Recurrence of Infantile Cortical Hyperostosis
Bibliographic record
Abstract
OBJECTIVE: Also named Caffey disease, infantile cortical hyperostosis is a rare disease that usually affects children of a few weeks of age. The clinical picture is that of irritability, soft tissue swelling at various sites (mandible, clavicle, limbs) with local warmth, and pain on palpation. Radiographs demonstrate an important cortical thickening of the affected bony structures. There is generally spontaneous resolution of the inflammatory signs within the first few months or years of life, without future recurrence. We report the case of a recurrence in the adolescent period and conducted a thorough review of the literature to confirm this rare possibility. METHODS: We report the case of a 12-year-old female patient, presenting symptoms of pain and swelling of both forearms and legs, similar to her initial clinical picture at 1 month of age when the diagnosis of Caffey disease was made. Genetic analysis confirmed the COL1A1 mutation, the classic mutation described in the familial form of this disease. Radiologic investigation revealed new periosteal bone formation of the right and left ulnae and the left fibula, suggesting a recurrence of cortical hyperostosis. A thorough review of the literature was conducted, using the Medline database between 1948 and 2011, to confirm this hypothesis. RESULTS: The literature confirms the possibility but also the rarity of a recurrence of cortical hyperostosis in an adolescent who was diagnosed with Caffey disease in infancy. We have identified less than a dozen cases in the literature describing such a recurrence and in the majority there are no medical photographs or radiographs to support the clinical diagnosis. Our case is well documented both clinically and radiologically regarding the initial presentation and the recurrence in adolescence. CONCLUSIONS: Although very rare and poorly recognized, the diagnosis of recurrent cortical hyperostosis must be evoked in a patient who suffered from Caffey disease in infancy, and presents similar clinical characteristics in the adolescent period. LEVEL OF EVIDENCE: Level V--case report.
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How this classification was reachedexpand
Full frame machine prediction
Teacher imitationNot calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.
Distilled classifier scores by category (both heads)
| Category | Codex | Gemma |
|---|---|---|
| Metaresearch | 0.000 | 0.004 |
| Meta-epidemiology (narrow) | 0.000 | 0.000 |
| Meta-epidemiology (broad) | 0.001 | 0.000 |
| Bibliometrics | 0.002 | 0.001 |
| Science and technology studies | 0.000 | 0.001 |
| Scholarly communication | 0.001 | 0.001 |
| Open science | 0.001 | 0.000 |
| Research integrity | 0.001 | 0.001 |
| Insufficient payload (model declined to judge) | 0.001 | 0.000 |
Machine scores (provisional)
The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.
Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.
score_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from itClassification
machine, unvalidatedMachine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.
How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".