]
- Paccalin, M.; Amoura, Z.; Wechsler, B.; Piette, J.C. Une calcification du cartilage auriculaire [Ear cartilage calcification]. Rev. Med. Interne 2000, 21, 295–296. [Google Scholar] [CrossRef] [PubMed]
- Wang, C.Y.; Chang, T.C.; Chen, F.W. Ossification of the auricles: A forgotten sign in adrenal insufficiency. J. Otolaryngol. 2002, 31, 52–54. [Google Scholar] [CrossRef]
- Agut Fuster, M.A.; Agulles Fornés, M.J.; Ferrer Rodríguez, A.; Ramos Martínez, M.J.; del Campo Biosca, J.; Viel Martínez, J.M. Calcification of auricular cartilages in adrenal insufficiency. Acta Otorrinolaringol. Esp. 2007, 58, 167–168. [Google Scholar] [CrossRef]
- Fukushima, K.; Sato, S.; Yamazaki, M.; Kaneko, A.; Oofusa, H.; Yahikozawa, H. Shock and stony hard ears. Lancet 2007, 369, 856. [Google Scholar] [CrossRef]
- Thomson, M.A.; Biggs, P.; Berth-Jones, J. Familial petrified ears associated with endocrinopathy. J. Am. Acad. Dermatol. 2007, 57 (Suppl. S5), S121–S122. [Google Scholar] [CrossRef] [PubMed]
- Machado, A.; Lopes, M.; Ferreira, C. Petrified auricular cartilages pointing the diagnosis of post-partum hypopituitarism in an encephalopathic patient. Eur. Arch. Otorhinolaryngol. 2009, 266, 305–307. [Google Scholar] [CrossRef]
- Mastronikolis, N.S.; Zampakis, P.; Kalogeropoulou, C.; Stathas, T.; Siabi, V.; Geropoulou, E.; Goumas, P.D. Bilateral ossification of the auricles: An unusual entity and review of the literature. Head Face Med. 2009, 5, 17. [Google Scholar] [CrossRef] [PubMed]
- Richter, J.C.; Chappuis, B. Ohren “aus Stein”—Ungewöhnlicher Begleitbefund aufgrund einer untersubstituierten Nebenniereninsuffizienz [Ears “made of stone”—Unusual secondary phenomenon of undersubstituted adrenal insufficiency]. Dtsch. Med. Wochenschr. 2009, 134, 1869–1871. [Google Scholar] [CrossRef]
- Lee, M.T.; Wang, C.Y. Auricular ossification in adrenal insufficiency. J. Endocrinol. Metab. 2011, 1, 45–46. [Google Scholar] [CrossRef]
- Uthoff, H.; Wiesli, P. Images in clinical medicine. Auricular cartilage calcification and adrenal insufficiency. N. Engl. J. Med. 2011, 364, 157. [Google Scholar]
- Gogate, Y.; Gangadhar, P.; Walia, R.R.; Bhansali, A. “Petrified ears” with idiopathic adult-onset pituitary insufficiency. Indian J. Endocrinol. Metab. 2012, 16, 830–832. [Google Scholar] [CrossRef]
- Koning, M.V.; Struijs, A. Adrenal insufficiency presenting as bilateral rigid auricles: A case report. J. Med. Case Rep. 2014, 8, 302. [Google Scholar] [CrossRef]
- Calderón-Komáromy, A.; Córdoba, S.; Tardío, J.C.; Borbujo, J. Osificación bilateral del cartílago auricular (bilateral ossification of the auricular cartilage). Actas Dermosifiliogr. 2015, 106, 433–435. [Google Scholar]
- Goswami, S.; Chakraborty, P.P.; Ray, S.; Ghosh, S.; Chowdhury, S. “Petrified ears” in secondary adrenal insufficiency. J. Clin. Diagn. Res. 2016, 10, OD23–OD24. [Google Scholar] [CrossRef]
- James, H.A.; Wermers, R.A. “Petrified ears”: Auricular ossification in adrenal insufficiency. Mayo Clin. Proc. 2016, 91, 1487–1488. [Google Scholar] [CrossRef]
- Taguchi, T.; Yoshida, M.; Terada, Y. Petrified ear auricles with isolated adrenocorticotropic hormone deficiency. Intern. Med. 2017, 56, 3263–3264. [Google Scholar] [CrossRef]
- Sedhai, Y.R.; Basnyat, S. Petrified pinna and pericarditis in autoimmune polyendocrine syndrome. BMJ Case Rep. 2019, 12, e229369. [Google Scholar] [CrossRef]
- Kannenberg, S.; Meyhöfer, S.; Lehnert, H.; Schmid, S.M. Petrifying: Ears as hard as stone in adrenal insufficiency. Lancet Diabetes Endocrinol. 2021, 9, 406. [Google Scholar] [CrossRef] [PubMed]
- Ozer Ozturk, E.; Karatas, E. Auricular calcification: Case report of petrified ears in a woman. Ann. Medical Res. 2021, 28, 621–623. [Google Scholar] [CrossRef]
- Recalcati, S.; Fantini, F. Petrified ears: A clue for adrenal insufficiency. Dermatopathology 2021, 8, 45–48. [Google Scholar] [CrossRef]
- Recalcati, S.; Vassallo, A.; Villanova, M.; Lanzi, R.; Losa, M.; Patricelli, M.G.; Carrera, P.; Fantini, F. Growth hormone deficiency and secondary adrenal insufficiency in petrified ear syndrome: A case report and literature review. Dermatol. Rep. 2025, 17, 10263. [Google Scholar] [CrossRef] [PubMed]
- Choo, K.S.; Yew, J.; Tan, E.J.H.; Puar, T.H.K. Case Report: Hypercalcemia as a manifestation of acute adrenal crisis precipitated by fluconazole use, and a review of the literature. Front. Endocrinol. 2023, 14, 1168797. [Google Scholar] [CrossRef]
- Batson, J.M. Calcification of the ear cartilage associated with the hypercalcemia of sarcoidosis. Report of a case. N. Engl. J. Med. 1961, 265, 876–877. [Google Scholar] [CrossRef] [PubMed]
- Riikonen, R.; Simell, O.; Jääskeläinen, J.; Rapola, J.; Perheentupa, J. Disturbed calcium and phosphate homeostasis during treatment with ACTH of infantile spasms. Arch. Dis. Child. 1986, 61, 671–676. [Google Scholar] [CrossRef]
- Wexler, L.; Brundage, B.; Crouse, J.; Detrano, R.; Fuster, V.; Maddahi, J.; Rumberger, J.; Stanford, W.; White, R.; Taubert, K. Coronary artery calcification: Pathophysiology, epidemiology, imaging methods, and clinical implications. A statement for health professionals from the American Heart Association. Writ. Group Circ. 1996, 94, 1175–1192. [Google Scholar] [CrossRef]
- Verghese, A.; Charlton, B.; Kassirer, J.P.; Ramsey, M.; Ioannidis, J.P. Inadequacies of physical examination as a cause of medical errors and adverse events: A collection of vignettes. Am. J. Med. 2015, 128, 1322–1324.e3. [Google Scholar] [CrossRef] [PubMed]
- Ende, J.; Fosnocht, K.M. Clinical examination: Still a tool for our times? Trans. Am. Clin. Climatol. Assoc. 2002, 113, 137–155. [Google Scholar] [PubMed]
Adrenal Insufficiency | p-Value | |||||
---|---|---|---|---|---|---|
All | Primary | Central | Unclassified | |||
N | 40 | 18 | 20 | 2 | ||
Demographics | ||||||
Males:females | 38:2 | 18:0 | 18:2 | 2:0 | 0.5385 | |
Age ∆, years | 56 (44–67) | 55 (46–70) | 49 (37–62) | 60, 70 | 0.1917 | |
Concomitant endocrine conditions | 16 (40) | 3 (17) | 11 (55) | 2 (100) | 0.0070 | |
Hypothyroidism | 11 (28) | 3 (17) | 7 (35) | 1 (50) | 0.2672 | |
Type 2 diabetes mellitus | 1 (2.5) | 0 | 0 | 1 (50) | >0.9999 | |
Hypogonadism | 5 (13) | 0 | 5 (25) | 0 | 0.0366 | |
Growth hormone deficiency | 3 (7.5) | 0 | 3 (15) | 0 | 0.0734 | |
Hyperprolactinemia | 3 (7.5) | 0 | 3 (15) | 0 | 0.0734 | |
Temporal relationship with adrenal disease diagnosis | 0.0381 | |||||
Calcification before by ≥2 years | 4 (10) | 2 (11) | 1 (5.0) | 1 (50) | ||
Calcification and adrenal disease concurrently | 19 (48) | 3 (17) | 16 (80) | 0 | ||
Calcification after by ≥2 years | 17 (43) | 13 (72) | 3 (15) | 1 (50) | ||
Phosphocalcemic factors | ||||||
Hypercalcemia | 1 (2.5) | 0 | 1 (5.0) | 0 | >0.9999 | |
Hyperphosphatemia | 1 (2.5) | 0 | 1 (5.0) | 0 | >0.9999 | |
Parathyroid hormone level increased | 0 | 0 | 0 | 0 | >0.9999 | |
Vitamin D level increased | 0 | 0 | 0 | 0 | >0.9999 |
Disclaimer/Publisher’s Note: The statements, opinions and data contained in all publications are solely those of the individual author(s) and contributor(s) and not of MDPI and/or the editor(s). MDPI and/or the editor(s) disclaim responsibility for any injury to people or property resulting from any ideas, methods, instructions or products referred to in the content. |
© 2025 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
Share and Cite
Jörg, E.; Milani, G.P.; Lava, S.A.G.; Bianchetti, M.G.; Bronz, G.; Faré, P.B.; Santi, M. Petrified Ear in Adrenal Insufficiency: Systematic Literature Review. J. Clin. Med. 2025, 14, 6870. https://doi.org/10.3390/jcm14196870
Jörg E, Milani GP, Lava SAG, Bianchetti MG, Bronz G, Faré PB, Santi M. Petrified Ear in Adrenal Insufficiency: Systematic Literature Review. Journal of Clinical Medicine. 2025; 14(19):6870. https://doi.org/10.3390/jcm14196870
Chicago/Turabian StyleJörg, Elisa, Gregorio P. Milani, Sebastiano A. G. Lava, Mario G. Bianchetti, Gabriel Bronz, Pietro B. Faré, and Maristella Santi. 2025. "Petrified Ear in Adrenal Insufficiency: Systematic Literature Review" Journal of Clinical Medicine 14, no. 19: 6870. https://doi.org/10.3390/jcm14196870
APA StyleJörg, E., Milani, G. P., Lava, S. A. G., Bianchetti, M. G., Bronz, G., Faré, P. B., & Santi, M. (2025). Petrified Ear in Adrenal Insufficiency: Systematic Literature Review. Journal of Clinical Medicine, 14(19), 6870. https://doi.org/10.3390/jcm14196870