Jacob’s disease: report of a rare case and literature review

Abstract

Jacob’s disease is a rare condition consisting of pseudojoint formation between enlarged coronoid process and the inner surface of the zygoma. The authors report a 45-year-old female patient who presented with limited mouth opening (5 mm) and swelling of the left zygoma. No temporomandibular joint disease was diagnosed. 3D computed tomography images showed a mushroom-shaped coronoid growth with pseudojoint formation medial and superior to the zygomatic arch. Under general anaesthesia, coronoidectomy was carried out through a combined extraoral and intraoral approach. Histopathological examination revealed normal trabecular bone covered with hyaline cartilage. To the best of the authors’ knowledge this case is the fifth typical mushroom-shaped Jacob’s disease; only four similar cases have been reported. A review of the literature revealed only 39 histologically proved cases of Jacob’s disease cases up to 2010. Aggressive physical treatment was given to improve function and maximum mouth opening, which had increased to 40 mm at 3 months follow-up. The literature concerning this condition’s aetiology, pathogenesis, clinical characteristics, diagnosis, and treatment is reviewed.

Jacob’s disease or osteochondroma of the mandibular coronoid process, is a benign skeletal tumour that is rare in the oral and maxillofacial skeleton and even more rare in the coronoid process. Though first noted by Langenbeck in 1853 as an enlargement of the mandibular coronoid process, the first case report was described by J acob in 1899, who reported a pseudojoint between the coronoid and the zygomatic arch due to an osteochondroma formation of the coronoid process.

A review of the literature revealed only 39 histologically proved cases out of 50 reported cases ( Table 1 ). To the best of the authors’ knowledge this is the second case reported in India and it is the fifth typical mushroom-shaped Jacob’s disease; only four similar cases have been reported. The disease appears to involve males (63%) more often than females at a mean age of 30 years with an age range of 5–73 years. There is slight predilection for involvement of the left coronoid process. Initial signs and symptoms are a feeling of tightness in the joint region and gradual reduction in mouth opening. Later stages show a total trismus and appreciable swelling at the malar prominence. Pain is not a feature of the disease but deformity of the surrounding structures and facial deformity was found in most cases .

Table 1
Summary of the reported cases of osteochondroma of the coronoid process.
Case no Authors Year Gender Age Side Facial asymmetry Mouth opening in mm Surgical approach
1 S hackelford & B rown 1943 Male 15 Left Present 12.5 mm Extraoral
2 S hackelford & B rown 1943 Male 19 Right Present 10 mm Extraoral
3 S hackelford & B rown 1943 Male 38 Right Present Reduced Extraoral
4 B randt 1949 Female 37 Right Present 25 mm Extraoral
5 B railsford 1952 Male 29 Left Present
6 H olmes 1956 Male 36 Left 8 mm
7 L evine et al. 1957 Female 73 Left Present 6 mm
8 D ingman & N atvig 1957 Male 27 Right 8 mm Extraoral
9 A ntoni et al. 1958 Male 29 Left Present Adequate
10 P ap & F riedman 1958 Male 13 Left Present 10 mm Extraoral
11 L ebo 1961 Male 18 Left 14 mm Extraoral
12 M ohnac 1962 Male 18 Right 18 mm Extraoral
13 A llan & R eid 1967 Female 32 Right 16 mm Intraoral
14 A llan & R eid 1967 Male 22 Left Present 13 mm Intraoral
15 M eyer 1972 Female 10 Right None 20 mm Intraoral
16 J ames et al. 1974 Female 52 Right Present 5 mm Extraoral
17 C ooper & F inch 1974 Female 43 Left None 6 mm Intraoral
18 T akeda et al. 1975 Female 14 Left Present 10 mm Extraoral
19 S inger & S chultz 1976 Male 64 Right Present 5 mm Combined
20 R amon et al. 1977 Male 45 Right Present Reduced Combined
21 I to et al. 1981 Female 20 Left Present 3 mm Intraoral
22 I shii et al. 1983 Female 32 Left Present 7 mm Intraoral
23 I shii et al. 1983 Male 53 Right Present 20 mm Combined
24 R evington 1984 Male 24 Right None 9 mm Extraoral
25 T ucker et al. 1984 Male 16 Right Present 22 mm Intraoral
26 S chwartz & L iebel 1987 Male 15 Left Present 18 mm Extraoral
27 M acleod 1987 Female 46 Left Present 5 mm Intraoral
28 T otsuka et al. 1990 Female 37 Left Present 2 mm Intraoral
29 A sanami et al. 1990 Male 17 Left None 8 mm Combined
30 K erscher et al. 1993 Male 45 Left None 14 mm Intraoral
31 C onstantinides et al. 1997 Male 31 Right Present 10 mm Extraoral
32 C hen et al. 1998 Female 57 Left Present 14 mm Extraoral
33 M aganaro 1998 Female 26 Present
34 C hichareon et al. 1999 Male 5 Right Present
35 H ernandez et al. 2000 Male 22 Left Present 21 mm Extraoral
36 R oychoudhury et al. 2001 Male 32 Left Present 0 mm Extraoral
37 E scuder i de la T orre et al. 2001 Female 24 Bilateral Present Reduced Intraoral
38 E mekli 2002 Female 26 Right Present 10 mm Intraoral
39 E mekli 2002 Male 21 Right Present Reduced Extraoral
40 C apote 2005 Female 23 Present Reduced Intraoral
41 V illanueva 2006 Female 44 Left Present 30 mm Intraoral
42 A kan & M ehreliyeva 2006 Male 24 Bilateral None 15 mm Intraoral
43 D ede 2007 Male 20 Bilateral Present 6 mm Intraoral
44 F erro M artín 2008 Male 28 Bilateral Present 13 mm Intraoral
45 G opikiran 2009 Male 15 Bilateral Present 15 mm Intraoral
46 D’A mbrosio 2009 Male 39 Reduced
47 E toz O sman 2009 Female 43 Right Present 14 mm Intraoral
48 Z hong et al. 2009 Male 28 Left Present 13 mm Intraoral
49 W aseem 2010 Male 15 Right Present 10 mm Intraoral
50 A hmet et al. 2010 Male 16 Right Present 7 mm Extraoral
51 K rishna (present case) 2010 Female 45 Left Present 5 mm Combined
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Feb 5, 2018 | Posted by in Oral and Maxillofacial Surgery | Comments Off on Jacob’s disease: report of a rare case and literature review
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